{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,2,6]],"date-time":"2026-02-06T05:39:50Z","timestamp":1770356390200,"version":"3.49.0"},"reference-count":32,"publisher":"Journal of Neurosurgery Publishing Group (JNSPG)","issue":"19","license":[{"start":{"date-parts":[[2021,5,10]],"date-time":"2021-05-10T00:00:00Z","timestamp":1620604800000},"content-version":"unspecified","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2021,5,10]]},"abstract":"<jats:sec>\n                    <jats:title>BACKGROUND<\/jats:title>\n                    <jats:p>Adamantinomatous craniopharyngioma (ACP) is a highly morbid adult and pediatric brain tumor derived from epithelial remnants of the craniopharyngeal canal (Rathke\u2019s pouch), which gives rise to the anterior pituitary gland. Standard therapy includes maximal safe resection with or without radiation therapy. Systemic antitumor therapy remains elusive. Immune-related paracrine signaling involving the interleukin-6 receptor (IL-6R) may contribute to ACP pathogenesis. Tocilizumab, a recombinant humanized monoclonal antibody against IL-6R, is approved by the US Food and Drug Administration but does not cross an intact blood\u2013brain barrier.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>OBSERVATIONS<\/jats:title>\n                    <jats:p>In a phase 0 trial design, a single dose of tocilizumab was delivered intravenously before clinically indicated surgical intervention in 3 children with ACP. The presence of tocilizumab was assayed in plasma, tumor tissue, tumor cyst fluid, and cerebrospinal fluid (n = 1) using a novel enzyme-linked immunosorbent assay. Tocilizumab reached ACP tumor tissue and\/or cyst fluid after one systemic dose in every patient.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>LESSONS<\/jats:title>\n                    <jats:p>This finding helps explain extant data that indicate tocilizumab may contribute to ACP therapy. It further indicates that ACP does not reside behind an intact blood\u2013brain barrier, dramatically broadening the range of potential antitumor therapies against this tumor. This has substantial implications for the design of future clinical trials for novel therapies against ACP in both children and adults.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.3171\/case2150","type":"journal-article","created":{"date-parts":[[2021,5,17]],"date-time":"2021-05-17T15:50:42Z","timestamp":1621266642000},"source":"Crossref","is-referenced-by-count":4,"title":["Adamantinomatous craniopharyngioma associated with a compromised blood\u2013brain barrier: patient series"],"prefix":"10.3171","volume":"1","author":[{"given":"Eric W.","family":"Prince","sequence":"first","affiliation":[{"name":"Departments of Neurosurgery,"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Lindsey M.","family":"Hoffman","sequence":"additional","affiliation":[{"name":"Division of Hematology\/Oncology, Phoenix Children\u2019s Hospital, Phoenix, Arizona; and"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Trinka","family":"Vijmasi","sequence":"additional","affiliation":[{"name":"Departments of Neurosurgery,"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Kathleen","family":"Dorris","sequence":"additional","affiliation":[{"name":"Pediatrics,"},{"name":"Morgan Adams Foundation Pediatric Brain Tumor Research Program, Denver, Colorado"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Jennifer A.","family":"McWilliams","sequence":"additional","affiliation":[{"name":"Immunology and Microbiology, and"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Kimberly R.","family":"Jordan","sequence":"additional","affiliation":[{"name":"Immunology and Microbiology, and"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"David M.","family":"Mirsky","sequence":"additional","affiliation":[{"name":"Radiology, University of Colorado School of Medicine and Children\u2019s Hospital Colorado, Aurora, Colorado"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Todd C.","family":"Hankinson","sequence":"additional","affiliation":[{"name":"Departments of Neurosurgery,"},{"name":"Pediatrics,"},{"name":"Morgan Adams Foundation Pediatric Brain Tumor Research Program, Denver, Colorado"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"1754","reference":[{"issue":"1","key":"b1","series-title":"Nat Rev Dis Primers","doi-asserted-by":"publisher","first-page":"75","DOI":"10.1038\/s41572-019-0125-9","article-title":"Craniopharyngioma","volume":"5","author":"M\u00fcller HL","year":"2019","unstructured":"M\u00fcller HL, Merchant TE, Warmuth-Metz M,  Craniopharyngioma. 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