Neuropathic and Nociplastic Pain Profiles are Common in Adult Chronic Nonbacterial Osteitis (CNO)

Anne T Leerling; Marieke Niesters; Marcel Flendrie; Marije Tel; Natasha M Appelman-Dijkstra; Olaf M Dekkers; Elizabeth M Winter

doi:10.1007/s00223-024-01214-3

Neuropathic and Nociplastic Pain Profiles are Common in Adult Chronic Nonbacterial Osteitis (CNO)

Calcif Tissue Int. 2024 Apr 16. doi: 10.1007/s00223-024-01214-3. Online ahead of print.

Authors

Anne T Leerling^{1

2

3}, Marieke Niesters⁴, Marcel Flendrie⁵, Marije Tel^{1

2}, Natasha M Appelman-Dijkstra^{1

2}, Olaf M Dekkers^{1

3}, Elizabeth M Winter^{6

7}

Affiliations

¹ Division of Endocrinology, Department of Internal Medicine, Leiden University Medical Center, Leiden, The Netherlands.
² Center for Bone Quality, Leiden University Medical Center, Albinusdreef 2, 2333, ZA, Leiden, The Netherlands.
³ Department of Clinical Epidemiology, Leiden University Medical Center, Leiden, The Netherlands.
⁴ Department of Anesthesiology and Pain Medicine, Leiden University Medical Center, Leiden, The Netherlands.
⁵ Department of Rheumatology, Sint Maartenskliniek, Nijmegen, The Netherlands.
⁶ Division of Endocrinology, Department of Internal Medicine, Leiden University Medical Center, Leiden, The Netherlands. e.m.winter@lumc.nl.
⁷ Center for Bone Quality, Leiden University Medical Center, Albinusdreef 2, 2333, ZA, Leiden, The Netherlands. e.m.winter@lumc.nl.

PMID: 38627292
DOI: 10.1007/s00223-024-01214-3

Abstract

Chronic nonbacterial osteitis (CNO) is a rare musculoskeletal disease causing chronic bone pain. It is known that chronic musculoskeletal pain may involve other mechanisms than nociceptive pain only. We investigate the prevalence of neuropathic and nociplastic pain in adult CNO and their association with clinical characteristics and treatment outcomes. Survey study among the Dutch adult CNO cohort (n = 84/195 participated), including PAIN-detect for neuropathic pain, and the Central Sensitization Inventory (CSI), Fibromyalgia Rapid Screening Tool (FiRST), and ACTTION-APS Pain Taxonomy (AAPT) for nociplastic pain. Clinical characteristics and CNO-related bone pain scores were compared between patients with exclusive nociceptive pain and those with nociceptive pain plus neuropathic and/or nociplastic pain (mixed pain). 31% (95% CI 21-41) of patients classified as likely having neuropathic pain according to PAIN-detect. 53% (41-64) of patients displayed central sensitization on CSI, 61% (50-72) screened positive for fibromyalgia on FiRST and 14% (7-23) of patients fulfilled the AAPT criteria, all indicative of nociplastic pain. Mixed pain was associated with longer diagnostic delay (mean difference 2.8 years, 95% CI 0.4-5.2, p = 0.023), lower educational level (72% versus 20%, p < 0.001), and opioid use (37% versus 13%, p = 0.036). Despite comparable disease severity and extent, patients with mixed pain reported significantly higher CNO-related bone pain scores. This study demonstrates the high prevalence of mixed pain in adult CNO, in which neuropathic and nociplastic pain exist alongside nociceptive inflammatory bone pain. Disease burden in CNO may extend beyond inflammatory activity, highlighting the need for a multifaceted management approach.

Keywords: Central pain; Central sensitization; Chronic nonbacterial osteomyelitis; Fibromyalgia; Neuropathic pain; Nociceptive pain; Nociplastic pain; Osteitis; Pain; Patient reported outcomes; SAPHO.