Suppr超能文献

在二倍体/三倍体嵌合体情况下,伴有线状体(杆状体)的中央核心病的新表现。

Novel presentation of central core disease with nemaline bodies (rods) in the setting of diploid/triploid mosaicism.

作者信息

Shafi Nadiya Babar, Parker Joseph C, Atkinson James B, Parker John R

机构信息

Department of Pathology, University of Louisville Hospital, 530 South Jackson St., Louisville, KY 40292, USA.

出版信息

Ann Clin Lab Sci. 2007 Spring;37(2):177-81.

Abstract

Diploid/triploid mosaicism is an uncommon malformation syndrome thought to result from incorporation of the second polar body into a blastomere nucleus of the developing embryo. Clinical manifestations include mental and growth retardation, truncal obesity, body asymmetry, hypotonia, syndactyly, clino-/camptodactyly, malformed low-set ears, and small phallus. Although muscular atrophy has been documented in 35% of cases of diploid/triploid mosaicism, to our knowledge histologic evidence of myopathy has not been reported. We present a novel case of diploid/triploid mosaicism with evidence of central core disease and nemaline bodies (rods). The histologic and ultrastructural features are described. A review of the literature is provided, including discussion of the various theories regarding the co-expression of central cores and nemaline rods.

摘要

二倍体/三倍体嵌合体是一种罕见的畸形综合征,被认为是由于第二极体并入发育中胚胎的卵裂球核所致。临床表现包括智力和生长发育迟缓、躯干肥胖、身体不对称、肌张力减退、并指/弯指畸形、低位畸形耳及小阴茎。虽然在35%的二倍体/三倍体嵌合体病例中记录有肌肉萎缩,但据我们所知,尚未有关于肌病组织学证据的报道。我们报告了一例二倍体/三倍体嵌合体新病例,有中央轴空病和线状体(杆状体)的证据。描述了其组织学和超微结构特征。提供了文献综述,包括关于中央轴空和线状体共同表达的各种理论的讨论。

相似文献

3
Congenital myopathies.
Curr Neurol Neurosci Rep. 2004 Jan;4(1):68-73. doi: 10.1007/s11910-004-0015-7.
4
Diploid/triploid mosaicism in dysmorphic patients.
Clin Genet. 2002 Nov;62(5):376-82. doi: 10.1034/j.1399-0004.2002.620504.x.
5
Beyond LGMD1A: myotilin is a component of central core lesions and nemaline rods.
Neuromuscul Disord. 2003 Aug;13(6):451-5. doi: 10.1016/s0960-8966(03)00064-6.
6
Somatic mosaicism in TPM2-related myopathy with nemaline rods and cap structures.
Acta Neuropathol. 2013 Jan;125(1):169-71. doi: 10.1007/s00401-012-1049-6. Epub 2012 Sep 27.
8
Three different origins for apparent triploid/diploid mosaics.
Prenat Diagn. 2003 Jul;23(7):529-34. doi: 10.1002/pd.634.

文献AI研究员

20分钟写一篇综述,助力文献阅读效率提升50倍。

立即体验

用中文搜PubMed

大模型驱动的PubMed中文搜索引擎

马上搜索

文档翻译

学术文献翻译模型,支持多种主流文档格式。

立即体验