Intensive, Interdisciplinary Pain Treatment in Fibrous Dysplasia/McCune-Albright Syndrome
Abstract Pain in fibrous dysplasia/McCune-Albright syndrome (FD/MAS) remains inadequately treated. The pilot study was aimed to determine the feasibility (ie, completion, retention, and acceptability) and explore the effectiveness of an intensive interdisciplinary pain treatment (IIPT) using a biopsychosocial approach in patients with FD/MAS. Five patients (4 Females, 1 Male; 22-30 yr old) with FD/MAS, with baseline pain severity of 4+ (0-10 scale) were enrolled. A three-week intervention (4 d/wk; 3-4 h/d) integrating psychological, physical, and occupational therapies was administered to patients. Study measures included patient-reported outcomes at treatment admission (week 0), discharge (week 3), and follow-up (week 15). Physician global assessments (PGA) at weeks 0 and 3 were also collected. All five patients completed the 3-wk intervention and follow-up assessment and reported a high degree of acceptability, appropriateness, and feasibility of the intervention. Average pain severity decreased after treatment, with mean changes of −3.0 and − 3.2 points at weeks 3 and 15, respectively. Patients demonstrated improvements in pain interference, external pain expression (measured with the pain behavior scale), and pain catastrophizing. Trends of decreased anxiety and depression were observed. Overall health-related quality of life improved at treatment discharge, which was consistent with the PGA (ie, +2 to +3) scores. This pilot study demonstrates that IIPT was feasible and showed initial clinical benefit across multiple dimensions in a small cohort of patients with FD/MAS. Larger-scale, controlled trials are needed for further validation.
Authors
- Ingrid A. Holm (ORCID: https://orcid.org/0000-0003-4712-8821)
- Navil F. Sethna (ORCID: https://orcid.org/0000-0003-2200-9826)
- Julie Shulman (ORCID: https://orcid.org/0000-0002-5530-9692)
- Jaymin Upadhyay (ORCID: https://orcid.org/0000-0002-8682-163X)
- Catherine E. Stewart (ORCID: https://orcid.org/0000-0003-1216-0450)
- Boyu Ren (ORCID: https://orcid.org/0000-0002-5300-1184)
- Amanda Q. Cao (ORCID: https://orcid.org/0000-0003-0963-634X)
- Ella Trumper
- Jennifer Christofferson (ORCID: https://orcid.org/0000-0003-3892-9424)
- Zachary S. Peacock (ORCID: https://orcid.org/0000-0001-7683-3111)
- Michael Mannstadt (ORCID: https://orcid.org/0000-0003-0867-9778)
- Camryn Berry
- S R O'Donnell
- Kevin Zirko
- Edin Randall (ORCID: https://orcid.org/0000-0001-5402-7215)
- Eleanor Powell (ORCID: https://orcid.org/0009-0001-8598-0700)
- Ana Gallotto (ORCID: https://orcid.org/0009-0008-5162-4899)
Institutions
- Boston Children's Hospital (US)
- McLean Hospital (US)
- Massachusetts General Hospital (US)
- Center for Pain and the Brain (US)
Publication Details
- Journal
- JBMR Plus
- Published
- 2026-06-29
- DOI
- https://doi.org/10.1093/jbmrpl/ziag107
- Primary Topic
- Bone Tumor Diagnosis and Treatments
- Type
- article
- Field-Weighted Citation Impact
- 0.00