{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,8]],"date-time":"2026-09-08T00:09:25Z","timestamp":1788826165808,"version":"build-2803163510"},"reference-count":20,"publisher":"Springer Science and Business Media LLC","issue":"1","license":[{"start":{"date-parts":[[2026,6,17]],"date-time":"2026-06-17T00:00:00Z","timestamp":1781654400000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"},{"start":{"date-parts":[[2026,9,8]],"date-time":"2026-09-08T00:00:00Z","timestamp":1788825600000},"content-version":"vor","delay-in-days":83,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"}],"content-domain":{"domain":["link.springer.com"],"crossmark-restriction":false},"short-container-title":["Pediatr Rheumatol"],"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Background<\/jats:title>\n                    <jats:p>Tumour necrosis factor (TNF) receptor-1 associated periodic syndrome (TRAPS) is an autoinflammatory condition. Most treatment options require regular injections, posing challenges for individuals with needle phobia.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Findings<\/jats:title>\n                    <jats:p>We reviewed the medical records of two siblings, a now 14-year-old female, and her 10-year-old brother, both diagnosed with TRAPS in early infancy. Both children responded to on-demand oral corticosteroid therapy, but due to frequent flares, persistent biochemical inflammation and poor growth, steroid-sparing therapies were required. Non-standard first-line therapies were subsequently selected as the eldest child had developed a needle phobia, and she was commenced on an oral Janus kinase (JAK) inhibitor. The younger brother was treated with tocilizumab, reflecting parental preference to minimise frequent injections. Over the last two years, both children have demonstrated significant improvement, with almost no further TRAPS flares, normalisation of baseline inflammatory markers and improvements in their growth trajectories. No adverse effects were reported on either agent.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusions<\/jats:title>\n                    <jats:p>JAK inhibitor therapy and tocilizumab were effective in our two cases of TRAPS. To our knowledge, this is the first report describing the use of a JAK inhibitor in TRAPS, while also adding to the limited published experience with tocilizumab in TRAPS.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1186\/s12969-026-01225-4","type":"journal-article","created":{"date-parts":[[2026,6,17]],"date-time":"2026-06-17T03:46:03Z","timestamp":1781667963000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["The use of JAK inhibitors and tocilizumab in the management of TRAPS: a sibling case study"],"prefix":"10.1186","volume":"24","author":[{"given":"David","family":"Thai","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Georgina","family":"Tiller","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jonathan","family":"Akikusa","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mark","family":"Taranto","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kathryn","family":"Shepherd","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Seth L.","family":"Masters","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sam","family":"Mehr","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2026,6,17]]},"reference":[{"key":"1225_CR1","doi-asserted-by":"publisher","first-page":"133","DOI":"10.1016\/s0092-8674(00)80721-7","volume":"97","author":"MF McDermott","year":"1999","unstructured":"McDermott MF, Aksentijevich I, Galon J, McDermott EM, Ogunkolade BW, Centola M, et al. Germline Mutations in the Extracellular Domains of the 55\u00a0kDa TNF Receptor, TNFR1, Define a Family of Dominantly Inherited Autoinflammatory Syndromes. Cell. 1999;97:133\u201344. https:\/\/doi.org\/10.1016\/s0092-8674(00)80721-7.","journal-title":"Cell"},{"key":"1225_CR2","doi-asserted-by":"publisher","first-page":"783","DOI":"10.1016\/j.jaip.2020.10.053","volume":"9","author":"R Papa","year":"2021","unstructured":"Papa R, Lane T, Minden K, Touitou I, Cantarini L, Cattalini M, et al. INSAID Variant Classification and Eurofever Criteria Guide Optimal Treatment Strategy in Patients with TRAPS: Data from the Eurofever Registry. 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Eur J Rheumatol. 2015;2:35\u20136. https:\/\/doi.org\/10.5152\/eurjrheumatol.2014.14053.","journal-title":"Eur J Rheumatol"},{"key":"1225_CR5","doi-asserted-by":"publisher","first-page":"1908","DOI":"10.1056\/nejmoa1706314","volume":"378","author":"FD Benedetti","year":"2018","unstructured":"Benedetti FD, Gattorno M, Anton J, Ben-Chetrit E, Frenkel J, Hoffman HM, et al. Canakinumab for the Treatment of Autoinflammatory Recurrent Fever Syndromes. N Engl J Med. 2018;378:1908\u201319. https:\/\/doi.org\/10.1056\/nejmoa1706314.","journal-title":"N Engl J Med"},{"key":"1225_CR6","doi-asserted-by":"publisher","first-page":"907","DOI":"10.1136\/annrheumdis-2021-221801","volume":"81","author":"M Romano","year":"2022","unstructured":"Romano M, Arici ZS, Piskin D, Alehashemi S, Aletaha D, Barron KS, et al. 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