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Published on: December 31, 2017
Chronic nonbacterial osteomyelitis and inflammatory bowel disease in children: A French national cohort
Valérie Bertrand1,2, Bénédicte Pigneur3,4, Marie-Louise Frémond5,6
1Département de pédiatrie, Hôpital Jacques Monod, BP 24, Le Havre, 76083, France.
Insights
The rare association between inflammatory bowel disease (IBD) and chronic nonbacterial osteomyelitis (CNO) in children is described. Anti-tumor necrosis factor-alpha therapies showed efficacy in managing this condition, with an estimated incidence of 1:20,000 pediatric IBD patients.
Area of Science:
- Pediatric rheumatology and gastroenterology
- Rare disease associations
- Inflammatory bowel disease (IBD) and chronic nonbacterial osteomyelitis (CNO)
Background:
- The co-occurrence of IBD and CNO is infrequently recognized, potentially leading to underdiagnosis.
- Increased clinical awareness is crucial for identifying this rare pediatric association.
Purpose of the Study:
- To characterize a French national pediatric cohort with IBD-CNO association.
- To estimate the incidence of CNO in pediatric patients with IBD.
Main Methods:
- Retrospective data collection from pediatric gastroenterology and rheumatology units (2000-2024).
- Analysis of demographic, clinical, and treatment data.
- Incidence estimation using the French EPIMAD cohort data.
Main Results:
- Thirty-seven pediatric patients with IBD-CNO were identified, with a median onset age of 13.5 years.
- CNO and IBD diagnoses occurred concurrently or sequentially in most cases; lower limbs and pelvis were common CNO sites.
- Anti-tumor necrosis factor-alpha (anti-TNF-α) therapy was associated with high remission rates (82.1%) in IBD-CNO patients.
Conclusions:
- This study presents a significant national cohort of pediatric IBD-CNO.
- Whole-body MRI is recommended for IBD patients with bone pain; multidisciplinary care is essential.
- Anti-TNF-α therapies appear effective in managing IBD-CNO, potentially preventing bone complications.
Objectives:
Inflammatory bowel disease (IBD) and chronic nonbacterial osteomyelitis (CNO) is a rare association. Its prevalence is probably underestimated due to low awareness among clinicians. The aim of this study was to describe the French pediatric national cohort and to estimate the incidence of CNO among IBD.
Methods:
We retrospectively collected all reports of IBD-CNO association from pediatric gastroenterological and rheumatologic units between 2000 and 2024. We analyzed demographic data; age of the onset; IBD type; CNO localizations; dermatological lesions; explorations results; medications; follow-up. Incidence was estimated using French regional inception population-based cohort EPIMAD data.
Results:
Thirty-seven patients were identified. Median age at the onset was 13.5 years (interquartile range [IQR] 11.5-14.5). CNO diagnosis preceded IBD in 11 cases, followed it in 13 cases, while 13 IBD-CNO cases were diagnosed simultaneously (median interval 1 year). IBD included 25 Crohn's disease, 10 ulcerative colitis, 2 IBD-unclassified. Bone localizations predominated in lower limbs and pelvis (75.3%). Fourteen children (37.8%) had dermatological lesions. At last follow-up (median 3 years, IQR 1.25-4.75), 28 patients (75.7%) were in IBD-CNO remission, having mostly anti-tumor necrosis factor (TNF)-α (p = 0.041, 82.1% remission, 44.4% relapse) in monotherapy (60.7%). We estimate the incidence of this association among French pediatric IBD at 1 case for 20,000 patients-year.
Conclusion:
This study describes a large national cohort of pediatric IBD-CNO association. Gastroenterologist should request a whole body-magnetic resonance imaging when IBD patients present chronic bone pain. Multidisciplinary management involving rheumatologists and dermatologists is recommended. Most patients responded to anti-TNF-α antibodies, which could avoid bone complications.
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