ArticleClinical endocrinology2018
Growth hormone-Insulin-like growth factor 1 axis hyperactivity on bone fibrous dysplasia in McCune-Albright Syndrome.
Article in Clinical endocrinology, 2018. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 12 papers.
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Who cites it
12 citing papers in PubMed, 28 citations in OpenAlex.
- Multidisciplinary approach and clinical management of McCune-Albright syndrome.Frontiers in endocrinology · 2026Review
- 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
- Wnt pathway inhibition with the porcupine inhibitor LGK974 decreases trabecular bone but not fibrosis in a murine model with fibrotic bone.JBMR plus · 2024Article
- Craniofacial Fibrous Dysplasia: Clinical and Therapeutic Implications.Current osteoporosis reports · 2023Review
- Current concepts of craniofacial fibrous dysplasia: pathophysiology and treatment.Archives of craniofacial surgery · 2023Article
- 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
- Auxological and Endocrinological Features in Children With McCune Albright Syndrome: A Review.Frontiers in endocrinology · 2020Review
- Improved Molecular Diagnosis of McCune-Albright Syndrome and Bone Fibrous Dysplasia by Digital PCR.Frontiers in genetics · 2019Article
- The Clinical Spectrum of McCune-Albright Syndrome and Its Management.Hormone research in paediatrics · 2019Review
- Short stature and growth hormone deficiency: unexpected manifestations of McCune-Albright syndrome.BMJ case reports · 2018Article
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Authors and funding
7 authors at 3 institutions in 3 countries.
Funding
Abstract
contextIn fibrous dysplasia (BFD), normal bone and bone marrow are replaced by fibro-osseous tissue, leading to fracture, deformity and pain. BFD may be isolated, or in association with cutaneous hyperpigmentation and/or hyperfunctioning endocrinopathies, termed McCune-Albright syndrome (MAS). GH hypersecretion has been described in 10%-20% of MAS-BFD patients. Aim of the study was to determine the impact of GH-insulin like growth factor 1 (IGF1) axis hyperactivity on MAS-BFD morbidities and the efficacy of GH excess therapy. DESIGN AND PATIENTS: A multicentric cross-sectional analysis was conducted on three different MAS cohorts. From 195 MAS patients, 37 subjects (19%) with GH excess were identified and compared with 34 MAS controls without GH hypersecretion.
resultsMean head circumference SDS was significantly higher in GH excess: 4.025 SDS vs 0.683 SDS (P < .0001). The risk of optic neuropathy (Odds ratio 4.231; P = .039), hearing deficit (Odds ratio 2.961; P = .0481), facial asymmetry (Odds ratio 6.563; P = .0192), malignancies (Odds ratio 15.24; P = .0173) were higher in GH excess group. Overall, pharmacotherapy (octreotide alone 10-30 mg/mo or with pegvisomant 10-20 mg/d) was effective in IGF1 normalization (IGF1 Z-score between -2 and +2 SDS) in 21/29 patients (72.4%) with good compliance to the regimen. Late diagnosis and GH excess treatment after 16 years old of age was associated with an increased risk of optic neuropathy (Odds ratio 4.500; P = .0491) and growth of pituitary adenomas (Odds ratio 7.846; P = .050).
conclusionsGH-IGF1 hyperactivity increases risk of morbidities in MAS. Medical therapy is effective in normalizing IGF1 in most patients, and early treatment during paediatric age is associated with a decreased risk of optic neuropathy and GH-secreting adenomas growth.
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