Startseite Medizin A family with Camurati-Engelman disease. The role of the missense p.R218C mutation in TGFB1 in bones and endocrine glands
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A family with Camurati-Engelman disease. The role of the missense p.R218C mutation in TGFB1 in bones and endocrine glands

  • Meropi Toumba EMAIL logo , Vassos Neocleous , Christos Shammas , Violetta Anastasiadou , Jeremy Allgrove , Leonidas A. Phylactou und Nicos Skordis
Veröffentlicht/Copyright: 17. Mai 2013

Abstract

Objectives: Camurati-Engelmann disease (CED) is a rare form of progressive bone dysplasia due to mutations in the transforming factor gene TGFB1 on chromosome 19q13.1-q13.3. Endocrine complications such as osteoporosis, vitamin D deficiency, delayed puberty and hypogonadotrophic hypogonadism may be present.

Methods and results: Genetic analysis of the TGFB1 gene revealed a heterozygous missense mutation p.R218C in exon 4 of chromosome 19q13.1-q13.3 in a 14-year-old girl who presented with typical symptoms of CED, hyperprolactinaemia and menstrual irregularity. The patient responded well to prednisone 5 mg/kg per day as well as calcium and vitamin D supplements.

Conclusions: The role of p.R218C in TGFB1 on the mechanism of the disease itself and the complications of it in bones and endocrine glands remain unclear. Early recognition as well as a detailed understanding of the pathogenesis of the disease is important for future treatment options and better quality of life of such patients.


Corresponding author: Meropi Toumba, MD, Department of Paediatrics, IASIS Hospital, 8036, Paphos, Cyprus, Phone: +357 99573633, Fax: +357 26848300, E-mail:

Conflict of interest statement

The authors have no conflict of interests.

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Received: 2013-1-20
Accepted: 2013-4-18
Published Online: 2013-05-17
Published in Print: 2013-10-01

©2013 by Walter de Gruyter Berlin Boston

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