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Europ. J. Obstet. Gynec. reprod. Biol.

, 1980,10/S, 415-463 415


0 Elsevier/North-Holland Biomedical Press

REVIEW ARTICLE

CHRONIC PELVIC PAIN WITHOUT OBVIOUS PATHOLOGY IN WOMEN


Personal observations and a review of the problem

M. RENAER
(in collaboration with P. Nijs, A. Van Assche and H. Vertommen)
Departments of Obstetrics and Gynecology and Psychology, Catholic University of
Leuven, Belgium

Accepted for publication 23 January 1980

CONTENTS

A. INTRODUCTION 416

B. CHRONIC PELVIC PAIN WITHOUT OBVIOUS PATHOLOGY (CPPWOP) 418


I. The syndrome as it is described in the literature 418
II. Personal observations 419
1. Material and methods 419
2. Results 421

C. PATHOGENESIS OF CHRONIC PELVIC PAIN WITHOUT OBVIOUS


PATHOLOGY 423
I. Do many cases with CPPWOP coincide with the Allen and Masters syn-
drome? 423
1. A critical perusal of the literature 423
2. Our own observations 424
3. Conclusion 425
II. To what extent is CPPWOP due to circulatory disturbances? 425
1. Active or passive hyperemia? 425
2. The search for pelvic circulatory disturbances by phlebography and
laparoscopy 426
III. Attempts at a histopathological explanation of the pain 433
1. A short summary of the literature 433
2. Personal investigations 437
3. Discussion 439
IV. The psychological characteristics of women with CPPWOP 441
1. A review of recent publications 441
2. Personal investigations 445
V. Pathogenesis of CPPWOP - Conclusion 454
1. General considerations 454
2. A working hypothesis 455

D. FOLLOW-UP OF PATIENTS WITH CPPWOP 456

E. THERAPEUTIC STRATEGY 457


416

A. INTRODUCTION

In many medical specialities one encounters one or more clinical syn-


dromes, whose name is a matter of dispute, whose pathogenesis is ill-known
and whose treatment is less than satisfactory. The cardiologists have, for
example, their ‘neurocirculatory asthenia’, the gastroenterologists have the
‘irritable bowel syndrome’, and gynecologists who are sufficiently interested
in chronic pain note a number of patients who complain of chronic lower
abdominal pain, apparently of gynecological origin, for which they do not
find a definite lesion or cause. Depending on the author’s view as to the
pathogenesis, this clinical syndrome has received a variety of names, denot-
ing the divergent opinions of the authors on its pathogenesis. During recent
years it has been called the ‘syndrome of pelvic congestion and fibrosis’
by Taylor (1949a--c), ‘Pelipathia vegetativa’ by Gauss (1949), ‘pelvic
sympathetic syndrome’ by Theobald (1951), and ‘pelvic neurodystonia’
by Schockaert (1956).
On the other hand, several authors deny the existence of this syndrome.
Atlee (1966) discards it as “diagnostic garbage”. Novak et al. (1975) hardly
believe that there is such a thing as the “so-called generalized pelvic conges-
tion phenomenon”. Jeffcoate (1975) admits that there are cases of pelvic
congestion syndrome. What he calls ‘congestive dysmenorrhea’ is a periodi-
cal exacerbation of a constant discomfort; it takes the form of a diffuse, dull
ache in the pelvis, and is thought to be the result of increasing tension in the
pelvic tissues associated with premenstrual engorgement. But he does not
believe in the condition which has, since Young (1938), been called ‘broad
ligament neuritis’. “This term, like pelvic sympathetic syndrome, is some-
times used to denote pelvic pain for which there is not a proved basis of
infection. The trouble in these cases is psychological” (Jeffcoate, 1975). He
also does not believe in the condition which, since Martius (1939), has been
called ‘spastic posterior parametritis’.
If there is such a syndrome, it is to be supposed that few will quarrel with
the name ‘chronic pelvic pain without obvious pathology’. But, before
admitting the existence of such a syndrome in its own right, one will have to
show (i) that the patient’s pain is not due to a non-gynecological cause; (ii)
that the pain has characteristics of a pain of gynecological origin; (iii) that
the syndrome is not due to one of the acknowledged causes of gynecological
pain; (iv) that no definite recognized lesion or cause can be found.
As the very existence of this syndrome is called in question, these 4 condi-
tions are well worth emphasizing.

1. Non-gynecological causes of chronic lower abdominal pain and backache


Before accepting the diagnosis of chronic pelvic pain without obvious
pathology (CPPWOP), it is necessary to discard, by the appropriate diagnos-
tic means, non-gynecological causes of pain. Diseases or dysfunctions which
are most apt to be confused with chronic pain provoking causes of gynecol-
ogic origin are: gastroenterologic disorders such as irritable bowel syndrome,
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diverticulitis and, less frequently, Crohn’s disease and cancer of the sigmoid
colon; orthopedic disorders such as the hyperlordosis syndrome of the
lumbar spine, or instability or spondylarthrosis of the lumbar area; urologic
diseases such as chronic or recurrent cystitis, ureteral stones, ptosis of a
kidney with obstruction of the urinary flow. Neurological diseases will only
rarely create diagnostic problems. Psychopathological disturbances should
not be omitted, for they can enhance the symptomatology or prolong the
duration of gynecological disorders; they can, moreover, give rise to chronic
lower abdominal pain complaints by purely psychogenic mechanisms.

2. Does the pain present characteristics of a pain of gynecological origin?


We will not dwell here on those characteristics whose importance as an aid
to diagnosis we have stressed in earlier publications (Renaer, 1971, 1973).

3. Are there no organic lesions of the genital organs which could explain the
pain ?
The absence of organic lesions like, for example, endometriosis, chronic
pelvic inflammatory disease or sequelae of acute PID, adenomyosis, has to be
demonstrated by careful clinical and laparoscopic, and sometimes by
hysterographic, investigations.

4. Other possible causes


To better circumscribe the concept of CPPWOP, it is useful to mention
other possible causes of gynecologic pain which should be discarded in a
clinical evaluation of CPPWOP, in order to make the series of cases studied
as homogeneous as possible.
Although a mobile uterine retroversion which causes pain is admittedly
rare, we believe that it exists. Proof of the causal relationship between the
pain and the mobile retroversion relies on the disappearance of the lower
abdominal and/or sacrogluteal pain within one or two days after the replace-
ment and maintenance of the uterus with a Smith or Hodge pessary. As there
is no general agreement on the pathogenesis of the pain in the rare cases
which are painful, we could classify this condition as CPPWOP. It was, how-
ever, considered preferable not to include those cases in this study as the
condition is, at the moment, recognized as a possible cause of chronic pelvic
pain. We also do not include cases where the pain is apparently due to a
painful scar in one of the lateral vaginal fornices after a deep cervical tear.
We believe, as do McClure Browne (1973), Novak et al. (1975) and
Jeffcoate (1975), that, although chronic cervicitis is not painful by itself it
can, in some instances, provoke spontaneous pelvic pain mostly accompanied
by deep dyspareunia, when the inflammatory process has given rise to
chronic cellulitis or posterior parametritis (Jeffcoate), or pelvic lymphangitis
(Novak), or “a tracking back of the infection along the sacrouterine liga-
ments” (McClure Browne). Although the pathological data on this condition
are scant, it seems reasonable to admit its existence when a woman starts
complaining of lower abdominal pain right after a delivery and presents, on
418

gynecological examination, a more or less severely tom cervix, agglutinated


leucocytes in the cervical mucus and one or two sensitive sacrouterine liga-
ments. The diagnosis (posterior parametritis due to a chronic cervicitis)
becomes all the more plausible if both the spontaneous and the provoked
pain disappear a short time after treatment with antibiotics and the use of
hot sitzbaths. Such cases have also been eliminated.
Contrary to what is said in some publications (Josimovich, 1975), it is
felt that the premenstrual tension syndrome should not be regarded as an
example of CPPWOP; it is, indeed, characterized by a great diversity of
symptoms, but the patients mostly complain of abdominal bloating and dis-
comfort rather than of lower abdominal pain.
We believe that in gynecological practice one encounters a limited number
of patients who fulfil the 4 above-mentioned conditions. It is the purpose of
this paper to study in a series of patients the clinical symptoms, the patho-
genesis and the treatment of this syndrome, and to give a review of the more
recent literature.

B. CHRONIC PELVIC PAIN WITHOUT OBVIOUS PATHOLOGY

I. The syndrome as it is described in the literature

Before describing our own clinical observations, we recall the descriptions


of the symptomatology as they are found in the literature.
The most important symptom is lower abdominal pain and, less fre-
quently, low back pain. The lower abdominal pain may be localized either in
the whole lower abdomen or in both iliac fossae, or in one iliac fossa only.
The low back pain may be felt over the whole breadth of the upper sacro-
gluteal zone or over one gluteal zone only (Renaer, 1973). The pain is
generally more severe for several days before the menstrual period and abates
on the first or second day of menstruation, although not infrequently the
pain is felt continuously. The pain also increases when standing upright for
some time and when the patient runs or jumps or sits down abruptly. It is
alleviated by horizontal rest. Dyspareunia on deep penetration is very
common.
Besides the pain symptoms, other complaints such as leucorrhea, men-
strual disturbances, constipation, cystalgia, asthenia, depression or anxiety
are mentioned with varying frequency, depending on the authors.
Most patients complaining of the syndrome are married, and the symp-
toms frequently start a short time after a delivery. The age of the patients is
generally between 20 and 40 years; the syndrome is seldom seen in the pre-
menopause and never after menopause.
On gynecological examination, the uterus and adnexa may be tender;
there is very frequently marked tenderness of the posterior parametrium.
Examination of the cervix, it is said, may show either cervicitis or leucorrhea
or a normal cervix. As stated above, we prefer to exclude the cases with
419

cervicitis and tenderness of the posterior parametrium *, as we classify them


as probable “posterior parametritis due to cervicitis”. When a chronic pain
syndrome has lasted several months and medical treatment has not relieved
it, it is indicated to perform a laparoscopy in order to look for non-palpable
lesions such as endometriosis or sequelae of pelvic inflammatory disease
which might cause the pain. In chronic pelvic pain cases without obvious
pathology, those lesions are not found, by definition. We will later dwell on
some conditions which may be found at laparoscopy and which may or may
not explain the pain.
Among the patients with CPPWOP, one can distinguish two varieties or
clinical forms. The symptoms of the first are suggestive of circulatory
disturbances; in the second there is essentially a marked tenderness of the
posterior parametrium.
The first one seems to be the least frequent. The pain has a symmetrical
localization in the lower abdomen; it is especially severe during the premen-
strual period and tends to disappear on the first day of menstruation. The
uterus and the adnexa are tender on palpation, and there may or may not be
tenderness of the sacrouterine ligaments or the posterior parametrium. Some
authors stress that inspection of the cervix reveals a cyanotic external
cervical mucosa. Some patients present the same pain syndrome during
several days in the paraovulatory period; in these instances the syndrome
could also be considered as a prolonged episode of Mittehchmerz.
In the second form the pain may have either a symmetrical or a unilateral
localization; it does not always disappear on the first day of menstruation
and, on gynecological examination, there is always tenderness of the poste-
rior parametrium. One frequently finds painful anteroposterior condensa-
tions or densifications in the posterior parametrium, and sometimes the
posterior parametrium is shortened, so that the cervix is found in retro-posi-
tion. When pressure or traction is exerted on the sacrouterine ligaments this
maneuvre generally reproduces the spontaneous pain and the deep dys-
pareunia .

II. Personal 0 bserva tions

The first part of this contribution describes in detail a series of patients


whose symptoms and signs fulfil the 4 conditions cited in the Introduction.

1. Material and methods


We have studied two series of consecutive cases of chronic pelvic pain
without obvious pathology. By ‘consecutive’ is meant that all patients were
included who were admitted into the clinic for extensive investigation. The
first subseries comprised 15 patients, the second, 16; i.e., a total of 31. All

* We make a diagnosis of cervicitis when, after cleansing the cervix with a swab, we find
purulent mucus in the cervical canal or we find, in the paraovulatory period, many
agglutinated ieucocytes in the endocervical mucus.
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patients had been complaining for at least 6 months, but most of them for
several years, of lower abdominal pain with or without low back pain.
The first series of patients were examined between June 1971 and
December 1973. During this period 200 laparoscopies were performed in the
Department of Obstetrics and Gynecology, 108 of which were done on
patients with chronic lower abdominal pain syndromes. Most of those
patients had been examined several times as outpatients. They were admitted
into the clinic either because the diagnosis was unclear or because of the
poor results of medical treatment. Disturbances of the gastrointestinal tract
were looked for either by a G.I.-series and a barium enema, or by a barium
enema alone. Almost all patients had an intravenous urography. All under-
went a laparoscopy.
When the complaints had characteristics of a pain of orthopedic origin,
the opinion was asked of an orthopedic surgeon, who generally ordered a
radiological examination of the lumbosacral spine. In all cases in which the
complaints could not be explained by an organic cause, the opinion was
requested of a psychiatrist (P. Nijs), who is a senior staff member of the
department and who is in charge of the patients with psychosomatic prob-
lems. The psychological characteristics of the patients with CPPWOP are
examined in detail in a separate chapter. The final diagnosis in the 108
chronic pain cases is given in Table I.
Out of the 28 cases of this first series who were considered as CPPWOP
we retained, for analysis of the pain syndrome, 15 cases in whom any other
diagnosis could be discarded with a high probability.
In the second series there were 16 more cases of chronic pelvic pain with-
out obvious pathology. They were part of a consecutive series of 54 patients
who were investigated for chronic pelvic pain in 1975 and 1976. Besides
these 54 patients, there were 15 control patients, who did not complain of
pain but who underwent a laparoscopy for infertility. The 54 cases of the
second series were evaluated along the same lines as those of the first series,
but, in order to study various factors which are considered important in the
pathogenesis of the syndrome, we took advantage of the examination under
anesthesia and of the laparoscopy:
- to compare the condition of the sacrouterine ligaments and the posterior
parametrium before the general anesthesia and under anesthesia;
- to observe the frequency of a ‘mottled’ appearance of the uterus;
- to look for the presence or absence of varicosities in the infundibulopelvic
ligaments and along the lower part of the broad ligaments;

TABLE I Diagnosis in 108 consecutive patients complaining of chronic pain

CPPWOP 28
Chronic PID (or sequels of acute salpingitis) 23
Endometriosis 22
Various pain syndromes 35
421

- to measure the amount of peritoneal fluid in the pelvis, and to perform a


cytological examination of the fluid;
- to observe the presence or absence of tears in the sacrouterine ligaments
or in the posterior leaf of the broad ligaments, as described in the Allen and
Masters syndrome.
In the 15 control cases, the same protocol was followed.

2. Results
(a) Some characteristics of the patients with CPPWOP. Twenty-six of
the 31 patients were between 20 and 40 years of age; 5 were more than 40
years old; no patient was postmenopausal; 30 patients were married; 25 of
the 31 had one or several children. Most of the patients were workers or
wives of workers; a few were lower-middle class.
(b) Clinical history. All patients complained of chronic lower abdomi-
nal pain with or without low back pain. The low back pain, if any, was
always localized at the sacrogluteal level, the zone where backache of
gynecological origin is nearly always felt (Renaer, 1973). The localization of
the pain is given in Table II.
The frequency of deep dyspareunia was 17 times in 31 patients. Other
complaints, such as frequency of micturition, urgency of micturition and
leucorrhea, which are described as frequent symptoms by several authors,
did not strike us as particularly common. It should be noted, however, that
we are only mentioning symptoms the patient was complaining of sponta-
neously. In 12 cases out of 31 there were several complaints, including one
or more which are generally termed ‘functional’, such as headache, asthenia,
palpitations, nausea, epigastric discomfort, irritability, depression. These
patients may be called ‘polysymptomatic’; most oligosymptomatic patients
were merely complaining of spontaneous pain and deep dyspareunia.
(c) Clinical examination. On gynecological examination we seldom
found a striking tenderness of the uterus or the adnexa. Palpation almost
never revealed an oversized and tender uterus, as has been described in some
papers. The volume of the cervix was seldom augmented, and it only rarely
had a cyanotic appearance. Unilateral or bilateral tenderness of the sacro-
uterine ligaments or the posterior parametrium was, however, found fre-
quently. In a few cases this tenderness was accompanied by shortening of the

TABLE II Localization of the pain in 31 patients with CPPWOP

Lower abdominal 13
Lower abdominal and sacrogluteal 7
Left iliac fossa 5
Left iliac fossa and left sacrogluteal zone 1
Right iliac fossa 4
Right iliac fossa and right sacrogluteal zone 1
Total 31
422

TABLE III Tenderness of the sacrouterine (SU) ligaments or of the posterior para-
metrium

Bilateral tenderness 18
Unilateral, right 1
Unilateral, left 2
Tender and shortened SU ligaments 5
Ligaments not tender 5
Total 31

posterior parametrium and densifications in the anteroposterior direction.


The absence of endometriotic lesions had been verified by laparoscopy; the
tenderness of the sacrouterine ligaments did not seem to be due to an actual
lymphatic propagation of a cervical infection, as any cervical infection had
been excluded by microscopic examination of the cervical mucus. It was not
due to scar tissue either, because the cases in which there was a cervical
laceration with extension in a fornix had been eliminated. Table III shows
the frequency of tenderness of the sacrouterine ligaments or the posterior
parametrium.
Tenderness of the lower abdominal wall was never found on elective pal-
pation of the abdominal wall; the lower abdominal pain could therefore not
be attributed to the abdominal wall itself.
(d) Laparoscopic examination. All patients had a laparoscopy, but in
the 16 patients of the second series more detailed observations were made.
Table IV gives the laparoscopic observations in the 31 patients with
CPPWOP.
We will return to the volume of peritoneal fluid and to the pelvic vari-
cosities in the chapter on the pathogenesis of the syndrome.

TABLE IV Laparoscopic observations in 31 patients with CPPWOP

1st series (15 cases)


Laparoscopy negative 13
Laparoscopy negative; mottled uterus 1
Laparoscopy negative; tear or depression in the posterior leaf of a broad ligament 1
2nd series (16 patients)
Laparoscopy negative 6
Laparoscopy negative ; varicose veins in the pelvis 8*
Laparoscopy negative; tear or depression in posterior leaf of a broad ligament 2

* In one case with pelvic varicosities a mottled uterus was found.


423

C. PATHOGENESIS OF CHRONIC PELVIC PAIN WITHOUT OBVIOUS PATHOLOGY

I. Do many cases with CPPWOP coincide with the Allen and Masters syn-
drome?

1. A critical perusal of the literature


When looking at the ‘presenting complaints’ in Allen and Masters’ paper
(1955) on ‘Traumatic laceration of uterine support’, one is struck by the fact
that the majority of the patients are polysymptomatic, and present symp-
toms which are frequently of a functional nature. Table V shows the pelvic
pathology found in the 28 cases of Allen and Masters (1955).
In the description of the 28 cases of third-degree retroversion, it is noted
“that the volume of the uterus was usually increased, that the uterus was
soft, that it had the purplish-red cast so frequently noted in hemostasis, and
that it shrunk in size, when it was brought forward”. The various procedures
performed in Allen and Masters’ paper on the same patients make deductions
as to the reasons for the good results of the operation difficult. In at least
23 cases the positive result could be explained by the uterine suspension
operation as well as by repair of the laceration.
True and Musset (1973) discuss the syndrome and give an extensive
review of the literature. The majority of the papers they cite are, however,
of French origin, as the syndrome seems to be rather seldom diagnosed in
Germany and the United States, and very rarely in Great Britain. A few
points are well worth mentioning. Even in France there is a great differ-
ence in the frequency with which the syndrome is found or diagnosed.
Serment (1972) has operated 132 cases in a few years and Keller et al.
(1972), 108, whereas others rarely see the syndrome. Palmer (1973) found it
3 times in 400 laparoscopies performed for pain. True and Musset (1973),
after a diligent search for 12 years, found only 10 cases, 4 of whom they
operated. This divergent frequency may, in part, be due to the fact that the
name ‘Allen and Masters syndrome’ seems to be given to different pathologi-
cal lesions or conditions. The drawings in the paper by Allen and Masters
(1955) show a tear of the posterior leaf of a broad ligament and one in a
broad ligament and a sacrouterine ligament, but in publications by others the
lesion is not really a tear but a depression of the posterior leaf of a broad

TABLE V Pelvic pathology in the 28 cases of Allen and Masters (1955)

Pathology demonstrated Total cases with positive report

1, Third degree retroversion of the uterus 28


2. Serous fluid in the pelvis (30-80 ml) 27
3. Bilateral broad ligament laceration 22
4. Unilateral broad ligament laceration 6
5. Unilateral sacrouterine ligament laceration 5
6. Bilateral sacrouterine ligament laceration 1
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ligament, and in the depth of this depression varicose veins are said to be
found frequently. Sometimes the name is given to several small grouped
depressions.
True and Musset, and we ourselves, have found the described depressions
in patients who consulted either for sterility or for another reason, but not
for pain. True and Musset (1973, p. 233) grouped excerpts from 31 papers in
a table to show that in some papers a uterine retroversion was present in 80%
and in others in up to 100% of the cases, and that in a majority of them the
retroversion was accompanied by pelvic varicosities. As in the Allen and
Masters paper, surgical treatment in the majority of the papers consisted not
only in suturing the rent or the depression but also in replacing the retrover-
sion, so that one is at a loss to know what made the pain disappear, if it dis-
appeared.

2. Our own observations


As stated above, the presence of tears or depressions in the posterior
leaves of the broad ligament or of the sacrouterine ligaments was looked for
during laparoscopy in 69 patients; 54 were complaining of chronic pelvic
pain, 15 were controls.
The final diagnoses for the 54 pain patients were: CPPWOP, 16; endo-
metriosis, 19; sequels of acute salpingitis, 7; cause of pain not clear, 12.
In none of the 54 cases was a real tear of the sacrouterine ligaments
found. In 5 cases a depression was found of the peritoneum of the posterior
leaf of a broad ligament, but, owing to the regular edges of these depressions,
one wonders whether they were due to tears in the peritoneal covering or to
slight congenital structural defects. Two depressions out of 5 were found in
the 19 patients in whom the chronic pain could be explained by the presence
of endometriotic lesions, 2 were found in the 16 patients in whom no
obvious pathology was discovered, and 1 in a patient whose pain had appar-
ently no gynecologic but a gastroenterologic origin. In the 15 control
patients, who did not complain of pain, we found one depression of a sacro-
uterine ligament: this means that one sacrouterine ligament was divided into
two parts with, in between, an anteroposterior depression.
Following are some details about the two patients with CPPWOP who
presented a depression. Mrs. De Cr. had been complaining for 15 months of
lower abdominal pain, which was most pronounced in the right iliac fossa.
Her right adnexum was tender on palpation and the right sacrouterine liga-
ment even more so; at laparoscopy, 4 tiny depressions were found on the
posterior leaf of the left broad ligament. Mrs. Sa. had, since the birth of her
child, been complaining periodically of pain in the lower abdomen but
especially in the right iliac fossa, with exacerbations during the first two days
of menstruation. The uterus and adnexa were normal on palpation, the
posterior parametrium was tender, not shortened, and no denser parts were
palpated. At laparoscopy, varicosities were found in the left infundibulo-
pelvic ligament and two depressions in the posterior leaf of the left broad
ligament. According to the literature those tears or depressions almost
425

always originate during a delivery. Although the last delivery of Mrs. De Cr.
had occurred 14 years before, her pain had only begun 15 months before.
Mrs. Sa.‘s pain began right after her only delivery, but it had appeared
periodically and could therefore hardly be ascribed to a depression, i.e. a
structural lesion.

3. Conclusion
Neither a study of the literature nor our own observations could convince
us of a causal relationship between pelvic pain and a depression in a posterior
leaf of a broad ligament or in the peritoneal covering of a sacrouterine liga-
ment. It is not certain that those depressions are due to tears resulting from a
traumatic delivery: indeed, amongst 108 cases described by Keller et al.
(1972), 11 were found in nulliparae.
A real tear in a sacrouterine ligament may perhaps give rise to pain, but
then, in more than 1000 laparoscopies, several hundred of which were
performed for chronic lower abdominal pain, we only once saw what may
have been a tear in a sacrouterine ligament.
Therefore, in our opinion, the role of the so-called Allen and Masters
syndrome is utterly negligible in the explanation of chronic pelvic pain with-
out obvious pathology. Also, one wonders whether the symptomatology in
most of the operated cases described in the literature was not essentially that
of a painful mobile retroversion.

II. To what extent is CPPWOP due to circulatory disturbances?

Many gynecologists, relying on clinical examination of the uterus and the


cervix, on observations at laparoscopy or laparotomy or on phlebographic
evidence, admit that circulatory disturbances, essentially in the form of
passive congestion, are an important pathogenetic factor in chronic pelvic
pain. There are, indeed, several reasons to admit that circulatory factors or
anomalies can provoke pain in the internal genital organs.

1. Active or passive hyperemia?


It is well known that the first modification to be observed in the uterine
wall of castrated female animals after the administration of estrogens is an
increase in blood flow and a retention of water (Reynolds, 1949; Greiss and
Anderson, 1970; Makowski, 1977).
The lower abdominal pain some postmenopausal women experience a few
days after starting an estrogen treatment can probably be explained by an
increased tension in the uterine wall due to a sharp increase in blood flow.
The same mechanism could also explain some cases of prolonged Mittel-
schmerz, whose duration seems to coincide with the paraovulatory estrogen
peak, unless it is due to an increased basal tone of the uterine contractions
during this period (Hein et al., 1972). One should immediately add that
increased blood flow cannot be the sole explanation, as this sort of pain is
only rarely seen in the beginning of pregnancy, where we must assume there
426

is an important rise in the myometrial blood flow (Makowski, 1977).


Several authors (Cotte, 1949; Taylor, 1949a-c; Jeffcoate, 1975) believe
that in some women lower abdominal pain may be due to an increased
intramural pressure caused by passive congestion during the premenstrual
period. The French authors call this ‘pelvic congestion’, whereas the English
call it ‘congestive dysmenorrhea’. Taylor (1949 a-c, 1961) ascribes the
lower abdominal pain of many cases of CPPWOP to a phenomenon of pelvic
congestion, in whose natural history he distinguishes “an early congestive
phase leading to a later one of fibrous tissue proliferation, due to prolonged
vascular stasis”. On page 218 of Taylor’s paper (1949a), a specimen is
shown of a hypertrophic uterus which is said to be the result of prolonged
vascular stasis. It may be mentioned, however, that, at the time we were
still treating many such patients by hysterectomy, most of the excised uteri
were not hypertrophic. Moreover, uterine hypertrophy is not frequently
seen at laparoscopies performed for CPPWOP.
Passive or venous congestion is, in our opinion, the most probable cause of
pain in the rare cases of painful mobile retroversion. In those cases the size
of the uterus is frequently increased; the uterus is soft and tender on palpa-
tion and it becomes more firm and less tender, with disappearance of the
spontaneous pain, within 2 or 3 days of replacement of the retroversion. It is
important to note, however, that it is generally the uterine corpus rather
than the posterior parametrium which is tender in those cases, whereas the
reverse is observed in most cases of CPPWOP where there is no uterine
retroversion. Another factor which seems to plead for a ‘circulatory’
explanation is the fact that the syndrome is almost never seen after meno-
pause.
As long as we cannot perform sufficiently exact measurements of the
uterine blood flow, we will not be able to prove whether circulatory distur-
bances play a role in the pathogenesis of some or all cases of CPPWOP. At
the moment, we can only approach the problem by indirect methods.
Several authors have used uterine phlebography to study the venous circu-
lation of the myometrium and of the pelvis. We have not used this technique,
as we are not sure of being able to obtain reproducible results, and as it is
difficult to study control cases, i.e. patients without pain, by phlebography.
We therefore studied the pelvic veins by laparoscopy.
In many papers on chronic pelvic pain it is stated that one generally finds
rather great amounts of straw-colored fluid in the pelvis at laparotomy or
laparoscopy. It is implicitly thought that this fluid is a transudate from the
internal genital organs, and that the amount of transudate may be an indirect
measure of the degree of congestion in the pelvic organs.

2. The search for pelvic circulatory disturbances by phlebography and


laparoscopy
(a) A look at the literature. An association between the pelvic conges-
tion syndrome and pelvic varicosities has been assumed by many authors
(Allen and Masters, 1955; Chidekel and Edlundh, 1968; Allen, 1971). Pelvic
421

veins and varicosities have been studied by various venographic methods:


-the transuterine route (Guilhem and Baux, 1954; Topolanski-Sierra, 1958;
Wegryn and Harron, 1960; Hughes and Curtis, 1962; Heinen and Schiissler,
1963; PLli et al., 1963; Giraud and Lasdgue, 1964; Hammen, 1965; Ver-
meersch et al., 1971). When using this route, the contrast medium should
preferably be injected into the myometrium by means of 2 (Hammen) or 4
(Heinen and Schiissler) cannulae. Indeed, injecting with one single cannula
often results in asymmetric contrast-filling of the visceral pelvic veins;
-retrograde injection of the left ovarian vein by selective cannulation of
the left renal vein (Chidekel and Edlundh, 1968);
-the vulvar route, by cannulation of vulvar veins or vulvar varicosities
(Craig and Hobbs, 1974);
-by introducing polythene catheters into each of the femoral veins and
injecting the pelvic veins in a retrograde direction after total compression
of the inferior vena cava.
Helander and Lindbom (1960) studied the pelvic veins using the last
method, having totally compressed the inferior vena cava. Their study was
performed on 300 patients, the majority of whom had cancer of the cervix.
In 17 cases they found a large venous communication between the utero-
vaginal plexus and the ovarian veins. As this connection had a diameter of
more than 0.5 cm, these 17 cases were considered as examples of varicocele
of the broad ligament. The authors do not give the symptomatology of the
patients in detail, but they state that 8 cases out of 17 were detected
incidentally, while searching for metastases along the pelvic walls. We there-
fore suppose that these patients had not complained of lower abdominal
pain. This argues that varicocele of the broad ligament does not necessarily
cause pain.
Hughes and Curtis (1962) performed phlebographies by injecting 20 ml of
Renografin into the myometrium of 105 patients. A film was taken immedi-
ately after the injection and a second film 20 set later. With this second film
they intended to examine the disappearance rate of the dye, a moderate to
marked amount of dye present at the 20th set being interpreted as conges-
tion. Only 1 out of 11 normal cases showed a delay in outflow of the dye.
All 22 patients with the pelvic congestion syndrome had such a delay; 15 of
these patients came to hysterectomy, and no pathology other than mild
chronic cervicitis was found. In 11 out of 14 patients with cervicitis and
erosion, moderate to marked congestion was seen. After adequate treatment
of the cervicitis, the phlebograms returned to normal in 2 cases in which a
control phlebography was performed. Unfortunately, the authors do not
state whether, in the cases with cervicitis and erosion, the patients were
complaining of pain. It is interesting to mention that 6 postpartum patients
also showed delay in the outflow of dye injected into the fundus uteri. Since
those last patients apparently did not complain of pain, this paper shows
that ‘delay in outflow’ does not necessarily mean pain. If, then, passive
pelvic congestion may cause lower abdominal pain, as a comparison between
the control group and the 22 patients with the pelvic congestion syndrome
428

suggests, there must be other factors at work besides the congestion.


Giraud and Lassegue (1964) state that the most interesting indication for
uterine phlebography is the search for tubo-ovarian varicosities, whose
presence may, according to them, explain some cases of lower abdominal
pain with ‘normal’ internal genital organs. But they warn caution in inter-
pretation of the films. Indeed, several authors feel that a sudden stop on the
course of a vein is due to a pelvic thrombosis, which may lead, upstream of
this stop, to the formation of varicosities. The existence of a thrombosis
could, however, not be confirmed by surgical exploration of cases with this
kind of ‘venous obliteration’.
Vermeersch et al. (1971) studied the uterine and pelvic venous circulation
by transmyometrial phlebography. The symmetry of the spread of dye in
the myometrium was verified after an initial injection of 1 ml of dye, and
the spread itself was followed by intermittent photofluorography during
injection and disappearance of the dye. In this way the venous circulation
can be visualized and an idea of the velocity of the venous flow obtained.
Normally the dye has disappeared after 20 set; the authors admit that there
is ‘pelvic congestion’ when the dye persists after 30 sec. In fact, in the cases
they considered as pelvic congestion, the dye mostly persisted for more than
2 min. They studied 8 control cases and 39 patients with unexplained lower
abdominal pain. In 12 of these 39 patients the phlebographic findings were
normal, and in 27 they were found to be abnormal; amongst those 27 they
found 11 with pelvic congestion and 10 with varicocele. They consider
varicocele as a severe stage of pelvic congestion. The radiological diagnoses
were confirmed either by laparoscopy or laparotomy in 8 cases out of 9
with pelvic congestion and in 5 out of 6 with pelvic varicocele. For the sake
of clarity the authors should have stated how their 8 control cases were
selected and whether their cases with unexplained pain had had a laparos-
copy. Their paper shows that, amongst the patients with unexplained pelvic
pain, about a third did not present any radiological anomalies of the venous
circulation, whereas the other two-thirds did.
Craig and Hobbs (1974) examined 12 patients, 10 of whom were believed
to be suffering from the pelvic congestion syndrome, by percutaneous vulval
phlebography. The authors feel that this method is to be preferred over
transuterine phlebography and catheterization of the left renal vein, as by
the two latter routes not all the visceral veins are visualized. A careful study
of their cases does, however, not show a good correlation between the varied
presenting complaints of the patients and the location of the varicosities
visualized by their method.
Having reviewed a few of the numerous papers on pelvic phlebography, we
conclude: (i) that a good percentage of the patients with CPPWOP present
radiologic signs of passive pelvic congestion; (ii) that a certain percentage do
not present those signs; (iii) that persons who do not complain of pain may
also have radiologic signs of passive pelvic congestion, so that, apart from
passive pelvic congestion, other factors must intervene to explain the pain.
429

(b) Personal observations.


(i) Material and methods:
We examined by laparoscopy 3 series of patients: (1) 16 patients with
CPPWOP; (2) 16 patients who were complaining of chronic pelvic pain, but
whose pain was almost certainly due to endometriosis; (3) 15 patients who
did not complain of pain and underwent a laparoscopy for the evaluation
of their infertility.
In all those patients we looked for: the presence or absence of a large
mottled uterus, and the presence or absence of varicosities in the infundibu-
lopelvic ligaments and in the uterine veins at the basis of the broad liga-
ments.
- We aspirated and measured the fluid found in the pouch of Douglas.
- The aspirated fluid underwent a careful cytologic examination.
Examination of the pelvic veins during laparoscopy necessitates, on the
one hand, a steep Trendelenburg position and, on the other, elevation of the
uterus by means of an intrauterine cannula. It generally takes l-2 min for
the pelvic veins to empty themselves, so there is ample time to examine
them. It should be noted, however, that sometimes, even when one has not
found pelvic varicosities at laparoscopy, one may find varicosities at a sub-
sequent laparotomy; this means that one has to reckon with a certain
number of errors, ‘which can only be corrected by examining an experimental
and a control series of patients. We admit the presence of varicosities when
we find very large veins, with a diameter of 5 mm or more, or an impressive
bundle of veins either along the base of the broad ligaments or in the infun-
dibulopelvic ligaments.
(ii) Results:
Volume and color of the uterus
In none of the 16 patients with CPPWOP did we see an enlarged uterus; it
should be recalled, however, that enlarged, painful retroverted uteri were
not included in this series. A mottled appearance of the uterus was found
once. It was never seen in the 16 cases with pain due to endometriosis or in
the 15 control cases without pain. This mottled appearance of the uterus is,
however, seen occasionally in patients who do not complain of pain.
Varicosities
Table VI gives the frequency of varicosities in the 3 groups of patients.
From Table VI it would seem that varicosities are more frequent in
CPPWOP cases than in the other groups. There are, however, two variables

TABLE VI Frequency of varicosities in the 3 groups of patients

No. of patients No. with varicosities

1. CPPWOP 16 8
2. Chronic pain due to endometriosis 16 3
3. Control patients 15 4
430

which might possibly explain this difference and which are unevenly distrib-
uted amongst the 3 groups: the frequency of use of contraceptive steroids
and the parity of the patients. More patients of the group with chronic pain
had been taking estroprogestogens than in the other groups, and the parity
of the patients was not comparable. The prolonged use of steroids may
perhaps increase the frequency of pelvic varicosities, and it is most probable
that parous women tend to have pelvic varicosities more frequently than
nulliparae. Therefore, in order to obtain a comparable control series, we have
examined the frequency of varicosities in 20 consecutive patients, who
underwent a laparoscopic tubal ring ligature. All those patients were parous
and all were using oral contraceptives at the time of the sterilization. Table
VII gives the frequency of varices in the patients with CPPWOP and in the
control patients,
There is, thus, no striking difference in the frequency of varices between
CPPWOP and control patients when we compare 2 series of patients who are
on steroids and are comparable as far as parity is concerned.
Quantity of peritoneal fluid
Studying the quantity of peritoneal fluid in comparable series of patients
is rather difficult. Maathuis et al. (1978) have shown that the amount of
peritoneal fluid in the pouch of Douglas varies greatly, depending on the
phase of the menstrual cycle. The amount gradually increases during the
proliferative phase, is maximal during the early secretory phase, and
decreases towards very small amounts in the late secretory and menstrual
phase. Most of our patients (12/16) were taking oral contraceptives at the
time of laparoscopy. It must be noted that the use of oral contraceptives
causes a significant reduction in the quantity of peritoneal fluid, and there is
but slight variability during the artificial cycle (Koninckx et al., 1980), and
since we have but 4 patients with CPPWOP who were not taking oral con-
traceptives, we cannot compare their volume of peritoneal fluid with our
control group without pain. We can therefore only compare the amount of
peritoneal fluid between the CPPWOP patients who were taking oral con-
traceptives and the series of 20 parous women who did not complain of
pain and were also using oral contraceptives. Table VIII gives the mean
quantity of peritoneal fluid in a series of CPPWOP patients and a control
series without pain. All patients were taking combined oral contraceptives.
There is, thus, apparently a greater volume of peritoneal fluid in CPPWOP

TABLE VII Frequency of varices in the patients with CPPWOP and in the control
patients

No. of patients taking oral No. of parous No. with


contraceptives patients varices

CPPWOP 12 9 6
Control series 20 20 10
431

TABLE VIII Mean quantity of peritoneal fluid in a series of CPPWOP patients and
control series without pain; all the patients were taking oral contraceptives

No. of patients Mean volume of peritoneal fluid

CPPWOP 12 8.66 ml (’ 4.96)


Control series 20 5.4 ml (+ 3.69)

Student’s t-test: P = 0.04

patients on contraceptive steroids than in a control series of patients without


pain.
(iii) Discussion:
- In our series, from which patients with painful retroverted uteri were
discarded, we did not find any enlarged uteri, and saw a mottled aspect of
the uterus only once. It should be noted, on the other hand, that, on the
occasion of laparotomies for patients who do not complain of pain, one may
sometimes find a mottled appearance of the uterus. We can therefore not
attribute any importance to this sign.
- As far as pelvic congestion and pelvic varicosities are concerned, data
culled from our various control series show that the frequency of varicosities
depends to a large extent on the parity of the patients, whether they have
pain or not. There is no striking difference in the frequency of pelvic vari-
cosities between CPPWOP patients and parous control patients without pain
(Table VII). Admittedly laparoscopy gives no information on the venous
circulation in the uterus itself, but, if the belief that varicosities are an
advanced stage of pelvic congestion is justified, the morphologic evidence in
favor of pelvic congestion is not very impressive. Even those who found fre-
quent phlebographic evidence of passive pelvic congestion found it in only
some of the patients with chronic pelvic pain without obvious pathology. We
saw earlier that, if passive pelvic congestion, as demonstrated by uterine
phlebography, intervenes in the mechanism of chronic pelvic pain, there
must be other factors at work besides the circulatory stasis. These factors
might be either an increase in intratissular pressure, or they might be
psychologic in nature.
- According to Table VIII, 12 CPPWOP patients taking combined pills had a
mean peritoneal fluid volume of 8.66 ml (?4.96), whereas 20 control
patients without pain who were also taking combined pills had a mean vol-
ume of 5.4 ml (k3.69). The difference between these means is significant:
P = 0.04 (Student’s t-test).
In a recent clinical investigation, Koninckx et al. (1980) compared the
amount of peritoneal fluid found during the luteal phase of the menstrual
cycle in women who underwent a laparoscopy for infertility with the volume
found in women who were taking combined pills and underwent a laparos-
topic sterilization. None of these two series of patients complained of lower
abdominal pain. The presence or absence of pelvic varicosities (0, +, ++) was
also looked for.
LUTEAL PHASE COMBINED PILL

. ..XXX

. .

-x-

x x

. ..X .
.X
...
.X .
. x
.
.. .

. . ..XXX x
.
. ..XX .
x

.m .
. . ..XX

.
.X x
n:l& n:9 n-s

...
..XX .
xx . ..
..XXX .
..XXX> . .
. .
. .. -.X-

.XXX .

xx
x
” : 78 n = 10 n:s
0 + 0 + ++

“A;COSITIES
Fig. 1. Peritoneal fluid volume found at laparoscopy during the luteal phase of the cycle
(left) or on combined pills (right). The cases have been classified according to the absence
(0) or presence (+ or ++) of varicosities of the pelvic veins.

As can be seen from Figure 1, there is a significant difference between the


volume of peritoneal fluid during the luteal phase on the one hand, and
under combined pills on the other. The presence or absence of varicosities
had no influence on the amount of peritoneal fluid.
Although Table VIII shows a significant difference in mean volume, we
feel, for various reasons, that it would be hazardous to conclude that the
peritoneal fluid volume is greater in patients with CPPWOP than in normal
women.
433

Indeed
- The amount of peritoneal fluid found in our patients has certainly been
modified under the influence of the combined pills.
-If we compare the amount of fluid in our 4 patients with CPPWOP who
did not take combined pills with that of 4 patients without pain and without
pills, at comparable dates of their cycle, we find:

CPPWOP: (1) day 11, 5 ml controls: (1) day 10, 5 ml


(2) day 14, 4 ml (2) day 12,15 ml
(3) day 17,lO ml (3) day 16,lO ml
(4) day 22, 50 ml (4) day 20,50 ml

It is therefore possible that there would simply have been no difference if we


had been able to make a comparison between patients and controls who
were not taking contraceptive pills.
- According to Figure 1 the presence of pelvic varices does not increase the
amount of peritoneal fluid.
In the normal menstrual cycle the amount of fluid seems essentially due
to hormonally determined changes in vascular permeability, leading to a
change in the equilibrium between inflow and outflow across the peritoneal
membrane, and to some extent to fluid transfer after and possibly before
follicular rupture at the level of the ovarian surface (Maathuis et al., 1978).
The hormones which enhance this permeability are most probably estrogens
(Koninckx et al., 1980).
It therefore remains to be demonstrated that in CPPWOP there is a greater
amount of peritoneal fluid. Although it is probable that the amount of fluid
is influenced by circulatory factors, a great amount of fluid is not necessar-
ily proof of vascular stasis.
(iv) Conclusion:
As we saw at the beginning of this chapter, there is good circumstantial
evidence suggesting that circulatory factors may give rise to chronic or
intermittent lower abdominal pain. There is some phlebographic evidence for
the presence of passive pelvic congestion in some cases of chronic pelvic pain
without obvious pathology, but this passive pelvic congestion does not seem
to be the sole factor at work in causing the pain. A clear-cut demonstration
of the importance of circulatory factors in the pain mechanism will have to
await better methods for the evaluation of pelvic circulation. From a practi-
cal point of view, it may be concluded that the presence of indirect signs
pointing towards pelvic congestion should not lead too rapidly to such
surgical interventions as, for example, suspension operations, ligature of
varicosities or hysterectomy.

III. Attempts at a histopathological explanation of the pain

1. A short summary of the literature


Many attempts have been made at finding a pathological basis for this
434

syndrome. An explanation has been looked for in the uterus, in the ovaries,
and in the pericervical tissues or parametrium.
(a) The uterus. Out of 105 cases Taylor (1949a-c) found the uterus
enlarged in 32 cases, normal in size in 70 and small in 3.
In what he calls ‘congestion utero-annexielle’, Cotte (1949) generally
found the volume of the uterus increased to a variable degree. The cervix
appears blue and congested; it is often eroded and enlarged, and shows
abundant secretion. In describing the clinical findings in cases of ‘the pelvic
congestion syndrome’, several authors note that the fundus is frequently felt
in retroversion and seems soft, boggy and definitely enlarged (Montgomery,
1966; Parsons and Sommers, 1978).
We found these features only in a minority of our cases. In the majority
the uterus was normal in size and firm on palpation. We did not include any
cases in our series in which the pain could be due to a painful retroversion,
although painful mobile retroversions and some cases of CPPWOP may well
have common pathogenetic factors.
Stearns and Sneeden (1966) studied the pathology of the pelvic conges-
tion syndrome in a series of 69 uteri obtained by hysterectomy during a
period of 32 months. They found marked blood and lymph vessel ectasia
and edema. They felt that these features were characteristic of the syn-
drome. It should be pointed out, however, that their series was heteroge-
neous. Indeed 49 patients complained of severe menometrorrhagia, 14 of
pelvic discomfort and sore breasts, and 16 of severe dyspareunia. This means
that only a minority had either spontaneous or provoked pelvic pain, and
“the morphologic features in the cases the authors are presenting are those
of uteri removed for excessive and uncontrolled bleeding” (Stearns and
Sneeden, 1966). This paper does not show that the dilated veins and lymph
channels and the localized edema are the causes of the pelvic vein.
(b) The ovaries. Many French authors have for years considered the so-
called ‘sclerocystic dystrophy of the ovaries’ as a rather frequent cause of
chronic pelvic pain (Netter, 1953). This dystrophy was said to be the cause
of uni- or bilateral pain and to be accompanied frequently by irregularities
in the menstrual cycle and sometimes by intermenstrual bleeding.
This form of ‘ovarian dystrophy’ is virtually unknown in the Anglo-Saxon
and German literature. Dubreuil (1947) writes about this ‘sclerocystic
dystrophy’: “The sclerotic changes observed by the pathologist in the so-
called sclerocystic ovaries mostly are normal physiologic features: those
changes may become more pronounced over the years but they do not
interfere with normal ovarian function. The cystic follicles are but the usual
mode of regression of follicles which reached a rather great diameter without
proceeding to ovulation”.
From the point of view of pathogenesis of lower abdominal pain, it should
be added that, for all the morphologic changes they undergo during the
reproductive period of a woman’s life, ovaries are rather insensitive organs.
(c) Pathology of paracervical tissues or parametrium.
(i) Clinical features:
435

Most authors who have studied CPPWOP have stressed tenderness on


examination of the uterosacral ligaments or of the tissues of the posterior
parametrium. The names by which the syndrome was designated in the
earlier German literature, ‘Parametritis posterior chronica’, ‘Parametritis
posterior’, ‘Parametropathia spastica’, bear witness to this. Taylor (1949a-c)
states that the single most characteristic sign of pelvic congestion is tender-
ness of the uterosacral ligaments; this sign was present in 98 out of 105 of
his cases and absent or questionable in only 7.
In the clinical description of our own 31 cases, we noted that 26 pre-
sented a striking tenderness of the posterior parametrium when it was
stretched during a vaginal or rectovaginal examination. In most patients the
pain provoked on stretching the posterior parametrium reproduced the pain
they experienced spontaneously or during intercourse.
(ii) Inf2ammato~y process or spasm ?
In the earlier German literature, tenderness of the posterior parametrium
was believed to be due to an inflammatory process and, since the work of
Opitz (1922), Louros (1927), Neumann (1933) and Martius (1939), this
tenderness has generally been ascribed to a spastic contraction of the smooth
muscle fibers of the paracervical tissue.
During the last half of the 19th century Schultze (1885) and in the first
half of the present century W.A. Freund (1903), R. Freund (1933) and
Martin (1944) attributed the syndrome either to a diffuse or a localized
phlegmonous inflammation of the parametrium. In most cases, the process
was localized in the posterior parametrium and was considered either as a
result of an acute phlegmonous episode or as a primary chronic process.
Opitz (1922), Louros (1927) and Martius (1939) showed, however, that,
although an infectious process may be responsible in some, there were no
signs of infection in the majority of the cases. In 14 cases of persistent
‘posterior parametritis’, Louros (1927) excised a 5 mm broad strip from the
uterosacral ligaments and did not find, on histological examination, any sign
of inflammation. Opitz (1922) states to have cured many patients by
massage and stretching of the uterosacral ligaments under anesthesia.
According to Knorr et al. (1972), the ‘spasm of the ligaments’ disappears
under anesthesia, and Prill(l964) finds that it disappears under the influence
of relaxation and reassurance.
Neumann (1933) repeatedly performed microscopic examinations of
thickened ‘spastic’ uterosacral ligaments. The tissues were rather edematous;
there was an increased amount of fibrous tissue, but the number of elastic
fibers and smooth muscle fibers had decreased. The blood vessels were fre-
quently distended by blood. Neumann never found any signs of inflamma-
tion. It is difficult to draw conclusions from this description, as no control
cases have been studied and as it is known that the histological composition
of the paracervical tissue varies widely depending on the age, the parity, and
the amount of physical labor performed by the patient.
(iii) Neuritis? Recurring pelvic peritonitis?
Amongst patients with CPPWOP, Cotte (1949) distinguished between
436

those with pelvic congestion and those with what he called ‘pelvic neuralgia’.
The last term was based on histological studies performed by Dechaume
(Cotte and Dechaume, 1931) on 22 specimens of the superior hypogastric
plexus excised during ‘Cotte operations’. In 10 cases out of 21, Dechaume
found distinct lesions. It is, however, hardly possible to draw any conclusions
from this work: (i) because the series of patients whose plexus hypogastricus
was examined was not clinically homogeneous; (ii) because different lesions
were found in different patients; (iii) because the lesions were found in only
10 patients out of 21 and (iv) because no mention is made of a control series
of patients without pain.
De Brux et al. (1968) examined 25 patients with CPPWOP. In 17 of these
cases a laceration of the broad ligament was found, whereas it was absent in
8. Large biopsies were taken for histological study from the peritoneum of
the broad ligaments, the uterosacral ligaments and the pouch of Douglas.
Out of the 8 cases without a laceration of the broad ligament, they observed
hemangiomas and plexiform angiomas in two cases, endometriotic lesions in
4 and areas of cytonecrosis in 2. Besides these histologic features the
phenomena of congestion, exudation and progressive transformation of the
connective tissue sometimes resulting in a mutilating sclerosis, as described by
Taylor (1949a-c), were constant findings. In those same 8 cases there was
practically always an excess of clear fluid in the pouch of Douglas, and
cytologic study of this fluid revealed a marked inflammatory reaction with
mesothelial cells and lymphocytes. Microscopic study of the pelvic tissues in
the 17 cases with a laceration of the broad ligament disclosed: endometriotic
lesions of minimal size in 8 cases, a glomus tumor in 1 case, a neuroma in 1,
a foreign body reaction in 1 and areas of cytonecrosis in 6. These data are
interesting but in this paper, as in others, there is a lack of a control series of
patients of the same age and reproductive characteristics who do not com-
plain of pain. Moreover, one should be cautious of ascribing complaints of
pain to microscopic foci of endometriosis, as it is well-known that even large
foci of endometriosis may remain painless.
(iv) Immunoallergic phenomena?
Lbzlo and Gyiiry (1959, 1963) examined biopsy specimens from the
parametria of 80 patients operated for CPPWOP. The specimens were
examined by various staining techniques in order to study the blood vessels
and the periuterine connective tissue. They also examined the periuterine
system of ligaments in 15 young and middle-aged women who had died of
other diseases, as well as that of 20 patients who were operated upon for
various other gynecologic diseases. They found serious damage of the con-
nective tissue and of the vessels in 82% of the patients with pelvic pain.
Routine staining methods revealed in the tissues of patients with pelvic pain a
swelling, disintegration, fragmentation and curling of the collagenous con-
nective tissue fibers in some instances and necrosis, cicatrization and shrink-
age in others. They felt the changes in the collagenous, elastic and argyrophil
reticular fibers and in the ground substance, demonstrated by the more
specific staining techniques, the granulomatous character of the inflamma-
437

tory foci, as well as their association with nervous elements, to be histologic


manifestations of immunoallergic phenomena. As those histologic features
were very frequent in the pain cases and infrequent in the control cases, they
took those immunoallergic phenomena to play a significant role in the
pathogenesis of the disorder.
It is regrettable that the authors did not give more details on the clinical
symptoms and signs which justified the inclusion of the patients in the
‘experimental group’. It would also have been interesting to know the age
distribution of the experimental and the control group, as many of the
changes found in the experimental group could also be explained as part of
the aging process. Moreover, they did not present valid morphological evi-
dence to support the hypothesis of an inappropriate immunological reac-
tion as an explanation of the pain syndrome.

2. Personal investigations
To study the role of the pericervical tissues in the pathogenesis of
CPPWOP, we compared the frequency of tenderness of the posterior para-
metrium in CPPWOP patients to that in patients who did not complain of
pain. We examined the changes, if any, in the posterior parametrium in
patients with CPPWOP both before and under general anesthesia. A histo-
logic investigation of the cervix and the posterior parametrium was per-
formed after hysterectomy in a group of patients with CPPWOP and in a
group of patients who did not complain of pain.
(a) The importance of tenderness of the posterior parametrium on palpa-
tion or on traction. As shown in Table III, there was tenderness of the
posterior parametrium in 26 out of 31 cases with CPPWOP. We looked for
tenderness with or without shortening of the posterior parametrium in a
control series of 44 patients (Table IX). None of the patients complained of
pain, and almost all had come for a prophylactic out-patient examination.
All the patients were married; all of them had one or more children; none
had had a traumatic delivery. Their age distribution was between 30 and 45
years. No patient had a history of acute PID or findings compatible with
endometriosis. A microscopic examination was made of the endocervical
mucus, and no patients who presented numerous agglutinated leucocytes
were included. In this way it was intended to eliminate cases with an actual

TABLE IX Clinical examination of the posterior parametrium in 44 patients without


pain

Posterior parametrium

Negative Slightly tender Shortened, Tender Shortened and


no tenderness tender
21 7 1 10 5

29 15
438

or presumed pericervical lymphatic extension of a cervical infection.


In all the patients the uterus and the adnexa were normal on palpation.
Tenderness of the posterior parametrium with or without condensations or
shortening was tested by vaginal examination and by forward displacement
of the cervix on vaginal examination.
The posterior parametrium is distinctly tender in roughly one-third of
the patients who did not complain of pain (cf. Table IX). As we did not
perform a laparoscopy on the control series there may, however, have
been a few patients whose tender parametrium was attributable to a non-
recognized endometriosis.
Besides women who do not complain of any pain, either spontaneous
or provoked, but whose posterior parametrium is tender, there are also
patients with a tender and sometimes shortened posterior parametrium, who
have no cervicitis and apparently no endometriosis, who do not complain
of spontaneous pain but who regularly experience deep dyspareunia, and
whose pain can be reproduced by traction on the sacrouterine ligaments.
The problem therefore arises as to why some patients with a tender poste-
rior parametrium do not complain of any pain, why some patients experi-
ence only deep dyspareunia, and why some patients have deep dyspareunia
and spontaneous pain probably related to the tender posterior parametrium.
(b) Is tenderness of the posterior parametrium or the uterosacral ligaments
due to ‘spasm ‘? Out of the 31 cases of our series of CPPWOP we examined
18 whose posterior parametrium was distinctly tender on pressure or on
traction. Those patients were examined both before and after the induction
of general anesthesia at the occasion of a laparoscopy.
In 12 cases out of 18 there was only tenderness of the parametrium or the
uterosacral ligaments, but in 6 cases there were, besides the tenderness,
anteroposterior cord-like indurations or condensations in the posterior
parametrium. In 5 cases out of 6 there was, on palpation under anesthesia,
no modification of the consistency of the condensed part of the posterior
parametrium, nor was there a noticeable lengthening of the uterosacral liga-
ments in the 3 cases in which they were not only indurated but also
shortened. We therefore did not find under anesthesia a relaxation of a
‘spasm’ of the uterosacral ligament, as described by several German authors.
As the cord-like induration of the uterosacral ligaments may be unilateral
and as the uni- or bilateral condensations do not disappear or change in the
majority of the cases under general anesthesia, we doubt whether the
‘induration’ is due to a spasm. It is more probably due to an organic modifi-
cation in the texture of the posterior parametrium, although we have not
been able to identify such a modification on microscopic examination of
uteri resected with part of the posterior parametrium (vide infra).
(c) Pathology of cervix and posterior parametrium in CPPWOP. In the
laboratory, one of us (A.V.A.) has examined the pathology of the cervix
and of the posterior parametrium in 8 cases of CPPWOP and in 6 cases who
did not complain of pain and who underwent a hysterectomy either for
menorrhagia due to fibroids or for metrorrhagia due to dysfunctional bleed-
439

ing. The patients had the same age distribution in both groups. The 8 cases
with CPPWOP had been complaining of lower abdominal pain for many
months; they had had various medical treatments and underwent a hysterec-
tomy at a time when we still resorted more frequently to operative treat-
ment than we do now. The operation was, in both groups, a total hysterec-
tomy with resection of the anterior part of the posterior parametrium.
A histologic examination of the cervix and the posterior parametrium was
performed. The specimens were cut into sections perpendicular to the long
axis of the cervix. The tissues were studied after hematoxylin-eosin stain-
ing, after Masson connective tissue staining and orcein staining for elastic
fibers, Other sections were studied after PAS staining. The nerves of the
posterior parametrium were examined after a Jaboneiro staining.
When performing the histologic examination of the specimens, A. Van
Assche did not know which ones were from patients with pain and which
from control patients. The 14 specimens were studied for signs of infection,
for the presence and degree of interstitial edema, for the presence of PAS-
positive material in connective tissue, and for the characteristics of the
elastic fibers. The walls of the blood vessels were examined for PAS-positive
material. The nerve plexuses were carefully studied by Professor Brucher, a
neuropathologist.
No significant differences could be found in any of the aspects studied
between the specimens from the pain cases and those from the control cases.
Interstitial edema was more pronounced in the cervix and in the posterior
parametrium in 3 pain cases; this may, however, be due to the fact that those
3 patients had been taking 5 mg ethinylestrenol for several weeks before
surgery. A. Van Assche could, therefore, not find a clear morphologic
explanation either for the tenderness or for the painful condensations fre-
quently found in the posterior parametrium in CPPWOP.

3. Discussion
(a) Tenderness of the posterior parametrium can explain pelvic pain in
most cases of CPPWOP. In Table III we saw that, in 26 out of 31 cases of
CPPWOP, tenderness of the posterior parametrium was the most frequent
anomaly found after extensive investigation; and it has struck most writers
on the subject as being the principal symptom of the syndrome. In patients in
whom this tenderness is found, the uterus and adnexa generally are not
particularly tender. On the other hand, pressure or traction on the posterior
parametrium regularly produces the same sensation as the spontaneous
pain, and also the deep dyspareunia which is a frequent symptom. This
tenderness can also explain why in many patients the pain is enhanced while
running or being jolted.
The pain derived from tender tissues is known to increase under the
influence of circulatory changes; this could explain why the spontaneous
pain in the CPPWOP cases frequently increases during the premenstrual
period and the beginning of menstruation.
We know of several circumstances in which tenderness of the posterior
440

parametrium explains a similar pain syndrome. It is a sign of parametritis,


or lymphatic spread of infection in some cases of chronic cervicitis. It is also
found in many cases of endometriosis of the uterosacral ligaments, and also
in some - now, in fact, rare - cases with a scar in the vaginal cul de sac after
a cervicovaginal tear. Very rarely, a real tear of a uterosacral ligament may
explain spontaneous lower abdominal pain and dyspareunia.
Plausible as this explanation may be for most cases of CPPWOP, there are,
however, a few important restrictions:
- As tenderness of the posterior parametrium is not present in all cases of
CPPWOP, it does not offer a universal explanation of the symptoms. It is
probable that, in a minority of cases, there is primarily a circulatory distur-
bance, either in the form of an active or a passive congestion. Proof of this
hypothesis awaits, however, an adequate technique for the measurement of
uterine or pelvic blood flow.
-A painful posterior parametrium does not explain the various other
symptoms many polysymptomatic patients complain of.
-There is, moreover, the problem of why some patients with a tender
posterior parametrium on examination only complain of deep dyspareunia
and no spontaneous pain, and why some of those persons do not complain
of pain at all,
(b) What causes the tenderness with or without shortening of the poste-
rior parametrium? Several German authors, amongst whom Opitz (1922),
Freund (1933), Neumann (1933) and Martius (1939), believe that the
tenderness and the condensations in the posterior parametrium may in some
cases be due to inflammation but are, in the main, due to a spasm of the
smooth muscle fibers.
One has to agree that inflammation is a possible cause, but it should be
recalled that any patient who has a tender posterior parametrium and
cervicitis should be treated for her cervicitis and her possible parametritis
with antibiotics and with local heat, e.g. with hot sitzbaths; she should, how-
ever, not be included for study in a series of chronic pain without obvious
pathology. As this kind of patient was discarded, it is clear from the above-
mentioned histologic investigations that, in most cases of CPPWOP, inflam-
mation cannot be entertained as the cause of the tenderness.
As we did not find any modification in the length or in the texture of the
posterior parametrium when examined before and under anesthesia, and as
shortening and condensation of the uterosacral ligaments may sometimes be
unilateral, we have difficulty in accepting that this shortening is due to a
spasm. Moreover, in most cases, the tenderness is not accompanied either by
shortening or by condensation.
The histologic studies referred to in this chapter did not show any definite
or characteristic anomaly, if we except the venous congestion and the
increase in interstitial edema found by Neumann (1933) and LCzlo and
Gyory (1963). Venous congestion and interstitial edema are, however,
features which are difficult to quantify by histologic techniques. The
patients with CPPWOP are nearly all married women, and the pain fre-
441

quently starts after a delivery or a miscarriage. One therefore wonders


whether the process leading to the chronic pain situation does not begin as
postpartum cervicitis with posterior parametritis, which in most cases dis-
appears spontaneously or under an appropriate treatment, but which in some
patients leaves a tender parametrium. This might persist without causing
any pain or it might provoke only deep dyspareunia, or it might lead, in
some predisposed people, to a state of chronic pelvic pain without any
distinct remaining pathology.
(c) What determines that some patients will complain of pain whereas
others will not? The answer may be searched for either in a difference in
peripheral pain stimuli, or in a difference in the patient’s reaction to given
stimuli, or in a combination of both; in the absence of obvious peripheral
stimuli it may also be found in psychologic mechanisms.
The histologic studies mentioned in this chapter did not reveal a clear-cut
organic cause, except perhaps circulatory disturbances in some cases. Com-
parative histologic studies will probably never be able to demonstrate a
difference between pain-provoking and non-pain-provoking conditions in the
clinical situations referred to above. At the present time we do not know
any adequate technique for the comparative study of uterine or pelvic
blood flow. For this reason, a psychologic evaluation of a series of patients
with CPPWOP was undertaken in order to enquire about the life situations
of patients with CPPWOP and to measure a series of personality variables:
of patients with CPPWOP; of patients with chronic pain due to an organic
cause; and of patients without pain complaints.

IV. The psychological characteristics of women with chronic pelvic pain


without obvious pathology

1. A review of recent publications


The importance of psychological factors in the pathogenesis of chronic
pelvic pain without obvious pathology has been stressed by many authors;
a review of some recent papers follows.
In a detailed description of ‘Pelvic Congestion and Fibrosis’, Taylor
(1949a-c) drew attention to psychiatric anomalies in several patients out of
his series of 105. In 1952, Duncan and Taylor gave the results of a
psychosomatic study of 36 patients with pelvic congestion. Most of those
patients had not experienced a secure family life during their childhood;
most of them displayed inability to function as women, either sexually or
maternally. Emotional immaturity and strong dependent needs were other
common traits. Although other personality characteristics varied consider-
ably amongst the group, almost all the patients studied were psychologically
ill. In 24 out of the 36 patients, the onset of the symptoms of ‘pelvic conges-
tion’ was temporally related to a stressful life situation. In the same study
Duncan and Taylor also made a continuous record of the thermal con-
ductance of the lateral vaginal wall in 10 patients during a long interview,
and regularly found a correlation between emotional episodes elicited by the
442

interview and increased blood flow in the vaginal wall. As the stressful situa-
tions were generally antecedent to the appearance of the symptoms, and as
stressful episodes can enhance hyperemia, the authors postulated that this
hyperemia, one of the somatic counterparts of emotional stress, ultimately
leads to congestion, edema and pain. This work calls for a few critical
remarks. First, the diagnosis of ‘pelvic congestion’ was apparently established
by clinical examination, and not checked by laparoscopy; a certain number
of organic causes of lower abdominal pain, such as chronic PID and
endometriosis, may have been missed. Second, the correlation between
stressful situations and the onset of symptoms was made in a retrospective
manner, but, as is shown by Rabkin and Struening (1976), one should be
cautious in establishing retrospectively relationships between stressful life
situations and illness. Third, although vascular congestion and hyperemia
may well be important pathogenetic factors in many cases of chronic pelvic
pain without obvious pathology, it is probable that circulatory disturbances
are not the determining cause in the majority of cases.
The psychologic characteristics of patients with pelvic pain without
obvious pathology were again studied in 1960, by a psychiatrist and a
psychologist, in patients followed by Taylor (Gidro-Frank et al., 1960).
They studied 40 patients with pelvic pain and 25 control patients using
repeated interviews and psychology tests such as figure drawing, the
thematic aperception test and the Rohrschach test. Fifteen of the 40
patients with pelvic pain exhibited signs and symptoms of chronic pelvic
congestion; the 25 others did not present the typical symptoms of chronic
pelvic congestion but suffered from pelvic pain in the absence of demonstra-
ble pelvic pathology. The psychological evaluation of the patients did, how-
ever, not show any difference between the two subgroups. With the excep-
tion of two, all the patients with pelvic pain had psychiatric disturbances
covering a whole gamut from schizophrenia (4), to borderline psychosis (lo),
severe neurosis (20), moderate neurosis (3) and mild neurosis (1). Although
in the control group the psychiatric disturbances were less pronounced; there
were 8 patients with borderline psychosis, 4 with severe neurosis, 7 with
moderate neurosis and 5 with mild neurosis. Contrary to Duncan and Taylor
(1952), Gidro-Frank et al. were only rarely successful in tracing the develop-
ment of pain to its origins in a patient’s life history. The authors postulate
“that patients with pelvic pain were unable to establish and preserve that
(unconscious) sense of feminine identity which permits the unhampered exe-
cution of feminine functions”.
The following comments can be made on this paper: the absence of
demonstrable pathology in the pain patients had apparently not been ver-
ified by laparoscopy or laparotomy. It is interesting to remark, in passing,
that amongst the 40 patients only 15 were classified as typical cases of
chronic pelvic congestion and 25 as ‘atypical cases’ of pelvic pain. As all the
controls had either borderline psychosis or severe, or at least mild, neurosis,
there was a virtual absence of ‘normal persons’ in the control group; there
rernains, however, an appreciable difference in the degree of disturbance
443

between the members of the two groups. The conclusion about the inability
of pelvic pain patients to establish and preserve their feminine identity is
somewhat at variance with the conclusion of other workers on this subject.
Benson et al. (1959) studied 35 women with chronic disabling pelvic
distress who presented essentially normal physical findings. These cases were
not sequential but selected over a 3-year period. No patient had only one or
two complaints; many had half a dozen or more, among which ‘psychoso-
matic’ symptoms such as nervousness, nausea and vomiting, dizziness and
fainting, weakness and fatigue were very common. Only in a minority of
patients was a diagnosis of pelvic congestion made. All the patients were
evaluated by repeated interviews taken by the psychiatrists of the team; 29
were diagnosed as psychoneurotic (almost all being hysterical) and 6 were
found to be psychotic (schizophrenic). The patients were also evaluated by
4 different objective tests. It is postulated that the pathogenesis and the
location of the pain is based upon a psychophysiological conditioning
mechanism that is initiated by psychodynamic factors connected with con-
flicts of the patient over her femininity and her independence of her mother.
This paper is based on 35 selected cases who had been examined
repeatedly and extensively, so that one can assume that there were no
organic lesions in most of the patients; it would, however, have been prefer-
able to ascertain by laparoscopy that there were indeed no lesions. It is
interesting to note that sensitivity or condensation of the posterior para-
metrium are not mentioned on gynecological examination. Under the
subtitle ‘Case material’, it is stated that the 35 cases were chosen as definite
and striking examples of the problem of unexplained pelvic pain; numerous
mild and borderline cases were eliminated. This leaves the impression that
the group is to some extent biased towards the polysymptomatic variety of
the syndrome, in which one is more apt to find psychologically disturbed
patients.
Since Gauss (1949) and von Jaschke (1950), chronic pelvic pain without
obvious pathology has been called ‘Pelipathia vegetativa’ by many German
authors. Prill (1964) summarizes his findings on the psychopathologic
evaluation of 163 patients with Pelipathia vegetativa. In 68 of them he found
a neurosis, either endo- or exogenous; 34 patients had psychopathic or
psychasthenic personality traits, and in 61 patients he did not find any
evidence of psychogenic factors. Unlike some American authors, Prill could
not find, in the patients who presented psychiatric disturbances, a psycho-
logical problem or conflict situation which could be considered a common
denominator to all cases. Apart from the patients in whom a neurosis was
found, Prill lists a whole series of different psychological problems. Accord-
ing to Prill, it would be unwise to try to find one single etiology for a syn-
drome with such a broad range of symptoms. In Prill’s work, as in the former
papers, there is no mention of a laparoscopic verification of the absence of
organic causes for the lower abdominal pain; but even so it is interesting
that, in a percentage of his cases, Prill did not find any psychiatric distur-
bances or psychological problems.
444

Castelnuovo-Tedesco and Krout (1970) examined 3 groups of patients


from a gynecologic and a psychiatric point of view: (a) 40 women of lower
socioeconomic class with chronic pelvic pain. The presence or absence of
specific pelvic pathology was ascertained by culdoscopy or laparotomy. In
15 patients there was no demonstrable organic pathology; in the 25 others a
variety of organic pathology was found, but it was not at all certain that the
pathology explained the pain complaint; (b) 27 women of lower socio-
economic class, all with pelvic pathology but without pelvic pain; (c) 25
women of lower middle class, some with and some without organic pathol-
ogy, but all with chronic pelvic pain.
The group (a) patients were examined by psychiatric interviews and
psychometric tests, whereas the group (b) and (c) patients only received
psychometric tests. Pelvic pain patients, regardless of the presence or absence
of organic pathology, almost all presented, besides their pain, other ‘psycho-
somatic complaints’ such as chronic fatigue, tension, sleep and appetite
disturbances, headache. They presented a striking degree of psychopathology
and social disorganization. On the psychometric tests (MMPI) patients with-
out pain and of the same socio-economic class had a more normal composite
MMPI profile than those with pain. Among patients with pelvic pain, social
class had little apparent bearing on the intensity of the psychopathologic
disturbances.
These data are instructive but the starting point was the complaint ‘pelvic
pain either with or without an organic origin’, and therefore the conclusions
are less useful for the solution of our problem: “Is there a difference
between the psychological characteristics of patients with chronic pelvic pain
without obvious pathology and those of patients with chronic pelvic pain
due to organic pathology?“.
Beard et al. (1977) studied 35 patients who were complaining of pelvic
pain: 18 were found not to present any pelvic abnormality at laparoscopy
and 17 had some form of pelvic abnormality, which could be a cause of
pelvic pain. There was a control group of 9 persons without any gynecologic
complaint. Patients and controls were asked to complete the following
questionnaires: (a) the Eysenck Personality Inventory (EPI), a screening test
for neuroticism and extraversion; (b) the Middlesex Hospital Questionnaire
(MHQ), used to measure anxiety, obsessional traits, somatic aspects of
anxiety, depression and hysterical symptoms; (c) a semantic differential test,
used to measure attitudes towards certain concepts which may have
influence on the life styles of individuals.
On the EPI the mean N (neuroticism) score of the laparoscopy negative
group was significantly higher than that of the control group. The mean N
score of the laparoscopy positive group fell midway between those of the
laparoscopy negative group and the controls. E (extraversion) scores did not
differ significantly between groups. Analysis of variance applied to the scores
from the subtest of the MHQ revealed no differences between the 3 groups.
The authors conclude that women who complain of pelvic pain without a
demonstrable organic cause are, as a group, psychologically different from
445

women without pelvic pain. From the point of view of clinical practice, they
feel that their results allow the assumption that a symptom such as pain,
which is usually regarded as evidence of organic disease, may stem from an
emotional disturbance. In the clinical assessment of the patients, no symp-
tom or symptom complex clearly distinguished the laparoscopy negative
from the laparoscopy positive group; the incidence of associated symptoms
tended to be higher in the laparoscopy positive group.
It strikes the reader that the sensitivity of the posterior parametrium,
elicited by moving the cervix forward, was absent in 16 patients out of
17 in whom it was looked for, although this symptom is found very fre-
quently in patients studied in most publications on the subject. The value
of the findings might have been enhanced had the laparoscopy positive group
been more homogeneous instead of comprising patients with some form of
pelvic abnormality which could possibly be the cause of pelvic pain. It would
also have been helpful if the duration of the symptoms had been mentioned;
there are indeed reasons for believing that there may be a difference in the
psychological characteristics of patients with chronic pain of organic origin
of, for example, more than 6 months’ duration and patients whose pain has
only lasted for a few weeks (Sternbach, 1974).

2. Personal investigations
In the first part of this report (C. IV, 2a) on our own work the complaints
of a group of patients with chronic pelvic pain without obvious pathology
(CPPWOP) are compared with those of a group of patients in whom endo-
metriosis had been found and in whom the pain was most probably due to
endometriosis. As stated earlier, all patients had been complaining of pain
for more than 6 months and most of them for several years. All patients had
been extensively investigated by clinical, radiological and laparoscopic
examinations. In the second part (C. IV, 2b) a psychiatric evaluation is
presented of the group of patients with CPPWOP. In the third part (C. IV,
2c) a psychological evaluation is made, by means of several psychometric
tests, of a group of patients with CPPWOP, of a group of patients whose pain
was due to endometriosis, and a control group of gynecological patients
without any pain complaint.
(a) Comparison of the complaints of a group of patients in whom an
organic cause was found for the pain with a group of patients without
obvious pathology. The symptoms of 24 patients with CPPWOP were
noted and of 22 patients whose pain was most probably due to endometrio-
sis. A causal relationship between endometriotic lesions and pain complaints
can be established with a reasonable degree of probability (Renaer, 1971).
The symptoms noted are the spontaneous complaints of the patients; they
were in no way elicited. The number of complaints are shown in Table X.
Whereas only one patient out of 22 with pain due to endometriosis had
4 complaints or more, half of the patients with CPPWOP, i.e. 12 out of 24,
had 4 complaints or more, and, in most cases, a patient with a greater
number of complaints, in other words a polysymptomatic patient, generally
446

TABLE X Comparison of the complaints of a group of patients with CPPWOP with


those of a group whose pain was due to endometriosis

Patients with CPPWOP Patients with pain due- to endometriosis


(n = 24) (n = 22)

No. of No. of No. of No. of


complaints patients complaints patients

1 3 1 9
2 7 2 8
3 2 3 4
4 6 4 -
5 4 5 1
6 1
7 1

had several complaints which are usually considered as functional: by


‘functional’ being meant a symptom which has no clear organic cause. The
marked difference in the number of the complaints and in their nature
between the two groups of patients suggests that at least some of the com-
plaints of many patients with CPPWOP may be due to an underlying
psychogenic mechanism. But the fact that the patient has only one or two
complaints does not necessarily mean that the pain does not have a psychic
component. Indeed, Merskey and Spear (1967) state that there is no hard
and fast rule to differentiate a pain complaint with an organic origin from
one with a psychic origin: “Patients with psychological illness seen in a gen-
eral medical clinic did not describe their pain in unduly bizarre terms and in
this respect, they resemble patients with organic illness”.
(b) Psychiatric evaluation of the group of patients with chronic pain with-
out obvious pathology.
(i) Patients and methods:
A psychopathologic evaluation of 22 out of the 24 patients with CPPWOP
has been done by a psychiatrist (P. Nijs), who is a senior staff member in the
Department of Obstetrics and Gynecology, or by one of the registrars under
his supervision. The diagnostic evaluation generally took l-3 sessions of 45
minutes each; in some patients it was followed by several sessions of psycho-
therapeutic counselling. There is no control series for this evaluation.
(ii) Discussion:
These evaluations have shown that the psychological characteristics of this
group of patients can not be reduced to a common denominator. Only two
patients admitted a happy marriage relationship. For most patients marriage
started under stressful conditions, either because the woman was pregnant
(4 cases) or because her husband was not accepted by the in-laws (5 cases),
or the patient married right after the death of her father or mother (3 cases)
447

or married to escape a stressful family environment (6 cases). Sexual dys-


function was frequent (13 cases).
Analysis of the individual case histories shows that several patients had a
neurotic behavior; some of them had difficulty in assuming their female
identity. There were, on the other hand, many patients who labored under
stressful life situations. In 3 patients, the complaint clearly was psychologi-
cally determined: once by cancerophobia and twice by the pursuit of uncon-
scious secondary gain. In most cases the stressful situations were antecedent
to the complaints.
As there is no control group, we have no compelling evidence that this
group is significantly different from a section of the general population of
the same age and the same socio-economic level; but, according to our
psychiatrists, these patients certainly differ as a group from the great group
of patients they have seen for an evaluation of their request to be sterilized.
These persons rarely complain of pain, infrequently present psychosomatic
problems and generally enjoy satisfactory marriage and family relationships.
We can, then, conclude that patients with CPPWOP present a gamut of
diverse psychological problems, either due to neurotic behavior or to diffi-
cult physical or psychological life situations. These problems are either
related to disturbed interpersonal relations in their marriage or in their
family life (with children, parents or in-laws) or to difficulties in their
professional life or to unresolved personal problems either about their own
sexual identity or their own health, or about life in general.
It would be hazardous to infer from the absence of an organic cause and
the presence of psychological difficulties that there is a direct relationship
between those difficulties and the pain complaints. One cannot go further
than to postulate that the psychological problems may be considered as
contributing to a multifactorial pathogenesis of the syndrome.
(c) Psychometric evaluation of patients with CPPWOP.
(i) Patient groups and methods:
In a consecutive series of 108 patients complaining of chronic pelvic pain,
who were admitted in the clinic for an extensive investigation (always
including a laparoscopy), 28 were diagnosed as ‘chronic pelvic pain without
obvious pathology’; in 22 patients the pain was most probably due to endo-
metriosis.
The intention was to administer a battery of psychometric tests to the 28
patients with CPPWOP, to the 22 patients with endometriosis and to 23 con-
trol patients. The control cases were patients admitted to the gynecological
clinic for benign non-painful conditions that apparently were not due to any
psychogenic factor, e.g. patients with a benign tumor of the ovary found
during a routine examination, a number of patients who underwent a
curettage for inter-menstrual blood loss occurring either spontaneously or
under ovulation inhibitors, patients with a slight degree of bladder descent
without urinary incontinence, etc. In the selection of the control cases, we
attempted to find patients of about the same age, the same civil status and
educational level as the first two groups.
448

Whereas all patients with endometriosis (group II) and all the control
patients (group III) who were invited to participate in this clinical research
completed the questionnaires, only 15 patients out of the 28 with chronic
pelvic pain without clear organic cause (group I) completed the psycho-
metric tests, The psychic functioning of these 3 groups of patients was
evaluated by the following psychological tests:
The Minnesota Multiphasic Personality Inventory (MMPI) of Hathaway
and Kinley (1951): the Dutch adjustment of the test by Nuttin and Beuten
(1963) was used. The MMPI gives the following list of clinical variables: Hs,
hypochondriasis; D, depression; Hy, hysteria; Pd, psychopathic deviation; Mf,
masculinity/feminity; Pa, paranoia; Pt, psychasthenia; SC, schizophrenia;
Ma, hypomania; Si, social introversion.
As validity scales, to check the honesty of the answers, were included: L,
lie; F, validity; K, correction. At the end of the test were added: Es, ego-
strength scale; R, repression scale; A, anxiety scale (Barron, 1953; Welsh,
1965).
The Amsterdam Biographical Questionnaire (ABV-V) of Wilde (1963)
with 4 scales: N, neurotic instability as manifested by the presence of
psychoneurotic complaints; NS, neurotic instability as manifested by the
presence of functional somatic complaints; E, social extraversion; T, test-
taking attitude.
Interpersonal Relations Questionnaire (VIR) of Vertommen and Rochette
(1978): This questionnaire examines the male-female relationship. Our
research concerned only the woman’s view of her marriage. On both the
reality and desired levels we examined how the wife felt about her husband
(I + husband) and what the wife supposed that the husband felt about her
(my husband -+ me). The variables used and the results obtained with this
questionnaire can be found in an earlier paper by Renaer et al. (1979).
The purpose of these tests was to explore the psychic background of the
3 groups of patients and at the same time to make a quantitative addition to
the psychopathological evaluations described in part C. IV, 2b.
A multivariate variance analysis was used to evaluate our findings, namely
the MANOVA computer program (Cooley and Lohnes, 1971). This analysis
compares the dispersions and centroids of the different groups. By rejecting
the nul-hypothesis concerning the centroid equality, one can, using the
univariate F values, determine which of the test variables best differentiates
between the various groups. Where appropriate, the Student’s t-test was done
for significance of difference.
When the results from the various groups were known, a comparison with
the means of the standardization group of the test was also done. That
standardization group is called here group IV, For comparisons which refer
to group IV no multivariate test could be performed, as we do not possess
the required detailed information. We thus tested the means by Fischer’s
t-test for uncorrelated means (Guilford and Fruchter, 1973).
In Table XI will be found the composition of the 3 groups regarding age,
civil status, and educational level of the patient.
449

TABLE XI Composition of the 3 groups:


I. Patients with chronic pelvic pain without obvious pathology
II. Patients whose pain was due to endometriosis
III. Control patients

Group I Group II Group III


(n = 15) (n = 22) (n = 23)

Age (in years)


<20 0 0 0
20-29 5 4 4
30-39 6 11 11
40-49 4 5 8
50+ 0 2 0
Civil status
Married 13 21 22
Unmarried 2 1 0
Divorced 0 0 1
Educational level
Grammar school 7 5 7
Junior high school 3 7 3
Senior high school 2 8 5
University 3 2 8

Group I : -
Group II : o----o
Group 111 : &_.._..d

Group IV : horizontal l8nc

20 I 1 1 I I I I I I I I I I I I I

L F K Hs D Hg Pd Mf Pa Pt SC Ma Si Es R A
M.M.P.I.-scales
Fig. 2. Graphic presentation of the means of the groups on the MMPI scales in t values.
450

(ii) Results:
The results for each psychological test will be discussed separately.
MMPI
Figure 2 gives a graphic presentation of the results. For group IV the mean
is 50 with a standard deviation of 10. The means and the standard deviations
on the MMPI for the different groups are given in an earlier paper (Renaer et
al., 1979), as are the multivariate variance analysis and t-test on the MMPI
for the different groups.
Multivariate analysis and t-test on the results show that groups I and II do
not differ significantly on the clinical scales of the MMPI, while both groups
I and II do differ from group III as well as from group IV (the standardization
group). The two control groups (III and IV) also appear to differ from each
other.
Figure 2 clearly shows similar profiles for groups I and II, the two pain
groups. The inflated scores on hypochondriasis, depression and hysteria
clearly contrast with the two control groups, while the former groups (I and
II) also display, when compared with group III, higher scores on Pt (psychas-
thenia), Si (social introversion) and A (anxiety), as well as lower scores on
Es (ego-strength). The scores of the two pain groups (I and II) clearly indi-
cate a neurotic behavior.
The difference between the two control groups lies principally in higher
scores for the patient control group (group III) on Hs (hypochondriasis),
and R (repression), and the lower score on Pd (psychopathic deviation) or A
(anxiety) and on the scales that rather denoted tendencies toward psychotic
behavior.
ABV-V
In Table XII are given the means and standard deviations in raw scores and
percentiles for the various groups, while these results are expressed in
percentile form in Figure 3. As stated above, the comparison cannot include
group IV, as the necessary data are not available.
From multivariate variance analysis on ABV-V for the different groups, it
appears that the two pain groups I and II do not differ from each other on
the ABV-V scales, nor do they differ from the patient control group (group

TABLE XII Means, standard deviations and percentiles on the ABV-V for the different
groups

Scales Group I Group II Group III


(n = 15) (n = 22) (n = 23)

x SD Pert. F SD Pert. K SD Pert.

N 76.8 28.7 80 73.8 22.7 77 64.4 23.6 68


NS 30.9 8.1 92 25.8 9.3 82 23.5 8.3 76
E 46.9 19.6 38 45.8 19.6 32 53.7 17.4 53
T 43.4 10.3 46 47.9 9.4 63 44.8 7.8 57
451

90 -

60 -

60 -

30 -
Group I : -
Group II: O-----o
20-
Group 111’ ib..-.. -.A
* Group IV: horizontal line
lo-
I I I
NS E T
ABVV-scales
Fig. 3. Graphic presentation of the results of Table XII in percentile form.

III). At a univariate level there is a significant difference between groups I


and II on the NS scale. Patients with pelvic pain without clear organic cause
display a stronger neurotic liability manifested by functional somatic com-
plaints than the patient control group (F = 6.73; significant at the 5% level).
Figure 3 clearly shows that the profiles from the two pain groups are very
nearly parallel, though there is a tendency for group I to be more neurotic
and less defensive than group II. However, this difference is not significant.
According to Figure 3, the patient control group differs from the norm
group by a heightened neurotic lability (N and NS). We were, however, not
able to test this difference for significance. As on the MMPI, the two pain
groups did not differ from each other significantly, while there is a differ-
ence between the two pain groups and the norm group. On the other hand,
in this test a difference between the patient control group and the norm
group is also indicated. We’ can thus conclude that also on the ABV-V the
pain groups answer as do neurotic patients, while the patient control group
shows a certain neurotic lability.
(iii) Discussion:
The two pain groups
It was our intention to administer the various tests to 28 patients with
chronic pelvic pain without clear organic cause. Whereas the patients of
groups II and III were very cooperative, several patients among the 28 of
group I were not, and only 15 completed the questionnaires. This leaves us
with the interesting problem as to precisely why patients of this group
showed such resistance to psychological examination. It is possible that the
patients who did not comply with our request had more psychological prob-
452

lems than those who completed the questionnaires. Greater numbers,


especially in group I, might have permitted the discovery of differences
between more variables in the various groups than those uncovered by the
statistical analysis, but this does not detract from the value of the present
results. It appears that our expectation: “The test will probably show a
significant difference between the variables concerning the psychic function-
ing of patients with chronic pain without a clear organic cause and those
with chronic pain with an organic cause” was not realized.
It is difficult to admit that chronic pain for which an organic cause has
been found occurs predominantly in patients with neurotic personality struc-
tures or neurotic reactions. We must therefore suppose that chronic pain
with an organic origin can lead to neurotic reactions. In fact the same con-
clusion has been drawn by a few earlier authors.
Bond (1971) studied 52 women with cervical carcinoma, who were sub-
divided into 3 groups. Patients of group I had no pain and did not receive
analgesics; group II patients complained of pain and did not receive
analgesics; group III had pain and requested and received analgesics. All the
patients completed the Eysenck Personality Inventory (EPI), the Cornell
Medical Index (CMI), which refers to past and present physical and emo-
tional symptoms, and the Whiteley Index, which measures the patient’s
attitudes towards his illness and environment. Pain-free patients (group I)
had low N (neuroticisms) and high E (extraversion) scores; group II had
high N and low E scores; group III had high N and E scores. The results
showed that neuroticism increases with chronic pain on an organic basis.
Woodforde and Merskey (1972) asked two groups of patients complaining
of chronic pain to complete the Middlesex Hospital Questionnaire (MHQ)
and the EPI. There were 27 patients with a known organic neurologic basis for
their pain (0 group) and 16 patients lacking evidence of organic disease
related to their symptoms (P or psychiatric group). On the MHQ, there were
no significant differences between the 0 and the P groups in total scores.
Both groups obtained scores comparable to those of psychoneurotic out-
patients. The neuroticism of the patients in both groups 0 and P is again
confirmed by the scores from N and E on the EPI, which were very similar
to those quoted by Eysenck and Eysenck (1964) for neurotic patients.
On the other hand, Merskey (1972) did not find that, by itself, the
presence of a complaint of pain raised neuroticism scores of psychiatric
patients on the Maudsley Personality Inventory. There is thus evidence
which suggests that pain of organic, but not of psychological, origin tends
to be associated with a relative increase in neuroticism. Amongst the various
hypotheses which may explain the results obtained by Woodforde and
Merskey in their 1972 paper, the authors believe that one has to admit that
chronic pain of organic origin causes emotional disturbance.
To demonstrate that there is a causal relationship between chronic pain
and emotional disturbance, or to show that pain is more likely to become
chronic in patients with previously existing neurotic personality structures,
would require personality testing before and after the onset of the pain,
453

which is almost impossible in practice. Therefore Sternbach and Timmer-


mans (1975) studied personality changes associated with reduction of pain
in 113 patients with pain lasting more than 6 months and attributable to
somatic lesions; 29 patients received surgery for pain relief and 84 did not;
both groups received psychological treatment and rehabilitation. The surgery
patients showed significantly greater reduction on the MMPI hysteria and
hypomania scales and on the invalidism scale of a Health Index, and this was
associated with a significantly greater reduction of pain. These results sup-
port the hypothesis that the neuroticism associated with chronic pain is the
result of it, and may be reversible when the pain is reduced or abolished.
Another paper indirectly substantiates the hypothesis of a causal relation-
ship between chronic pain of organic origin and neuroticism. Using the
MMPI, Sternbach et al. (1973) compared patients complaining of chronic
low back pain due to objective disorders with patients complaining of
chronic low back pain without clear organic cause. They did not find signifi-
cant differences between the two groups; they found, however, that patients
whose low back pain had lasted for less than 6 months displayed a lower
degree of neuroticism than those whose low back pain had lasted more than
6 months.
The conclusions of these various papers confirm Sternbach’s opinion
(1976) that, although the psychometric tests are useful aids in evaluating the
patient’s personality, “nothing in the battery of pain tests and personality
examinations can indicate whether a patient’s pain is psychogenic or
somatogenic”.
Our pain groups versus our control groups
When we examine the scores of our control group, we see that this group
differs on several variables from the group on which the instruments were
standardized. Apparently, group III should be considered as a control group
in a restrictive sense, i.e. in so far as these patients did not complain of pain.
To obtain an appropriate control group one must go outside the patient
population group. For this reason the scores of the 3 groups of our study
were compared with the scores of the standardization groups of the MMPI
adapted by Nuttin and Beuten (1963) and of the ABV-V.
The comparison between groups I and IV shows more clearly that group I
exhibits a neurotic symptomatology, with heightened scores on hypochon-
driasis, depression, hysteria and repression, and lower scores on Es (ego-
strength, MMPI) and with heightened scores on neurotic instability (ABV-V).
Similar results are found when group II is compared with group IV. The
comparison between our patient control group (group III) and group IV is
very interesting; in the patient control group we find a tendency towards
high scores on Hs (hypochondriasis) and low scores on Pd (psychopathic
deviation) and, more strikingly, clearly lower scores on tests that indicate
psychotic deviation: Pt (psychasthenia), SC (schizophrenia), Ma (hypomania),
Si (social introversion). On the other hand there was an increase on repres-
sion (MMPI). The deviations of group III from the norm group IV seem to
indicate that the exposure to a clinical situation, even for symptoms whose
454

non-malignity is stressed by the doctor, but which nonetheless call for a sur-
gical operation, promotes a person to ‘patient’ status with the attending fear,
uncertainty and denial (MMPI). The selection of control groups when study-
ing psychosomatic problems is therefore especially delicate.
(iu) Conclusion:
Evaluation of personality and behavior of patients with CPPWOP revealed
psychological problems or psychiatric disorders in most of them. According
to the psychometric findings of several authors, including our own, this
correlation does not allow to conclude that the psychological problems or
disturbances are primary and the pain complaints mere consequences. The
psychiatric interviews conducted on several series of cases have shown, how-
ever, that many patients had a family and personal history which made them
more prone to adopt an abnormal illness behavior (Pilowsky, 1978), or a
maladaptation to the ordinary or extraordinary stresses of their professional
or family life. The multifarious functional complaints presented by many
patients also point towards important psychogenic influences in the patho-
genesis of their complaints. On the other hand, the psychiatric interviews
conducted by Prill (1964) and by P. Nijs (this study) have shown that some
patients’ reactions and behaviors could not be considered abnormal.

V. Pa thogenesis of CPPWOP - Conclusion

1. General considerations
Any gynecologist who is interested in the pain mechanism of cases where
the pelvic pain almost certainly has an organic origin, as, for example, in
endometriosis, or adenomyosis or PID cases, wonders why certain patients
complain of severe pain whereas others with comparable lesions do not
complain at all. The solution of this problem would probably be a great
stride towards understanding the pathogenesis of CPPWOP. In pelvic pain
without obvious pathology there is an understandable tendency to look for
a psychogenic explanation; the more so as stressful life situations and
psychopathological problems are indeed commonly found in those patients’
histories.
There are, however, a few reasons militating against a too ready adhesion
to a simple psychosomatic explanation: indeed, correlation does not mean
cause. A causal relationship between a pain complaint and a particular
psychological situation may seem plausible in some instances but in many
cases of chronic illness “it is clearly recognized that illness onset is the out-
come of multiple characteristics of the individual interacting with a number
of interdependent factors in the individual’s social context in the presence
of a disease agent” (Rabkin and Struening, 1976).
We should beware of attributing too readily to psychogenic factors any
complaint for which we do not find a satisfactory physical cause. Primary
dysmenorrhea is an example of a condition which has frequently been
considered as having a psychogenic origin, although the evidence in favor
455

of this hypothesis is rather flimsy, and it now seems that this syndrome may
have various causes, there being good reasons to admit that the primary
mechanisms are pathophysiologic rather than psychogenic (Lennane and
Lennane, 1973). One also wonders whether the ‘irritable colon syndrome’
will in the end not fall apart into various forms determined by different
pathophysiologic factors: “It must be anticipated that with increasing
knowledge further examples of ‘organic’ disease will be separated off from
the irritable bowel syndrome” (Fielding, 1977). Stembach (1978) rightly
draws attention to the complexity of pain experience and expression, and
emphasizes that “the causative labels psychogenic and somatogenic need not
be, and frequently are not, mutually exclusive”.
Even if it is plausible that a patient’s complaints are to a great extent
influenced by psychologic issues, we still have to explain why precisely she
has lower abdominal pain and low-back pain. This localization may, in the
absence of a clear organic cause, have a psychological origin, for example
by the mechanism of identification or in symbolic terms; but in actual
practice purely psychogenic mechanisms seem to leave many cases of chronic
pelvic pain unexplained.

2. A working hypothesis
The tendency to look for a psychological explanation of CPPWOP is based
on the absence of clear organic or pathophysiological causes, on the high
prevalence of psychopathologic disturbances, and on the poor results of
various operations which have been performed on many of these patients.
It would, however, seem to us that the pendulum has swung too far when all
cases of CPPWOP are stamped as “mere neurotics”.
There are indeed many patients presenting with chronic pelvic pain and
with an array of functional symptoms in whom a meticulous search for a
somatogenic cause remains negative and in whom one finds, on the other
hand, personality and behavior disturbances. But, when repeated psychologic
evaluations by a gynecologist and one or more interviews conducted by a
psychiatrist do not demonstrate any personality disorder or maladaptive
behavior, one has to admit that the cause of the pain is not primarily
psychogenic but most probably organic or pathophysiologic, even though
for the time being the pain mechanism is not clear. Between those extremes
there are oligosymptomatic patients, who have had a dreadful life history
or who are burdened by a stressful life situation, where one is wondering
which factors, organic or psychologic, are preponderant in the genesis of the
complaint.
We also saw that some patients with a tender and even shortened posterior
parametrium do not present with spontaneous pain but only complain of
deep dyspareunia.
In order to explain those diverse observations, we suggest that patients
with CPPWOP are a heterogeneous population presenting a spectrum of
miscellaneous conditions. At one end there seem to be patients with very
little or even no peripheral noxious stimulation and a preponderance of
456

psychogenic factors due either to a psychopathologic personality or to actual


adverse life circumstances; in those patients the complaints would to a large
extent have a psychological explanation. The other extreme would be made
up by persons with rather intense peripheral noxious stimulation, whose
nature may be either a pelvic circulatory disturbance or a tenderness of the
posterior parametrium and, less often, uterine cramps or a tear in a utero-
sacral ligament, and few or no psychologic influences. Between these two
extremes will be found many cases in whom the complaints are conditioned
by less-pronounced peripheral noxious stimulation combined with one or
more of the great variety of psychophysiologic mechanisms which may
contribute to the complex stimulus constellations that induce complaints
and care-seeking behavior (Rabkin and Struening, 1976; Chapman, 1978;
Sternbach, 1978).
D. FOLLOW-UP OF PATIENTS WITH CPPWOP

The follow-up of the 24 patients studied in part C. IV, 2a is interesting


both from the point of view of pathogenesis and of results of management.
All those patients have been followed for at least 1: years and many of them
for 4 years or more. Follow-up of chronic pain patients after any form of
treatment should, indeed, last for longer than a few months, in order to be
able to discount the temporary placebo effect which may result from any
medical treatment.
Thirteen patients out of the 24 were symtomless or felt distinctly better;
in 3 out of these 13 the pain had disappeared since their stay in the clinic
after their having been told that all objective explorations had been negative.
The 10 others became distinctly better under conservative medical treat-
ment. Six patients kept complaining of pain, but their subsequent behavior
confirmed that the symptoms had essentially a psychogenic origin. One
patient, no. 20, had been better for one year under conservative medical
treatment, but started complaining again later; she underwent a hyster-
ectomy and has felt better since this operation. Patient no. 21 went on
complaining of lower abdominal pain for several years; when the pain locali-
zation shifted to the left iliac fossa, a left ovarian tumor was discovered
and a left salpingo-oophorectomy was performed for an endometriotic
ovarian cyst; this endometriotic lesion does not seem to have been the cause
of the earlier complaints. Another patient, no. 22, could only be contacted
by mail; 6 years after the diagnostic laparoscopy, she still is complaining of
lower abdominal pain, and she insists on having a hysterectomy. Patient no.
22 had a total non-radical hysterectomy elsewhere some 2 years after her
exploration in our clinic; 8 months after this operation her dyspareunia was
better, but she still had the same spontaneous pain as before. Patient no. 24
underwent a hysterectomy elsewhere shortly after her stay at the clinic and
felt better for the next 2 years; later on she felt a bearable pain in the lower
abdomen “as if the uterus was still there”. Compared with the results we
used to obtain when we performed hysterectomies much more readily, we
are rather pleased with these results.
451

E. THERAPEUTIC STRATEGY

The concept ‘therapeutic strategy’ (Almy, 1977) is preferred to the term


‘treatment’ as it stresses the complexity of management of CPPWOP. In our
review of the pathogenesis it was concluded that the syndrome frequently
seems to have a plurifactorial pathogenesis. Some doctors consider these
patients as “mere neurotics”; others feel that there is a simple organic
explanation for the pain, and tend to apply a ‘straightforward’ therapy
to erase or correct the supposed anomaly. Both opinions disregard the com-
plexity of the situation.
In order to evaluate the results of any treatment, medical or surgical, of
chronic pain syndromes the patients should be followed for at least 1 or 2
years. It is indeed well known that patients with this condition who ask for
an operation are usually pleased with the results for the first few months,
only to start presenting the same or other complaints after some time.

1. Establishing an efficient therapeutic relationship


A doctor who is not prepared to take the pain symptoms and the other
complaints seriously will not be able to help these patients. He may either
traumatize them by saying or intimating that the symptoms are “just
nerves”, or frighten and discourage them by stating that there is “an inflam-
mation” or “varices” or “a tear” which will probably have to be operated
upon “because they rarely disappear without an operation”.
It is indispensable to take time to listen to the patient’s description of her
symptoms and her problems. Most patients have already consulted one or
more physicians. It will generally be useful to see these patients more than
once, in order to verify whether the symptoms remain the same or vary and
whether, apart from the complaints at the first visit, there are any others
which modify the clinical picture. These visits will also enable the doctor to
carefully study their behavior and personality. It is most helpful if one can
obtain information from their general practitioner concerning family and
personal history, marital life and general behavior. Enquiries should be made
about the diagnosis formulated and the treatment proposed or performed
by other doctors.
It is imperative to look for any non-gynecologic or any organic gyneco-
logic disorder which could explain the symptoms. When conservative medical
treatment prescribed on a probability diagnosis has remained unsuccessful,
a laparoscopy should be performed in order to establish a correct diagnosis
and to alleviate more efficiently the anxiety of the patient. This diagnostic
exploration sometimes suffices to reassure the patient and to raise her
“complaining threshold”. It should be emphasized, however, that in many
patients “the reassurance that cancer or other serious disease are not present,
has a short half-life” (Almy, 1977).
This multiphasic diagnostic exploration will serve to discover the prob-
lems, the personality and the reactions of the patient and to try to locate her
on the spectrum of possibilities between a purely psychogenic syndrome and
458

one which seems to have a definite, albeit at present unclear, pathophysio-


logic explanation.

2. Psychologic evaluation and treatment


As CPPWOP is frequently associated with psychological problems or with
personality disorders, it is important to examine the relationship between
psychologic factors and the pain complaints. Evaluating this relationship
may sometimes be easy, but is usually complicated. A few examples of the
mechanisms which may come into play follow. In 3 out of the 24 patients
referred to under ‘follow-up’, the pain disappeared in two and became more
bearable in one after the laparoscopy which confirmed the absence of
lesions. It is probable that in those cases the pain experience was to a large
extent conditioned by anxiety. In another patient it seemed to be influenced
by an identification reaction with a friend who had died of cancer after
having suffered from chronic lower abdominal pain.
There is abundant evidence that pain complaints may be closely related
to psychiatric illness (Merskey and Spear, 1967; Merskey, 1972; Pilowsky,
1978); the most frequent associations are with depression or with anxiety.
Pain is not infrequently used as a hysterical conversion symptom; it may
also be a manifestation of hypochondriasis. Whenever such an association is
probable, treatment should address itself to the psychiatric condition. This
may be attempted by the gynecologist, or the patient may be referred to a
psychiatrist. Such a referral should, however, not be resorted to too readily
because it may result in a breach of the therapeutic relationship between the
patient and the gynecologist. When a referral is deemed necessary it is in the
best interest of the patient for a close collaboration to be maintained
between the gynecologist and the psychiatrist.

3. Medical treatment
Signs of cervical infection should be treated with systemic antibiotics. If
there is a concurrent tender posterior parametrium, there is the possibility
that one is not dealing with CPPWOP in a stricter sense, but with a posterior
parametritis due to cervicitis; local heat in the form of hot sitzbaths or
diathermy will frequently be useful.
Some patients have been helped with cyclic estroprogestogens, others by
a hypoestrogenic amenorrhea obtained by continuously administering
progestogens, e.g. lynestrenol 5 mg/day or norethisterone acetate 5 mg/day
over several months. If the pain disappears under continuous progestogen
treatment, one may try to bring the daily dose down to 2.5 mg/day in order
to reduce the anabolic effect of the drug.
Even if the spontaneous pain disappears under estroprogestogens or
progestogens, deep dyspareunia may persist in those cases which present
a tender posterior parametrium; it will frequently be alleviated by avoidance
of deep penetration during intercourse. One may, for example, suggest to
the patient that she adduct her thighs after penetration, or one may
recommend the lateral vis-a-vis or the lateral atergo position for intercourse.
459

Several German and some Anglo-Saxon authors (Young, 1947) have used
infiltrations of the uterosacral ligaments either with a local anesthetic or
with a depot anesthetic in order to block the inferior hypogastric plexus or
the hypogastric nerves. Many years ago we tried this treatment on many
patients but have abandoned it, as infiltration of the tender tissues is pain-
ful and usually provides only short-lived pain relief.

4. Surgical treatment
Although surgical interruption of the pain-conducting nerve tracts would
a priori seem logical, few gynecologists have obtained lasting results either
with the resection of the superior hypogastric plexus (Cotte’s operation,
1931) or with the transection of the uterosacral ligaments in order to inter-
rupt the hypogastric nerves (Doyle, 1955). Having hardly ever seen a true
tear in a uterosacral or a broad ligament, we have only once performed a
suture of a ‘depression’ in a posterior leaf of a broad ligament, with poor
results.
We have never ligated any varicosities of the infundibulopelvic ligaments
or of the uterine veins. Several gynecologists who have used this conservative
sort of operation have been less than pleased with the results, and have
stopped performing it (personal communications E. De Muylder, Brussels,
and H. Frangenheim, Konstanz).
Wedge resections of so-called ‘sclerocystic ovaries’ have but seldom
relieved chronic lower abdominal pain; they have frequently been followed
by recurrence of the pain, and subsequent scars and adhesions have reduced
the fertility chances of countless women. Wedge resection with this ‘indica-
tion’ is therefore mentioned only to be condemned.
Painful mobile uterine retroflexion has deliberately been eliminated from
our discussion of the subject. A suspension operation will but rarely be
indicated; a causal relationship between the retroversion and the pain will
only be recognized by disappearance of the pain after bimanual reduction of
the retroverted uterus; this should be maintained by a Smith or Hodge
pessary .
In cases of CPPWOP with a unilateral pain localization a salpingo-oopho-
rectomy will only rarely be efficient; therefore a normal-looking ovary
should but very seldom, if ever, be excised for chronic or recurrent unilateral
pain. On the other hand, total hysterectomy has been commonly used by
many gynecologists. Taylor (1961) performed a hysterectomy in 9 cases out
of 114 with chronic pelvic pain without evidence of pathology and obtained
good results in all of them; he stressed, however, that several conditions
should be fulfilled before resorting to this operation. According to Mills
(1978), a hysterectomy can be strongly recommended in severe cases of
CPPWOP: it gives “immediate, dramatic and persistent relief”. Before the
era of the contraceptive pills and the oral progestogens, we used to perform
many hysterectomies for chronic lower abdominal pain only to become dis-
couraged by the rather low percentage of lasting good results. As many
others, we have progressively become more reluctant to perform a hysterec-
460

tomy in these cases. At present, we only perform a total (never a sub-total!)


hysterectomy under the following conditions: (1) the patient should have
completed her family; (2) she should have been treated conservatively for
several months or years; (3) she should be oligosymptomatic; (4) no reserva-
tions should have been formulated by our psychiatrists.
Conclusions may only be drawn after a long-term follow-up. Our own
follow-up of a series of 24 patients may not constitute an unqualified
success, but the 6 patients who kept complaining during our trials of con-
servative therapy would most probably not have been cured by total hyster-
ectomy either.

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