{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,25]],"date-time":"2026-10-25T03:46:48Z","timestamp":1792900008461,"version":"4.5.0"},"reference-count":17,"publisher":"American Academy of Pediatrics (AAP)","issue":"2","content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2016,8,1]]},"abstract":"<jats:p>Chronic granulomatous disease (CGD) is a primary immunodeficiency that leads to severe recurrent infection and inflammatory complications that are usually difficult to diagnose and treat. Several hyperinflammation mechanisms, such as decreased neutrophil apoptosis, toll-like receptor activation imbalance, Th17 cell induction, Nrf2 activity deficiency, and inflammasome activation, have been described in CGD patients However, there have been no reports of chronic recurrent multifocal osteomyelitis as an inflammatory complication in CGD, and the differential diagnosis of this condition with infectious osteomyelitis is challenging. Thalidomide has been used to treat several inflammatory manifestations in CGD patients with good clinical results. Here, we report the case of a previously asymptomatic 11-year-old boy who consulted for difficulty walking and pain at the back of the right thigh, with increased inflammatory markers. Multifocal bone involvement was seen on bone scintigraphy, and acute-phase reactants were elevated. On the basis of a suspected diagnosis of infectious osteomyelitis, broad-spectrum antibiotic therapy was started, with no clinical response. Bone biopsy and microbiological tests yielded negative results; at that point, chronic recurrent multifocal osteomyelitis was suspected. The patient was unresponsive to nonsteroidal antiinflammatory drugs and corticosteroids. Thalidomide was started, and within 6 months, clinical and radiologic resolution of the condition was achieved with no adverse effects. More than 1 year after stopping thalidomide, the patient remained free of symptoms and inflammatory parameters are within normal levels. Thalidomide has a favorable safety profile compared with other alternatives and could be considered a feasible therapeutic option for this type of condition in selected patients.<\/jats:p>","DOI":"10.1542\/peds.2015-4017","type":"journal-article","created":{"date-parts":[[2016,7,19]],"date-time":"2016-07-19T09:10:11Z","timestamp":1468919411000},"source":"Crossref","is-referenced-by-count":1,"title":["Chronic Recurrent Multifocal Osteomyelitis and Thalidomide in Chronic Granulomatous Disease"],"prefix":"10.1542","volume":"138","author":[{"given":"Andrea","family":"Mart\u00edn-Nalda","sequence":"first","affiliation":[{"name":"Pediatric Infectious Diseases and Immunodeficiencies Unit,"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Isabel","family":"Roca","sequence":"additional","affiliation":[{"name":"Department of Nuclear Medicine,"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Cesar Galo","family":"Fontecha","sequence":"additional","affiliation":[{"name":"Unit of Pediatric Orthopaedic Surgery,"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Aurora","family":"Fern\u00e1ndez-Polo","sequence":"additional","affiliation":[{"name":"Department of Pharmacy,"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ignasi","family":"Barber","sequence":"additional","affiliation":[{"name":"Department of Pediatric Radiology, and"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"M\u00f3nica","family":"Martinez-Gallo","sequence":"additional","affiliation":[{"name":"Division of Immunology, Hospital Universitari Vall d\u2019Hebron, Institut de Recerca Vall d\u2019Hebron, Universitat Aut\u00f2noma de Barcelona, Barcelona, Spain"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Pere","family":"Soler-Palacin","sequence":"additional","affiliation":[{"name":"Pediatric Infectious Diseases and Immunodeficiencies Unit,"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"417","published-online":{"date-parts":[[2016,8,1]]},"reference":[{"issue":"3","key":"2021120904513111600_B1","doi-asserted-by":"crossref","first-page":"155","DOI":"10.1097\/00005792-200005000-00003","article-title":"Chronic granulomatous disease. Report on a national registry of 368 patients.","volume":"79","author":"Winkelstein","year":"2000","journal-title":"Medicine (Baltimore)"},{"issue":"4","key":"2021120904513111600_B2","doi-asserted-by":"crossref","first-page":"e5234","DOI":"10.1371\/journal.pone.0005234","article-title":"Chronic granulomatous disease: the European experience.","volume":"4","author":"van den Berg","year":"2009","journal-title":"PLoS One"},{"issue":"11","key":"2021120904513111600_B3","doi-asserted-by":"crossref","first-page":"924","DOI":"10.1590\/1414-431X20143735","article-title":"Chronic granulomatous disease: why an inflammatory disease?","volume":"47","author":"Roxo-Junior","year":"2014","journal-title":"Braz J Med Biol Res"},{"issue":"3","key":"2021120904513111600_B4","doi-asserted-by":"crossref","first-page":"655","DOI":"10.1016\/j.jaci.2014.04.014","article-title":"Inflammatory manifestations in a single-center cohort of patients with chronic granulomatous disease.","volume":"134","author":"Magnani","year":"2014","journal-title":"J Allergy Clin Immunol"},{"issue":"4","key":"2021120904513111600_B5","doi-asserted-by":"crossref","first-page":"407","DOI":"10.1007\/s00281-015-0488-2","article-title":"New discoveries in CRMO: IL-1\u03b2, the neutrophil, and the microbiome implicated in disease pathogenesis in Pstpip2-deficient mice.","volume":"37","author":"Ferguson","year":"2015","journal-title":"Semin Immunopathol"},{"issue":"3","key":"2021120904513111600_B6","doi-asserted-by":"crossref","first-page":"120","DOI":"10.4103\/1450-1147.136737","article-title":"Chronic recurrent multifocal osteomyelitis in children: a multidisciplinary approach is needed to establish a diagnosis.","volume":"12","author":"Alshammari","year":"2013","journal-title":"World J Nucl Med"},{"issue":"1","key":"2021120904513111600_B7","doi-asserted-by":"crossref","first-page":"59","DOI":"10.1016\/S0046-8177(99)90301-5","article-title":"Chronic recurrent multifocal osteomyelitis in children: diagnostic value of histopathology and microbial testing.","volume":"30","author":"Girschick","year":"1999","journal-title":"Hum Pathol"},{"issue":"4","key":"2021120904513111600_B8","doi-asserted-by":"crossref","first-page":"997","DOI":"10.1016\/j.jaci.2013.04.059","article-title":"Efficacy and safety of thalidomide in patients with inflammatory manifestations of chronic granulomatous disease: a retrospective case series.","volume":"132","author":"Noel","year":"2013","journal-title":"J Allergy Clin Immunol"},{"issue":"4","key":"2021120904513111600_B9","doi-asserted-by":"crossref","first-page":"1069","DOI":"10.1038\/ajg.2009.56","article-title":"Thalidomide as a treatment for refractory CGD colitis.","volume":"104","author":"Sokol","year":"2009","journal-title":"Am J Gastroenterol"},{"key":"2021120904513111600_B10","unstructured":"European public assessment report (EPAR) product information: Thalidomide Celgene: EPAR\u2014Conditions imposed on member states for safe and effective use\u2014Annex IV. London, United Kingdom: European Medicines Agency; February 25,2009. Available at: http:\/\/www.ema.europa.eu\/ema\/index.jsp?curl=pages\/medicines\/human\/medicines\/000823\/human_med_001090.jsp&mid=WC0b01ac058001d124. Accessed August 3, 2015"},{"issue":"1\u20132","key":"2021120904513111600_B11","doi-asserted-by":"crossref","first-page":"72","DOI":"10.1007\/s00439-004-1208-5","article-title":"Characterization of six novel mutations in the CYBB gene leading to different sub-types of X-linked chronic granulomatous disease.","volume":"116","author":"Stasia","year":"2005","journal-title":"Hum Genet"},{"issue":"9","key":"2021120904513111600_B12","doi-asserted-by":"crossref","first-page":"1688","DOI":"10.1093\/rheumatology\/kev013","article-title":"Chronic recurrent multifocal osteomyelitis in children: nine years\u2019 experience at a statewide tertiary paediatric rheumatology referral centre.","volume":"54","author":"Walsh","year":"2015","journal-title":"Rheumatology (Oxford)"},{"issue":"138","key":"2021120904513111600_B13","first-page":"252460","article-title":"Current concepts of hyperinflammation in chronic granulomatous disease.","volume":"2012","author":"Rieber","year":"2012","journal-title":"Clin Dev Immunol"},{"issue":"3","key":"2021120904513111600_B14","doi-asserted-by":"crossref","first-page":"217","DOI":"10.1038\/sj.ejhg.5200789","article-title":"Chronic recurrent multifocal osteomyelitis (CRMO): evidence for a susceptibility gene located on chromosome 18q21.3\u201318q22.","volume":"10","author":"Golla","year":"2002","journal-title":"Eur J Hum Genet"},{"issue":"6","key":"2021120904513111600_B15","doi-asserted-by":"crossref","first-page":"e39155","DOI":"10.1371\/journal.pone.0039155","article-title":"TLR9-dependent and independent pathways drive activation of the immune system by Propionibacterium acnes.","volume":"7","author":"Tchaptchet","year":"2012","journal-title":"PLoS One"},{"issue":"6","key":"2021120904513111600_B16","doi-asserted-by":"crossref","first-page":"853","DOI":"10.1086\/338875","article-title":"Propionibacterium acnes chest infections in patients with chronic granulomatous disease: case reports.","volume":"34","author":"Bourdeaut","year":"2002","journal-title":"Clin Infect Dis"},{"issue":"9","key":"2021120904513111600_B17","doi-asserted-by":"crossref","first-page":"3526","DOI":"10.1073\/pnas.1322831111","article-title":"IL-1 receptor blockade restores autophagy and reduces inflammation in chronic granulomatous disease in mice and in humans.","volume":"111","author":"de Luca","year":"2014","journal-title":"Proc Natl Acad Sci USA"}],"container-title":["Pediatrics"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/publications.aap.org\/pediatrics\/article-pdf\/doi\/10.1542\/peds.2015-4017\/1099021\/peds_20154017.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"syndication"},{"URL":"https:\/\/publications.aap.org\/pediatrics\/article-pdf\/doi\/10.1542\/peds.2015-4017\/1099021\/peds_20154017.pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2021,12,9]],"date-time":"2021-12-09T21:47:36Z","timestamp":1639086456000},"score":1,"resource":{"primary":{"URL":"https:\/\/publications.aap.org\/pediatrics\/article\/138\/2\/e20154017\/52411\/Chronic-Recurrent-Multifocal-Osteomyelitis-and"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2016,8,1]]},"references-count":17,"journal-issue":{"issue":"2","published-online":{"date-parts":[[2016,8,1]]},"published-print":{"date-parts":[[2016,8,1]]}},"URL":"https:\/\/doi.org\/10.1542\/peds.2015-4017","relation":{},"ISSN":["0031-4005","1098-4275"],"issn-type":[{"value":"0031-4005","type":"print"},{"value":"1098-4275","type":"electronic"}],"subject":[],"published":{"date-parts":[[2016,8,1]]},"article-number":"e20154017"}}