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活产二倍体胎儿合并部分性葡萄胎,伴有子痫前期、母体贫血及看似巨大的胎盘:一例罕见的局限性胎盘嵌合体病例及文献综述

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.

作者信息

Kawasaki Kaoru, Kondoh Eiji, Minamiguchi Sachiko, Matsuda Fumihiko, Higasa Koichiro, Fujita Kohei, Mogami Haruta, Chigusa Yoshitsugu, Konishi Ikuo

机构信息

Department of Gynecology and Obstetrics, Kyoto University, Kyoto, Japan.

Department of Diagnostic Pathology, Kyoto University, Kyoto, Japan.

出版信息

J Obstet Gynaecol Res. 2016 Aug;42(8):911-7. doi: 10.1111/jog.13025. Epub 2016 May 26.

Abstract

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.

摘要

部分性葡萄胎几乎总是因三倍体胎儿而以流产告终。我们描述了一例罕见的单胎部分性葡萄胎病例,其胎盘看似巨大,并持续至分娩出一个活产二倍体婴儿。一名27岁的初产妇患有严重子痫前期和进行性贫血。子宫相对于孕周明显增大。由于孕25周时母亲状况恶化,遂行剖宫产,胎盘娩出时同时涌出超过3000毫升血液。组织学检查显示蜕膜充血,提示绒毛间隙母体静脉回流受阻。胎盘和新生儿的染色体核型分别为69,XXX和46,XX。我们还回顾了所有已发表的单胎部分性葡萄胎病例。文献检索得到18例单胎二倍体胎儿合并部分性葡萄胎的病例。分娩时的平均孕周为24.5±6.2周,仅4例(22.2%)胎儿存活至宫外。有趣的是,既往报道中有10例胎盘为二倍体,还有5例未对胎盘进行核型分析,提示这些病例可能包含双胎妊娠中的完全性葡萄胎或胎盘间充质发育异常。总之,这是首例出现腹部过度膨隆和母体严重贫血表现的胎盘肿大病例,也是第二例经染色体分析确诊的单胎部分性葡萄胎并分娩出二倍体活婴的病例。

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