Determinants of Quality of Life in Adult Patients with Chronic Non-Bacterial Osteomyelitis (CNO) of the Sternocostoclavicular Region (SCCH): A Dutch Single Center Study

Abstract

Sternocostoclavicular hyperostosis (SCCH), the main clinical manifestation of chronic non-bacterial osteomyelitis (CNO) in adults, is associated with various degrees of chronic pain and restricted shoulder girdle function. We evaluated the impact of CNO/SCCH on quality of life (QoL) and its determinants in 136 adult patients with this rare auto-inflammatory bone disorder using the Short Form 36, Brief Pain Inventory, Brief Illness Perception, Utrecht Coping List, and Shoulder Rating questionnaires. Data were compared with those of the general Dutch population, patients with chronic pain, fibrous dysplasia, or osteoarthritis. Eighty-six (64%) predominantly female (85%) patients with completed questionnaires were included in the study. Sixty-four (75%) had isolated CNO/SCCH. Mean delay in diagnosis was 3.0 ± 5.5 (SD) years, 90% had variable pain, and 84% limited shoulder function. Compared to healthy and chronically diseased reference populations, CNO/SCCH patients demonstrated significant impairments in almost all aspects of QoL, maladaptive illness perceptions, and ineffective coping strategies. For patients with >5-year delay in diagnosis, higher pain scores and limited shoulder function were identified as determinants for impaired QoL. Patients with CNO/SCCH reported significant impairments in QoL associated with clinical and psychological determinants. Clinical measures such as shortening delay in diagnosis, effective pain management, and psychosocial interventions targeting these factors should help minimize the negative impact of CNO/SCCH on QoL.

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

“…These findings suggest that disease burden in CNO-largely dictated by pain-involves more than just nociceptive pain signals resulting from inflammation. Therefore, multiangled rather than just anti-inflammatory treatments may offer opportunity to reduce the high burden of disease [6]. This notion is gaining recognition in other musculoskeletal diseases too [10,17,[31][32][33][34], with growing understanding that nociceptive pain can intermingle with both neuropathic pain and nociplastic pain and integrated management can significantly improve patient outcomes [35].…”

“…The disease spectrum is centrally characterized by relapsing–remitting sterile bone inflammation, causing bone pain, stiffness, compromised joint mobility and bone deformation [ 3 , 4 ]. Despite ongoing efforts for timely diagnosis, patients with CNO still face an average diagnostic delay of 5 years during which they may be exposed to chronic pain [ 5 , 6 ]. After diagnosis, chronic pain remains the main contributor to overall disease burden, reflected in frequent work absence and decreased quality of life [ 6 – 8 ].…”

“…Despite ongoing efforts for timely diagnosis, patients with CNO still face an average diagnostic delay of 5 years during which they may be exposed to chronic pain [ 5 , 6 ]. After diagnosis, chronic pain remains the main contributor to overall disease burden, reflected in frequent work absence and decreased quality of life [ 6 – 8 ]. Clinical management of CNO therefore focuses on alleviating pain through targeting the osteitis with various anti-inflammatory and anti-resorptive treatments, all of which are off-label due to lack of supportive evidence [ 1 , 2 , 9 ].…”

Defining the imaging diagnostic criteria for adult chronic non-bacterial osteitis

“…Several patients with this disorder remain thus to date underdiagnosed and untreated, particularly in the early stages of the disease, with prolonged delays in diagnosis often associated with the development of chronic changes, persistent pain and limitation of function, significantly impacting on several aspects of quality of life. 16 This further stresses the need for a clear set of diagnostic imaging criteria for the disease.…”

Garrè Sclerosing Osteomyelitis of the Clavicle: Clinical Results after Clavicular Resection

“…These physical impairments are often associated with psychological sequelae, which may significantly impact various aspects of a patient's quality of life [7]. The main identified determinants of impaired QoL in adult CNO/SCCH were delay in diagnosis, chronic pain of moderate to high severity, and restricted shoulder girdle function [8].…”