RANK-L inhibitor as a promising agent for refractory extensive craniofacial fibrous dysplasia: A case report.

Autor: Abouammo MD; Department of Otolaryngology - Head and Neck Surgery, Tanta University, Tanta, Egypt.; Department of Otolaryngology - Head and Neck Surgery, The Ohio State University Wexner Medical Center, Columbus, Ohio, USA., Alsavaf MB; Department of Otolaryngology - Head and Neck Surgery, The Ohio State University Wexner Medical Center, Columbus, Ohio, USA.; Department of Neurological Surgery, The Ohio State University Wexner Medical Center, Columbus, Ohio, USA., Prevedello DM; Department of Otolaryngology - Head and Neck Surgery, The Ohio State University Wexner Medical Center, Columbus, Ohio, USA.; Department of Neurological Surgery, The Ohio State University Wexner Medical Center, Columbus, Ohio, USA., Ghalib L; Department of Endocrinology, Diabetes, and Metabolism, The Ohio State University Wexner Medical Center, Columbus, Ohio, USA., Boyce AM; Metabolic Bone Disorders Unit, National Institute of Dental and Craniofacial Research, National Institutes of Health, Bethesda, Maryland, USA., Carrau RL; Department of Otolaryngology - Head and Neck Surgery, The Ohio State University Wexner Medical Center, Columbus, Ohio, USA.; Department of Neurological Surgery, The Ohio State University Wexner Medical Center, Columbus, Ohio, USA.
Jazyk: angličtina
Zdroj: Head & neck [Head Neck] 2024 Jan; Vol. 46 (1), pp. E1-E5. Date of Electronic Publication: 2023 Oct 12.
DOI: 10.1002/hed.27546
Abstrakt: Background: McCune-Albright syndrome is a rare disorder characterized by polyostotic fibrous dysplasia (FD), café-au-lait skin pigmentation, and endocrine dysfunction. Extensive FD in the craniofacial region can present significant challenges in terms of disease control and carries a high risk of permanent visual impairment.
Methods: We present a case of medically and surgically resistant FD that required nine optic nerve decompressions.
Results: The condition was ultimately controlled with the use of the denosumab agent.
Conclusion: The case highlights the importance and potential efficacy of denosumab in resistant FD management, particularly in cases involving sensitive organs.
(© 2023 The Authors. Head & Neck published by Wiley Periodicals LLC.)
Databáze: MEDLINE
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