{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,27]],"date-time":"2026-08-27T17:17:28Z","timestamp":1787851048681,"version":"build-2784847793"},"reference-count":13,"publisher":"SAGE Publications","license":[{"start":{"date-parts":[[2026,8,24]],"date-time":"2026-08-24T00:00:00Z","timestamp":1787529600000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/journals.sagepub.com\/page\/policies\/text-and-data-mining-license"}],"content-domain":{"domain":["journals.sagepub.com"],"crossmark-restriction":true},"short-container-title":["Clin Pediatr (Phila)"],"abstract":"<jats:p>Central nervous system (CNS) tuberculosis (TB) carries high mortality and neurologic sequelae, especially when unrecognized inborn errors of immunity are present. We report a young woman with refractory TB meningitis, multiple intracranial tuberculomas, hydrocephalus, and intracranial hypertension despite 10 months of standard anti-TB therapy and corticosteroids. Severe vomiting led to poor adherence and severe weight loss. As immunologic evaluation suggested atypical combined immunodeficiency (CID); monthly intravenous immunoglobulin (IVIG) was started. Neurosurgery did not give indication for shunt surgery. Given the severity and presumed impaired antimycobacterial immunity, adjuvant interferon-\u03b3 (IFN-\u03b3; 50 \u00b5g\/m\u00b2 3 times weekly) was added to therapy with steroids. Vomiting resolved, weight improved, and treatment was tolerated. Serial magnetic resonance imaging showed reduction in leptomeningeal enhancement and tuberculoma burden without surgery; no neurologic sequelae developed. Next-generation sequencing identified a homozygous NHEJ1 variant, not confirmed by Sanger, but findings supported CID. In refractory CNS-TB with suspected immunodeficiency, IFN-\u03b3, steroids, IVIG in addition to anti-TB therapy may improve outcomes and avoid neurosurgical intervention. Early immunological evaluation and host-directed therapy should be considered.<\/jats:p>","DOI":"10.1177\/00099228261474403","type":"journal-article","created":{"date-parts":[[2026,8,25]],"date-time":"2026-08-25T06:33:45Z","timestamp":1787639625000},"update-policy":"https:\/\/doi.org\/10.1177\/sage-journals-update-policy","source":"Crossref","is-referenced-by-count":0,"title":["Successful Therapy of CNS Tuberculosis With Granulomatous Inflammation in Immunodeficiency"],"prefix":"10.1177","author":[{"given":"Deniz","family":"Cagdas","sequence":"first","affiliation":[{"name":"Division of Pediatric Immunology, Department of Pediatrics, Hacettepe University Faculty of Medicine, Ankara, Turkey"},{"name":"Section of Immunology, Institute of Child Health, Ankara, Turkey"},{"name":"\u0130hsan Do\u011framac\u0131 Children\u2019s Hospital, Hacettepe University Faculty of Medicine, Ankara, Turkey"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Rahsan","family":"Gocmen","sequence":"additional","affiliation":[{"name":"Department of Radiology, Hacettepe University Faculty of Medicine, Ankara, Turkey"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0001-3488-0283","authenticated-orcid":false,"given":"Gamze","family":"Sonmez","sequence":"additional","affiliation":[{"name":"Hacettepe University Faculty of Medicine, Ankara, Turkey"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Aysegul","family":"Akarsu","sequence":"additional","affiliation":[{"name":"Division of Pediatric Immunology, Department of Pediatrics, Hacettepe University Faculty of Medicine, Ankara, Turkey"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Cagkan","family":"Inkaya","sequence":"additional","affiliation":[{"name":"Department of Infectious Diseases, Hacettepe University Faculty of Medicine, Ankara, Turkey"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ilkay","family":"Isikay","sequence":"additional","affiliation":[{"name":"Department of Neurosurgery, Hacettepe University Faculty of Medicine, Ankara, Turkey"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"179","published-online":{"date-parts":[[2026,8,24]]},"reference":[{"key":"e_1_3_3_2_2","doi-asserted-by":"publisher","DOI":"10.1016\/S1473-3099(24)00158-0"},{"key":"e_1_3_3_3_2","doi-asserted-by":"publisher","DOI":"10.1128\/CMR.00042-07"},{"key":"e_1_3_3_4_2","doi-asserted-by":"publisher","DOI":"10.1016\/j.jctube.2024.100414"},{"key":"e_1_3_3_5_2","doi-asserted-by":"publisher","DOI":"10.1007\/s10875-021-01002-w"},{"key":"e_1_3_3_6_2","doi-asserted-by":"publisher","DOI":"10.24953\/turkjped.2016.04.002"},{"key":"e_1_3_3_7_2","doi-asserted-by":"publisher","DOI":"10.1016\/j.jneuroim.2022.577952"},{"key":"e_1_3_3_8_2","doi-asserted-by":"publisher","DOI":"10.1016\/j.tube.2020.102016"},{"key":"e_1_3_3_9_2","volume-title":"Tuberculosis Surveillance and Monitoring in Europe 2019\u20132017 Data","author":"World Health Organization; Regional Office for Europe; European Centre for Disease Prevention and Control","year":"2019","unstructured":"World Health Organization; Regional Office for Europe; European Centre for Disease Prevention and Control. Tuberculosis Surveillance and Monitoring in Europe 2019\u20132017 Data. WHO; 2019."},{"key":"e_1_3_3_10_2","doi-asserted-by":"publisher","DOI":"10.1016\/j.jocn.2010.01.006"},{"key":"e_1_3_3_11_2","doi-asserted-by":"publisher","DOI":"10.1016\/j.eclinm.2023.101900"},{"key":"e_1_3_3_12_2","doi-asserted-by":"publisher","DOI":"10.1111\/sji.13084"},{"key":"e_1_3_3_13_2","doi-asserted-by":"publisher","DOI":"10.1007\/s10875-022-01342-1"},{"key":"e_1_3_3_14_2","doi-asserted-by":"publisher","DOI":"10.1002\/eji.201948190"}],"container-title":["Clinical Pediatrics"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/journals.sagepub.com\/doi\/pdf\/10.1177\/00099228261474403","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/journals.sagepub.com\/doi\/full-xml\/10.1177\/00099228261474403","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/journals.sagepub.com\/doi\/pdf\/10.1177\/00099228261474403","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,8,27]],"date-time":"2026-08-27T16:20:34Z","timestamp":1787847634000},"score":1,"resource":{"primary":{"URL":"https:\/\/journals.sagepub.com\/doi\/10.1177\/00099228261474403"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026,8,24]]},"references-count":13,"alternative-id":["10.1177\/00099228261474403"],"URL":"https:\/\/doi.org\/10.1177\/00099228261474403","relation":{},"ISSN":["0009-9228","1938-2707"],"issn-type":[{"value":"0009-9228","type":"print"},{"value":"1938-2707","type":"electronic"}],"subject":[],"published":{"date-parts":[[2026,8,24]]},"article-number":"00099228261474403"}}