• 文献检索
  • 文档翻译
  • 深度研究
  • 学术资讯
  • Suppr Zotero 插件Zotero 插件
  • 邀请有礼
  • 套餐&价格
  • 历史记录
应用&插件
Suppr Zotero 插件Zotero 插件浏览器插件Mac 客户端Windows 客户端微信小程序
定价
高级版会员购买积分包购买API积分包
服务
文献检索文档翻译深度研究API 文档MCP 服务
关于我们
关于 Suppr公司介绍联系我们用户协议隐私条款
关注我们

Suppr 超能文献

核心技术专利:CN118964589B侵权必究
粤ICP备2023148730 号-1Suppr @ 2026

文献检索

告别复杂PubMed语法,用中文像聊天一样搜索,搜遍4000万医学文献。AI智能推荐,让科研检索更轻松。

立即免费搜索

文件翻译

保留排版,准确专业,支持PDF/Word/PPT等文件格式,支持 12+语言互译。

免费翻译文档

深度研究

AI帮你快速写综述,25分钟生成高质量综述,智能提取关键信息,辅助科研写作。

立即免费体验

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.

DOI:10.2350/09-11-0743-CR.1
PMID:20429643
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。

相似文献

1
Myeloid proliferation without GATA1 mutations in a fetus with Down syndrome presenting in utero as a pericardial effusion.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。
Pediatr Dev Pathol. 2010 Sep-Oct;13(5):423-6. doi: 10.2350/09-11-0743-CR.1. Epub 2010 Apr 29.
2
Megakaryocyte hyperproliferation without GATA1 mutation in foetal liver of a case of Down syndrome with hydrops foetalis.一例患有胎儿水肿综合征的唐氏综合征胎儿肝脏中巨核细胞过度增殖且无GATA1突变
Br J Haematol. 2008 Oct;143(2):300-3. doi: 10.1111/j.1365-2141.2008.07332.x. Epub 2008 Aug 10.
3
Severe TMD/AMKL with GATA1 mutation in a stillborn fetus with Down syndrome.患有唐氏综合征的死产胎儿中伴有GATA1突变的严重短暂性骨髓异常增生症/急性巨核细胞白血病。
Nat Clin Pract Oncol. 2007 Jul;4(7):433-8. doi: 10.1038/ncponc0876.
4
Morphologic and GATA1 sequencing analysis of hematopoiesis in fetuses with trisomy 21.三体 21 胎儿造血系统的形态学和 GATA1 测序分析。
Hum Pathol. 2014 May;45(5):1003-9. doi: 10.1016/j.humpath.2013.12.014. Epub 2014 Jan 17.
5
Blasts in transient leukaemia in neonates with Down syndrome differentiate into basophil/mast-cell and megakaryocyte lineages in vitro in association with down-regulation of truncated form of GATA1.唐氏综合征新生儿短暂性白血病中的爆裂细胞在体外向嗜碱性粒细胞/肥大细胞和巨核细胞谱系分化,同时伴有 GATA1 截断形式的下调。
Br J Haematol. 2010 Mar;148(6):898-909. doi: 10.1111/j.1365-2141.2009.08038.x. Epub 2010 Jan 11.
6
Isolated pericardial effusion in the human fetus: a report of three cases.
Prenat Diagn. 2003 Mar;23(3):193-7. doi: 10.1002/pd.563.
7
[Myeloid proliferations related to Down syndrome].[与唐氏综合征相关的髓系增殖性疾病]
Rinsho Ketsueki. 2019;60(9):1299-1307. doi: 10.11406/rinketsu.60.1299.
8
A rare case of GATA1 negative chemoresistant acute megakaryocytic leukemia in an 8-month-old infant with trisomy 21.一例罕见的 GATA1 阴性化疗耐药性急性巨核细胞白血病发生于一名 8 月龄伴有 21 三体的婴儿。
Pediatr Blood Cancer. 2010 Jul 1;54(7):1048-9. doi: 10.1002/pbc.22331.
9
GATA1 mutations in acute leukemia in children with Down syndrome.唐氏综合征患儿急性白血病中的GATA1突变
Cancer Genet Cytogenet. 2006 Apr 15;166(2):112-6. doi: 10.1016/j.cancergencyto.2005.10.008.
10
GATA1 as a new target to detect minimal residual disease in both transient leukemia and megakaryoblastic leukemia of Down syndrome.GATA1作为检测唐氏综合征短暂性白血病和巨核细胞白血病微小残留病的新靶点。
Leuk Res. 2005 Nov;29(11):1353-6. doi: 10.1016/j.leukres.2005.04.007.

引用本文的文献

1
Acute Megakaryocytic Leukemia.急性巨核细胞白血病。
Cold Spring Harb Perspect Med. 2020 Feb 3;10(2):a034884. doi: 10.1101/cshperspect.a034884.
2
Fetal/Neonatal Pericardial Effusion in Down's Syndrome: Case Report and Review of Literature.唐氏综合征胎儿/新生儿心包积液:病例报告及文献综述
AJP Rep. 2018 Oct;8(4):e301-e306. doi: 10.1055/s-0038-1675337. Epub 2018 Oct 29.
3
A dysmorphic newborn with petechiae and a 'Big Heart'.一名患有瘀点和“大心脏”的畸形新生儿。
BMJ Case Rep. 2014 Apr 4;2014:bcr2014204195. doi: 10.1136/bcr-2014-204195.
4
Tumorigenesis in Down's syndrome: big lessons from a small chromosome.唐氏综合征中的肿瘤发生:从小染色体中获得的重要教训。
Nat Rev Cancer. 2012 Oct;12(10):721-32. doi: 10.1038/nrc3355. Epub 2012 Sep 21.