| Literature DB >> 30462379 |
Shinji Takeyari1, Takuo Kubota1, Kei Miyata1, Kenichi Yamamoto1,2, Hirofumi Nakayama1,3, Keiko Yamamoto1, Yasuhisa Ohata1,4, Taichi Kitaoka1, Kumiko Yanagi5, Tadashi Kaname5, Keiichi Ozono1.
Abstract
Cole-Carpenter syndrome is a rare skeletal dysplasia associated with low-bone mass or an osteogenesis imperfecta (OI)-like syndrome. Only 3 and 6 variants in SEC24D have been reported in patients with Cole-Carpenter syndrome type 2 and autosomal recessive OI, respectively. We describe a 15-year-old Japanese boy with short stature of the short-trunk type and craniofacial abnormalities including ocular proptosis, marked frontal bossing, midface hypoplasia, and micrognathia. These features were consistent with a diagnosis of Cole-Carpenter syndrome. He had low-bone mineral density and basilar impression. Whole exome sequencing analysis identified biallelic variants in SEC24D (p.Arg484* and p.Arg313His) in the patient. We will report a patient with compound heterozygous variants of SEC24D causing Cole-Carpenter syndrome type 2.Entities:
Keywords: zzm321990SEC24D; Cole-carpenter syndrome; osteogenesis imperfecta
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Year: 2018 PMID: 30462379 DOI: 10.1002/ajmg.a.40643
Source DB: PubMed Journal: Am J Med Genet A ISSN: 1552-4825 Impact factor: 2.802