Literature DB >> 934161

[A new (brachymelic) type of primordial dwarfism (author's transl)].

F Majewski, J Spranger.   

Abstract

Second report of an apparently new type of primordial dwarfism characterized by severe intrauterine and postnatal growth retardation, striking craniofacial deformities and a peculiar osteochondrodysplasia. The major clinical findings are microcephaly, hypotrichosis, bulging eyes, prominent nose, micrognathia and short extremities. The osteochondrodysplasia is characterized by short and bowed humeri and femora with absent ossification of the femoral necks, small and dysplastic iliac wings, strikingly retarded ossification of the epiphyses and shortened metacarpal I and middle phalanges II-V. Possible this condition is caused by the homozygous state of mutant gene. The brachymelic type of primordial dwarfism differs from other forms of primordial dwarfism (particularly from case I of Seckel) by its skeletal abnormalities. The cases described by Seckel (and sometimes referred to as Seckel or bird-headed dwarfism) are heterogenous: Seckel dwarfism apparently does not exist as a nosologic entity.

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Year:  1976        PMID: 934161

Source DB:  PubMed          Journal:  Monatsschr Kinderheilkd


  8 in total

Review 1.  International classification of osteochondrodysplasias. The International Working Group on Constitutional Diseases of Bone.

Authors:  J Spranger
Journal:  Eur J Pediatr       Date:  1992-06       Impact factor: 3.183

2.  Microcephalic osteodysplastic primordial dwarfism type I with biallelic mutations in the RNU4ATAC gene.

Authors:  R Nagy; H Wang; B Albrecht; D Wieczorek; G Gillessen-Kaesbach; E Haan; P Meinecke; A de la Chapelle; J A Westman
Journal:  Clin Genet       Date:  2011-08-28       Impact factor: 4.438

3.  Case report. Microcephalic osteodysplastic primordial dwarfism type II: a child with unusual symptoms and clinical course.

Authors:  S Spranger; G Tariverdian; F K Albert; D Sontheimer; J Zöller; M Weber; J Tröger
Journal:  Eur J Pediatr       Date:  1996-09       Impact factor: 3.183

4.  Autozygosity mapping of a seckel syndrome locus to chromosome 3q22. 1-q24.

Authors:  J Goodship; H Gill; J Carter; A Jackson; M Splitt; M Wright
Journal:  Am J Hum Genet       Date:  2000-07-11       Impact factor: 11.025

5.  Mutations in U4atac snRNA, a component of the minor spliceosome, in the developmental disorder MOPD I.

Authors:  Huiling He; Sandya Liyanarachchi; Keiko Akagi; Rebecca Nagy; Jingfeng Li; Rosemary C Dietrich; Wei Li; Nikhil Sebastian; Bernard Wen; Baozhong Xin; Jarnail Singh; Pearlly Yan; Hansjuerg Alder; Eric Haan; Dagmar Wieczorek; Beate Albrecht; Erik Puffenberger; Heng Wang; Judith A Westman; Richard A Padgett; David E Symer; Albert de la Chapelle
Journal:  Science       Date:  2011-04-08       Impact factor: 47.728

6.  Seckel syndrome: an overdiagnosed syndrome.

Authors:  E Thompson; M Pembrey
Journal:  J Med Genet       Date:  1985-06       Impact factor: 6.318

7.  A new familial intrauterine growth retardation syndrome the "3-M syndrome".

Authors:  J Spranger; J M Opitz; A Nourmand
Journal:  Eur J Pediatr       Date:  1976-09-01       Impact factor: 3.183

8.  Lowry-Wood syndrome: further evidence of association with RNU4ATAC, and correlation between genotype and phenotype.

Authors:  Ivan Shelihan; Sophie Ehresmann; Cinzia Magnani; Francesca Forzano; Chiara Baldo; Nicola Brunetti-Pierri; Philippe M Campeau
Journal:  Hum Genet       Date:  2018-10-27       Impact factor: 4.132

  8 in total

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