Literature DB >> 1342874

New autosomal recessive chondrodysplasia--pseudohermaphrodism syndrome.

A Nivelon1, J L Nivelon, J P Mabille, P Maroteaux, J P Feldman, S Douvier, S Aymé.   

Abstract

Two siblings with a previously undescribed syndrome are presented. They both have severe dwarfism, antenatal in origin, with generalized chondrodysplasia, severe microcephaly with cerebellar vermis hypoplasia, a hypoplastic iris and a papillous coloboma (Coloboma of the optic disc). The first sibling has a 46,XY karyotype despite normal female internal and external genitalia. She has moderate mental retardation. Gestation of the second sibling was interrupted after antenatal diagnosis. The fetus was 46,XX and very similar to the first case.

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Year:  1992        PMID: 1342874

Source DB:  PubMed          Journal:  Clin Dysmorphol        ISSN: 0962-8827            Impact factor:   0.816


  3 in total

Review 1.  The Smith-Lemli-Opitz syndrome.

Authors:  R I Kelley; R C Hennekam
Journal:  J Med Genet       Date:  2000-05       Impact factor: 6.318

2.  Hedgehog acyl-transferase-related multiple congenital anomalies: Report of an additional family and delineation of the syndrome.

Authors:  Shruti Pande; Periyasamy Radhakrishnan; Naveenchandra M Shetty; Anju Shukla; Katta M Girisha
Journal:  Am J Med Genet A       Date:  2021-03-22       Impact factor: 2.578

3.  Loss of function mutation in the palmitoyl-transferase HHAT leads to syndromic 46,XY disorder of sex development by impeding Hedgehog protein palmitoylation and signaling.

Authors:  Patrick Callier; Pierre Calvel; Armine Matevossian; Periklis Makrythanasis; Pascal Bernard; Hiroshi Kurosaka; Anne Vannier; Christel Thauvin-Robinet; Christelle Borel; Séverine Mazaud-Guittot; Antoine Rolland; Christèle Desdoits-Lethimonier; Michel Guipponi; Céline Zimmermann; Isabelle Stévant; Françoise Kuhne; Béatrice Conne; Federico Santoni; Sandy Lambert; Frederic Huet; Francine Mugneret; Jadwiga Jaruzelska; Laurence Faivre; Dagmar Wilhelm; Bernard Jégou; Paul A Trainor; Marilyn D Resh; Stylianos E Antonarakis; Serge Nef
Journal:  PLoS Genet       Date:  2014-05-01       Impact factor: 5.917

  3 in total

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