First-year follow-up of children with chronic nonbacterial osteomyelitis—an analysis of the German National Pediatric Rheumatologic Database from 2009 to 2018

Abstract Objective To assess the first-year features of patients with chronic nonbacterial osteomyelitis (CNO). Methods Patients with a diagnosis of CNO, disease duration of under 13 months, and first registration in the German National Pediatric Rheumatologic Database (NPRD) between 2009 and 2018 w...

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Autores principales:	Christiane Reiser, Jens Klotsche, Anton Hospach, Rainer Berendes, Anja Schnabel, Annette F. Jansson, Markus Hufnagel, Nadine Grösch, Martina Niewerth, Kirsten Minden, Hermann Girschick
Formato:	article
Lenguaje:	EN
Publicado:	BMC 2021
Materias:	Chronic nonbacterial osteomyelitis Pediatric rheumatology Autoinflammation Chronic nonbacterial multifocal osteomyelitis Diseases of the musculoskeletal system RC925-935
Acceso en línea:	https://doaj.org/article/5b740b28a4ed42948593af61e3bdbfa0
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spelling	oai:doaj.org-article:5b740b28a4ed42948593af61e3bdbfa02021-11-14T12:07:10ZFirst-year follow-up of children with chronic nonbacterial osteomyelitis—an analysis of the German National Pediatric Rheumatologic Database from 2009 to 201810.1186/s13075-021-02658-w1478-6362https://doaj.org/article/5b740b28a4ed42948593af61e3bdbfa02021-11-01T00:00:00Zhttps://doi.org/10.1186/s13075-021-02658-whttps://doaj.org/toc/1478-6362Abstract Objective To assess the first-year features of patients with chronic nonbacterial osteomyelitis (CNO). Methods Patients with a diagnosis of CNO, disease duration of under 13 months, and first registration in the German National Pediatric Rheumatologic Database (NPRD) between 2009 and 2018 were included in this cross-sectional analysis. Results Of 774 documented patients, 62.8% were female, and all patients had a median age of 11 years. The most affected clinical sites were the tibia (29.7%), pelvis (28.0%), and femur (27.8%). HLA-B27 was positive in 48 of 314 analyzed patients (15.3%). In 406 patients, an X-ray was performed at the first visit; X-ray results showed osteosclerosis/−lysis in 34.0% and hyperostosis in 14.5% of the patients. MRI scans (focal and whole-body scans) were performed in 648 patients, and 81.5% showed a positive TIRM/STIR signal. A total of 84.7% of the patients were administered nonsteroidal anti-inflammatory drugs, 9.6% were administered oral glucocorticoids, 10.8% were administered disease-modifying anti-rheumatic drugs (DMARDs), and 6.1% were administered bisphosphonates. An evaluation of the patient’s questionnaire showed an overall well-being (NRS 0–10) of 2.0. The PedCNO disease “activity” score revealed a 70% improvement in variables in 43% of patients in the initial 1-year follow-up. Copresentation with diagnostic criteria of pediatric enthesitis-related arthritis was rare. Conclusion To our knowledge, the NPRD cohort seemed to be the largest cohort of children and adolescents suffering from CNO worldwide. Most patients were treated effectively with NSAIDs, and only a small group of patients was administered additional medication. The patient-defined measures of disease activity had a moderate impact on patients’ daily lives. Trial registration Not applicable.Christiane ReiserJens KlotscheAnton HospachRainer BerendesAnja SchnabelAnnette F. JanssonMarkus HufnagelNadine GröschMartina NiewerthKirsten MindenHermann GirschickBMCarticleChronic nonbacterial osteomyelitisPediatric rheumatologyAutoinflammationChronic nonbacterial multifocal osteomyelitisDiseases of the musculoskeletal systemRC925-935ENArthritis Research & Therapy, Vol 23, Iss 1, Pp 1-12 (2021)
institution	DOAJ
collection	DOAJ
language	EN
topic	Chronic nonbacterial osteomyelitis Pediatric rheumatology Autoinflammation Chronic nonbacterial multifocal osteomyelitis Diseases of the musculoskeletal system RC925-935
spellingShingle	Chronic nonbacterial osteomyelitis Pediatric rheumatology Autoinflammation Chronic nonbacterial multifocal osteomyelitis Diseases of the musculoskeletal system RC925-935 Christiane Reiser Jens Klotsche Anton Hospach Rainer Berendes Anja Schnabel Annette F. Jansson Markus Hufnagel Nadine Grösch Martina Niewerth Kirsten Minden Hermann Girschick First-year follow-up of children with chronic nonbacterial osteomyelitis—an analysis of the German National Pediatric Rheumatologic Database from 2009 to 2018
description	Abstract Objective To assess the first-year features of patients with chronic nonbacterial osteomyelitis (CNO). Methods Patients with a diagnosis of CNO, disease duration of under 13 months, and first registration in the German National Pediatric Rheumatologic Database (NPRD) between 2009 and 2018 were included in this cross-sectional analysis. Results Of 774 documented patients, 62.8% were female, and all patients had a median age of 11 years. The most affected clinical sites were the tibia (29.7%), pelvis (28.0%), and femur (27.8%). HLA-B27 was positive in 48 of 314 analyzed patients (15.3%). In 406 patients, an X-ray was performed at the first visit; X-ray results showed osteosclerosis/−lysis in 34.0% and hyperostosis in 14.5% of the patients. MRI scans (focal and whole-body scans) were performed in 648 patients, and 81.5% showed a positive TIRM/STIR signal. A total of 84.7% of the patients were administered nonsteroidal anti-inflammatory drugs, 9.6% were administered oral glucocorticoids, 10.8% were administered disease-modifying anti-rheumatic drugs (DMARDs), and 6.1% were administered bisphosphonates. An evaluation of the patient’s questionnaire showed an overall well-being (NRS 0–10) of 2.0. The PedCNO disease “activity” score revealed a 70% improvement in variables in 43% of patients in the initial 1-year follow-up. Copresentation with diagnostic criteria of pediatric enthesitis-related arthritis was rare. Conclusion To our knowledge, the NPRD cohort seemed to be the largest cohort of children and adolescents suffering from CNO worldwide. Most patients were treated effectively with NSAIDs, and only a small group of patients was administered additional medication. The patient-defined measures of disease activity had a moderate impact on patients’ daily lives. Trial registration Not applicable.
format	article
author	Christiane Reiser Jens Klotsche Anton Hospach Rainer Berendes Anja Schnabel Annette F. Jansson Markus Hufnagel Nadine Grösch Martina Niewerth Kirsten Minden Hermann Girschick
author_facet	Christiane Reiser Jens Klotsche Anton Hospach Rainer Berendes Anja Schnabel Annette F. Jansson Markus Hufnagel Nadine Grösch Martina Niewerth Kirsten Minden Hermann Girschick
author_sort	Christiane Reiser
title	First-year follow-up of children with chronic nonbacterial osteomyelitis—an analysis of the German National Pediatric Rheumatologic Database from 2009 to 2018
title_short	First-year follow-up of children with chronic nonbacterial osteomyelitis—an analysis of the German National Pediatric Rheumatologic Database from 2009 to 2018
title_full	First-year follow-up of children with chronic nonbacterial osteomyelitis—an analysis of the German National Pediatric Rheumatologic Database from 2009 to 2018
title_fullStr	First-year follow-up of children with chronic nonbacterial osteomyelitis—an analysis of the German National Pediatric Rheumatologic Database from 2009 to 2018
title_full_unstemmed	First-year follow-up of children with chronic nonbacterial osteomyelitis—an analysis of the German National Pediatric Rheumatologic Database from 2009 to 2018
title_sort	first-year follow-up of children with chronic nonbacterial osteomyelitis—an analysis of the german national pediatric rheumatologic database from 2009 to 2018
publisher	BMC
publishDate	2021
url	https://doaj.org/article/5b740b28a4ed42948593af61e3bdbfa0
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First-year follow-up of children with chronic nonbacterial osteomyelitis—an analysis of the German National Pediatric Rheumatologic Database from 2009 to 2018

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