A child with Chronic Nonbacterial Osteomyelitis and celiac disease: accidental association or two different aspects of the same condition?
Abstract Background Chronic Nonbacterial Osteomyelitis (CNO) is a rare auto-inflammatory disease that mainly affects children, and manifests with single or multiple painful bone lesions. Due to the lack of specific laboratory markers, CNO diagnosis is a matter of exclusion from different conditions,...
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2025-01-01
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| Series: | Italian Journal of Pediatrics |
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| Online Access: | https://doi.org/10.1186/s13052-025-01842-x |
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| author | Grazia Bossi Maria Sole Prevedoni Gorone Luca Lungarotti Francesco Pelillo Amelia Mascolo Matteo Naso Daniele Veraldi Francesca Olivero Costanza Chirico Maria Vittoria Marino Cristina Dutto Gian Luigi Marseglia |
| author_facet | Grazia Bossi Maria Sole Prevedoni Gorone Luca Lungarotti Francesco Pelillo Amelia Mascolo Matteo Naso Daniele Veraldi Francesca Olivero Costanza Chirico Maria Vittoria Marino Cristina Dutto Gian Luigi Marseglia |
| author_sort | Grazia Bossi |
| collection | DOAJ |
| description | Abstract Background Chronic Nonbacterial Osteomyelitis (CNO) is a rare auto-inflammatory disease that mainly affects children, and manifests with single or multiple painful bone lesions. Due to the lack of specific laboratory markers, CNO diagnosis is a matter of exclusion from different conditions, first and foremost bacterial osteomyelitis and malignancies. Whole Body Magnetic Resonance (WBMR) and bone biopsy are the gold standard for the diagnosis. Although the association with Inflammatory Bowel Disease (IBD) has been reported in the literature, cases of CNO in celiac patients have never been described before. Case presentation We report about a girl of 3 years and 8 months of age who presented with severe bone pain, slight increase of inflammatory markers, micro-hematuria and high calprotectin values. Her personal medical history was uneventful, apart from low weight growth. She had never complained of abdominal pain or other gastro-intestinal symptoms. WBMR showed the classical features of multifocal CNO, and biopsy confirmed the diagnosis. Celiac disease (CD) was suspected on the basis of antibody screening, and confirmed by gut biopsy. With gluten-free diet the patient achieved rapid and complete symptom remission together with healing of all the bone lesions proven by WBMR. Three years after the onset of the disease the girl is healthy and totally asymptomatic, still on clinical and radiological follow-up. Conclusions Based on our experience, the diagnostic work-up of new cases of CNO should include the screening test for CD and, according to the literature, the possibility of IBD should also be properly ruled out. When CNO and CD coexist, gluten-free diet, combined with antinflammatory therapy, could be able to completely reverse bone lesions, shortening the duration of medical treatment. Because the diseases’ onset is seldom simultaneous, patients with CNO and IBD deserve a properly extended follow-up. Finally, the analysis of the relationship between CNO and autoimmune intestinal diseases provides a unique opportunity to understand the pathophysiological pro-inflammatory network underlying both types of disorders and it is necessary to make the most suitable therapeutic choice. |
| format | Article |
| id | doaj-art-6de0bf79c49b438698b172580d6fc39b |
| institution | Kabale University |
| issn | 1824-7288 |
| language | English |
| publishDate | 2025-01-01 |
| publisher | BMC |
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| series | Italian Journal of Pediatrics |
| spelling | doaj-art-6de0bf79c49b438698b172580d6fc39b2025-02-02T12:34:55ZengBMCItalian Journal of Pediatrics1824-72882025-01-0151111010.1186/s13052-025-01842-xA child with Chronic Nonbacterial Osteomyelitis and celiac disease: accidental association or two different aspects of the same condition?Grazia Bossi0Maria Sole Prevedoni Gorone1Luca Lungarotti2Francesco Pelillo3Amelia Mascolo4Matteo Naso5Daniele Veraldi6Francesca Olivero7Costanza Chirico8Maria Vittoria Marino9Cristina Dutto10Gian Luigi Marseglia11Department of Pediatrics, IRCCS Policlinico San Matteo FoundationDepartment of Diagnostic and Interventional Radiology and Neuroradiology, IRCCS Policlinico San Matteo FoundationDepartment of Diagnostic and Interventional Radiology and Neuroradiology, IRCCS Policlinico San Matteo FoundationOrthopedics and Traumatology Clinic, IRCCS Policlinico San Matteo FoundationDepartment of Pediatrics, IRCCS Policlinico San Matteo FoundationPediatric School of Specialization, University of PaviaPediatric School of Specialization, University of PaviaPediatric School of Specialization, University of PaviaPediatric School of Specialization, University of PaviaPediatric School of Specialization, University of PaviaPediatric School of Specialization, University of PaviaDepartment of Pediatrics, IRCCS Policlinico San Matteo FoundationAbstract Background Chronic Nonbacterial Osteomyelitis (CNO) is a rare auto-inflammatory disease that mainly affects children, and manifests with single or multiple painful bone lesions. Due to the lack of specific laboratory markers, CNO diagnosis is a matter of exclusion from different conditions, first and foremost bacterial osteomyelitis and malignancies. Whole Body Magnetic Resonance (WBMR) and bone biopsy are the gold standard for the diagnosis. Although the association with Inflammatory Bowel Disease (IBD) has been reported in the literature, cases of CNO in celiac patients have never been described before. Case presentation We report about a girl of 3 years and 8 months of age who presented with severe bone pain, slight increase of inflammatory markers, micro-hematuria and high calprotectin values. Her personal medical history was uneventful, apart from low weight growth. She had never complained of abdominal pain or other gastro-intestinal symptoms. WBMR showed the classical features of multifocal CNO, and biopsy confirmed the diagnosis. Celiac disease (CD) was suspected on the basis of antibody screening, and confirmed by gut biopsy. With gluten-free diet the patient achieved rapid and complete symptom remission together with healing of all the bone lesions proven by WBMR. Three years after the onset of the disease the girl is healthy and totally asymptomatic, still on clinical and radiological follow-up. Conclusions Based on our experience, the diagnostic work-up of new cases of CNO should include the screening test for CD and, according to the literature, the possibility of IBD should also be properly ruled out. When CNO and CD coexist, gluten-free diet, combined with antinflammatory therapy, could be able to completely reverse bone lesions, shortening the duration of medical treatment. Because the diseases’ onset is seldom simultaneous, patients with CNO and IBD deserve a properly extended follow-up. Finally, the analysis of the relationship between CNO and autoimmune intestinal diseases provides a unique opportunity to understand the pathophysiological pro-inflammatory network underlying both types of disorders and it is necessary to make the most suitable therapeutic choice.https://doi.org/10.1186/s13052-025-01842-xPediatricsChronic nonbacterial osteomyelitisAutoimmune intestinal diseasesBone inflammation |
| spellingShingle | Grazia Bossi Maria Sole Prevedoni Gorone Luca Lungarotti Francesco Pelillo Amelia Mascolo Matteo Naso Daniele Veraldi Francesca Olivero Costanza Chirico Maria Vittoria Marino Cristina Dutto Gian Luigi Marseglia A child with Chronic Nonbacterial Osteomyelitis and celiac disease: accidental association or two different aspects of the same condition? Italian Journal of Pediatrics Pediatrics Chronic nonbacterial osteomyelitis Autoimmune intestinal diseases Bone inflammation |
| title | A child with Chronic Nonbacterial Osteomyelitis and celiac disease: accidental association or two different aspects of the same condition? |
| title_full | A child with Chronic Nonbacterial Osteomyelitis and celiac disease: accidental association or two different aspects of the same condition? |
| title_fullStr | A child with Chronic Nonbacterial Osteomyelitis and celiac disease: accidental association or two different aspects of the same condition? |
| title_full_unstemmed | A child with Chronic Nonbacterial Osteomyelitis and celiac disease: accidental association or two different aspects of the same condition? |
| title_short | A child with Chronic Nonbacterial Osteomyelitis and celiac disease: accidental association or two different aspects of the same condition? |
| title_sort | child with chronic nonbacterial osteomyelitis and celiac disease accidental association or two different aspects of the same condition |
| topic | Pediatrics Chronic nonbacterial osteomyelitis Autoimmune intestinal diseases Bone inflammation |
| url | https://doi.org/10.1186/s13052-025-01842-x |
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