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Clinical and genetic diagnosis of autosomal dominant osteopetrosis type II in a Chinese family: A case report.
Gong, Hong-Ping; Ren, Yan; Zha, Pan-Pan; Zhang, Wen-Yan; Zhang, Jin; Zhang, Zhi-Wen; Wang, Chun.
Afiliación
  • Gong HP; International Medical Center Ward, General Practice Medical Center, Sichuan University West China Hospital, Chengdu 610041, Sichuan Province, China.
  • Ren Y; Department of Endocrinology and Metabolism, Sichuan University West China Hospital, Chengdu 610041, Sichuan Province, China.
  • Zha PP; Department of Endocrinology and Metabolism, Sichuan University West China Hospital, Chengdu 610041, Sichuan Province, China.
  • Zhang WY; Department of Endocrinology and Metabolism, Sichuan University West China Hospital, Chengdu 610041, Sichuan Province, China.
  • Zhang J; Department of Pathology, West China Hospital, Sichuan University, Chengdu 610041, Sichuan Province, China.
  • Zhang ZW; Department of Endocrinology and Metabolism, The People's Hospital of Leshan, Leshan 614003, Sichuan Province, China.
  • Wang C; Department of Endocrinology and Metabolism, The People's Hospital of Leshan, Leshan 614003, Sichuan Province, China.
World J Clin Cases ; 11(3): 700-708, 2023 Jan 26.
Article en En | MEDLINE | ID: mdl-36793634
BACKGROUND: Osteopetrosis is a rare genetic disorder characterized by increased bone density due to defective bone resorption of osteoclasts. Approximately, 80% of autosomal dominant osteopetrosis type II (ADO-II) patients were usually affected by heterozygous dominant mutations in the chloride voltage-gated channel 7 (ClCN7) gene and present early-onset osteoarthritis or recurrent fractures. In this study, we report a case of persistent joint pain without bone injury or underlying history. CASE SUMMARY: We report a 53-year-old female with joint pain who was accidentally diagnosed with ADO-II. The clinical diagnosis was based on increased bone density and typical radiographic features. Two heterozygous mutations in the ClCN7 and T-cell immune regulator 1 (TCIRG1) genes by whole exome sequencing were identified in the patient and her daughter. The missense mutation (c.857G>A) occurred in the CLCN7 gene p. R286Q, which is highly conserved across species. The TCIRG1 gene point mutation (c.714-20G>A) in intron 7 (near the splicing site of exon 7) had no effect on subsequent transcription. CONCLUSION: This ADO-II case had a pathogenic CLCN7 mutation and late onset without the usual clinical symptoms. For the diagnosis and assessment of the prognosis for osteopetrosis, genetic analysis is advised.
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Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Tipo de estudio: Diagnostic_studies / Prognostic_studies Idioma: En Revista: World J Clin Cases Año: 2023 Tipo del documento: Article País de afiliación: China Pais de publicación: Estados Unidos

Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Tipo de estudio: Diagnostic_studies / Prognostic_studies Idioma: En Revista: World J Clin Cases Año: 2023 Tipo del documento: Article País de afiliación: China Pais de publicación: Estados Unidos