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Journal of Obstetrics and Gynaecology Research
ISSN 1341-8076 · DOI: 10.1111/jog.16089
Received 11 October 2024
Accepted 28 November 2024
Published 18 December 2024
Kavitha Subramaniam¹, Nalini Devi¹, Rakesh Singh², Priya Vijayan³
¹ Department of Obstetrics and Gynaecology, JIPMER, Puducherry 605006, India
² Department of Radiology, JIPMER, Puducherry 605006, India
³ Department of Anaesthesiology, JIPMER, Puducherry 605006, India
Correspondence: Dr. Kavitha Subramaniam, Department of Obstetrics and Gynaecology, JIPMER, Puducherry 605006. Email: kavithaobs@jipmer.edu.in
Background: Heterotopic pregnancy — the simultaneous occurrence of intrauterine and ectopic pregnancy — has an estimated incidence of 1 in 30,000 in spontaneous conceptions, rising to 1 in 100–500 with assisted reproductive technologies. Interstitial (cornual) ectopic pregnancy is particularly dangerous, accounting for 2–4% of all ectopics, with rupture causing massive haemorrhage and a mortality rate up to 2.5%. We report a rare case of spontaneous heterotopic interstitial ectopic pregnancy managed laparoscopically with successful preservation of the intrauterine pregnancy.
Case presentation: A 28-year-old G2P1L1 with a naturally conceived dichorionic diamniotic twin pregnancy at 7 weeks presented with acute lower abdominal pain and haemoperitoneum. Ultrasound revealed an intrauterine twin gestation alongside a separate mass in the right cornual region with a cardiac activity. Emergency laparoscopic cornuostomy was performed. The intrauterine twins survived and were delivered at 36 weeks as healthy neonates.
Conclusions: Spontaneous heterotopic interstitial ectopic pregnancy is a life-threatening obstetric emergency that requires high clinical suspicion. Laparoscopic cornuostomy with potassium chloride injection to the ectopic sac can achieve definitive treatment while preserving the intrauterine pregnancy. Early diagnosis by transvaginal ultrasound and timely surgical intervention are key to foetal and maternal survival.
Heterotopic pregnancy refers to the simultaneous coexistence of intrauterine and extrauterine gestation. In the era before assisted reproductive technologies (ART), the incidence was estimated at approximately 1 in 30,000 naturally conceived pregnancies. However, with widespread use of ART, this incidence has risen dramatically to 1 in 100–500 IVF cycles. The rising use of ovulation induction agents in India has contributed to an increasing clinical encounter with this entity.
Interstitial (cornual) ectopic pregnancy, accounting for 2–4% of all ectopics, carries particular danger: the interstitial portion of the fallopian tube is surrounded by myometrium and can accommodate a larger gestational sac before rupture, reaching 7–8 weeks at the time of presentation. When rupture occurs, the surrounding myometrial vascularity produces catastrophic haemorrhage — mortality has been reported up to 2.5%, approximately 7 times higher than ampullary ectopic pregnancies.
The diagnostic difficulty in heterotopic pregnancy lies in the false reassurance provided by the intrauterine pregnancy on initial ultrasound, leading to delayed diagnosis of the concurrent ectopic component. We report a case of spontaneous heterotopic interstitial ectopic pregnancy in a patient without ART, successfully managed by laparoscopic cornuostomy with full salvage of the intrauterine twin gestation.
A 28-year-old G2P1L1 woman (previous full-term normal vaginal delivery) presented to the gynaecological emergency of JIPMER, Puducherry with a 6-hour history of acute, severe lower abdominal pain with mild vaginal bleeding. The last menstrual period was 7 weeks prior. She had conceived spontaneously without any fertility treatments, ovulation induction, or ART. There was no history of previous pelvic inflammatory disease, previous ectopic pregnancy, intrauterine device use, or tubal surgery.
On examination: pulse 112/min, BP 90/60 mmHg (hypotensive), pallor +. Abdomen revealed generalised tenderness with guarding, particularly in the right iliac fossa. Bowel sounds were reduced. Per vaginum: cervical os was closed, uterus bulky, right adnexal mass palpable with marked tenderness, cervical motion tenderness present. Free fluid on posterior fornix aspiration (serosanguinous).
Serum β-hCG: 18,420 mIU/mL.
Transvaginal ultrasonography (TVUS): Uterine cavity: a dichorionic diamniotic twin intrauterine pregnancy identified at 7 weeks 2 days with cardiac activity in both sacs (FHR 142 and 138 bpm). Right cornual region: a separate, eccentric gestational sac measuring 2.8 × 2.6 cm with a foetal pole and cardiac activity (FHR 128 bpm), located in the right interstitial portion of the fallopian tube. The sac was surrounded by a thin rim of myometrium (<5 mm) — the 'interstitial line sign' and 'myometrial mantle sign' confirmed the interstitial location. Significant haemoperitoneum (free fluid in Morrison's pouch and pelvis, estimated 400 mL).
Diagnosis: Spontaneous heterotopic pregnancy — intrauterine twin gestation (viable) + interstitial (cornual) ectopic pregnancy (right side, unruptured but haemoperitoneum present).
The patient was resuscitated with IV fluids, 2 units packed red cells, and taken for emergency diagnostic laparoscopy under general anaesthesia.
Laparoscopic findings: approximately 500 mL of haemoperitoneum. Right cornual region showed a 3 cm gestational sac bulging from the interstitial portion with early serosanguinous surface ooze (impending rupture).
Procedure: (1) Haemoperitoneum evacuated by suction irrigation. (2) Vasopressin 20 IU in 100 mL NS injected into the right cornual myometrium (to reduce bleeding). (3) Cornuostomy incision made with monopolar hook. (4) Ectopic sac carefully evacuated with alligator forceps. (5) Intracornual injection of potassium chloride 2 mEq/2 mL under laparoscopic vision to ensure devascularisation of ectopic trophoblast. (6) Cornuostomy closed with figure-of-8 vicryl sutures. (7) Bilateral tubal patency confirmed by chromopertubation. The intrauterine twin pregnancy was not disturbed.
Postoperative course was uncomplicated. Serial β-hCG measured every 48 hours showed declining trend (Day 2: 16,200; Day 5: 12,800; Day 10: 8,400 mIU/mL) confirming resolution of ectopic trophoblast. Fortnightly obstetric ultrasound confirmed continued viability and normal growth of both intrauterine twins.
The patient was delivered at 36 weeks gestation by elective LSCS (due to previous uterine surgery and twin gestation) — two healthy female neonates, birth weights 2,320 g and 2,180 g, APGAR scores 9/10 and 8/9. Both neonates are well at 6-month follow-up.
The incidence of spontaneous heterotopic pregnancy without ART is cited as 1:30,000 but may be underreported in India due to incomplete ultrasound assessment of adnexa when an intrauterine pregnancy is identified. A retrospective audit from a Kolkata centre (Chakraborty et al., JOGI 2021) identified 8 heterotopic pregnancies over 5 years, of which 3 were spontaneous — suggesting a higher-than-reported incidence in Indian patients, possibly related to subclinical tubal disease from prior pelvic infection.
Diagnosis is the greatest challenge. The TVUS "interstitial line sign" (echogenic line extending from the uterine cavity to the cornual sac) has a sensitivity of 80% and specificity of 98% for interstitial ectopic pregnancy and should be specifically sought in all early pregnancies with adnexal pathology. An asymmetric uterine contour and eccentric gestational sac location are additional discriminatory features.
Management options for the ectopic component include: (1) laparoscopic cornuostomy (preferred, fertility-preserving), (2) laparoscopic cornual resection, (3) hysteroscopic evacuation, and (4) systemic methotrexate (contraindicated here due to concurrent IUP). Intrasac potassium chloride injection under laparoscopic guidance, as performed in our case, selectively devascularises the ectopic trophoblast without systemic foetal risk. This technique has been reported to successfully preserve co-existing IUP in heterotopic cases with a success rate exceeding 85% (Fernandez et al., 2019).
The outcome in our case — delivery of two healthy neonates at 36 weeks after laparoscopic cornual surgery — is exceptional and adds to the limited published literature on successful IUP salvage in spontaneous heterotopic interstitial ectopic pregnancy.
This case demonstrates that spontaneous heterotopic interstitial ectopic pregnancy, while rare and life-threatening, can be successfully managed by laparoscopic cornuostomy with intrauterine pregnancy salvage. Systematic adnexal assessment on TVUS in every early pregnancy — even with a confirmed IUP — is essential to avoid delayed diagnosis. Timely surgery by an experienced laparoscopic surgeon, combined with intraoperative potassium chloride injection, can achieve excellent maternal and foetal outcomes.
Learning Points / Take-Away Lessons
Patient Consent
Written informed consent was obtained from the patient for case publication and use of clinical images. Consent includes publication of obstetric outcomes.
Ethics
Approved by JIPMER Ethics Committee, Reference JIPMER/SC/2024/OBG/CR/041, in accordance with the Declaration of Helsinki.
Competing Interests
No conflicts of interest declared.
Funding
No funding received.
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