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Asynchronous Bilateral Ovarian Torsion. A Case

Report and Mini Review

Article in Journal of pediatric and adolescent gynecology · September 2013

DOI: 10.1016/j.jpag.2013.06.016 · Source: PubMed

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Mini-Review

Asynchronous Bilateral Ovarian Torsion. A Case Report and Mini Review

Emel Kurtoglu MD 1, Arif Kokcu MD 1,*, Murat Danaci MD 2
1
Department of Obstetrics and Gynecology of Medicine, University of Ondokuz Mayis, Samsun, Turkey
2
Department of Radiology, School of Medicine, University of Ondokuz Mayis, Samsun, Turkey

a b s t r a c t
Background: Adnexal torsion is a serious condition and delay in surgical intervention may result in loss of ovary. Children and adolescents
who have suffered from uterine adnexal torsion may be at risk for asynchronous torsion of the contralateral adnexa.
Case: We report the case of asynchronous bilateral ovarian torsion in a 9-year-old girl, resulting in right and subsequently left salpingo-
oophorectomy.
Conclusion: The diagnosis of ovarian torsion often is delayed. When ovarian torsion is suspected, laparoscopy should be performed without
delay, and conservative management should be strongly considered to prevent surgical castration. Oophoropexy of the ipsilateral and
contralateral ovary should be considered to prevent a recurrent torsion.
Key Words: Adolescent, Asynchronous, Bilateral, Girl, Ovarian torsion

Introduction necrosis in both adnexa. Emphasize the importance of early

Adnexal torsion often occurs due to adnexal pathology
such as ovarian cysts and tumors and is rare in young girls
while torsion of normal ovaries is more commonly seen in
children and adolescents. Once a child has lost one ovary,
there is a risk of asynchronous torsion of contralateral ovary
which may result in catastrophic sequelae.1e3 Bilateral
ovarian torsion was first reported by Warnek in 1895.4 Since
that time there have been 16 reported cases of bilateral
torsion.5 Asynchronous bilateral adnexal torsion, first
described by Baron in 1934, is a very rare entity in childhood
and adolescence.3 We searched the PubMed/Medline
databases for the previous case reports using the key words
"Asynchronous", "Bilateral" and "Ovarian torsion." We
found a total of 22 cases of asynchronous bilateral ovarian
torsion reported between 1934 and 2008. Ozcan et al1
reviewed the English-language literature in point of asyn-
chronous bilateral adnexal torsion in children and adoles-
cents and they were able to document 17 such cases. The
age of patients ranged from 3 to 12 years on first presen-
tation, and from 6 to 17 years on second presentation.
Beaunoyer et al6 reported four cases with asynchronous
bilateral adnexal torsion. The mean age at presentation was
10.6 years and the mean interval between the two episodes
of ovarian torsion was 15.7 months. Svensson et al7 re-
ported a 6-year-old girl with asynchronous bilateral ovarian
torsion. Interval between the two episodes was 3 years.
Herein, we present an extremely rare case of asynchro-
nous bilateral ovarian torsion in an adolescent who was
surgically castrated because of the gross evidence of total
The authors indicate no conflicts of interest.
* Address correspondence to: Arif Kokcu, MD, University of Ondokuz Mayis, School
of Medicine, Department of Obstetrics and Gynecology, Kurupelit, 55139 Samsun,
Turkey; Phone þ90 0362 3121919-2452; fax: þ90 362-4576029
E-mail address: arifkokcu@yahoo.com (A. Kokcu).
1083-3188/$ - see front matter Ó 2014 North American Society for Pediatric and Adolescent Gynecology. Published by Elsevier Inc.
http://dx.doi.org/10.1016/j.jpag.2013.06.016
diagnosis and conservative management of ovarian torsion
in adolescents or children.
Case Report
We report a 12-year-old who presented to our gynecology
clinic due to absence of secondary sex characteristics:
At the age of 9, she was admitted to the hospital emer-
gency service with abdominal pain, nausea, and vomiting,
existing for 3 days. She had a history of right salpingooo-
phorectomy and appendectomy due to torsion of a “normal
ovary” a month before at another hospital. On physical
examination, there were rebound and tenderness in all
quadrants of the abdomen. Laboratory tests including
hemogram, biochemistry, and urine investigations, all were
within normal limits except slightly elevated white blood
cell count and anemia. Pelvic ultrasonography revealed the
left ovary measuring 58
46
48 mm with anechoic cysts,
the largest of which was 16
12 mm in diameter, located in
the periphery of the ovary. Color Doppler ultrasonography
showed absence of circulation in the left ovary, supporting
the diagnosis of ovarian torsion.
The patient underwent exploratory laparotomy by
pediatric surgeons. At abdominal exploration, the left ovary
was found to be torsed twice, cyanotic, and necrotic.
Detorsion and “hot application” was performed but no sign
of circulation was observed, so the patient underwent
left salpingooophorectomy. There were no postoperative
complications and she was discharged on postoperative
day 2.
Three years later, at the age of 12 years, she presented to
our gynecology clinic due to absence of secondary sex
characteristics. On physical examination, she did not have
signs of development of secondary sex characteristics,
including telarche, pubarche, and menarche. The absence of
ovaries was imaged by magnetic resonance imaging. Her
 E. Kurtoglu et al. / J Pediatr Adolesc Gynecol 27 (2014) 122e124
FSH and estradiol levels were 94.53 mu/mL and 5 pg/ml
respectively. Hormone replacement therapy was initiated
with an oral drug containing estradiol valerat (2 mg/day)
and norgestrel (0.5 mg/day).
Discussion
The mechanism of adnexal torsion is not known with
certainty. Most cases are secondary to adnexal pathology.
Although excess rotation of the adnexa can be explained
readily to be caused by large masses of associated ovarian
pathology, torsion of the normal uterine adnexa is less
comprehensible. In contrast to the adult, the pediatric
patient may have normal ovaries in up to 25% of the
cases.3 Excess mobility of the adnexa because of an
abnormally long fallopian tube, mesosalpinx, or meso-
varium, adnexal venous congestion as in premenarchal
activity, and jarring movement of the body are possible
mechanisms that have been suggested in torsion of
normal (uterine) adnexa. Adnexa appear to be particularly
prone to torsion in the early pubertal years. Functional
ovarian cysts are very common during these peripubertal
years and may be a predisposing cause of adnexal
twisting.1e3
Torsion of the normal adnexa is a challenging entity to
diagnose. At the time of surgery most children are sus-
pected of having acute appendicitis. Both appendicitis and
ovarian torsion can present with lower quadrant pain,
peritonitis, and leukocytosis. Patients with ovarian torsion
can be febrile, particularly in cases with tissue necrosis. The
right adnexa is more frequently involved than the left in
torsion by about 3:2. Most likely the sigmid colon’s mass
effect helps prevent left adnexal twisting. One historical
difference between torsion and appendicitis is that half of
the patients with torsion had suffered episodes of similar
abdominal pain in the past. Most of adnexal torsions occur
in the 7 to 10 year age range. Functional ovarian cysts are
very common during these peripubertal years and may be
a predisposing factor for adnexal torsion.8 Early diagnosis
and high index of suspicion may permit salvage of more
twisted ovaries. Hence, every girl presenting with nonspe-
cific lower abdominal pain should be evaluated for ovarian
torsion. It is a diagnosis based on clinical and radiologic
findings. Ultrasonographic scan remains the most useful
investigation, but blood flow on Doppler ultrasonography
scan does not rule out an ovarian torsion.6 Although color
Doppler ultrasonography can detect the status of adnexal
circulation, presence of normal flow does not exclude
torsion. Thus, this confusion may lead to delay in early
diagnosis and gonadal loss. Patients with ovarian torsion
have an echogenic mass observed on ultrasonography.
Although the echogenic mass may be confused with an
appendiceal abscess, improved ultrasonography now allows
a more confident preoperative diagnosis of ovarian torsion.
Torsion of the adnexa apparently disrupts venous and
lymphatic flow, which can result in ovarian enlargement
secondary to edema. Ovarian edema is a stage in the process
of ovarian torsion. If twisting does not resolve spontane-
ously, necrosis of the ovary occurs. Ultrasonographically
visualized cul-de-sac fluid is usually a late manifestation of
123
torsion and is associated with hemorrhagic nonviable
ovarian tissue.3,8
Although traditional treatment advocates removal of the
twisted adnexa, the treatment in this manner is not always
desirable for children and adolescents. More recent litera-
ture contains cases of conservative therapy. The impact of
bilateral oophorectomy on the future physical, social, and
emotional development of a prepubescent girl cannot be
underestimated.5,9 Further, the decision to perform salpingo-
oophorectomy should not be based on the color and con-
sistency of the adnexa; dark hemorrhagic edematous
appearance of the torsed adnexa is the result of ovarian
engorgement, secondary to venous stasis. Complete arterial
obstruction probably does not occur in most cases.2 Detorsion
and oophoropexy in unilateral torsion in children even in
delayed diagnosis are recommended. Various methods of
oophoropexy have been performed. The ovary is either
attached directly to the pelvic sidewall, the posterior aspect of
the uterus, or the uterosacral ligament, or the utero-ovarian
ligament is foreshortened via plication. The ovary may be
fixed with absorbable suture. Also, the use of permanent
suture has been suggested for fixation of the ovary to the
pelvic sidewall.8 Oophoropexy must be considered at the
time of the first oophorectomy to prevent torsion of the
remaining enlarged ovary. Contralateral oophoropexy in
addition to detorsion and pexy of the affected ovary must be
performed to prevent sequential bilateral adnexal torsion.
After detorsion of the ovary, the key point is detection of
the viability. Serial color Doppler ultrasonography can be
used to confirm the viability of the detorsioned ovary.2,5
Svensson et al7 suggested that hyperbaric oxygen could be
an adjunct in the treatment of the ischemic adnexa after
oophoropexy and detorsion in selected cases. They stated
that hyperbaric oxygen therapy induces vasodilatation and
increased perfusion in injured tissues. Increased tissue
oxygen tension also leads to improved function of leukocytes
and general antiinflammatory effects. Styer and Laufer10
suggested the bivalving procedure in the case of severely
hemorrhagic and edematous adnexa. For the bivalving
procedure, a linear incision is made along the antimesenteric
aspect of the affected ovary after untwisting. This procedure
helps to identify viable tissue within hemorrhagic, ischemic
areas, and releases the increased pressure of the edematous
ovarian capsule to facilitate lymphatic and venous drainage
and allow adequate arterial flow.
The most commonly encountered complication of
conservative management is postoperative fever. This can
be managed by antipyretics and resolves spontaneously in
a few days after the operation. Proponents of adnexectomy
have cited a theoretical risk of thromboembolic events at
the time of unrotating a torsioned adnexa. However, this
complication has not been seen in multiple studies to date.
Another particular concern with conservative management
is the possibility of leaving a malignancy in situ. However, if
there is no tumor seen at exploration, the ovary can be left
in place, or biopsies can be performed if there is any
suspicious macroscopic appearance of a tumor.1,5
We have presented an extremely rare case of asynchro-
nous bilateral ovarian torsion in a child who underwent
oophorectomy. Strong consideration should be given for
124 E. Kurtoglu et al. / J Pediatr Adolesc Gynecol 27 (2014) 122e124
oophoropexy of the ipsilateral and contralateral ovary by
laparoscopy. Macroscopic appearance of the ovary is not
a reliable indicator of the degree of ischemia. Early diag-
nosis maximizes the success of conservative therapy.
Although the detection of normal flow by Doppler sonog-
raphy does not exclude an ovarian torsion, it still is the most
useful noninvasive diagnostic modality, which could lead to
early operative intervention.
References
1. Ozcan C, Celik A, Ozok G, et al: Adnexal torsion in children may have
a catastrophic sequel: asynchronous bilateral torsion. J Pediatr Surg 2002; 37:
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2. Celik A, Ergu € n O, Aldemir H, et al: Long-term results of conservative
management of adnexal torsion in children. J Pediatr Surg 2005; 40:704
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