| Literature DB >> 29884795 |
Gizem Ürel-Demir1, Pelin Ozlem Simsek-Kiper2, Özlem Akgün-Doğan2, Rahşan Göçmen3, Zheng Wang4,5, Naomichi Matsumoto6, Noriko Miyake6, Gülen Eda Utine2, Gen Nishimura7, Shiro Ikegawa4, Koray Boduroglu2.
Abstract
Spondylo-meta-epiphyseal dysplasia, short limb-abnormal calcification type, is a rare autosomal recessive disorder of the skeleton characterized by disproportionate short stature with narrow chest and dysmorphic facial features. The skeletal manifestations include platyspondyly, short flared ribs, short tubular bones with abnormal metaphyses and epiphyses, severe brachydactyly, and premature stippled calcifications in the cartilage. The abnormal calcifications are so distinctive as to point to the definitive diagnosis. However, they may be too subtle to attract diagnostic attention in infancy. Homozygous variants in DDR2 cause this disorder. We report on a 5-year-old girl with the classic phenotype of SMED, SL-AC in whom a novel homozygous nonsense mutation in DDR2 was detected using exome sequencing.Entities:
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Year: 2018 PMID: 29884795 DOI: 10.1038/s10038-018-0473-4
Source DB: PubMed Journal: J Hum Genet ISSN: 1434-5161 Impact factor: 3.172