Literature DB >> 31865341

The Clinical Spectrum of McCune-Albright Syndrome and Its Management.

Tiahna Spencer1, Kristen S Pan1, Michael T Collins1, Alison M Boyce2.   

Abstract

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.
© 2019 S. Karger AG, Basel.

Entities:  

Keywords:  Fibroblast growth factor 23; Fibrous dysplasia; Gαs; Metabolic bone disease; Mosaicism

Mesh:

Substances:

Year:  2019        PMID: 31865341      PMCID: PMC7302983          DOI: 10.1159/000504802

Source DB:  PubMed          Journal:  Horm Res Paediatr        ISSN: 1663-2818            Impact factor:   2.852


  74 in total

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2.  Burosumab Therapy in Children with X-Linked Hypophosphatemia.

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Journal:  N Engl J Med       Date:  2018-05-24       Impact factor: 91.245

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Authors:  Roberta Elisa Rossi; Sara Massironi
Journal:  Expert Rev Gastroenterol Hepatol       Date:  2018-10-08       Impact factor: 3.869

Review 4.  GNAS Spectrum of Disorders.

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Journal:  Curr Osteoporos Rep       Date:  2015-06       Impact factor: 5.096

5.  Cushing syndrome in the McCune-Albright syndrome.

Authors:  Rebecca J Brown; Marilyn H Kelly; Michael T Collins
Journal:  J Clin Endocrinol Metab       Date:  2010-02-15       Impact factor: 5.958

6.  Effect of pamidronate treatment in children with polyostotic fibrous dysplasia of bone.

Authors:  Horacio Plotkin; Frank Rauch; Leonid Zeitlin; Craig Munns; Rose Travers; Francis H Glorieux
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7.  Sinonasal disease in polyostotic fibrous dysplasia and McCune-Albright Syndrome.

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8.  Activating mutations of the stimulatory G protein in the McCune-Albright syndrome.

Authors:  L S Weinstein; A Shenker; P V Gejman; M J Merino; E Friedman; A M Spiegel
Journal:  N Engl J Med       Date:  1991-12-12       Impact factor: 91.245

9.  Fulvestrant treatment of precocious puberty in girls with McCune-Albright syndrome.

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Review 10.  Fibrous dysplasia for radiologists: beyond ground glass bone matrix.

Authors:  Yevgeniya S Kushchayeva; Sergiy V Kushchayev; Tetiana Y Glushko; Sri Harsha Tella; Oleg M Teytelboym; Michael T Collins; Alison M Boyce
Journal:  Insights Imaging       Date:  2018-11-27
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Review 1.  Physical Examination for Endocrine Diseases: Does It Still Play a Role?

Authors:  Andrea Crafa; Rosita A Condorelli; Rossella Cannarella; Antonio Aversa; Aldo E Calogero; Sandro La Vignera
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2.  McCune-Albright syndrome - A case report with transmission electron microscopy.

Authors:  Victor Garcia Neto; Hiram Larangeira de Almeida; Claúdia Fernandes Lorea; Valéria Magalhães Jorge; Antônia Larangeira de Almeida
Journal:  An Bras Dermatol       Date:  2021-11-25       Impact factor: 1.896

Review 3.  Cutaneous mosaicism: Special considerations for women.

Authors:  Katharine T Ellis; Diana Ovejero; Keith A Choate
Journal:  Int J Womens Dermatol       Date:  2021-10-27

Review 4.  Coexistence of craniopharyngioma and cranial fibrous dysplasia: a case series of clinicopathological study.

Authors:  Yang-Hua Fan; Zhi Li
Journal:  Orphanet J Rare Dis       Date:  2022-03-18       Impact factor: 4.123

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6.  McCune-Albright Syndrome in Infant with Growth Hormone Excess.

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Journal:  Genes (Basel)       Date:  2022-07-27       Impact factor: 4.141

7.  Otalgia revealing McCune-Albright syndrome: A case report.

Authors:  Achraf Amine Sbai; FatimZahra Es-Salhi; Adil Abdenbi Tsen
Journal:  Ann Med Surg (Lond)       Date:  2022-09-20

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  8 in total

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