ArticleThe Journal of clinical endocrinology and metabolism2013
Optic neuropathy in McCune-Albright syndrome: effects of early diagnosis and treatment of growth hormone excess.
Article in The Journal of clinical endocrinology and metabolism, 2013. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 34 papers.
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Who cites it
34 citing papers in PubMed, 80 citations in OpenAlex.
- Impact of endoscopy and related technologies on indications for transsphenoidal pituitary surgery for growth hormone-producing pituitary neuroendocrine tumors in McCune-Albright syndrome: illustrative case.Journal of neurosurgery. Case lessons · 2026Article
- Genetics of familial acromegaly and pituitary gigantism.The Journal of clinical endocrinology and metabolism · 2026Review
- Somatotropic axis drives bone turnover heterogeneity independently of gonadotropin synergy in girls with central precocious puberty.Frontiers in endocrinology · 2026Article
- Burosumab treatment for FGF23-related hypophosphatemia in a two-year-old girl with McCune-Albright syndrome.Clinical pediatric endocrinology : case reports and clinical investigations : official journal of the Japanese Society for Pediatric Endocrinology · 2026Article
- Article
- Safe Continuation of Pegvisomant During Pregnancy in a Patient With Fibrous Dysplasia/McCune-Albright Syndrome.JCEM case reports · 2025Article
- Clinical spectrum and uncommon features of McCune-Albright syndrome in children: a cohort study from a National Referral Center.Frontiers in endocrinology · 2025Article
- Lesion Expansion in Gnathic Fibrous Dysplasia: Natural History, Indicators of Progression, and Response to Bisphosphonates.Journal of bone and mineral research : the official journal of the American Society for Bone and Mineral Research · 2023Article
- Current concepts of craniofacial fibrous dysplasia: pathophysiology and treatment.Archives of craniofacial surgery · 2023Article
- Craniofacial Fibrous Dysplasia: Clinical and Therapeutic Implications.Current osteoporosis reports · 2023Review
- Clinical Biology of the Pituitary Adenoma.Endocrine reviews · 2022Review
- Genetic and Epigenetic Pathogenesis of Acromegaly.Cancers · 2022Review
- Rare Diseases of the Nose, the Paranasal Sinuses, and the Anterior Skull Base.Laryngo- rhino- otologie · 2021Review
- Article
- Clinical Characteristics and Management of Patients With McCune-Albright Syndrome With GH Excess and Precocious Puberty: A Case Series and Literature Review.Frontiers in endocrinology · 2021Review
- Fibrous Dysplasia/McCune-Albright Syndrome: A Rare, Mosaic Disease of Gα s Activation.Endocrine reviews · 2020Review
- Best practice management guidelines for fibrous dysplasia/McCune-Albright syndrome: a consensus statement from the FD/MAS international consortium.Orphanet journal of rare diseases · 2019Review
- Fibrous Dysplasia of Bone and McCune-Albright Syndrome: A Bench to Bedside Review.Calcified tissue international · 2019Review
- Fibrous Dysplasia of Bone and McCune-Albright Syndrome: A Bench to Bedside Review.Calcified tissue international · 2019Review
- The Clinical Spectrum of McCune-Albright Syndrome and Its Management.Hormone research in paediatrics · 2019Review
Corrections and comments
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Authors and funding
11 authors at 6 institutions in 1 country.
Funding
Abstract
contextGH excess is a serious complication of McCune-Albright syndrome (MAS) and has been associated with craniofacial morbidity.
objectiveThe aim of the study was to determine whether early diagnosis and treatment of MAS-associated GH excess prevents optic neuropathy and hearing impairment, the major morbidities associated with GH excess. DESIGN AND
settingA retrospective cross-sectional analysis was conducted at a clinical research center. PATIENTS: Twenty-two subjects with MAS-associated GH excess and 21 control MAS subjects without GH excess were included in the study.
interventionBiochemical testing included random GH, nadir GH after glucose load, nadir GH on frequent sampling, and IGF-I Z-score. Subjects underwent imaging, ophthalmological, audiological, and otolaryngological assessment. Treatment included octreotide, pegvisomant, transphenoidal surgery, and/or radiotherapy as indicated.
main outcome measureAssociation of optic neuropathy and hearing impairment to age at GH excess diagnosis/treatment was measured.
resultsOf 129 MAS subjects, 26 (20%) were diagnosed with GH excess based on elevation of two measures of GH function. Of these, 22 subjects were candidates for pharmacological intervention. Optic neuropathy was significantly correlated with intervention status, with no cases in the early intervention group (diagnosed/treated before age 18) or the control group, and four of seven (57%) in the late intervention group (diagnosed/treated after age 18) (Fisher's exact test; odds ratio, 0.027; P = 0.0058). Early diagnosis/intervention was not associated with reduction in hearing deficits (odds ratio, 1.25; P = 1.00). Mean head circumference SD score was significantly higher in the late (6.08; range, 2.70 to 22.56) than the early intervention (2.67; range, -0.65 to 6.72) or control groups (2.13; range, -2.06 to 7.79) (P = 0.003).
conclusionsEarly diagnosis/treatment of GH excess in MAS is important to prevent optic neuropathy and craniofacial expansion. The relationship between hearing deficits and GH excess remains less clear and requires further study.
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