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Diaphanospondylodysostosis and ischiospinal dysostosis, evidence for one disorder with variable expression in a patient who has survived to age 9 years
Journal article   Open access   Peer reviewed

Diaphanospondylodysostosis and ischiospinal dysostosis, evidence for one disorder with variable expression in a patient who has survived to age 9 years

Janet M Legare, Kristin Seaborg, Jennifer Laffin and Philip F Giampietro
American journal of medical genetics. Part A, v 173(10), pp 2808-2813
Oct 2017
PMID: 28815954
url
https://doi.org/10.1016/j.mtla.2020.100872View
Accepted (AM)Maybe Open Access (Publisher Bronze) Open

Abstract

Carrier Proteins - genetics Child Craniofacial Abnormalities - genetics Craniofacial Abnormalities - pathology Dysostoses - genetics Dysostoses - pathology Humans Ischium - pathology Male Mutation Prognosis Ribs - abnormalities Ribs - pathology Spine - abnormalities Spine - pathology
Diaphanospondylodysostosis (DSD) and ischiospinal dysostosis (ISD) are both rare skeletal dysplasias consisting of abnormal axial skeletal development but normal appendicular skeletal development. Both disorders recently have been found to result from mutations in the BMPER gene. We report a patient with one deletion and one mutation of the BMPER gene who has features most consistent with DSD but who has survived to age 9 years. Survival suggests that DSD and ISD reflect a spectrum of severity of one disease process.

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Collaboration types
Domestic collaboration
Web of Science research areas
Genetics & Heredity
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