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. 2017 Dec;12(1):21.
doi: 10.1007/s11657-016-0271-6. Epub 2017 Feb 27.

Improving patient outcomes in fibrous dysplasia/McCune-Albright syndrome: an international multidisciplinary workshop to inform an international partnership

A M Boyce  1   2   3 , A Turner  4 , L Watts  4 , L Forestier-Zhang  4 , A Underhill  5 , R Pinedo-Villanueva  4 , F Monsell  6 , D Tessaris  7 , C Burren  6 , L Masi  8 , N Hamdy  9 , M L Brandi  8 , R Chapurlat  10 , M T Collins  1 , Muhammad Kassim Javaid  11
Affiliations

Improving patient outcomes in fibrous dysplasia/McCune-Albright syndrome: an international multidisciplinary workshop to inform an international partnership

A M Boyce et al. Arch Osteoporos. 2017 Dec.

Abstract

To develop consensus on improving the management of patients, we convened an international workshop involving patients, clinicians, and researchers. Key findings included the diagnostic delay and variability in subsequent management with agreement to develop an international natural history study. We now invite other stakeholders to join the partnership.

Purpose: The aim of this study was develop a consensus on how to improve the management of patients with fibrous dysplasia and prioritize areas for research METHODS: An international workshop was held over 3 days involving patients, clinicians, and researchers. Each day had a combination of formal presentations and facilitated discussions that focused on clinical pathways and research.

Results: The patient workshop day highlighted the variability of patients' experience in getting a diagnosis, the knowledge of general clinical staff, and understanding long-term outcomes. The research workshop prioritized collaborations that improved understanding of the contemporary natural history of fibrous dysplasia/McCune-Albright syndrome (FD/MAS). The clinical workshop outlined the key issues around diagnostics, assessment of severity, treatment and monitoring of patients.

Conclusions: In spite of advances in understanding the genetic and molecular underpinnings of fibrous dysplasia/McCune-Albright syndrome, clinical management remains a challenge. From the workshop, a consensus was reached to create an international, multi-stakeholder partnership to advance research and clinical care in FD/MAS. We invite other stakeholders to join the partnership.

Keywords: Epidemiology; Fibrous dysplasia; McCune-Albright syndrome; Patient-centered care.

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References

    1. Albright F, Butler MD, Hampton MD, Smith P. Syndrome characterized by osteitis fibrosa disseminata, areas of pigmentation and endocrine dysfunction, with precocious puberty in females— report of five cases. N Engl J Med. 1937;216:727–746.
    1. Weinstein LS, Shenker A, Gejman PV, Merino MJ, Friedman E, Spiegel AM. Activating mutations of the stimulatory G protein in the McCune-Albright syndrome. N Engl J Med. 1991;325(24):1688–1695. - PubMed
    1. Kelly MH, Brillante B, Kushner H, Gehron Robey P, Collins MT. Physical function is impaired but quality of life preserved in patients with fibrous dysplasia of bone. Bone. 2005;37(3):388–394. - PubMed
    1. Kelly MH, Brillante B, Collins MT. Pain in fibrous dysplasia of bone: age-related changes and the anatomical distribution of skeletal lesions. Osteoporos Int: J Establish Result Coop Eur Found Osteoporos Ntnl Osteoporos Found USA. 2008;19(1):57–63. - PubMed
    1. Bousson V, Rey-Jouvin C, Laredo JD, Le Merrer M, Martin-Duverneuil N, Feydy A, et al. Fibrous dysplasia and McCune-Albright syndrome: imaging for positive and differential diagnoses, prognosis, and follow-up guidelines. Eur J Radiol. 2014;83(10):1828–1842. - PubMed

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