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.

Keywords: diagnosis; ectopic; heterotopic; pregnancy.

Figure 1

PRISMA flowchart of systematic review article selection process.

Figure 2

Assessment of selected articles using the CARE guidelines. Diagnostic challenges (8b), changes in therapeutic interventions (9c), adverse and unanticipated events (10d), and patient perspectives (12) are grossly underreported in the identified case reports. Legend: (1) Title—The diagnosis or intervention of primary focus followed by the words “case report”; (2) Key Words—2 to 5 key words that identify diagnoses or interventions in this case report (including “case report”); (3a) Introduction—What is unique about this case and what does it add to the scientific literature; (3b) The patient’s main concerns and important clinical findings; (3c) The primary diagnoses, interventions, and outcomes; (3d) Conclusion—“Take-away” lessons from this case report? (4) Introduction—Briefly summarizes why this case is unique and may include medical literature references; (5a) De-identified patient specific information; (5b) Primary concerns and symptoms of the patient; (5c) Medical, family, and psychosocial history including relevant genetic information; (5d) Relevant past interventions and their outcomes; (6) Describe significant physical examination (PE) and important clinical findings; (7) Historical and current information from this episode of care organized as a timeline (figure or table); (8a) Diagnostic methods (PE, laboratory testing, imaging, surveys); (8b) Diagnostic challenges; (8c) Diagnosis (including other diagnoses considered); (8d) Prognostic characteristics when applicable; (9a) Types of therapeutic intervention (pharmacologic, surgical, preventive); (9b) Administration of therapeutic intervention (dosage, strength, duration); (9c) Changes in therapeutic interventions with explanations; (10a) Clinician- and patient-assessed outcomes if available; (10b) Important follow-up diagnostic and other test results; (10c) Intervention adherence and tolerability; (10d) Adverse and unanticipated events; (11a) Strengths and limitations in this case; (11b) Discussion of the relevant medical literature; (11c) The rationale for the conclusions; (11d) The primary “take-away” lessons from this case report; (12) The patient should share their perspective on the treatment(s) they received; (13) The patient should give informed consent.