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

نویسندگان

  • Nadiya Babar Shafi
  • Joseph C Parker
  • James B Atkinson
  • John R Parker
چکیده

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.

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عنوان ژورنال:
  • Annals of clinical and laboratory science

دوره 37 2  شماره 

صفحات  -

تاریخ انتشار 2007