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DOI 10.1002/ajmg.a.63422

Медицинская библиотека · American journal of medical genetics. Part A · 2023

Indian patients with CHST3-related chondrodysplasia with congenital joint dislocations.

Singh Swati, Jacob Prince, Patil Siddaramappa J, Muranjan Mamta, Shah Hitesh, Girisha Katta M, Bhavani Gandham SriLakshmi

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Аннотация

CHST3-related chondrodysplasia with congenital joint dislocations (CDCJD, #MIM 143095), is a rare genetic skeletal disorder caused by biallelic loss of function variants in CHST3. CHST3 is critical for the sulfation of chondroitin sulfate. This study delineates the clinical presentation of nine individuals featuring the key symptoms of CDCJD; congenital joint (knee and elbow) dislocations, short trunk short stature progressive vertebral anomalies, and metacarpal shortening. Additional manifestations include irregular distal femoral epiphysis, supernumerary carpal ossification centers, bifid humerus, club foot, and cardiac abnormalities. Sanger sequencing was carried out to investigate molecular etiology in eight patients and exome sequencing in one. Genetic testing revealed five homozygous variants in CHST3 (four were novel and one was previously reported). All these variants are located on sulfotransferase domain of CHST3 protein and were classified as pathogenic/ likely pathogenic. We thus report on nine individuals with CHST3-related chondrodysplasia with congenital joint dislocations from India and suggest monitoring the health of cardiac valves in this condition.

Для цитирования: Singh Swati, Jacob Prince, Patil Siddaramappa J, Muranjan Mamta, Shah Hitesh, Girisha Katta M, Bhavani Gandham SriLakshmi. Indian patients with CHST3-related chondrodysplasia with congenital joint dislocations.. American journal of medical genetics. Part A. 2023. DOI: 10.1002/ajmg.a.63422.

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