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DOI 10.1590/1678-4685-gmb-2022-0335

Медицинская библиотека · Genetics and molecular biology · 2024

An adult with cystathionine beta-synthase deficiency, camptodactyly-arthropathy-coxa vara-pericarditis syndrome, and deafness: A case report.

Donis Karina Carvalho, Kalil Marco Antônio Baptista, Poswar Fabiano, Kok Fernando, Kohem Charles Lubianca, Poloni Soraia, Borsatto Taciane, Vairo Filippo Pinto E, Pinheiro Franciele Cabral, Schwartz Ida Vanessa Doederlein

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Аннотация

Massive sequencing platforms allow the identification of complex clinical phenotypes involving more than one autosomal recessive disorder. In this study, we report on an adult patient, born to a related couple (third degree cousins), referred for genetic evaluation due to ectopia lentis, deafness and previous diagnosis of juvenile idiopathic arthritis. He was biochemically diagnosed as having Classic Homocystinuria (HCU); Sanger sequencing of the CBS gene showed the genotype NM_000071.2(CBS):c.[833T>C];[833T>C], compatible with the diagnosis of pyridoxine-responsive HCU. As he also had symptoms not usually associated with HCU, exome sequencing was performed. In addition to the variants found in the Sanger sequencing, the following variants were identified: NM_001256317.1(TMPRSS3):c.[413C>A];[413C>A]; and the NM_005807.6(PRG4):c.[3756dup]:[3756dup], confirming the diagnosis of autosomal recessive nonsyndromic deafness and Camptodactyly-Arthropathy-Coxa Vara-Pericarditis Syndrome (CACP), respectively. Genomic analysis allowed the refinement of the diagnosis of a complex case and improvement of the patient's treatment.

Для цитирования: Donis Karina Carvalho, Kalil Marco Antônio Baptista, Poswar Fabiano, Kok Fernando, Kohem Charles Lubianca, Poloni Soraia, Borsatto Taciane, Vairo Filippo Pinto E, Pinheiro Franciele Cabral, Schwartz Ida Vanessa Doederlein. An adult with cystathionine beta-synthase deficiency, camptodactyly-arthropathy-coxa vara-pericarditis syndrome, and deafness: A case report.. Genetics and molecular biology. 2024. DOI: 10.1590/1678-4685-gmb-2022-0335.
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