Literature DB >> 21068148

The jumping SHOX gene--crossover in the pseudoautosomal region resulting in unusual inheritance of Leri-Weill dyschondrosteosis.

Sarina G Kant1, Hetty J van der Kamp, Marjolein Kriek, Egbert Bakker, Boudewijn Bakker, Mariette J V Hoffer, Patrick van Bunderen, Monique Losekoot, Saskia M Maas, Jan M Wit, Gudrun Rappold, Martijn H Breuning.   

Abstract

CONTEXT: During meiosis I, the recombination frequency in the pseudoautosomal region on Xp and Yp (PAR1) in males is very high. As a result, mutated genes located within the PAR1 region can be transferred from the Y-chromosome to the X-chromosome and vice versa. PATIENTS: Here we describe three families with SHOX abnormalities resulting in Leri-Weill dyschondrosteosis or Langer mesomelic dysplasia.
RESULTS: In about half of the segregations investigated, a transfer of the SHOX abnormality to the alternate sex chromosome was demonstrated.
CONCLUSIONS: Patients with an abnormality of the SHOX gene should receive genetic counseling as to the likelihood that they may transmit the mutation or deletion to a son as well as to a daughter.

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Year:  2010        PMID: 21068148     DOI: 10.1210/jc.2010-1505

Source DB:  PubMed          Journal:  J Clin Endocrinol Metab        ISSN: 0021-972X            Impact factor:   5.958


  6 in total

1.  A novel intronic mutation in SHOX causes short stature by disrupting a splice acceptor site: direct demonstration of aberrant splicing by expression of a minigene in HEK-293T cells.

Authors:  Jennifer Danzig; Michael A Levine
Journal:  J Pediatr Endocrinol Metab       Date:  2012       Impact factor: 1.634

Review 2.  SHOX Haploinsufficiency as a Cause of Syndromic and Nonsyndromic Short Stature.

Authors:  Maki Fukami; Atsuhito Seki; Tsutomu Ogata
Journal:  Mol Syndromol       Date:  2016-03-15

3.  Rare inheritance of Leri-Weill Syndrome due to crossover of short stature Homeobox Gene (SHOX) Deletions between X and Y Chromosomes: a case report.

Authors:  Marisa Censani; Kwame Anyane-Yeboa; Ronald Wapner; Erica Spiegel; Edwin Guzman; Sharon E Oberfield
Journal:  Int J Pediatr Endocrinol       Date:  2013-06-28

Review 4.  A Track Record on SHOX: From Basic Research to Complex Models and Therapy.

Authors:  Antonio Marchini; Tsutomu Ogata; Gudrun A Rappold
Journal:  Endocr Rev       Date:  2016-06-29       Impact factor: 19.871

5.  Phenotypic characterization of patients with deletions in the 3'-flanking SHOX region.

Authors:  Sarina G Kant; Sander J Broekman; Caroline C de Wit; Marloes Bos; Sitha A Scheltinga; Egbert Bakker; Wilma Oostdijk; Hetty J van der Kamp; Erik W van Zwet; Annemieke H van der Hout; Jan M Wit; Monique Losekoot
Journal:  PeerJ       Date:  2013-02-19       Impact factor: 2.984

6.  Human Spermatogenesis Tolerates Massive Size Reduction of the Pseudoautosomal Region.

Authors:  Maki Fukami; Yasuko Fujisawa; Hiroyuki Ono; Tomoko Jinno; Tsutomu Ogata
Journal:  Genome Biol Evol       Date:  2020-11-03       Impact factor: 3.416

  6 in total

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