Spontaneous Heterotopic Triplet Pregnancy with a Two Viable Intrauterine Embryos and an Ectopic One with Right Tubal Rupture

Abstract

Heterotopic pregnancy (HP) is defined as the simultaneous development of an intra- and an extra uterine gestation. The occurrence of a spontaneous triplet HP is an exceptionally rare medical condition. We report the case of a young woman with spontaneous heterotopic triplets at 8 weeks of gestation, with a misdiagnosis of topic twins and acute appendicitis. The ectopic tubal pregnancy was ruptured and a salpingectomy was performed by laparotomy. The intrauterine pregnancy progressed uneventfully. The two healthy babies were delivery by cesarean section at 36 ± 2 weeks of gestation. Heterotopic triplets with ruptured tubal ectopic pregnancy represent a special diagnostic and therapeutic challenge for the obstetrician. A high rate of clinical suspicion and timely treatment by laparotomy or laparoscopy can preserve the intrauterine gestation with a successful outcome of the pregnancy.

Resumo

A gravidez heterotópica é definida como o desenvolvimento simultâneo de uma gestação intra- e extra-uterina. A ocorrência de gravidez tripla heterotópica espontânea é uma condição médica excepcionalmente rara. Relatamos o caso de uma jovem com gravidez tripla espontânea, às 8 semanas de gestação, com um diagnóstico errôneo de gêmeos tópicos e apendicite aguda. A gravidez tubária ectópica estava rota e uma salpingectomia foi realizada por laparotomia. A gravidez intrauterina progrediu sem intercorrências. Os bebês nasceram saudáveis por cesariana realizada às 36 semanas de gestação. A gravidez de heterotópicos com ectopia e rotura tubária é um desafio diagnóstico e terapêutico. Um alto índice de suspeita e tratamento oportuno por laparotomia ou laparoscopia podem preservar a gestação intrauterina com um resultado bem sucedido da gravidez tópica.

Keywords

obstetrical surgery - pregnancy complications - heterotopic pregnancy - multiple pregnancy - tubal pregnancy

Palavras-chave

cirurgia obstétrica - complicações na gravidez - gravidez heterotópica - gravidez múltipla - gravidez tubária

• References

• 1 Basile F, Di Cesare C, Quagliozzi L. , et al. Spontaneous heterotopic pregnancy, simultaneous ovarian, and intrauterine: a case report. Case Rep Obstet Gynecol 2012; 2012: 509694 . Doi: 10.1155/2012/509694
• 2 Felekis T, Akrivis C, Tsirkas P, Korkontzelos I. Heterotopic triplet pregnancy after in vitro fertilization with favorable outcome of the intrauterine twin pregnancy subsequent to surgical treatment of the tubal pregnancy. Case Rep Obstet Gynecol 2014; 2014: 356131 . Doi: 10.1155/2014/356131
• 3 Sun SY, Araujo Júnior E, Elito Júnior J. , et al. Diagnosis of heterotopic pregnancy using ultrasound and magnetic resonance imaging in the first trimester of pregnancy: a case report. Case Rep Radiol 2012; 2012: 317592 . Doi: 10.1155/2012/317592
• 4 Selo-Ojeme DO, GoodFellow CF. Simultaneous intrauterine and ovarian pregnancy following treatment with clomiphene citrate. Arch Gynecol Obstet 2002; 266 (04) 232-234 . Doi: 10.1007/s004040100213
• 5 Bugatto F, Quintero-Prado R, Kirk-Grohar J, Melero-Jiménez V, Hervías-Vivancos B, Bartha JL. Heterotopic triplets: tubal ectopic and twin intrauterine pregnancy. A review of obstetric outcomes with a case report. Arch Gynecol Obstet 2010; 282 (06) 601-606 . Doi: 10.1007/s00404-010-1577-z
• 6 Kiely JL, Kiely M. Epidemiological trends in multiple births in the United States, 1971-1998. Twin Res 2001; 4 (03) 131-133 . Doi: 10.1375/twin.4.3.131
• 7 Marshal GB. Case of ruptured tubal gestation associated with twin pregnancy in uterus. J Obstet Gynaecol 1903; 4: 448-450 . Doi: 10.1111/j.1471-0528.1903.tb11815.x
• 8 Divry V, Hadj S, Bordes A, Genod A, Salle B. Case of progressive intrauterine twin pregnancy after surgical treatment of cornual pregnancy. Fertil Steril 2007; 87 (01) 190.e1-190.e3 . Doi: 10.1016/j.fertnstert.2006.04.053
• 9 Inion I, Gerris J, Joostens M, De Vree B, Kockx M, Verdonk P. An unexpected triplet heterotopic pregnancy after replacement of two embryos. Hum Reprod 1998; 13 (07) 1999-2001 . Doi: 10.1093/humrep/13.7.1999
• 10 Nikolaou DS, Lavery S, Bevan R, Margara R, Trew G. Triplet heterotopic pregnancy with an intrauterine monochorionic diamniotic twin pregnancy and an interstitial pregnancy following in vitro fertilisation and transfer of two embryos. J Obstet Gynaecol 2002; 22 (01) 94-95 . Doi: 10.1080/01443610211123
• 11 Cholkeri-Singh A, LaBarge A. Spontaneous heterotopic triplets: a case report. Fertil Steril 2007; 88 (04) 968.e5-968.e7 . Doi: 10.1016/j.fertnstert.2006.12.034
• 12 Pan HS, Chuang J, Chiu SF. , et al. Heterotopic triplet pregnancy: report of a case with bilateral tubal pregnancy and an intrauterine pregnancy. Hum Reprod 2002; 17 (05) 1363-1366 . Doi: 10.1093/humrep/17.5.1363
• 13 Bataille P, Reynard A, Ducarme G. Spontaneous heterotopic triplets - A review of literature. J Gynecol Obstet Hum Reprod 2017; 46 (08) 657-659 . Doi: 10.1016/j.jogoh.2017.05.008
• 14 Alsunaidi MI. An unexpected spontaneous triplet heterotopic pregnancy. Saudi Med J 2005; 26 (01) 136-138
• 15 Simsek T, Dogan A, Simsek M, Pestereli E. Heterotopic triplet pregnancy (twin tubal) in a natural cycle with tubal rupture: case report and review of the literature. J Obstet Gynaecol Res 2008; 34 (4 Pt 2): 759-762 . Doi: 10.1111/j.1447-0756.2008.00921.x
• 16 Jeong HC, Park IH, Yoon YS. , et al. Heterotopic triplet pregnancy with bilateral tubal and intrauterine pregnancy after spontaneous conception. Eur J Obstet Gynecol Reprod Biol 2009; 142 (02) 161-162 . Doi: 10.1016/j.ejogrb.2008.10.013
• 17 Arsala L, Danso D. Spontaneous heterotopic triplet pregnancy with tubal rupture: a case report and literature review. J Investig Med High Impact Case Rep 2014; 2 (02) 2324709614531556 . Doi: 10.1177/2324709614531556
• 18 Nguyen-Tran C, Toy EC. Case 3: obstetrical. Heterotopic pregnancy: viable twin intrauterine pregnancy with a viable right tubal ectopic pregnancy. J Ultrasound Med 2000; 19 (05) 355