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The Clinical Spectrum of McCune-Albright Syndrome and Its Management.
Spencer, Tiahna; Pan, Kristen S; Collins, Michael T; Boyce, Alison M.
Afiliação
  • Spencer T; Skeletal Disorders and Mineral Homeostasis Section, National Institute of Dental and Craniofacial Research, National Institutes of Health, Bethesda, Maryland, USA.
  • Pan KS; Skeletal Disorders and Mineral Homeostasis Section, National Institute of Dental and Craniofacial Research, National Institutes of Health, Bethesda, Maryland, USA.
  • Collins MT; Skeletal Disorders and Mineral Homeostasis Section, National Institute of Dental and Craniofacial Research, National Institutes of Health, Bethesda, Maryland, USA.
  • Boyce AM; Skeletal Disorders and Mineral Homeostasis Section, National Institute of Dental and Craniofacial Research, National Institutes of Health, Bethesda, Maryland, USA, boyceam@mail.nih.gov.
Horm Res Paediatr ; 92(6): 347-356, 2019.
Article em En | MEDLINE | ID: mdl-31865341
McCune-Albright syndrome (MAS) is a rare, mosaic disorder presenting along a broad clinical spectrum. Disease arises from somatic-activating GNAS mutations, leading to constitutive Gαs activation and ligand-independent signaling of the Gαs-coupled protein receptor. The phenotype is largely determined by location and extent of tissues in which the GNAS mutation is expressed, as well as the pathophysiologic effects of Gαs activation within these tissues. Patients pre-sent clinically with a variable combination of fibrous dysplasia of bone (FD), café-au-lait skin macules, and hyperfunctioning endocrinopathies. In bone, Gαs leads to impaired differentiation of skeletal stem cells and formation of discrete, expansile FD lesions, resulting in fractures, pain, and functional impairment. A systematic approach to diagnosis and management is critically important to optimize outcomes for patients with FD/MAS. There are no medical therapies capable of altering the disease course in FD; however, screening and treatment for endocrinopathies can mitigate some skeletal morbidities. This review summarizes current understanding of MAS pathophysiology, describes the spectrum of clinical features, and includes a detailed discussion of the recommended approach to diagnosis and management.
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Texto completo: 1 Base de dados: MEDLINE Assunto principal: Cromograninas / Subunidades alfa Gs de Proteínas de Ligação ao GTP / Displasia Fibrosa Poliostótica / Mutação Tipo de estudo: Diagnostic_studies Limite: Humans Idioma: En Ano de publicação: 2019 Tipo de documento: Article

Texto completo: 1 Base de dados: MEDLINE Assunto principal: Cromograninas / Subunidades alfa Gs de Proteínas de Ligação ao GTP / Displasia Fibrosa Poliostótica / Mutação Tipo de estudo: Diagnostic_studies Limite: Humans Idioma: En Ano de publicação: 2019 Tipo de documento: Article