Live‐born diploid fetus complicated with partial molar pregnancy presenting with pre‐eclampsia, maternal anemia, and seemingly huge placenta: A rare case of confined placental mosaicism and literature review

A partial molar pregnancy almost always ends in miscarriage due to a triploid fetus. We describe a rare case of a singleton, partial molar pregnancy with a seemingly huge placenta, which continued to delivery of a live‐born diploid baby. A 27‐year‐old primigravida suffered from severe pre‐eclampsia and progressive anemia. The uterus was enormously enlarged for the gestational age. A cesarean section was performed because of deterioration of maternal status at 25 weeks' gestation, when more than 3000 mL blood spouted concurrently with the delivery of the placenta. The histological examination showed congestion in the decidua, which indicated disturbance of maternal venous return from the intervillous space. The chromosome complement of the placenta and the neonate were 69,XXX and 46,XX, respectively. We also reviewed all published cases of a singleton, partial molar pregnancy. A literature search yielded 18 cases of a singleton, diploid fetus with partial molar pregnancy. The mean gestational age at delivery was 24.5 ± 6.2 weeks, and fetuses survived outside the uterus in only four cases (22.2%). Intriguingly, previous reports numbered 10 cases with diploid placenta as well as five cases with no karyotyping of the placenta, indicating that they may have included a complete mole in a twin pregnancy or placental mesenchymal dysplasia. In conclusion, this was the first case of placentomegaly that presented manifestations of excessive abdominal distension and maternal severe anemia, and the second case of a singleton, partial molar pregnancy confirmed by chromosome analysis resulting in a diploid living baby.

[1]  J. Kingdom,et al.  Prenatal diagnosis, clinical outcomes, and associated pathology in pregnancies complicated by massive subchorionic thrombohematoma (Breus' mole) , 2013, Prenatal diagnosis.

[2]  A. Shiozaki,et al.  A triploid partial mole placenta from paternal isodisomy with a diploid fetus derived from one sperm and one oocyte may have caused angiogenic imbalance leading to preeclampsia-like symptoms at 19 weeks of gestation. , 2013, Placenta.

[3]  T. Shiozawa,et al.  Placental Mesenchymal Dysplasia: Chronological Observation of Placental Images during Gestation and Review of the Literature , 2013, Gynecologic and Obstetric Investigation.

[4]  U. Nayeri,et al.  Systematic review of sonographic findings of placental mesenchymal dysplasia and subsequent pregnancy outcome , 2013, Ultrasound in obstetrics & gynecology : the official journal of the International Society of Ultrasound in Obstetrics and Gynecology.

[5]  H. Soydinç,et al.  Diploid karyotype partial mole coexisting with live term fetus--case report and review of the world literature. , 2012, Ginekologia polska.

[6]  M. Post Placenta weight percentile curves for singleton and twins deliveries , 2012 .

[7]  N. Papantoniou,et al.  Partial molar pregnancy with a chromosomically and phenotypically normal embryo: presentation of an extremely rare case and review of literature , 2011, The journal of maternal-fetal & neonatal medicine : the official journal of the European Association of Perinatal Medicine, the Federation of Asia and Oceania Perinatal Societies, the International Society of Perinatal Obstetricians.

[8]  D. Sahota,et al.  Massive subchorionic thrombohematoma: a series of 10 cases , 2010, Acta obstetricia et gynecologica Scandinavica.

[9]  Y. Taketani,et al.  Elevated serum soluble fms-like tyrosine kinase 1 (sFlt1) level in women with hydatidiform mole. , 2010, Fertility and sterility.

[10]  A. Minguela,et al.  Partial Mole with a Diploid Fetus: Case Study and Literature Review , 2009, Fetal Diagnosis and Therapy.

[11]  U. Surti,et al.  P57KIP2 immunostaining and molecular cytogenetics: combined approach aids in diagnosis of morphologically challenging cases with molar phenotype and in detecting androgenetic cell lines in mosaic/chimeric conceptions. , 2008, Human pathology.

[12]  S. Dilbaz,et al.  Partial molar pregnancy and coexisting fetus with diploid karyotype , 2007, The journal of maternal-fetal & neonatal medicine : the official journal of the European Association of Perinatal Medicine, the Federation of Asia and Oceania Perinatal Societies, the International Society of Perinatal Obstetricians.

[13]  S. Reynolds,et al.  Partial molar pregnancy with severe pre-eclampsia at 19 weeks' gestation , 2006, Journal of obstetrics and gynaecology : the journal of the Institute of Obstetrics and Gynaecology.

[14]  Z. Hagay,et al.  Twin pregnancy consisting of a complete hydatidiform mole and co-existent fetus: report of two cases and review of literature. , 2005, Gynecologic oncology.

[15]  K. Devriendt Hydatidiform mole and triploidy: the role of genomic imprinting in placental development. , 2005, Human reproduction update.

[16]  D. Kalousek,et al.  Spontaneous abortion and confined chromosomal mosaicism , 1992, Human Genetics.

[17]  K. Oka,et al.  Coexisting true hermaphroditism and partial hydatidiform mole developing metastatic gestational trophoblastic tumors. A case report , 2002, Virchows Archiv.

[18]  R. Fraser,et al.  ASSOCIATION BETWEEN BREUS' MOLE AND PARTIAL HYDATIDIFORM MOLE: CHANCE OR CAN HYDROPIC VILLI PRECIPITATE PLACENTAL MASSIVE SUBCHORIONIC THROMBOSIS? , 2002, Pediatric pathology & molecular medicine.

[19]  K. Benirschke,et al.  Diploid/triploid mosaic placenta with fetus. Towards A better understanding of 'partial moles'. , 2000, Early human development.

[20]  B. Seckin,et al.  Partial hydatidiform mole with diploid karyotype in a live fetus , 2000, International journal of gynaecology and obstetrics: the official organ of the International Federation of Gynaecology and Obstetrics.

[21]  T. Hung,et al.  Delivery of a severely anaemic fetus after partial molar pregnancy: clinical and ultrasonographic findings. , 1999, Human reproduction.

[22]  D. Kalousek,et al.  Partial molar pregnancy with fetal survival: An unusual example of confined placental mosaicism , 1993, Obstetrics and gynecology.

[23]  H. Ngan,et al.  Persistent gestational trophoblastic disease after a diploid partial hydatidiform mole coexisting with a normal living fetus. , 1992, International journal of gynaecology and obstetrics: the official organ of the International Federation of Gynaecology and Obstetrics.

[24]  H. Saal,et al.  Molar pregnancy coexisting with a normal fetus: a case report. , 1989, Gynecologic oncology.

[25]  J. Hobbins,et al.  SONOGRAPHIC DIAGNOSIS OF A PREGNANCY WITH A DIFFUSE HYDATIDIFORM MOLE AND COEXISTENT 46,XX FETUS: A CASE REPORT , 1988, Obstetrics and gynecology.

[26]  E. J. Thomas,et al.  The Prospective Management of a Coexistent Hydatidiform Mole and Fetus , 1987, The Australian & New Zealand journal of obstetrics & gynaecology.

[27]  H. Geijn,et al.  A partial hydatidiform mole, dispersed throughout the placenta, coexisting with a normal living fetus. Case report , 1985, British journal of obstetrics and gynaecology.

[28]  N. Teng,et al.  Partial hydatidiform mole with diploid karyotype: report of three cases. , 1984, American journal of obstetrics and gynecology.

[29]  S. de la Garza,et al.  [Chorionic tumors]. , 1961, Ginecologia y obstetricia de Mexico.