Chronic Nonbacterial Osteomyelitis and Inflammatory Bowel Disease: A Literature Review-Based Cohort

Background: Chronic nonbacterial osteomyelitis (CNO) is a rare autoinflammatory bone disorder that mainly involves children and adolescents. The association with other inflammatory disorders, such as inflammatory bowel disease (IBD), psoriasis, and arthritis, has been reported in the literature. In...

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Main Authors: Stefania Costi, Sabino Germinario, Marco Pandolfi, Maria Rosa Pellico, Andrea Amati, Maurizio Gattinara, Cecilia Beatrice Chighizola, Roberto Caporali, Achille Marino
Format: Article
Language:English
Published: MDPI AG 2023-03-01
Series:Children
Subjects:
Online Access:https://www.mdpi.com/2227-9067/10/3/502
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author Stefania Costi
Sabino Germinario
Marco Pandolfi
Maria Rosa Pellico
Andrea Amati
Maurizio Gattinara
Cecilia Beatrice Chighizola
Roberto Caporali
Achille Marino
author_facet Stefania Costi
Sabino Germinario
Marco Pandolfi
Maria Rosa Pellico
Andrea Amati
Maurizio Gattinara
Cecilia Beatrice Chighizola
Roberto Caporali
Achille Marino
author_sort Stefania Costi
collection DOAJ
description Background: Chronic nonbacterial osteomyelitis (CNO) is a rare autoinflammatory bone disorder that mainly involves children and adolescents. The association with other inflammatory disorders, such as inflammatory bowel disease (IBD), psoriasis, and arthritis, has been reported in the literature. In particular, the relationship between bone and intestinal inflammation is still poorly understood. For this purpose, our review aims to describe the cases reported in the literature concerning this association and to compare them with data from our single-center cohort of patients. Methods: We conducted a literature review of published cases of CNO associated with IBD. Eligible articles were identified through a Medline search in the PubMed database until December 2022. We retrospectively reviewed medical records of patients with CNO referred to G. Pini Hospital and compared them with the literature-review-based cohort. Results: Fifty-seven patients with a defined diagnosis of CNO and associated IBD were described in the literature (female 55%). The median age of onset of the disease (CNO or IBD) was 11 years. In 32/53 (60%), a diagnosis of Crohn’s disease (CD) was made, while 18 (34%) patients were classified as suffering from ulcerative colitis (UC) and 3 (6%) from undifferentiated IBD. The diagnosis of CNO preceded the diagnosis of IBD in 59% of cases; while in 24%, IBD anticipated CNO; and in 17%, the two conditions appeared simultaneously. The median time between the two events was 24 months. In our Italian cohort (<i>n</i> = 23 patients), no diagnosis of IBD was made. No significant differences were found when comparing clinical and demographical characteristics of the Italian vs. review-based cohort, except for a significant involvement of rachis in the Italian group. Conclusions: The correlation between autoinflammatory bone disease and intestinal inflammation should be further investigated. It is essential to promote awareness among pediatric rheumatologists and gastroenterologists about this possible association to facilitate the diagnosis and better optimize treatment.
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spelling doaj.art-2020bde0b0c54c6f9bcc28412827fee12023-11-17T10:21:10ZengMDPI AGChildren2227-90672023-03-0110350210.3390/children10030502Chronic Nonbacterial Osteomyelitis and Inflammatory Bowel Disease: A Literature Review-Based CohortStefania Costi0Sabino Germinario1Marco Pandolfi2Maria Rosa Pellico3Andrea Amati4Maurizio Gattinara5Cecilia Beatrice Chighizola6Roberto Caporali7Achille Marino8Unit of Pediatric Rheumatology, ASST G. Pini-CTO, 20122 Milan, ItalyUniversity of Milan, 20122 Milan, ItalyUniversity of Milan, 20122 Milan, ItalyUniversity of Milan, 20122 Milan, ItalyUniversity of Milan, 20122 Milan, ItalyUnit of Pediatric Rheumatology, ASST G. Pini-CTO, 20122 Milan, ItalyUnit of Pediatric Rheumatology, ASST G. Pini-CTO, 20122 Milan, ItalyUnit of Pediatric Rheumatology, ASST G. Pini-CTO, 20122 Milan, ItalyUnit of Pediatric Rheumatology, ASST G. Pini-CTO, 20122 Milan, ItalyBackground: Chronic nonbacterial osteomyelitis (CNO) is a rare autoinflammatory bone disorder that mainly involves children and adolescents. The association with other inflammatory disorders, such as inflammatory bowel disease (IBD), psoriasis, and arthritis, has been reported in the literature. In particular, the relationship between bone and intestinal inflammation is still poorly understood. For this purpose, our review aims to describe the cases reported in the literature concerning this association and to compare them with data from our single-center cohort of patients. Methods: We conducted a literature review of published cases of CNO associated with IBD. Eligible articles were identified through a Medline search in the PubMed database until December 2022. We retrospectively reviewed medical records of patients with CNO referred to G. Pini Hospital and compared them with the literature-review-based cohort. Results: Fifty-seven patients with a defined diagnosis of CNO and associated IBD were described in the literature (female 55%). The median age of onset of the disease (CNO or IBD) was 11 years. In 32/53 (60%), a diagnosis of Crohn’s disease (CD) was made, while 18 (34%) patients were classified as suffering from ulcerative colitis (UC) and 3 (6%) from undifferentiated IBD. The diagnosis of CNO preceded the diagnosis of IBD in 59% of cases; while in 24%, IBD anticipated CNO; and in 17%, the two conditions appeared simultaneously. The median time between the two events was 24 months. In our Italian cohort (<i>n</i> = 23 patients), no diagnosis of IBD was made. No significant differences were found when comparing clinical and demographical characteristics of the Italian vs. review-based cohort, except for a significant involvement of rachis in the Italian group. Conclusions: The correlation between autoinflammatory bone disease and intestinal inflammation should be further investigated. It is essential to promote awareness among pediatric rheumatologists and gastroenterologists about this possible association to facilitate the diagnosis and better optimize treatment.https://www.mdpi.com/2227-9067/10/3/502chronic nonbacterial osteomyelitisinflammatory bowel diseasesbowel inflammationbone inflammation
spellingShingle Stefania Costi
Sabino Germinario
Marco Pandolfi
Maria Rosa Pellico
Andrea Amati
Maurizio Gattinara
Cecilia Beatrice Chighizola
Roberto Caporali
Achille Marino
Chronic Nonbacterial Osteomyelitis and Inflammatory Bowel Disease: A Literature Review-Based Cohort
Children
chronic nonbacterial osteomyelitis
inflammatory bowel diseases
bowel inflammation
bone inflammation
title Chronic Nonbacterial Osteomyelitis and Inflammatory Bowel Disease: A Literature Review-Based Cohort
title_full Chronic Nonbacterial Osteomyelitis and Inflammatory Bowel Disease: A Literature Review-Based Cohort
title_fullStr Chronic Nonbacterial Osteomyelitis and Inflammatory Bowel Disease: A Literature Review-Based Cohort
title_full_unstemmed Chronic Nonbacterial Osteomyelitis and Inflammatory Bowel Disease: A Literature Review-Based Cohort
title_short Chronic Nonbacterial Osteomyelitis and Inflammatory Bowel Disease: A Literature Review-Based Cohort
title_sort chronic nonbacterial osteomyelitis and inflammatory bowel disease a literature review based cohort
topic chronic nonbacterial osteomyelitis
inflammatory bowel diseases
bowel inflammation
bone inflammation
url https://www.mdpi.com/2227-9067/10/3/502
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