{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,27]],"date-time":"2026-10-27T13:13:25Z","timestamp":1793106805997,"version":"4.5.0"},"reference-count":51,"publisher":"The Endocrine Society","issue":"2","license":[{"start":{"date-parts":[[2020,11,25]],"date-time":"2020-11-25T00:00:00Z","timestamp":1606262400000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/academic.oup.com\/journals\/pages\/open_access\/funder_policies\/chorus\/standard_publication_model"}],"funder":[{"DOI":"10.13039\/100012524","name":"American Lebanese Syrian Associated Charities","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100012524","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100000024","name":"Canadian Institutes of Health Research","doi-asserted-by":"publisher","award":["FDN-148390"],"award-info":[{"award-number":["FDN-148390"]}],"id":[{"id":"10.13039\/501100000024","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2021,1,23]]},"abstract":"<jats:title>Abstract<\/jats:title>\n               <jats:sec>\n                  <jats:title>Context<\/jats:title>\n                  <jats:p>Pituitary blastoma is a rare, dysontogenetic hypophyseal tumor of infancy first described in 2008, strongly suggestive of DICER1 syndrome.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Objective<\/jats:title>\n                  <jats:p>This work aims to describe genetic alterations, clinical courses, outcomes, and complications in all known pituitary blastoma cases.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Design and Setting<\/jats:title>\n                  <jats:p>A multi-institutional case series is presented from tertiary pediatric oncology centers.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Patients<\/jats:title>\n                  <jats:p>Patients included children with pituitary blastoma.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Interventions<\/jats:title>\n                  <jats:p>Genetic testing, surgery, oncologic therapy, endocrine support are reported.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Outcome Measures<\/jats:title>\n                  <jats:p>Outcome measures included survival, long-term morbidities, and germline and tumor DICER1 genotypes.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Results<\/jats:title>\n                  <jats:p>Seventeen pituitary blastoma cases were studied (10 girls and 7 boys); median age at diagnosis was 11 months (range, 2-24 months). Cushing syndrome was the most frequent presentation (n = 10). Cushingoid stigmata were absent in 7 children (2 with increased adrenocorticotropin [ACTH]; 5 with normal\/unmeasured ACTH). Ophthalmoplegia and increased intracranial pressure were also observed. Surgical procedures included gross\/near-total resection (n = 7), subtotal resection (n = 9), and biopsy (n = 1). Six children received adjuvant therapy. At a median follow-up of 6.7 years, 9 patients were alive; 8 patients died of the following causes: early medical\/surgical complications (n = 3), sepsis (n = 1), catheter-related complication (n = 1), aneurysmal bleeding (n = 1), second brain tumor (n = 1), and progression (n = 1). Surgery was the only intervention for 5 of 9 survivors. Extent of resection, but neither Ki67 labeling index nor adjuvant therapy, was significantly associated with survival. Chronic complications included neuroendocrine (n = 8), visual (n = 4), and neurodevelopmental (n = 3) deficits. Sixteen pituitary blastomas were attributed to DICER1 abnormalities.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Conclusions<\/jats:title>\n                  <jats:p>Pituitary blastoma is a locally destructive tumor associated with high mortality. Surgical resection alone provides long-term disease control for some patients. Quality survival is possible with long-term neuroendocrine management.<\/jats:p>\n               <\/jats:sec>","DOI":"10.1210\/clinem\/dgaa857","type":"journal-article","created":{"date-parts":[[2020,11,25]],"date-time":"2020-11-25T08:48:27Z","timestamp":1606294107000},"page":"351-363","source":"Crossref","is-referenced-by-count":41,"title":["Clinical Outcomes and Complications of Pituitary Blastoma"],"prefix":"10.1210","volume":"106","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-8597-9128","authenticated-orcid":false,"given":"Anthony P Y","family":"Liu","sequence":"first","affiliation":[{"name":"Division of Neuro-Oncology, Department of Oncology, St. Jude Children\u2019s Research Hospital, Memphis, Tennessee, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Megan M","family":"Kelsey","sequence":"additional","affiliation":[{"name":"Department of Pediatrics, Section of Pediatric Endocrinology, Children\u2019s Hospital Colorado and University of Colorado Anschutz Medical Campus, Aurora, Colorado, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nelly","family":"Sabbaghian","sequence":"additional","affiliation":[{"name":"Department of Medical Genetics, The Lady Davis Institute, Segal Cancer Centre, Jewish General Hospital, Montreal, Quebec, Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sung-Hye","family":"Park","sequence":"additional","affiliation":[{"name":"Department of Pathology, Seoul National University, College of Medicine, Seoul, Republic of Korea"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Cheri L","family":"Deal","sequence":"additional","affiliation":[{"name":"Endocrinology and Diabetes Service, CHU-Sainte Justine and Department of Pediatrics, University of Montreal, Montreal, Quebec, Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Adam J","family":"Esbenshade","sequence":"additional","affiliation":[{"name":"Department of Pediatrics, Monroe Carell Jr. Children\u2019s Hospital, Nashville, Tennessee, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Oswald","family":"Ploner","sequence":"additional","affiliation":[{"name":"Diakonie-Klinikum, Stuttgart, Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Andrew","family":"Peet","sequence":"additional","affiliation":[{"name":"Institute of Cancer and Genomic Sciences, University of Birmingham, Birmingham, UK"},{"name":"Birmingham Children\u2019s Hospital NHS Foundation Trust, Birmingham, UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Heidi","family":"Traunecker","sequence":"additional","affiliation":[{"name":"Noah\u2019s Ark Children\u2019s Hospital for Wales, Cardiff, UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yomna H E","family":"Ahmed","sequence":"additional","affiliation":[{"name":"Royal College of Surgeons in Ireland-Bahrain, Bahrain"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-5562-6818","authenticated-orcid":false,"given":"Margaret","family":"Zacharin","sequence":"additional","affiliation":[{"name":"Department of Endocrinology and Diabetes, Royal Children\u2019s Hospital, Parkville, Melbourne, Victoria, Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Anatoly","family":"Tiulpakov","sequence":"additional","affiliation":[{"name":"Department and Laboratory of Inherited Endocrine Disorders, Endocrinology Research Centre, Moscow, Russia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Anastasia M","family":"Lapshina","sequence":"additional","affiliation":[{"name":"Department of Fundamental Pathomorphology, Endocrinology Research Centre, Moscow, Russia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Andrew W","family":"Walter","sequence":"additional","affiliation":[{"name":"duPont Hospital for Children, Wilmington, Delaware, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Pinaki","family":"Dutta","sequence":"additional","affiliation":[{"name":"Department of Endocrinology, Post Graduate Institute of Medical Education and Research, Chandigarh, India"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ashutosh","family":"Rai","sequence":"additional","affiliation":[{"name":"Department of Endocrinology, Post Graduate Institute of Medical Education and Research, Chandigarh, India"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"M\u00e1rta","family":"Korbonits","sequence":"additional","affiliation":[{"name":"Department of Endocrinology, Barts and the London School of Medicine, Queen Mary University of London, London, UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Leanne","family":"de Kock","sequence":"additional","affiliation":[{"name":"Harry Perkins Institute of Medical Research, QEII Medical Centre and UWA Centre for Medical Research, the University of Western Australia, Perth, Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kim E","family":"Nichols","sequence":"additional","affiliation":[{"name":"Division of Cancer Predisposition, Department of Oncology, St. Jude Children\u2019s Research Hospital, Memphis, Tennessee, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-7427-4651","authenticated-orcid":false,"given":"William D","family":"Foulkes","sequence":"additional","affiliation":[{"name":"Department of Medical Genetics, The Lady Davis Institute, Segal Cancer Centre, Jewish General Hospital, Montreal, Quebec, Canada"},{"name":"Department of Medical Genetics and Cancer Research Program, Research Institute McGill University Health Centre, Montreal, Quebec, Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"John R","family":"Priest","sequence":"additional","affiliation":[{"name":"Minneapolis, Minnesota, 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