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一名唐氏综合征胎儿在子宫内表现为心包积液,其髓系增殖但无GATA1突变。

Myeloid proliferation without GATA1 mutations in a fetus with Down syndrome presenting in utero as a pericardial effusion.

作者信息

Rougemont Anne-Laure, Makrythanasis Periklis, Finci Vildana, Billieux Marie-Hélène, Epiney Manuella, McKee Thomas A, Nizetic Dean, Fokstuen Siv

机构信息

Division of Clinical Pathology, Geneva University Hospitals, 1211 Geneva, Switzerland.

出版信息

Pediatr Dev Pathol. 2010 Sep-Oct;13(5):423-6. doi: 10.2350/09-11-0743-CR.1. Epub 2010 Apr 29.

Abstract

An isolated pericardial effusion was observed during a routine prenatal ultrasound in a fetus of 30 and 3/7 weeks gestation. Amniocentesis was performed and revealed a trisomy 21. After prenatal counseling, the parents opted for termination of the pregnancy at 32 weeks. Postmortem examination confirmed the presence of a pericardial effusion, without structural cardiac anomalies, and showed the development of ascites and subcutaneous edema. Histological examination showed an infiltrate of megakaryoblasts and irregular, dysplastic megakaryocytes confined to the epicardium, the pericardial lymph nodes, and the pancreas, consistent with a myeloid proliferation related to Down syndrome. Sequencing of exons 2 and 3 of the GATA1 gene from the umbilical cord blood and from megakaryoblast infiltrate showed no mutation. A high incidence of chromosomal abnormalities, in particular trisomy 21, has been described in fetuses with pericardial effusion. However, myeloid proliferation related to Down syndrome without GATA1 mutations is extremely rare. To our knowledge, only one such case has been reported to date. We present here a 2nd case, which further supports the hypothesis that hyperproliferation of megakaryocytes in a subset of Down syndrome patients may be initiated by events other than GATA1 mutations.

摘要

在一名妊娠30又3/7周胎儿的常规产前超声检查中发现了孤立性心包积液。进行了羊水穿刺,结果显示为21三体。经过产前咨询,父母选择在32周时终止妊娠。尸检证实存在心包积液,无心脏结构异常,并显示有腹水和皮下水肿形成。组织学检查显示巨核母细胞浸润以及局限于心外膜、心包淋巴结和胰腺的不规则、发育异常的巨核细胞,这与唐氏综合征相关的髓系增殖一致。对脐带血和巨核母细胞浸润部位的GATA1基因第2和第3外显子进行测序,未发现突变。心包积液胎儿中已报道有高发生率的染色体异常,尤其是21三体。然而,与唐氏综合征相关且无GATA1突变的髓系增殖极为罕见。据我们所知,迄今为止仅报道过1例此类病例。我们在此呈现第2例病例,这进一步支持了以下假说:唐氏综合征患者亚群中巨核细胞的过度增殖可能由GATA1突变以外的事件引发。

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