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PMID: 42458851 已发表 · aheadofprint 英语

Ocular and Systemic Findings in COL2A1 and COL11A1 Stickler Syndrome.

MacLachlan AG, Kraker JA, Morgan DJ, Schimmenti LA, Hartnett ME, Hwang ES

摘要

Stickler syndrome is most commonly caused by variants in COL2A1 and COL11A1 genes. The purpose of this study was to describe genetic variants and phenotypes in COL2A1 and COL11A1 Stickler syndrome. We performed a retrospective genotype-phenotype evaluation of COL2A1 and COL11A1 Stickler syndrome subjects. Thirty-two subjects with COL2A1 and 13 subjects with COL11A1 Stickler syndrome were last seen in clinic at a mean age of 17.2 ± 13.1 and 12.6 ± 3.8 years, respectively (p = 0.08). We found that 50% of COL2A1 subjects had lattice degeneration, whereas 8% of the COL11A1 subjects had lattice degeneration (p = 0.015). The most common type of variant for COL2A1 Stickler syndrome was a premature termination codon (12 out of 18 families, 67%); and the most common types of variants for COL11A1 Stickler syndrome were glycine missense variants (4 out of 10 families, 40%) and splice site variants (4 out of 10 families, 40%). There was a higher rate of lattice degeneration in COL2A1 patients compared with COL11A1 patients, a finding that could be influenced by a trend towards older age at last follow-up in the COL2A1 group.

关键词
COL11A1 COL2A1 Stickler syndrome lattice degeneration
文献信息
期刊
American journal of medical genetics. Part A
期刊简称
Am J Med Genet A
ISSN
1552-4833
发表日期
2026-07-15
语言
英语
国家/地区
United States
NLM ID
101235741
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