Spontaneous Heterotopic Pregnancy with Unaffected Intrauterine Pregnancy: Systematic Review of Clinical Outcomes

Abstract

Background and objective: Spontaneous heterotopic pregnancy (SHP) is a rare condition represented by the synchronous coexistence of an intrauterine and an ectopic pregnancy. It rarely occurs with natural conception and is usually a consequence of assisted reproductive techniques. Diagnosis of SHP can be a challenge for the clinician. The evolution of the intrauterine pregnancy is dependent on many factors, such as the location of the heterotopic pregnancy, gestational age at the time of diagnosis, the surgical procedure, the presence of other risk factors, early or delayed management. The aim of this systematic review of the literature was to extract existing evidence on spontaneous heterotopic pregnancy with otherwise unaffected intrauterine pregnancy. Materials and Methods: From a total of 1907 database entries identified in PubMed, EMBASE and Cochrane reviews, we selected 18 papers for narrative synthesis, for which we explored the diagnostic options, treatment, and outcome of these extremely rare epidemiologic occurrences. Manuscripts were assessed using the CARE guidelines for reporting case reports. Results: The main symptom was abdominal pain, and the preferred treatment approach was surgical, more precisely, using a laparoscopic approach. Most cases presented no risk factors, and the diagnosis was mostly made in the first semester. Conclusions: Normal follow-up and evolution of intrauterine pregnancy have been observed regardless of surgical approach (open or laparoscopic). Early diagnosis and treatment are advised, as they impact maternal and fetal outcomes. Evidence on this topic is scarce, predominantly comprised of case reports with variable degrees of adherence to dissemination guidelines. More studies on this topic are required to optimize care protocols for this type of pregnancy.

A unique case of coexisting intrauterine and abdominal pregnancy which progress to term with a positive birth outcome

“…Majority of heterotopic pregnancies are diagnosed during the first and early second trimester pregnancies [3,18,21,22]. According to a review done by Barrenetxea et al of published case reports from January 1994 to December 2004, out of 13 spontaneous heterotopic pregnancies, 74% of them are diagnosed early (between 5 and 8 weeks), the latest being at 20 weeks [21].…”

“…A systematic review of 18 spontaneous heterotopic pregnancies done by Oancea et al also revealed that the diagnosis of most heterotopic pregnancies was mainly established in the first trimester, the mean gestational age being 8 weeks [22]. They found only one article describing a diagnosis of heterotopic pregnancy in the second trimester, at 20 weeks of gestation [22].…”

Spontaneously conceived heterotopic pregnancy with abdominal pregnancy implanted on the vesicouterine pouch: A case report and literature review

“…Even if an intrauterine pregnancy is confirmed and progressing well, we should always consider the possibility of an ectopic pregnancy 1 . In addition, HP should be recognized as a disease that causes abdominal pain in early pregnancy, 1,29,31 and if severe abdominal pain is observed after the confirmation of intrauterine pregnancy, the rupture of ectopic pregnancy in HP should be considered. Postoperative management of HAP requires close follow‐up due to the high rate of miscarriage 1 and the possible proliferation of the residual villous tissue in the abdominal cavity, 32 especially in cases with collapsed AP.…”

A rare case of spontaneous heterotopic pregnancy with intrauterine gestational trophoblastic neoplasia and tubal ectopic pregnancy at a remote secondary care hospital

“…Many times diagnosis is difficult due to intrauterine pregnancy masking the ectopic pregnancy. [ 5 ] Transvaginal sonography can help in differentiating between intrauterine and extrauterine pregnancy in almost 90% of cases and if there is doubt about the diagnosis, diagnostic laparoscopy is ideal to perform to avoid delay in diagnosis and management of HP. [ 5 ] In this case report, we are reporting a rare case of spontaneous HP in a 40-year-old lady with intrauterine gestational trophoblastic neoplasia (GTN) and ruptured tubal ectopic pregnancy in a remotely located secondary care hospital.…”