{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,6]],"date-time":"2026-10-06T14:44:43Z","timestamp":1791297883014,"version":"4.1.0"},"reference-count":27,"publisher":"The Endocrine Society","issue":"1","content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2018,1,1]]},"abstract":"<jats:title>Abstract<\/jats:title>\n               <jats:sec>\n                  <jats:title>Context<\/jats:title>\n                  <jats:p>No therapies are approved for the treatment of metastatic and\/or recurrent pheochromocytoma or paraganglioma (PPGL) in the United States.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Objective<\/jats:title>\n                  <jats:p>To determine the maximum tolerated dose (MTD) of high-specific-activity I-131 meta-iodobenzylguanidine (MIBG) for the treatment of metastatic and\/or recurrent PPGL.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Design<\/jats:title>\n                  <jats:p>Phase 1, dose-escalating study to determine the MTD via a standard 3 + 3 design, escalating by 37 MBq\/kg starting at 222 MBq\/kg.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Setting<\/jats:title>\n                  <jats:p>Three centers.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Patients<\/jats:title>\n                  <jats:p>Twenty-one patients were eligible, received study drug, and were evaluable for MTD, response, and toxicity.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Intervention<\/jats:title>\n                  <jats:p>Open-label use of high-specific-activity I-131 MIBG therapy.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Main Outcome Measures<\/jats:title>\n                  <jats:p>Dose-limiting toxicities, adverse events, radiation absorbed dose estimates, radiographic tumor response, biochemical response, and survival.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Results<\/jats:title>\n                  <jats:p>The MTD was determined to be 296 MBq\/kg on the basis of two observed dose-limiting toxicities at the next dose level. The highest mean radiation absorbed dose estimates were in the thyroid and lower large intestinal wall (each 1.2 mGy\/MBq). Response was evaluated by total administered activity: four patients (19%), all of whom received &amp;gt;18.5 GBq of study drug, had radiographic tumor responses of partial response by Response Evaluation Criteria in Solid Tumors. Best biochemical responses (complete or partial response) for serum chromogranin A and total metanephrines were observed in 80% and 64% of patients, respectively. Overall survival was 85.7% at 1 year and 61.9% at 2 years after treatment. The majority (84%) of adverse events were considered mild or moderate in severity.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Conclusions<\/jats:title>\n                  <jats:p>These findings support further development of high-specific-activity I-131 MIBG for the treatment of metastatic and\/or recurrent PPGL at an MTD of 296 MBq\/kg.<\/jats:p>\n               <\/jats:sec>","DOI":"10.1210\/jc.2017-02030","type":"journal-article","created":{"date-parts":[[2017,11,1]],"date-time":"2017-11-01T21:59:33Z","timestamp":1509573573000},"page":"213-220","source":"Crossref","is-referenced-by-count":64,"title":["Phase 1 Study of High-Specific-Activity I-131 MIBG for Metastatic and\/or Recurrent Pheochromocytoma or Paraganglioma"],"prefix":"10.1210","volume":"103","author":[{"given":"Richard B","family":"Noto","sequence":"first","affiliation":[{"name":"Department of Diagnostic Imaging, Warren Alpert Medical School of Brown University, Providence, Rhode Island"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Daniel A","family":"Pryma","sequence":"additional","affiliation":[{"name":"Department of Radiology, University of Pennsylvania Health System, Philadelphia, Pennsylvania"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jessica","family":"Jensen","sequence":"additional","affiliation":[{"name":"Progenics Pharmaceuticals, Inc., New York, New York"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Tess","family":"Lin","sequence":"additional","affiliation":[{"name":"Progenics Pharmaceuticals, Inc., New York, New York"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nancy","family":"Stambler","sequence":"additional","affiliation":[{"name":"Progenics Pharmaceuticals, Inc., New York, New York"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Thomas","family":"Strack","sequence":"additional","affiliation":[{"name":"Progenics Pharmaceuticals, Inc., New York, New York"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Vivien","family":"Wong","sequence":"additional","affiliation":[{"name":"Progenics Pharmaceuticals, Inc., New York, New York"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Stanley J","family":"Goldsmith","sequence":"additional","affiliation":[{"name":"Radiology and Medicine, Weill Medical College of Cornell University, New York, New York"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"80","published-online":{"date-parts":[[2017,11,1]]},"reference":[{"key":"2020071700353167300_B1","unstructured":"National Institutes of Health\n          , National Cancer Institute. 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