ArticleThe Journal of clinical endocrinology and metabolism2024
Phenotyping Pain in Patients With Fibrous Dysplasia/McCune-Albright Syndrome.
Article in The Journal of clinical endocrinology and metabolism, 2024. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 10 papers.
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Who cites it
10 citing papers in PubMed, 12 citations in OpenAlex.
- Nociceptive responses and resting-state functional connectivity of the human midbrain tegmentum.Brain structure & function · 2026Article
- Individualized antiresorptive therapy in fibrous dysplasia and McCune-Albright syndrome: A retrospective cohort study.Bone reports · 2026Article
- Intensive, interdisciplinary pain treatment in fibrous dysplasia/McCune-Albright syndrome.JBMR plus · 2026Article
- Addressing the unmet challenge of pain in rare bone diseases: new insights from the RUDY UK registry.Orphanet journal of rare diseases · 2026Article
- Article
- Fibrous dysplasia/McCune-Albright syndrome: state-of-the-art advances, pathogenesis, and basic/translational research.Orphanet journal of rare diseases · 2025Review
- Bone pain in fibrous dysplasia does not rely on aberrant sensory nerve sprouting or neuroma formation.Journal of bone and mineral research : the official journal of the American Society for Bone and Mineral Research · 2025Article
- Pain in fibrous dysplasia: identifying nociceptive mechanisms in a preclinical model.Journal of bone and mineral research : the official journal of the American Society for Bone and Mineral Research · 2025Article
- A systematic literature review of the impact and measurement of mobility impairment in rare bone diseases.Therapeutic advances in musculoskeletal disease · 2025Review
- Pediatric Fibrous Dysplasia of the Skull Base: Update on Management and Treatment.Brain sciences · 2024Review
Corrections and comments
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Authors and funding
13 authors at 5 institutions in 1 country.
Funding
Abstract
contextPain is a poorly managed aspect in fibrous dysplasia/McCune-Albright syndrome (FD/MAS) because of uncertainties regarding the clinical, behavioral, and neurobiological underpinnings that contribute to pain in these patients.
objectiveIdentify neuropsychological and neurobiological factors associated with pain severity in FD/MAS.
designProspective, single-site study. PATIENTS: Twenty patients with FD/MAS and 16 age-sex matched healthy controls.
interventionAssessments of pain severity, neuropathic pain, pain catastrophizing (pain rumination, magnification, and helplessness), emotional health, and pain sensitivity with thermal quantitative sensory testing. Central nervous system (CNS) properties were measured with diffusion tensor imaging, structural magnetic resonance imaging, and functional magnetic resonance imaging.
main outcome measuresQuestionnaire responses, detection thresholds and tolerances to thermal stimuli, and structural and functional CNS properties.
resultsPain severity in patients with FD/MAS was associated with more neuropathic pain quality, higher levels of pain catastrophizing, and depression. Quantitative sensory testing revealed normal detection of nonnoxious stimuli in patients. Individuals with FD/MAS had higher pain tolerances relative to healthy controls. From neuroimaging studies, greater pain severity, neuropathic pain quality, and psychological status of the patient were associated with reduced structural integrity of white matter pathways (superior thalamic radiation and uncinate fasciculus), reduced gray matter thickness (pre-/paracentral gyri), and heightened responses to pain (precentral, temporal, and frontal gyri). Thus, properties of CNS circuits involved in processing sensorimotor and emotional aspects of pain were altered in FD/MAS.
conclusionThese results offer insights into pain mechanisms in FD/MAS, while providing a basis for implementation of comprehensive pain management treatment approaches that addresses neuropsychological aspects of pain.
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