{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,3,10]],"date-time":"2026-03-10T09:51:56Z","timestamp":1773136316358,"version":"3.50.1"},"reference-count":24,"publisher":"Journal of Neurosurgery Publishing Group (JNSPG)","issue":"22","license":[{"start":{"date-parts":[[2024,5,27]],"date-time":"2024-05-27T00:00:00Z","timestamp":1716768000000},"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":[[2024,5,27]]},"abstract":"<jats:sec>\n                    <jats:title>BACKGROUND<\/jats:title>\n                    <jats:p>Anaplastic lymphoma kinase (ALK)\u2013positive histiocytosis (ALK-H) is an emerging entity in the category of histiocytic neoplasms that was first reported as a multisystemic disease in three infants in 2008. The clinicopathological spectrum of ALK-H has been expanded to include localized disorders in specific organs, but the features of this subtype are not well known. The authors report a case of ALK-H localized in the central nervous system that was difficult to treat and review the relevant literature.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>OBSERVATIONS<\/jats:title>\n                    <jats:p>The authors reviewed archival histiocytic tumors at their institute and found a pediatric case of ALK-H localized in a cerebellar hemisphere that had previously been reported as histiocytic sarcoma. Chemotherapy (approximately 1 year), additional surgery, and conventional chemotherapy (approximately 2.5 years) led to clinical remission, and maintenance chemotherapy was continued (approximately 1.5 years). Three years after completing treatment, a high-grade glioma was found in a cerebral hemisphere, and the patient died of the glioma 2 years later.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>LESSONS<\/jats:title>\n                    <jats:p>Although the prognosis of ALK-H is generally good according to prior cases, the authors\u2019 case required long-term conventional chemotherapy, suggesting the tumor displayed aggressive characteristics. Early administration of ALK inhibitors may be necessary.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.3171\/case23466","type":"journal-article","created":{"date-parts":[[2024,5,27]],"date-time":"2024-05-27T08:02:00Z","timestamp":1716796920000},"source":"Crossref","is-referenced-by-count":2,"title":["Localized anaplastic lymphoma kinase\u2013positive histiocytosis in a cerebellar hemisphere with long-term treatment: illustrative case"],"prefix":"10.3171","volume":"7","author":[{"given":"Eita","family":"Kumagai","sequence":"first","affiliation":[{"name":"Department of Pathology, Yokohama City University Medical Center, Yokohama, Kanagawa, Japan;"},{"name":"Departments of Pathology"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Mio","family":"Tanaka","sequence":"additional","affiliation":[{"name":"Departments of Pathology"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Fuminori","family":"Iwasaki","sequence":"additional","affiliation":[{"name":"Hematology\/Oncology, and"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Masakatsu","family":"Yanagimachi","sequence":"additional","affiliation":[{"name":"Hematology\/Oncology, and"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Daisuke","family":"Hirokawa","sequence":"additional","affiliation":[{"name":"Neurosurgery, Kanagawa Children\u2019s Medical Center, Yokohama, Kanagawa, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Hiroaki","family":"Goto","sequence":"additional","affiliation":[{"name":"Hematology\/Oncology, and"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Yukichi","family":"Tanaka","sequence":"additional","affiliation":[{"name":"Departments of Pathology"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"1754","reference":[{"issue":"11","key":"b1","series-title":"Am J Surg Pathol.","first-page":"1372","article-title":"Primary histiocytic lymphoma of the central nervous system: a neoplasm frequently overshadowed by a prominent inflammatory component","volume":"25","author":"Cheuk W","year":"2001","unstructured":"Cheuk W, Walford N, Lou J, , et\u00a0al.. Primary histiocytic lymphoma of the central nervous system: a neoplasm frequently overshadowed by a prominent inflammatory component. Am J Surg Pathol. 2001;25(11):1372\u20131379."},{"issue":"9","key":"b2","series-title":"Am J Surg Pathol.","first-page":"1133","article-title":"Extranodal histiocytic sarcoma: clinicopathologic analysis of 14 cases of a rare epithelioid malignancy","volume":"28","author":"Hornick JL","year":"2004","unstructured":"Hornick JL, Jaffe ES, Fletcher CD Extranodal histiocytic sarcoma: clinicopathologic analysis of 14 cases of a rare epithelioid malignancy. Am J Surg Pathol. 2004;28(9):1133\u20131144."},{"issue":"5","key":"b3","series-title":"Mod Pathol.","first-page":"693","article-title":"Histiocytic sarcoma: a study of five cases including the histiocyte marker CD163","volume":"18","author":"Vos JA","year":"2005","unstructured":"Vos JA, Abbondanzo SL, Barekman CL, Andriko JW, Miettinen M, Aguilera NS Histiocytic sarcoma: a study of five cases including the histiocyte marker CD163. Mod Pathol. 2005;18(5):693\u2013704."},{"issue":"7","key":"b4","series-title":"Blood.","first-page":"2965","article-title":"ALK+ histiocytosis: a novel type of systemic histiocytic proliferative disorder of early infancy","volume":"112","author":"Chan JK","year":"2008","unstructured":"Chan JK, Lamant L, Algar E, , et\u00a0al.. ALK+ histiocytosis: a novel type of systemic histiocytic proliferative disorder of early infancy. Blood. 2008;112(7):2965\u20132968."},{"key":"b5","series-title":"Central Nervous System Tumours, Vol. 6: WHO Classification of Tumours Series","first-page":"349","article-title":"Haematolymphoid tumours involving the CNS: Introduction","author":"Soffietti R","year":"2021","unstructured":"Soffietti R Haematolymphoid tumours involving the CNS: Introduction. In: WHO Classification of Tumours Editorial Board, eds. Central Nervous System Tumours, Vol. 6: WHO Classification of Tumours Series, 5th ed. International Agency for Research on Center; 2021:349\u2013380."},{"issue":"2","key":"b6","series-title":"J Neurosurg Pediatr.","first-page":"126","article-title":"Primary cerebellar histiocytic sarcoma in a 17-month-old girl","volume":"10","author":"Gomi K","year":"2012","unstructured":"Gomi K, Tanaka M, Yoshida M, , et\u00a0al.. Primary cerebellar histiocytic sarcoma in a 17-month-old girl. J Neurosurg Pediatr. 2012;10(2):126\u2013129."},{"key":"b7","series-title":"Central Nervous System Tumours, Vol. 6: WHO Classification of Tumours Series","first-page":"77","article-title":"Diffuse paediatric-type high-grade glioma, H3-wildtype and IDH-wildtype","author":"Capper D","year":"2021","unstructured":"Capper D, Tabori U, Varlet P, Jones DTW Diffuse paediatric-type high-grade glioma, H3-wildtype and IDH-wildtype. In: WHO Classification of Tumours Editorial Board, eds. Central Nervous System Tumours, Vol. 6: WHO Classification of Tumours Series, 5th ed. International Agency for Research on Center; 2021:77\u201380."},{"issue":"2","key":"b8","series-title":"Blood.","first-page":"256","article-title":"ALK-positive\u00a0histiocytosis: a new clinicopathologic spectrum highlighting neurologic involvement and responses to ALK inhibition","volume":"139","author":"Kemps PG","year":"2022","unstructured":"Kemps PG, Picarsic J, Durham BH, , et\u00a0al.. ALK-positive\u00a0histiocytosis: a new clinicopathologic spectrum highlighting neurologic involvement and responses to ALK inhibition. Blood. 2022;139(2):256\u2013280."},{"issue":"6","key":"b9","series-title":"Neuropathol Appl Neurobiol.","first-page":"878","article-title":"ALK-rearranged histiocytosis: report of two cases with involvement of the central nervous system","volume":"47","author":"Rossi S","year":"2021","unstructured":"Rossi S, Gessi M, Barresi S, , et\u00a0al.. ALK-rearranged histiocytosis: report of two cases with involvement of the central nervous system. Neuropathol Appl Neurobiol. 2021;47(6):878\u2013881."},{"issue":"2","key":"b10","series-title":"Acta Neuropathol.","first-page":"335","article-title":"ALK-positive histiocytosis with KIF5B-ALK fusion in the central nervous system","volume":"138","author":"Lucas CG","year":"2019","unstructured":"Lucas CG, Gilani A, Solomon DA, , et\u00a0al.. ALK-positive histiocytosis with KIF5B-ALK fusion in the central nervous system. Acta Neuropathol. 2019;138(2):335\u2013337."},{"issue":"21","key":"b11","series-title":"Neurology.","first-page":"901","article-title":"Teaching NeuroImage: intradural ALK-positive histiocytosis with involvement of nerve roots and spinal dura","volume":"98","author":"Luo H","year":"2022","unstructured":"Luo H, Peng Y Teaching NeuroImage: intradural ALK-positive histiocytosis with involvement of nerve roots and spinal dura. Neurology. 2022;98(21):901\u2013902."},{"issue":"9","key":"b12","series-title":"Pediatr Blood Cancer.","first-page":"e30378","article-title":"Lorlatinib use in an infant with thalamic ALK-positive histiocytosis","volume":"70","author":"Eldem I","year":"2023","unstructured":"Eldem I, Picarsic J, Kumar A, , et\u00a0al.. Lorlatinib use in an infant with thalamic ALK-positive histiocytosis. Pediatr Blood Cancer. 2023;70(9):e30378."},{"issue":"11","key":"b1","series-title":"Am J Surg Pathol.","first-page":"1372","article-title":"Primary histiocytic lymphoma of the central nervous system: a neoplasm frequently overshadowed by a prominent inflammatory component","volume":"25","author":"Cheuk W","year":"2001","unstructured":"Cheuk W, Walford N, Lou J, , et\u00a0al.. Primary histiocytic lymphoma of the central nervous system: a neoplasm frequently overshadowed by a prominent inflammatory component. Am J Surg Pathol. 2001;25(11):1372\u20131379."},{"issue":"9","key":"b2","series-title":"Am J Surg Pathol.","first-page":"1133","article-title":"Extranodal histiocytic sarcoma: clinicopathologic analysis of 14 cases of a rare epithelioid malignancy","volume":"28","author":"Hornick JL","year":"2004","unstructured":"Hornick JL, Jaffe ES, Fletcher CD Extranodal histiocytic sarcoma: clinicopathologic analysis of 14 cases of a rare epithelioid malignancy. Am J Surg Pathol. 2004;28(9):1133\u20131144."},{"issue":"5","key":"b3","series-title":"Mod Pathol.","first-page":"693","article-title":"Histiocytic sarcoma: a study of five cases including the histiocyte marker CD163","volume":"18","author":"Vos JA","year":"2005","unstructured":"Vos JA, Abbondanzo SL, Barekman CL, Andriko JW, Miettinen M, Aguilera NS Histiocytic sarcoma: a study of five cases including the histiocyte marker CD163. Mod Pathol. 2005;18(5):693\u2013704."},{"issue":"7","key":"b4","series-title":"Blood.","first-page":"2965","article-title":"ALK+ histiocytosis: a novel type of systemic histiocytic proliferative disorder of early infancy","volume":"112","author":"Chan JK","year":"2008","unstructured":"Chan JK, Lamant L, Algar E, , et\u00a0al.. ALK+ histiocytosis: a novel type of systemic histiocytic proliferative disorder of early infancy. Blood. 2008;112(7):2965\u20132968."},{"key":"b5","series-title":"Central Nervous System Tumours, Vol. 6: WHO Classification of Tumours Series","first-page":"349","article-title":"Haematolymphoid tumours involving the CNS: Introduction","author":"Soffietti R","year":"2021","unstructured":"Soffietti R Haematolymphoid tumours involving the CNS: Introduction. In: WHO Classification of Tumours Editorial Board, eds. Central Nervous System Tumours, Vol. 6: WHO Classification of Tumours Series, 5th ed. International Agency for Research on Center; 2021:349\u2013380."},{"issue":"2","key":"b6","series-title":"J Neurosurg Pediatr.","first-page":"126","article-title":"Primary cerebellar histiocytic sarcoma in a 17-month-old girl","volume":"10","author":"Gomi K","year":"2012","unstructured":"Gomi K, Tanaka M, Yoshida M, , et\u00a0al.. Primary cerebellar histiocytic sarcoma in a 17-month-old girl. J Neurosurg Pediatr. 2012;10(2):126\u2013129."},{"key":"b7","series-title":"Central Nervous System Tumours, Vol. 6: WHO Classification of Tumours Series","first-page":"77","article-title":"Diffuse paediatric-type high-grade glioma, H3-wildtype and IDH-wildtype","author":"Capper D","year":"2021","unstructured":"Capper D, Tabori U, Varlet P, Jones DTW Diffuse paediatric-type high-grade glioma, H3-wildtype and IDH-wildtype. In: WHO Classification of Tumours Editorial Board, eds. Central Nervous System Tumours, Vol. 6: WHO Classification of Tumours Series, 5th ed. International Agency for Research on Center; 2021:77\u201380."},{"issue":"2","key":"b8","series-title":"Blood.","first-page":"256","article-title":"ALK-positive\u00a0histiocytosis: a new clinicopathologic spectrum highlighting neurologic involvement and responses to ALK inhibition","volume":"139","author":"Kemps PG","year":"2022","unstructured":"Kemps PG, Picarsic J, Durham BH, , et\u00a0al.. ALK-positive\u00a0histiocytosis: a new clinicopathologic spectrum highlighting neurologic involvement and responses to ALK inhibition. Blood. 2022;139(2):256\u2013280."},{"issue":"6","key":"b9","series-title":"Neuropathol Appl Neurobiol.","first-page":"878","article-title":"ALK-rearranged histiocytosis: report of two cases with involvement of the central nervous system","volume":"47","author":"Rossi S","year":"2021","unstructured":"Rossi S, Gessi M, Barresi S, , et\u00a0al.. ALK-rearranged histiocytosis: report of two cases with involvement of the central nervous system. Neuropathol Appl Neurobiol. 2021;47(6):878\u2013881."},{"issue":"2","key":"b10","series-title":"Acta Neuropathol.","first-page":"335","article-title":"ALK-positive histiocytosis with KIF5B-ALK fusion in the central nervous system","volume":"138","author":"Lucas CG","year":"2019","unstructured":"Lucas CG, Gilani A, Solomon DA, , et\u00a0al.. ALK-positive histiocytosis with KIF5B-ALK fusion in the central nervous system. Acta Neuropathol. 2019;138(2):335\u2013337."},{"issue":"21","key":"b11","series-title":"Neurology.","first-page":"901","article-title":"Teaching NeuroImage: intradural ALK-positive histiocytosis with involvement of nerve roots and spinal dura","volume":"98","author":"Luo H","year":"2022","unstructured":"Luo H, Peng Y Teaching NeuroImage: intradural ALK-positive histiocytosis with involvement of nerve roots and spinal dura. Neurology. 2022;98(21):901\u2013902."},{"issue":"9","key":"b12","series-title":"Pediatr Blood Cancer.","first-page":"e30378","article-title":"Lorlatinib use in an infant with thalamic ALK-positive histiocytosis","volume":"70","author":"Eldem I","year":"2023","unstructured":"Eldem I, Picarsic J, Kumar A, , et\u00a0al.. Lorlatinib use in an infant with thalamic ALK-positive histiocytosis. Pediatr Blood Cancer. 2023;70(9):e30378."}],"container-title":["Journal of Neurosurgery: Case Lessons"],"original-title":[],"link":[{"URL":"https:\/\/thejns.org\/view\/journals\/j-neurosurg-case-lessons\/7\/22\/article-CASE23466.xml","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/thejns.org\/downloadpdf\/journals\/j-neurosurg-case-lessons\/7\/22\/article-CASE23466.xml","content-type":"text\/html","content-version":"vor","intended-application":"syndication"},{"URL":"https:\/\/thejns.org\/downloadpdf\/journals\/j-neurosurg-case-lessons\/7\/22\/article-CASE23466.xml","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,2,5]],"date-time":"2026-02-05T14:26:02Z","timestamp":1770301562000},"score":1,"resource":{"primary":{"URL":"https:\/\/thejns.org\/view\/journals\/j-neurosurg-case-lessons\/7\/22\/article-CASE23466.xml"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2024,5,27]]},"references-count":24,"journal-issue":{"issue":"22"},"URL":"https:\/\/doi.org\/10.3171\/case23466","relation":{},"ISSN":["2694-1902"],"issn-type":[{"value":"2694-1902","type":"electronic"}],"subject":[],"published":{"date-parts":[[2024,5,27]]}}}