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Case report

Ruptured ovarian ectopic pregnancy presenting with


an acute abdomen
Louise Dunphy  ‍ ‍,1 Frances Wood,1 Joanne Hallchurch,2 Gill Douce,2 Shanthi Pinto1

1
Department of Obstetrics, SUMMARY a plethora of common gynaecological disorders
Leighton Hospital, Crewe, UK An ectopic pregnancy occurs in 2% of all pregnancies. can present with the aforementioned symptoms
2
Department of Histo-­Pathology, A primary ovarian ectopic (OP) is a rare entity and such as a ruptured ovarian cyst, a tubal ectopic
Royal Stoke University
occurs in <2% of all ectopic gestations. It may present pregnancy, spontaneous miscarriage or dysmen-
Hospital, University Hospitals
in those individuals who take ovulatory drugs, use orrhoea. Risk factors for an ovarian ectopic (OP)
of North Midlands NHS Trust,
Staffordshire, UK an intrauterine device or have undergone in vitro are similar to those for a tubal pregnancy, but
fertilisation or embryo transfer. Multiparity and a younger use of an intrauterine device (IUD) appears to be
Correspondence to age are other recognised risk factors. Diagnosing an disproportionately associated. An IUD prevents
Dr Louise Dunphy; OP pregnancy remains a challenge and it may be uterine implantation but does not provide protec-
​Louise.​Dunphy@​doctors.​org.​uk misdiagnosed as a bleeding luteal cyst, a haemorrhagic tion against ovarian implantation.3 Cheng’s multi-
ovarian cyst or a tubal pregnancy by ultrasound scan. centre, large sample, case–control study has shown
Accepted 18 October 2022 The diagnosis is often only established at laparoscopy that the risk of an ectopic pregnancy is increased
following histopathological examination. A ruptured following the failure of most contraceptives used
OP is a potentially life-­threatening condition due to its in the current cycle including IUDs (OR of 16.43),
potential for haemorrhage and hemodynamic collapse. oral contraceptive pills (OR 3.02), levonorgestrel
Hence, early diagnosis is crucial to prevent serious emergency contraception (OR 4.75) and female
morbidity and mortality. The authors present the case sterilisation (OR 4.73).4 An adnexal mass may be
of a multiparous woman in her late 30s presenting present on examination. Preoperative diagnosis is
with a seizure and lower abdominal pain at 6 weeks challenging as it shares a similar clinical presenta-
gestation. Her beta human chorionic gonadotropin was tion with a complicated ovarian cyst, a tubal ectopic
>9000 Miu/mL. A transvaginal ultrasound scan showed or a haemorrhaging corpus luteal cyst. Laparoscopy
no evidence of an intrauterine pregnancy. There was is the gold standard approach. The diagnosis is
free fluid in the pelvis. She was hemodynamically stable. established using the modified Spiegelberg criteria,
She underwent a diagnostic laparoscopy, which showed confirmation of non-­involvement of the fallopian
hemoperitoneum and a ruptured left OP pregnancy. She tube and histological evidence of chorionic villi in
underwent a left oophorectomy. Histology confirmed the ovary.5 Most cases terminate with rupture in
chorionic villi within the ovarian stroma. This case the first trimester. However, rare cases of an OP
demonstrates the challenges in preoperative diagnosis resulting in a live neonate have also been described.
of a ruptured OP pregnancy and acts as a cautionary
reminder that individuals can present with hemodynamic
CASE PRESENTATION
stability. Rarely, as in this case, an OP pregnancy can
A woman in her late 30s presented to the acute
occur without the presence of risk factors. Despite its
medicine unit at 6 weeks gestation by her last
rarity, a ruptured OP pregnancy should be considered
menstrual period with a seizure following alcohol
in the differential diagnosis of women of reproductive
withdrawal. Her medical history included anxiety,
age presenting to the emergency department with acute
depression and alcohol dependence. Her surgical
abdominal pain and a positive pregnancy test.
history was unremarkable. There was no gynae-
cological history of intrauterine contraceptive
devices, hormonal contraception, fertility treat-
BACKGROUND ment or assisted reproductive techniques. She
An ectopic pregnancy is the leading cause of denied a history of pelvic inflammatory disease,
maternal death during the first trimester of preg- endometriosis or previous ectopic pregnancy. She
nancy, accounting for 10% of all pregnancy-­related was gravida 6, parous 2 (3 miscarriages at <6
deaths.1 An ovarian pregnancy, a rare form of non-­ weeks gestation). Her obstetric history included
tubal ectopic pregnancy occurs when the gestation three normal deliveries at term. She had an irreg-
sac is implanted, grown and developed in the ovary. ular menstrual cycle. She was a smoker but denied
It was first described by Saint Maurice in 1682.2 smoking for 3 months prior to conception. She
© BMJ Publishing Group
Limited 2022. No commercial
Although its pathogenesis remains to be fully eluci- had a history of binge drinking alcohol. Following
re-­use. See rights and dated, possible hypothesis include interference with termination of her seizure with intramuscular
permissions. Published by BMJ. ovum release from the ruptured follicle, fallopian (IM) lorazepam, she commenced treatment with
tube malfunction or inflammatory thickening of chlordiazepoxide and pabrinex. Twenty-­four hours
To cite: Dunphy L, Wood F,
Hallchurch J, et al. BMJ Case the tunica albuginea. The majority of cases are following admission, she developed lower abdom-
Rep 2022;15:e252499. diagnosed by the seventh week of pregnancy when inal pain and right shoulder tip pain. There was
doi:10.1136/bcr-2022- the patient develops the classic clinical symptoms no history of vaginal bleeding. She was hemody-
252499 of abdominal pain and vaginal bleeding. However, namically stable. Physical examination showed no
Dunphy L, et al. BMJ Case Rep 2022;15:e252499. doi:10.1136/bcr-2022-252499 1
Case report

Table 1  Laboratory investigations on admission showed an


electrolyte imbalance and a raised BHCG
Full blood count Bone profile
Haemoglobin 123  g/L(115-­165) Calcium 2.31 mmol/L (2.20–2.60)
White cell count 12.2×109/L (4–11) Alkaline phosphatase 39 iu/L (30-­150)
Platelets 164×109/L (150–450) Phosphate 0.72 mmol/L (0.8–1.5)
Mean cell volume 102.5 fL (80–100)
Liver function tests
Renal function tests Albumin 44  g/L(35-­50)
Sodium 134  mmol/L(133-­146) Alanine transaminase 47 iu/L(10-­50)
Potassium 2.8 mmol/L (3.5–5.3) Total bilirubin 20 umol/L(0–21)
Urea 3.8 mmol/L (2.5–7.8) Amylase 118 iu/L(0–110)
Creatinine 118 umol/L (50–100) Figure 2  Ultrasound scan of the left ovary.
EGFR 51 mL/min/1.73 m2 (60–150) Magnesium 0.69 mmol/L(0.7–1.00)
BHCG 9436 mIU/mL (0–5)
TREATMENT
BHCG, beta human chorionic gonadotropin; eGFR, estimated glomerular filtration rate. She was transfused two units of packed red blood cells. An
indwelling urinary catheter was inserted. She received analgesia
and antiemetics.
adnexal excitation or mass. The cervical os was closed. There
was no evidence of vaginal bleeding. OUTCOME AND FOLLOW-UP
She underwent a diagnostic laparoscopy. An infraumbilical
INVESTIGATIONS incision was performed. Low pressure, high flow pneumo-
Laboratory investigations on admission showed electrolyte imbal- peritoneum was established. Hemoperitoneum was evident.
ances (K+ 2.8mmol/L, phosphate 0.72 mmol/L and magnesium Both fallopian tubes and the right ovary appeared normal.
0.69 mmol/L), which had been corrected. Her urine pregnancy The left ovary showed a ruptured ectopic with active bleeding
test- was positive and her beta human chorionic gonadotropin (figures 3 and 4). Saline wash and suction of the peritoneum was
(BHCG) was also elevated at 9,436 indicating a definitive preg- performed. Her BHCG (10, 141 iu/L) and ovarian findings were
nancy (table 1). Her haemoglobin was within normal range at consistent with a ruptured ovarian pregnancy. As there was no
123 g/L. An arterial blood gas confirmed a metabolic acidosis. evidence of haemorrhagic infiltration of the suspensory ligament
An early pregnancy viability scan (transvaginal ultrasound) or haematoma formation at the level of the broad ligament, a
was performed. There was no evidence of an intrauterine preg- left oophorectomy was performed using Vyant diathermy. The
nancy. The endometrium appeared regular with an estimated pedicle was checked for haemostasis. The estimated blood loss
thickness of 9.8 mm. There was extensive free fluid within the was 1.5 L. Her post-­transfusion haemoglobin was 97 g/L, there-
pelvis extending to the lower liver margin (figure 1). Hence, fore, she commenced treatment with oral iron tablets, ferrous
evaluation of the adnexa was difficult and the left ovary could sulphate 200 mg two times per day. On histological examina-
not be visualised optimally (figure 2). tion, the left ovary showed surface rupture with haemorrhage
measuring 18 mm in maximum dimension. No fetal parts were
DIFFERENTIAL DIAGNOSIS visualised macroscopically. Thrombus attached to the surface of
As the adnexa and the left ovary could not be visualised, the the ovary contained chorionic villi and the implantation site was
diagnosis of an ectopic pregnancy could not be excluded. A identified (figures 5 and 6). There was no evidence of tropho-
haemorrhagic cyst or a bleeding corpus luteal cyst was also blastic disease. Her recovery was uneventful. The remaining
considered in the differential diagnosis. Her recent BHCG was ovarian tissue should help preserve normal fertility.
10,141 iu/L (range: 0–5) and a repeat haemoglobin was 77 g/L
(range: 115–165), from an admission haemoglobin of 123 g/L. DISCUSSION
Therefore, the diagnosis of a ruptured ectopic pregnancy was An ectopic pregnancy occurs in up to 2% of all pregnancies.6
considered most likely. She remained hemodynamically stable. Indeed, it is among the leading cause of maternal morbidity

Figure 1  The ultrasound scan showed free fluid in the pelvis. Figure 3  Diagnostic laparoscopy showed hemoperitoneum.
2 Dunphy L, et al. BMJ Case Rep 2022;15:e252499. doi:10.1136/bcr-2022-252499
Case report

Figure 4  Diagnostic laparoscopy showed a left ovarian ectopic Figure 6  Chorionic villi were noted confirming the diagnosis of a
pregnancy. ruptured ovarian ectopic pregnancy.

An OP may present with a history of amenorrhoea, abdominal


and mortality. Approximately, 98% of ectopic pregnancies are pain, vaginal bleeding and a positive pregnancy test. Chronic
located in the fallopian tubes, with 70% occurring in the ampulla. pelvic pain frequently occurs. Individuals may present with
Primary OP pregnancy is the implantation of the gestation sac hypovolaemic shock secondary to an acute intra-­ abdominal
in the ovary. A secondary OP occurs if fertilisation takes place haemorrhage, although women may be hemodynamically stable,
in the tube with posterior regurgitation of the conceptus back as in this case. A ruptured ectopic may present with dizziness,
to the ovarian stroma.7 An OP pregnancy is a rare occurrence syncope or hemorrhagic shock. Speculum and bimanual exam-
with an estimated incidence of 0.5%–3% of all ectopic preg- ination may reveal an adnexal mass.
nancies.8 The first case was described by St. Maurice in 1682.2 Despite advances in modern sonography, a ruptured OP is still
The mean gestational age at diagnosis is 7 weeks.9 Ninety-­one difficult to diagnose on preoperative ultrasound scan. Indeed,
per cent of cases of OP are diagnosed and managed in the first the diagnosis of an OP is only established preoperatively in
trimester due to frequent ovarian rupture, 5.4% continue into between 5.3% and 25% of cases.14 Ultrasonography may show
the second trimester and 3.7% will reach the third trimester.10 a wide hyperechoic ring or mass formed by gestational tropho-
The risk factors for developing an OP include the use of an IUD cytes infiltrating into the surrounding tissues, which is greater
such as the mirena coil, a history of tubal surgery, a previous than the echogenicity of a normal ovary and corpus luteum.15
ectopic pregnancy, endometriosis, pelvic inflammatory disease Other findings may include a complex adnexal mass with or
or ovarian hyperstimulation during assisted reproductive tech- without fluid in the pouch of Douglas and ovarian enlargement.
nology.11 In vitro fertilisation is associated with 28.5% of OP, It is difficult to differentiate between a ruptured OP, a ruptured
thus supporting the idea of trans-­tubal reflux of the fertilised tubal pregnancy, a haemorrhagic corpus luteum or a chocolate
oocyte into the ovary.12 cyst due to the similar ultrasonographic appearances. Terzić et al
Its aetiology remains to be fully elucidated, but it appears to be have suggested that a ruptured OP sonographically is confused
secondary to reflux of the fertilised oocyte to the ovary.13 Inter- with a ruptured corpus luteum in 75% of cases.16
ference in the release of the ovum from the ruptured follicle, Surgery remains the mainstay of treatment with minimal access
malfunctioning of the fallopian tubes and inflammatory thick- surgery the preferred option. Spiegelberg described criteria for
ening of the ovarian tunica albuginea have also been suggested. establishing the diagnosis intraoperatively (box 1).5 17 The aim
Its pathogenesis includes fertilisation occurring outside of the of surgery is to remove the ectopic pregnancy, while preserving
fallopian tube, followed by implantation within the ovary. The as much ovarian tissue as possible, especially in women of repro-
surface of the ovary is covered by tunica albuginea. It lacks ductive age.18 Wedge resection and oophorectomy are commonly
muscle fibres. Loose connective tissue and blood vessels are preferred.
found inside the ovary. Therefore, early rupture may occur. There remains a paucity of reported cases regarding medical
management of an OP using methotrexate (MTX), probably
because an OP is diagnosed acutely when surgical management
remains the gold standard. The first case successfully treated
with MTX was described by Kudo et al, followed by Shamma
and Schwartz who used a single IM injection.19 20 Mittal was
the first to report an MTX injection directly into the gestational
sac.21 It has a high treatment failure rate and there is a risk of

Box 1  -­Spiegelberg's criteria for intraoperative


diagnosis.17

► An intact ipsilateral tube, clearly separate from the ovary.


► Gestational sac occupying the normal position of the ovary.
► Gestational sac connected to the uterus by the utero-­ovarian
ligament.
Figure 5  Histopathological examination confirmed the presence of
► Ovarian tissue in the wall of the gestation sac.
chorionic villi.
Dunphy L, et al. BMJ Case Rep 2022;15:e252499. doi:10.1136/bcr-2022-252499 3
Case report
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Funding  The authors have not declared a specific grant for this research from any
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funding agency in the public, commercial or not-­for-­profit sectors.
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Louise Dunphy http://orcid.org/0000-0001-5499-415X literature. J Gynecol Obstet Biol Reprod 1996;25:378–83.

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4 Dunphy L, et al. BMJ Case Rep 2022;15:e252499. doi:10.1136/bcr-2022-252499


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