Literature DB >> 9799317

Trichorhinophalangeal syndrome type I in monozygotic twins discordant for hip pathology. Report on the morphological evolution of cone-shaped epiphyses and the unusual pattern of skeletal maturation.

A Naselli1, M Vignolo, E Di Battista, V Papale, G Aicardi, S Becchetti, P Tomà.   

Abstract

A pair of monozygotic twin girls with trichorhinophalangeal syndrome type I (TRPS I), followed from 8.3 to 16.1 years of age, is described. Both showed typical dysmorphic features and severe short stature, but only one had Perthes-like changes in the right capital femoral epiphysis. The radiographic findings and evolutionary changes of phalangeal cone-shaped epiphyses (PCSE) of the hands are illustrated in this report. The unusual bone maturation and growth of the twins are also described. Both presented poor growth and delayed bone age until about 13 years, followed by marked acceleration of bone age and stunted pubertal height spurt.

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Year:  1998        PMID: 9799317     DOI: 10.1007/s002470050481

Source DB:  PubMed          Journal:  Pediatr Radiol        ISSN: 0301-0449


  5 in total

1.  Tricho-rhino-phalangeal syndrome type I in a Japanese boy.

Authors:  Tomohiro Kamoda; Akira Matsui
Journal:  Eur J Pediatr       Date:  2003-09-17       Impact factor: 3.183

2.  Genotypic and phenotypic spectrum in tricho-rhino-phalangeal syndrome types I and III.

Authors:  H J Lüdecke; J Schaper; P Meinecke; P Momeni; S Gross; H Hirche; M J Abramowicz; B Albrecht; C Apacik; H J Christen; U Claussen; K Devriendt; E Fastnacht; A Forderer; U Friedrich; T H Goodship; M Greiwe; H Hamm; R C Hennekam; G K Hinkel; M Hoeltzenbein; H Kayserili; F Majewski; M Mathieu; R McLeod; A T Midro; U Moog; T Nagai; N Niikawa; K H Orstavik; E Plöchl; C Seitz; J Schmidtke; L Tranebjaerg; M Tsukahara; B Wittwer; B Zabel; G Gillessen-Kaesbach; B Horsthemke
Journal:  Am J Hum Genet       Date:  2000-12-07       Impact factor: 11.025

3.  Severe brachydactyly and short stature resulting from a novel pathogenic TRPS1 variant within the GATA DNA-binding domain.

Authors:  Anara Karaca; Monica Reyes; Lauren T Shumate; Isilay Taskaldiran; Tulay Omma; Nese Ersoz Gulcelik; Murat Bastepe
Journal:  Bone       Date:  2019-03-23       Impact factor: 4.398

4.  Uncoupling of chondrocyte differentiation and perichondrial mineralization underlies the skeletal dysplasia in tricho-rhino-phalangeal syndrome.

Authors:  Dobrawa Napierala; Kathy Sam; Roy Morello; Qiping Zheng; Elda Munivez; Ramesh A Shivdasani; Brendan Lee
Journal:  Hum Mol Genet       Date:  2008-04-17       Impact factor: 6.150

5.  A novel TRPS1 gene mutation causing trichorhinophalangeal syndrome with growth hormone responsive short stature: a case report and review of the literature.

Authors:  Lina Merjaneh; John S Parks; Andrew B Muir; Doris Fadoju
Journal:  Int J Pediatr Endocrinol       Date:  2014-08-15
  5 in total

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