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Am J Med Genet A ; 188(11): 3343-3349, 2022 11.
Artigo em Inglês | MEDLINE | ID: mdl-35972031

RESUMO

Cutis laxa (CL) is a rare connective tissue disorder characterized by wrinkled, abundant and sagging skin, sometimes associated with systemic impairment. Biallelic alterations in latent transforming growth factor beta-binding protein 4 gene (LTBP4) cause autosomal recessive type 1C cutis laxa (ARCL1C, MIM #613177). The present report describes the case of a 17-months-old girl with cutis laxa together with a literature review of previous ARCL1C cases. Based on proband main clinical signs (cutis laxa and pulmonary emphysema), clinical exome sequencing (CES) was performed and showed a new nine base-pairs homozygous in-frame deletion in LTBP4 gene. RT-PCR and cDNA Sanger sequencing were performed in order to clarify its impact on RNA. This report demonstrates that a genetic alteration in the EGF-like 14 domain calcium-binding motif of LTBP4 gene is likely responsible for cutis laxa in our patient.


Assuntos
Cútis Laxa , Cálcio , Doenças das Cartilagens , Cútis Laxa/genética , DNA Complementar , Fator de Crescimento Epidérmico , Feminino , Gastroenteropatias , Humanos , Lactente , Proteínas de Ligação a TGF-beta Latente/genética , RNA , Doenças Respiratórias , Fator de Crescimento Transformador beta , Doenças Urológicas
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