Chronic Recurrent Multifocal Osteomyelitis - A Case Series from India

Rao, Anand Prahalad; Mallya, Pooja Prakash; Ranjani, Shashi; Raghuram, Jyothi

doi:10.4103/ortho.ijortho_464_17

Cited by 17 publications

(20 citation statements)

References 22 publications

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“…The diagnostic delay ranged from 1 to 72 months, suggesting that CNO is still sometimes not well recognized in our country. In term of clinical manifestation, bone pain is still the most frequency symptom, but the fever rate in our study, which up to 44.4%, is more higher than that in other studies [ 17 , 18 , 20 , 22 , 24 ]. This may be related to bone inflammation.…”

Section: Discussioncontrasting

confidence: 81%

“…Compared with previous reports, the clinical characteristics of our patients were a little different. (details in Table 4 ) [ 8 , 12 , 15 , 17 – 25 ] The female advantage in China is not such obvious as European countries [ 8 , 12 , 15 , 18 , 22 – 25 ]. The median age at diagnosis and diagnostic delays were 9.2 years and 10.2 months, respectively.…”

Section: Discussionmentioning

confidence: 99%

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Clinical characteristics and outcomes of chronic nonbacterial osteomyelitis in children: a multicenter case series

Liu

Tang

et al. 2022

Pediatr Rheumatol

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Objective The aim of this study was to evaluate demographic, clinical, laboratory, imaging, histopathology characteristics, and treatment responses of children with Chronic nonbacterial osteomyelitis (CNO). Methods Retrospective multi-center case series study of pediatric patients diagnosed with CNO treated at five tertiary centers in south China. Results Totally there were 18 patients diagnosed as CNO between 2014 and 2020. The median age of onset was 9.2 years (range 3.7–13.1) and 55.6% were female. Median delay in diagnosis was 10.9 months (range 1.0–72.0). The most frequent presenting symptoms were bone pain (100%) and fever (44.4%). Most patients had more than one lesion (median of 5, range 1–7). Most frequently affected bones were tibiofibula (88.9%) and femur (77.8%). The MRI characteristics mainly presented as bone edema and hyperintensity in bone marrow. Bone biopsy was conducted in 11 patients (61.1%) with inflammatory cells infiltration manifested as chronic osteomyelitis, and none showed bacterial infection or tumor. In treatment, non-steroid anti-inflamatory drugs (NSAIDs) is used as the first-line drug followed by steriods, methotexate (MTX), salazosulfadimidine (SASP), Bisphosphonates and TNF-α inhibitor. Two refractory cases received combination therapy with Bisphosphonates and TNF-α inhibitor, and achieved good therapeutic effect. Conclusions The present study described a multicenter series of CNO from south China and highlighted the clinical features, laboratory tests, imaging characteristics and treatment outcomes. Increasing awareness of this disease is important to decrease time to diagnosis, improve access to treatment, and reduce complications.

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Section: Discussioncontrasting

confidence: 81%

Section: Discussionmentioning

confidence: 99%

Clinical characteristics and outcomes of chronic nonbacterial osteomyelitis in children: a multicenter case series

Liu

Tang

et al. 2022

Pediatr Rheumatol

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“…The peak onset of CNO in studies from Turkey was between 9 and 10.2 years, and a male predominance was observed in 2 studies [12][13][14]. Likewise, two studies from India and Chile reported male predominance [15,16].…”

Section: Discussionmentioning

confidence: 99%

“…Açarı et al [14] reported lesion sites; femur (67.9%), tibia (57.1%), and pelvic bones (32.1%). Concha et al [16] observed lesions of 21% in the upper limb, 9% in the lower limb, and 36% in the axial skeleton, and the other studies reported sites of lesions under different titles and groups [11][12][13][14][15][16].…”

Section: Discussionmentioning

confidence: 99%

Chronic non-bacterial osteomyelitis in children: Presentation, diagnosis, outcomes, quality of life

Tekin

Gülleroğlu

Çelikel

et al. 2022

Preprint

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PurposeChronic non-bacterial osteomyelitis is a chronic sterile inflammatory condition of the bone. We aimed to describe the clinical and radiographic findings of patients and to evaluate their response to therapy and quality of life. MethodsThis cross-sectional study included 18 patients whose clinical, radiological features and outcomes were reviewed retrospectively. The quality of patients’ life after treatment was compared to healthy controls by using the Pediatric Quality of Life Inventory 4.0. ResultsThe median age of disease onset was 12 (IQR 10-14) years and 11 (61.1%) patients were male. The median follow-up duration was 15 (IQR 12-22) months. The persistent form of chronic non-bacterial osteomyelitis was the most common pattern in 15 (83.3%) patients and a recurrent pattern was defined in 3 (16.7%) patients. The lesions were multifocal in all patients and 15 (83.3%) patients had symmetric distribution in whole-body magnetic resonance imaging. The most common sites of arthritis were the knee and sacroiliac joints. Methotrexate was used in 16 (88.9%) patients as first-line therapy. However, some patients were unresponsive to first-line therapy and needed tumor necrosis factor-α inhibitors (55.6%) and bisphosphonates (16.7%). We observed remission in only 4 (22.2%) patients and 3 (16.7%) patients were unresponsive. The patients had a significantly poorer quality of life than controls (p=0.005).ConclusionsChronic non-bacterial osteomyelitis is an insidious disease that requires detailed analysis for diagnosis and whole-body magnetic resonance imaging is an effective tool for diagnosis. Despite the advanced treatment, patients with chronic non-bacterial osteomyelitis have a poor quality of life.

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“…The median age at diagnosis in pediatric patients is 10 years, with most cases reported between 8 and 13 years [ 4 , 5 , 6 , 7 , 8 ]. In European cohorts, females seem more likely to be affected, with a female-to-male ratio of 2:1 [ 5 , 6 , 7 , 8 , 9 , 10 ], while in Latin American and Indian series a male prevalence is reported [ 11 , 12 ]. Diagnostic delay is common in pediatric CNO, with a mean interval of 12 months between symptom onset and diagnosis in most pediatric studies [ 4 , 6 , 8 , 13 ], possibly related to the rareness of the disease, the discrete findings on examination, and the absence of pathognomonic laboratory studies.…”

Section: Incidence and Demographicsmentioning

confidence: 99%

Chronic Nonbacterial Osteomyelitis in Children

et al. 2021

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Chronic nonbacterial osteomyelitis (CNO) is an auto-inflammatory bone disorder with a wide spectrum of clinical manifestations, from unifocal to multifocal lesions. When it manifests with multifocal lesions, it is also referred to as chronic recurrent multifocal osteomyelitis (CRMO). CNO/CRMO can affect all age groups, with the pediatric population being the most common. Patients may present with systemic inflammation, but there is no pathognomonic laboratory finding. Magnetic resonance imaging (MRI) is the gold standard radiological tool for diagnosis. In the absence of validated diagnostic criteria, CNO/CRMO remains an exclusion diagnosis. Bone biopsy does not show a specific disease pattern, but it may be necessary in unifocal or atypical cases to differentiate it from malignancy or infection. First-line treatments are non-steroidal anti-inflammatory drugs (NSAIDs), while bisphosphonates or TNF-α blockers can be used in refractory cases. The disease course is unpredictable, and uncontrolled lesions can complicate with bone fractures and deformations, underlying the importance of long-term follow-up in these patients.

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Chronic Recurrent Multifocal Osteomyelitis - A Case Series from India

Cited by 17 publications

References 22 publications

Clinical characteristics and outcomes of chronic nonbacterial osteomyelitis in children: a multicenter case series

Clinical characteristics and outcomes of chronic nonbacterial osteomyelitis in children: a multicenter case series

Chronic non-bacterial osteomyelitis in children: Presentation, diagnosis, outcomes, quality of life

Chronic Nonbacterial Osteomyelitis in Children

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