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. 2024 Nov;115(5):533-541.
doi: 10.1007/s00223-024-01279-0. Epub 2024 Sep 30.

Causes of Musculoskeletal Pain in Paget's Disease of Bone

Kathryn Berg  1 , Dervil Dockrell  1 , Lesley Colvin  2 , William D Fraser  3   4 , Jonathan Cy Tang  3   4 , Terry Aspray  5 , Elaine Dennison  6 , Hrushikesh Divyateja  7 , Nazim Ghouri  8   9 , Esther Hanison  10 , Richard Keen  10 , Eugene McCloskey  11 , Terence W O'Neill  12 , Faizanur Rahman  13 , Mashood Siddiqi  14 , Stephen Tuck  15 , Jane Turton  16 , Stuart H Ralston  17
Affiliations

Causes of Musculoskeletal Pain in Paget's Disease of Bone

Kathryn Berg et al. Calcif Tissue Int. 2024 Nov.

Abstract

Paget's disease of bone (PDB) is characterised by increased and disorganised bone remodelling leading to various complications, such as bone deformity, deafness, secondary osteoarthritis, and pathological fracture. Pain is the most common presenting symptom of PDB, but it is unclear to what extent this is due to increased metabolic activity of the disease, complications, or unrelated causes. We conducted a cross-sectional study of 168 people with PDB attending secondary care referral centres in the UK. We documented the presence of musculoskeletal pain and sought to determine its underlying causes. Musculoskeletal pain was reported by 122/168 (72.6%) individuals. The most common cause was osteoarthritis of joints distant from an affected PDB site in 54 (44.3%), followed by metabolically active PDB in 18 (14.7%); bone deformity in 14 (11.4%); osteoarthritis of a joint neighbouring an affected site in 11 (9.0%), neuropathic pain in 10 (8.2%), and various other causes in the remainder. Pain was more common in women (p<0.019) and in older individuals (p<0.001). Circulating concentrations of macrophage colony-stimulating factor (M-CSF) were significantly higher in those with pain (p = 0.008), but there was no difference between groups of patients with and without pain in concentrations of interleukin-6 (IL-6) or biochemical markers of bone turnover. Pain is a common symptom in PDB but is most often due to osteoarthritis at an unaffected site. The study illustrates the importance of fully evaluating people with PDB to determine the underlying cause of pain so that management can be tailored appropriately.

Keywords: Macrophage colony stimulating factor; Osteoarthritis; Paget's Disease of bone; Pain.

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Conflict of interest statement

William D Fraser reports funding to his institution from Novartis, Takeda, NPS Pharma, Sanofi, Amolyt and Entera-Bio Pharma outside the submitted work and donation of assay materials and evaluation kits from Abbot Diagnostics outside the submitted work; Jonathan C Y Tang reports participating in advisory boards for NPS Pharma, Amolyt and Entera-Biopharma; having received assay materials from Abbott Diagnostics; having received funding to his institution from Novartis, Takeda, NPS Pharma and Sanofi; and honoraria from Novartis, Sanofi and Ascendis outside the submitted work. Eugene McCloskey reports receiving honoraria from Amgen, Thornton & Ross and Flynn Pharma outside the submitted work. Stuart H Ralston reports funding to his institution from Kyowa Kirin, UCB, and the Royal Osteoporosis Society outside the submitted work. The remaining authors have no conflicts of interest to report.

Figures

Fig. 1
Fig. 1
Causes of pain in the study population. The percentages of individuals with different causes of pain are shown. The percentages add up to more than 100% as several individuals had more than one cause of pain. See text for a breakdown of the individual causes of pain in the “other causes” group
Fig. 2
Fig. 2
Response of pain to previous bisphosphonate therapy. The values are the proportion of patients who reported that, following bisphosphonate treatment, their pain disappeared, improved a lot, improved a little, did not change, or worsened. The number of individuals in each group and shown on the x-axis
See this image and copyright information in PMC

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