Literature DB >> 31884116

The effect of growth hormone treatment in a child with tricho-rhino-phalangeal syndrome: A case report and review of the literature.

Yael Levy-Shraga1, Dalit Modan-Moses2, Shlomo Wientroub3, Dror Ovadia3, Leonid Zeitlin3.   

Abstract

Tricho-rhino-phalangeal syndrome (TRPS) is characterized by craniofacial and skeletal malformations including short stature, cone-shaped phalangeal epiphyses and Perthes-like changes of the hip. We describe the response to growth hormone (GH) treatment in a boy with TRPS. The patient presented at age 3.5 years for evaluation of short stature (-3.2SD). On physical examination, the characteristic facial phenotype of TRPS was noted. Radiographs showed cone-shaped phalangeal epiphyses and bilateral small and fragmented femoral heads. The diagnosis was confirmed by Sanger sequencing of the TRPS1 gene. Two GH stimulation tests revealed GH deficiency, and GH treatment was initiated. Subsequently, growth velocity improved, as did the radiographic appearance of the femoral epiphyses, as seen on sequential pelvis radiographs. This observation suggests the possibility of a beneficial effect of GH treatment on both height and epiphyses status in TRPS patient with GH deficiency. Further studies are needed to support the observation.
Copyright © 2019 Elsevier Masson SAS. All rights reserved.

Entities:  

Keywords:  Growth hormone; Hip dysplasia; TRPS

Year:  2019        PMID: 31884116     DOI: 10.1016/j.ejmg.2019.103830

Source DB:  PubMed          Journal:  Eur J Med Genet        ISSN: 1769-7212            Impact factor:   2.708


  2 in total

1.  A novel missense variant in TRAPPC2 causes X-linked spondyloepiphyseal dysplasia tarda: A case report.

Authors:  Li Zhang; Jinling Wang; Guanping Dong; Dingwen Wu; Wei Wu
Journal:  Medicine (Baltimore)       Date:  2021-03-19       Impact factor: 1.817

2.  Novel Loss-of-Function Mutations in NPR2 Cause Acromesomelic Dysplasia, Maroteaux Type.

Authors:  Jing Wu; Mengru Wang; Zhouyang Jiao; Binghua Dou; Bo Li; Jianjiang Zhang; Haohao Zhang; Yue Sun; Xin Tu; Xiangdong Kong; Ying Bai
Journal:  Front Genet       Date:  2022-03-16       Impact factor: 4.599

  2 in total

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