Shafi Nadiya Babar, Parker Joseph C, Atkinson James B, Parker John R
Department of Pathology, University of Louisville Hospital, 530 South Jackson St., Louisville, KY 40292, USA.
Ann Clin Lab Sci. 2007 Spring;37(2):177-81.
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.
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。