Clinical Presentation and Course of Pulmonary Involvement in Chronic Nonbacterial Osteomyelitis
Objective Pulmonary involvement in chronic nonbacterial osteomyelitis (CNO) is rare. Limited awareness results in diagnostic challenges, especially because malignancy or infection needs to be considered. Methods Based on a survey shared among centers participating in the Kerndokumentation Deutsches...
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2025-01-01
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Online Access: | https://doi.org/10.1002/acr2.11799 |
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author | Toni Hospach Friederike Blankenburg Anita Heinkele Kristina Rücklová Peter Müller‐Abt Frank Weller‐Heinemann Laura Buchtala Jan Maier Jasmin Kümmerle‐Deschner Christiane Reiser Ales Janda Andreas Urban Reiner Berendes Andrea Skrabl‐Baumgartner Jürgen Brunner Ralf Trauzeddel Johannes Peter Haas Annette Holl‐Wieden Henner Morbach Anja Schnabel Normi Brück Gabriele Hahn Wolfgang Emminger Isabella Valent Regine Borchers Christian Klemann Ilias Tsiflikas Jens Klotsche Kirsten Minden Christian M. Hedrich Thekla von Kalle |
author_facet | Toni Hospach Friederike Blankenburg Anita Heinkele Kristina Rücklová Peter Müller‐Abt Frank Weller‐Heinemann Laura Buchtala Jan Maier Jasmin Kümmerle‐Deschner Christiane Reiser Ales Janda Andreas Urban Reiner Berendes Andrea Skrabl‐Baumgartner Jürgen Brunner Ralf Trauzeddel Johannes Peter Haas Annette Holl‐Wieden Henner Morbach Anja Schnabel Normi Brück Gabriele Hahn Wolfgang Emminger Isabella Valent Regine Borchers Christian Klemann Ilias Tsiflikas Jens Klotsche Kirsten Minden Christian M. Hedrich Thekla von Kalle |
author_sort | Toni Hospach |
collection | DOAJ |
description | Objective Pulmonary involvement in chronic nonbacterial osteomyelitis (CNO) is rare. Limited awareness results in diagnostic challenges, especially because malignancy or infection needs to be considered. Methods Based on a survey shared among centers participating in the Kerndokumentation Deutsches Rheumaforschungszentrum (Germany), this study investigated clinical and imaging presentations, demographic features, treatment response and outcomes of pulmonary involvement in CNO (pCNO). Magnetic resonance imaging and computed tomography images were read centrally by an experienced pediatric radiologist. Results Twenty‐two patients with pCNO were included in this study. Among patients with CNO, pulmonary involvement was more common in girls (91% vs 62.8%, P = 0.006) and patients with multifocal bone lesions (95% vs 65%, P <0.001) but was not associated with systemic inflammation or additional organ involvement. Forty‐two pulmonary lesions were counted with a median of two per patient (two to six). They displayed a median size of 1.8 cm (0.3–4.0 cm) and followed mono‐ (40%) and oligo‐focal (60%) patterns representing consolidations or nodules, abutting the pleura in 50%. Although prominent hilar lymph nodes were present (in 19% of patients), no pathologic enlargement (>1 cm) was seen. When available (3 of 22 patients), histology revealed granulomatous inflammation with lymphocyte infiltration. Development and courses of pCNO did not associate with treatments chosen. Complete remission was reported in 60% of patients, partial remission in 20%. Conclusion pCNO is usually asymptomatic. Although more common in girls and patients with multifocal CNO, pCNO is not associated with systemic parameters of inflammation or specific organ involvement. Prognosis of pCNO is favorable, and most lesions resolve over time. Thus, a careful watch‐and‐wait strategy may be appropriate. |
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spelling | doaj-art-a2f2c93038ea424d8522fc9326adf4f72025-02-04T06:21:22ZengWileyACR Open Rheumatology2578-57452025-01-0171n/an/a10.1002/acr2.11799Clinical Presentation and Course of Pulmonary Involvement in Chronic Nonbacterial OsteomyelitisToni Hospach0Friederike Blankenburg1Anita Heinkele2Kristina Rücklová3Peter Müller‐Abt4Frank Weller‐Heinemann5Laura Buchtala6Jan Maier7Jasmin Kümmerle‐Deschner8Christiane Reiser9Ales Janda10Andreas Urban11Reiner Berendes12Andrea Skrabl‐Baumgartner13Jürgen Brunner14Ralf Trauzeddel15Johannes Peter Haas16Annette Holl‐Wieden17Henner Morbach18Anja Schnabel19Normi Brück20Gabriele Hahn21Wolfgang Emminger22Isabella Valent23Regine Borchers24Christian Klemann25Ilias Tsiflikas26Jens Klotsche27Kirsten Minden28Christian M. Hedrich29Thekla von Kalle30Olgahospital Klinikum Stuttgart GermanyOlgahospital Klinikum Stuttgart GermanyOlgahospital Klinikum Stuttgart GermanyOlgahospital, Klinikum Stuttgart, Germany, and Charles University Prague Czech RepublicOlgahospital Klinikum Stuttgart GermanyProf.‐Hess‐Kinderklinik Bremen GermanyProf.‐Hess‐Kinderklinik Bremen GermanyChildren Medical Practice Leinfelden‐Echterdingen GermanyUniversity of Tübingen Tübingen GermanyUniversity of Tübingen, Tübingen, Germany, and Landeskrankenhaus Bregenz Bregenz AustriaUniversity Medical Center, Ulm University Ulm GermanyKlinikum St Marien Amberg GermanyCenter for Pediatric Rheumatology Landshut GermanyMedical University Graz Graz AustriaMedical University of Innsbruck, Innsbruck, Austria, and Danube Private University Krems AustriaHelios Klinikum Berlin‐Buch Berlin GermanyGerman Center for Pediatric and Adolescent Rheumatology Garmisch‐Partenkirchen GermanyUniversity of Wuerzburg Wuerzburg GermanyUniversity of Wuerzburg Wuerzburg GermanyTechnische Universität Dresden GermanyTechnische Universität Dresden GermanyTechnische Universität Dresden GermanyUniversity of Vienna Vienna AustriaUniversity of Vienna Vienna AustriaUniversity of Augsburg Augsburg GermanyUniversity of Leipzig Leipzig GermanyUniversity of Tübingen Tübingen GermanyCharité Universitätsmedizin Berlin Berlin GermanyCharité Universitätsmedizin Berlin and Rheuma‐Forschungszentrum Berlin Berlin GermanyUniversity of Liverpool and Alder Hey Children's NHS Foundation Trust Liverpool United KingdomOlgahospital Klinikum Stuttgart GermanyObjective Pulmonary involvement in chronic nonbacterial osteomyelitis (CNO) is rare. Limited awareness results in diagnostic challenges, especially because malignancy or infection needs to be considered. Methods Based on a survey shared among centers participating in the Kerndokumentation Deutsches Rheumaforschungszentrum (Germany), this study investigated clinical and imaging presentations, demographic features, treatment response and outcomes of pulmonary involvement in CNO (pCNO). Magnetic resonance imaging and computed tomography images were read centrally by an experienced pediatric radiologist. Results Twenty‐two patients with pCNO were included in this study. Among patients with CNO, pulmonary involvement was more common in girls (91% vs 62.8%, P = 0.006) and patients with multifocal bone lesions (95% vs 65%, P <0.001) but was not associated with systemic inflammation or additional organ involvement. Forty‐two pulmonary lesions were counted with a median of two per patient (two to six). They displayed a median size of 1.8 cm (0.3–4.0 cm) and followed mono‐ (40%) and oligo‐focal (60%) patterns representing consolidations or nodules, abutting the pleura in 50%. Although prominent hilar lymph nodes were present (in 19% of patients), no pathologic enlargement (>1 cm) was seen. When available (3 of 22 patients), histology revealed granulomatous inflammation with lymphocyte infiltration. Development and courses of pCNO did not associate with treatments chosen. Complete remission was reported in 60% of patients, partial remission in 20%. Conclusion pCNO is usually asymptomatic. Although more common in girls and patients with multifocal CNO, pCNO is not associated with systemic parameters of inflammation or specific organ involvement. Prognosis of pCNO is favorable, and most lesions resolve over time. Thus, a careful watch‐and‐wait strategy may be appropriate.https://doi.org/10.1002/acr2.11799 |
spellingShingle | Toni Hospach Friederike Blankenburg Anita Heinkele Kristina Rücklová Peter Müller‐Abt Frank Weller‐Heinemann Laura Buchtala Jan Maier Jasmin Kümmerle‐Deschner Christiane Reiser Ales Janda Andreas Urban Reiner Berendes Andrea Skrabl‐Baumgartner Jürgen Brunner Ralf Trauzeddel Johannes Peter Haas Annette Holl‐Wieden Henner Morbach Anja Schnabel Normi Brück Gabriele Hahn Wolfgang Emminger Isabella Valent Regine Borchers Christian Klemann Ilias Tsiflikas Jens Klotsche Kirsten Minden Christian M. Hedrich Thekla von Kalle Clinical Presentation and Course of Pulmonary Involvement in Chronic Nonbacterial Osteomyelitis ACR Open Rheumatology |
title | Clinical Presentation and Course of Pulmonary Involvement in Chronic Nonbacterial Osteomyelitis |
title_full | Clinical Presentation and Course of Pulmonary Involvement in Chronic Nonbacterial Osteomyelitis |
title_fullStr | Clinical Presentation and Course of Pulmonary Involvement in Chronic Nonbacterial Osteomyelitis |
title_full_unstemmed | Clinical Presentation and Course of Pulmonary Involvement in Chronic Nonbacterial Osteomyelitis |
title_short | Clinical Presentation and Course of Pulmonary Involvement in Chronic Nonbacterial Osteomyelitis |
title_sort | clinical presentation and course of pulmonary involvement in chronic nonbacterial osteomyelitis |
url | https://doi.org/10.1002/acr2.11799 |
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