Literature DB >> 1915520

A family with severe X-linked arthrogryposis.

R C Hennekam1, P G Barth, W Van Lookeren Campagne, M De Visser, K P Dingemans.   

Abstract

Five males are reported with severe X-linked arthrogryposis. Main findings are marked respiratory insufficiency and feeding problems, multiple contractures, deformities of chest and vertebral column, and typical facies. Most of these findings can be explained by a pronounced prenatal and postnatal muscle weakness. The sole living child has severe psychomotor retardation. Several female carriers show mild features (clubfeet, contractures, hyperkyphosis, and slight muscle weakness). One manifesting carrier is affected more severely (multiple contractures, mental retardation, and various dysmorphic features). Additional investigations including muscle biopsy revealed none of the usual signs of denervation, and pointed to the presence of a degenerative muscle disorder.

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Mesh:

Year:  1991        PMID: 1915520     DOI: 10.1007/bf02072628

Source DB:  PubMed          Journal:  Eur J Pediatr        ISSN: 0340-6199            Impact factor:   3.183


  6 in total

1.  Congenital muscular dystrophy. A case report with autopsyfindings.

Authors:  J K SHORT
Journal:  Neurology       Date:  1963-06       Impact factor: 9.910

2.  Arthrogryposis multiplex congenita with Pierre-Robin syndrome.

Authors:  L RAMAKUMAR; S C SOOD
Journal:  Indian J Pediatr       Date:  1961-04       Impact factor: 1.967

3.  Arthrogryposis multiplex due to congenital muscular dystrophy.

Authors:  B Q BANKER; M VICTOR; R D ADAMS
Journal:  Brain       Date:  1957-09       Impact factor: 13.501

4.  X-linked infantile spinal muscular atrophy.

Authors:  F Greenberg; K R Fenolio; J F Hejtmancik; D Armstrong; J K Willis; E Shapira; H W Huntington; R L Haun
Journal:  Am J Dis Child       Date:  1988-02

5.  Specific involvement of muscle, nerve, and skin in late infantile and juvenile amaurotic idiocy.

Authors:  S Carpenter; G Karpati; F Andermann
Journal:  Neurology       Date:  1972-02       Impact factor: 9.910

6.  Three distinct types of X-linked arthrogryposis seen in 6 families.

Authors:  J G Hall; S D Reed; C I Scott; J G Rogers; K L Jones; A Camarano
Journal:  Clin Genet       Date:  1982-02       Impact factor: 4.438

  6 in total
  4 in total

1.  ZC4H2 mutations are associated with arthrogryposis multiplex congenita and intellectual disability through impairment of central and peripheral synaptic plasticity.

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Journal:  Am J Hum Genet       Date:  2013-04-25       Impact factor: 11.025

2.  Deleterious de novo variants of X-linked ZC4H2 in females cause a variable phenotype with neurogenic arthrogryposis multiplex congenita.

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Journal:  Hum Mutat       Date:  2019-08-21       Impact factor: 4.878

3.  Loss of Protein Function Causing Severe Phenotypes of Female-Restricted Wieacker Wolff Syndrome due to a Novel Nonsense Mutation in the ZC4H2 Gene.

Authors:  Jing-Jing Sun; Qin Cai; Miao Xu; Yan-Na Liu; Wan-Rui Li; Juan Li; Li Ma; Cheng Cai; Xiao-Hui Gong; Yi-Tao Zeng; Zhao-Rui Ren; Fanyi Zeng
Journal:  Genes (Basel)       Date:  2022-08-29       Impact factor: 4.141

4.  A novel de novo nonsense mutation in ZC4H2 causes Wieacker-Wolff Syndrome.

Authors:  Dan Wang; Dongjie Hu; Zhichao Guo; Rong Hu; Qunxian Wang; Yannan Liu; Mingjing Liu; Zijun Meng; Huan Yang; Yun Zhang; Fang Cai; Weihui Zhou; Weihong Song
Journal:  Mol Genet Genomic Med       Date:  2019-12-30       Impact factor: 2.183

  4 in total

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