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SOFT syndrome caused by compound heterozygous mutations of POC1A and its skeletal manifestation.
- Source :
-
Journal of human genetics [J Hum Genet] 2016 Jun; Vol. 61 (6), pp. 561-4. Date of Electronic Publication: 2016 Jan 21. - Publication Year :
- 2016
-
Abstract
- SOFT syndrome (MIM614813) is an extremely rare primordial dwarfism characterized by short stature, onychodysplasia, facial dysmorphism and hypotrichosis, which is caused by biallelic mutations in the POC1A gene. Only 19 patients with mutation-confirmed SOFT syndrome have been reported to date, all of whom carried homozygous variants that were strongly associated with consanguineous marriages. We report an 8.5-year-old boy with SOFT syndrome showing primordial dwarfism, no effect of growth-hormone therapy and skeletal dysplasia. This is the first report of compound heterozygous variants in POC1A, one previously reported and the other novel. A characteristic skeletal manifestation is reported.
- Subjects :
- Amino Acid Substitution
Cell Cycle Proteins
Cytoskeletal Proteins
DNA Mutational Analysis
Exome
High-Throughput Nucleotide Sequencing
Humans
Infant
Male
Pedigree
Syndrome
Abnormalities, Multiple diagnosis
Abnormalities, Multiple genetics
Heterozygote
Mutation
Phenotype
Proteins genetics
Skeleton pathology
Subjects
Details
- Language :
- English
- ISSN :
- 1435-232X
- Volume :
- 61
- Issue :
- 6
- Database :
- MEDLINE
- Journal :
- Journal of human genetics
- Publication Type :
- Academic Journal
- Accession number :
- 26791357
- Full Text :
- https://doi.org/10.1038/jhg.2015.174