Abstract
Chronic ectopic pregnancy is a variant of ectopic pregnancy presenting as chronic lower abdominal pain, menstrual irregularity and pelvic mass. Often, chronic ectopic may pose diagnostic conundrum due to unusual presentations. We are presenting an unusual case of chronic ectopic with negative urine pregnancy test, who presented with pain in right hypochondrium. The patient had bilateral adnexal mass with omental deposit on imaging masquerading adnexal malignancy, leading to decision for surgical management. Intraoperatively blood clots were present in supramesocolic compartment along with bilateral adnexal masses. Total abdominal hysterectomy and bilateral salpingo-oophorectomy with clots evacuation was done and postoperatively, patient had complete relief of her symptoms. Final histopathology came out as ectopic gestation. Culminating point is to keep ectopic pregnancy as differential in all reproductive age group women presenting with pain in abdomen regardless of other symptom particularly with pelvic mass.
Keywords
pregnancy, cancer - see oncology
Background
Ectopic pregnancy is a well known obstetrical condition, occurring in 1%–2% of all pregnancies. This condition often presents with an acute pelvic or lower abdominal pain, vaginal bleeding and fainting attack or sometimes as a hypovolaemic shock in case of rupture; though unusual presentations are not rare.1 There is one other presentation of tubal ectopic pregnancy, of which prevalence varies in literature—the chronic ectopic pregnancy. It is formed by multiple episodes of continuous, inconsequential bleeding in and through the fallopian tube causing chronic insult to the surrounding tissue resulting in chronic inflammation and scarring which leads to the formation of a pelvic mass and intra-abdominal adhesions.2 Most of the cases present with chronic pelvic pain, often misdiagnosed as complex pelvic mass by imaging and laboratory parameters due to low or undetectable serum β-human chorionic gonadotropin (β-HCG) value, as there is scant chorionic villi tissue.3
We are presenting a case of chronic ectopic pregnancy, where the patient presented with an unusual symptom of chronic right hypochondrium pain and pelvic mass without any obvious gynaecological report. The case was misdiagnosed as adnexal malignancy.
Case presentation
A 37-year-old, P3L3 woman presented to gynaecology outpatient department of our tertiary care hospital with an incidentally detected 3 cm endometrioma of right ovary on ultrasonography done elsewhere for mild pain in the right upper abdomen (right hypochondrium) and serum cancer antigen (CA-125) levels of 64 IU/mL. On further evaluation patient gave the history of dysmenorrhoea for the last 3–4 months with no changes in the menstrual pattern (cycles were regular with normal flow). Her abdominal examination was normal and vaginal examination revealed mild tenderness in the right fornix. Her urine pregnancy test was negative.
Based on all these findings which were suggesting towards endometrioma, patient was started on dienogest. Antacids were also started for her after consultation with gastroenterologist, done for pain in right hypochondrium. On her scheduled follow-up visit after 3 months, she presented with an increase in the intensity of pain in right hypochondrium and with relief in dysmenorrhoea. At that time on abdominal examination mild tenderness was present in the right hypochondrium and on vaginal examination there was a firm mass of around (~3×3 cm) felt in right adnexa with restricted mobility and a cystic mobile mass (~4×3 cm) in the left adnexa.
Investigations
At her follow-up visit (after 3 months), ultrasonography was repeated which showed a complex solid cystic lesion of around 3×4 cm in bilateral adnexa. In view of indeterminate ultrasonographic finding, patient was advised MRI for further evaluation along with tumour markers (CA-125, CA 19-9 and carcinoembryonic antigen (CEA)) and repeat gastroenterology consult.
MRI was suggestive of bilateral complex cystic adnexal masses (3.6×3.0 cm on right side and 4×3.6 cm on left side) with solid enhancing component (figures 1 and 2A) and two omental nodules adjacent to gall bladder of around 10 mm pointing towards malignancy (figure 2B). Among tumour marker only CA-125 was raised (141 U/mL).
Gastroenterologists advised her positron emission tomography (PET) scan and upper and lower gastrointestinal (GI) endoscopies to rule out any metastasis or primary GI malignancy as she had persistent symptom of right hypochondrium pain. In her PET scan there was hypermetabolic solid cystic mass in bilateral adnexa with low-grade fluorodeoxyglucose (FDG) avid fat stranding in adjoining mesentery, likely mitotic and low-grade FDG avid miliary mesenteric stranding with evident omental nodular deposit in right supramesocolic compartment. Her endoscopies were normal.
Differential diagnosis
Ovarian malignancy, endometrioma.
Treatment
Patient was given medical management for endometrioma first (tablet dienogest 2 mg once a day for 3 months along with antacids for pain in right hypochondrium). When her symptom did not resolve, further workup was done in which her imaging and clinical finding raised the suspicion of ovarian malignancy, in view of which patient was planned for an exploratory laparotomy. Procedure was properly explained to the patient along with all the possibilities and consequences. Intraoperatively there was a 3×3 cm solid mass in the right tube adhered with a 4×5 cm right ovarian haemorrhagic cyst and on left side there was a multiloculated cyst of 3×4 cm with haemorrhagic content and solid area. Based on these findings, a decision was taken to proceed with total abdominal hysterectomy and bilateral salpingo-oophorectomy, as the patient had asked to do the complete surgery even for the slightest uncertainty. There were old blood clots in the supramesocolic compartment, adhered to the liver surface adjacent to the gallbladder, subdiaphragmatic space and omentum, adhesiolysis done and all clots removed followed by infracolic omentectomy and peritoneal lavage as there were many minute haemorrhagic areas seen all over the mesentery and omentum.
Outcome and follow-up
Postoperative course was uneventful, patient got relief of her symptom and was discharged on post-operative day 5.
Patient followed up after 2 weeks of the surgery with no complaints. The histopathological report gave the relief to the patient at that moment as it was not malignancy; unexpectedly histopathology was suggestive of ectopic gestation of right adnexa. Histopathological reporting of other tissues were as follows—sections from left ovary showed ovarian parenchyma along haemosiderin-filled macrophages, the cyst on the right ovary was haemorrhagic corpus luteal cyst and her omentum showed fat necrosis along with old haemorrhage.
Discussion
The incidence of chronic ectopic pregnancy varies in literature from 6% of all ectopic pregnancies stated in one study to 20.3% in a series conducted over 62 patients.4 This variation may be due to different diagnostic criteria including clinical condition, ultrasonography, intraoperative finding and hormonal assays.2 Most common presentation for chronic ectopic is irregular vaginal bleeding of varying degree and lower abdominal pain along with the pelvic mass, although in only 50% of cases it is predicted by its signs and symptoms.4 5
As per best of our knowledge, we did not come across a case of tubal ectopic pregnancy in literature in which the patient presented with only complaint of pain in right hypochondrium. The pain may be attributed to friction created over the liver capsule by the blood clots, carried by sympathetic fibres from the coeliac ganglion or it may be referred liver pain transmitted by lower intercostal and subcostal nerves due to mechanical irritation of peritoneum and pleura.6 Although there are case reports of patient presenting with chest and shoulder pain, due to the irritation of phrenic nerve present in the diaphragm with blood.7
Other multiple case reports of unusual presentation of chronic ectopic pregnancy like intestinal perforation presented as a rectal bleeding, ureteral and intestinal obstruction due to adhesions, abdominal compartment syndrome due to haemoperitoneum and fistula with the abdominal wall have been reported in the literature.2 5 7
In this case pelvic mass was diagnosed incidentally on imaging done for pain in upper abdomen without any history of menstrual irregularity or pelvic pain and negative urine pregnancy test. This clinical presentation along with imaging finding led us to miss the diagnosis of chronic ectopic and raised the suspicious of adnexal malignancy for which surgery was done. Although there are cases reported in literature in which chronic ectopic has often been confused with the other adnexal and pelvic masses like malignancy especially germ cell tumour, endometrioma and fibroid due to sharing of same age group and finally diagnosed as ectopic pregnancy after histopathology report only,3 4 8 still there were many flaws in management of this case. As before surgery, even if we were considering adnexal malignancy, germ cell tumour marker which includes β-HCG (might be raised in chronic ectopic with negative urine pregnancy test) and also frozen section during the surgery could have been done, which could have prevented the hysterectomy with bilateral salpingo-oophorectomy for the patient.
Patient’s perspective.
I chose a tertiary care hospital after I didn’t get relief in my symptom even after going to multiple doctors and ultrasonography report of endometrioma. After so many investigations, I was counselled regarding the suspicion of malignancy and finally after the discussion with me, doctors planned for a surgery. I underwent surgery with no complication and got relief of the constant pain on right side of my upper abdomen which had persisted for so long. Although I was worrying for the final diagnosis as there was suspicion of malignancy intraoperatively also. When my final histopathology report came I was very much relieved at that time as it was not malignancy, though I am worried now about the future implication of such extensive surgery I went through.
Learning points.
This case gives us the insight that;
For any reproductive age group women who presents with a complaint of pain in abdomen regardless of its site, menstrual irregularity and urine pregnancy test; ectopic pregnancy should always be considered especially if associated with any kind of pelvic mass.
To consider serum β-human chorionic gonadotropin in investigation while evaluating any reproductive age patient with pelvic mass.
To consider frozen section intraoperatively, especially in young patient or whenever there is even any slightest diagnostic conundrum.
Footnotes
Contributors: SS, RV and AT were involved in the acquisition of idea, drafting and editing of the article.
Funding: The authors have not declared a specific grant for this research from any funding agency in the public, commercial or not-for-profit sectors.
Competing interests: None declared.
Patient consent for publication: Obtained.
Provenance and peer review: Not commissioned; externally peer reviewed.
References
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