{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,29]],"date-time":"2026-09-29T17:22:32Z","timestamp":1790702552735,"version":"4.1.0"},"reference-count":30,"publisher":"Wiley","issue":"24","license":[{"start":{"date-parts":[[2020,9,11]],"date-time":"2020-09-11T00:00:00Z","timestamp":1599782400000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"funder":[{"DOI":"10.13039\/100006058","name":"St. Baldrick's Foundation","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100006058","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["acsjournals.onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Cancer"],"published-print":{"date-parts":[[2020,12,15]]},"abstract":"<jats:sec>\n                    <jats:title>Background<\/jats:title>\n                    <jats:p>Lorvotuzumab mertansine (IMGN901) is an antibody\u2010drug conjugate linking an antimitotic agent (DM1) to an anti\u2010CD56 antibody (lorvotuzumab). Preclinical efficacy has been noted in Wilms tumor, rhabdomyosarcoma, and neuroblastoma. Synovial sarcoma, malignant peripheral nerve sheath tumor (MPNST), and pleuropulmonary blastoma also express CD56. A phase 2 trial of lorvotuzumab mertansine was conducted to assess its efficacy, recommended phase 2 dose, and toxicities.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>\n                      Eligible patients had relapsed after or progressed on standard therapy for their tumor type. Lorvotuzumab mertansine (110 mg\/m\n                      <jats:sup>2<\/jats:sup>\n                      per dose) was administered at the adult recommended phase 2 dose intravenously on days 1 and 8 of 21\u2010day cycles. Dexamethasone premedication was used. Pharmacokinetic samples, peripheral blood CD56\u2010positive cell counts, and tumor CD56 expression were assessed.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>Sixty\u2010two patients enrolled. The median age was 14.3 years (range, 2.8\u201029.9 years); 35 were male. Diagnoses included Wilms tumor (n\u00a0=\u00a017), rhabdomyosarcoma (n\u00a0=\u00a017), neuroblastoma (n\u00a0=\u00a012), synovial sarcoma (n\u00a0=\u00a010), MPNST (n\u00a0=\u00a05), and pleuropulmonary blastoma (n\u00a0=\u00a01). Five patients experienced 9 dose\u2010limiting toxicities: hyperglycemia (n\u00a0=\u00a01), colonic fistula (n\u00a0=\u00a01) with perforation (n\u00a0=\u00a01), nausea (n\u00a0=\u00a01) with vomiting (n\u00a0=\u00a01), increased alanine aminotransferase in cycle 1 (n\u00a0=\u00a02), and increased alanine aminotransferase in cycle 2 (n\u00a0=\u00a01) with increased aspartate aminotransferase (n\u00a0=\u00a01). Non\u2013dose\u2010limiting toxicities (grade 3 or higher) attributed to lorvotuzumab mertansine were rare. The median values of the maximum concentration, half\u2010life, and area under the curve from zero to infinity for DM1 were 0.87 \u00b5g\/mL, 35 hours, and 27.9 \u00b5g\/mL h, respectively. Peripheral blood CD56+ leukocytes decreased by 71.9% on day 8. One patient with rhabdomyosarcoma had a partial response, and 1 patient with synovial sarcoma achieved a delayed complete response.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusions<\/jats:title>\n                    <jats:p>\n                      Lorvotuzumab mertansine (110 mg\/m\n                      <jats:sup>2<\/jats:sup>\n                      ) is tolerated in children at the adult recommended phase 2 dose; clinical activity is limited.\n                    <\/jats:p>\n                  <\/jats:sec>","DOI":"10.1002\/cncr.33195","type":"journal-article","created":{"date-parts":[[2020,9,22]],"date-time":"2020-09-22T04:01:52Z","timestamp":1600747312000},"page":"5303-5310","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":37,"title":["ADVL1522: A phase 2 study of lorvotuzumab mertansine (IMGN901) in children with relapsed or refractory wilms tumor, rhabdomyosarcoma, neuroblastoma, pleuropulmonary blastoma, malignant peripheral nerve sheath tumor, or synovial sarcoma\u2014A Children's Oncology Group study"],"prefix":"10.1002","volume":"126","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-5181-116X","authenticated-orcid":false,"given":"James I.","family":"Geller","sequence":"first","affiliation":[{"name":"Division of Oncology Cincinnati Children's Hospital Medical Center, University of Cincinnati Cincinnati Ohio"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Joseph G.","family":"Pressey","sequence":"additional","affiliation":[{"name":"Division of Oncology Cincinnati Children's Hospital Medical Center, University of Cincinnati Cincinnati Ohio"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-9880-9876","authenticated-orcid":false,"given":"Malcolm A.","family":"Smith","sequence":"additional","affiliation":[{"name":"Cancer Therapy Evaluation Program National Institutes of Health Bethesda Maryland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Rachel A.","family":"Kudgus","sequence":"additional","affiliation":[{"name":"Mayo Clinic Comprehensive Cancer Center Rochester Minnesota"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mariana","family":"Cajaiba","sequence":"additional","affiliation":[{"name":"Lurie Children's Hospital of Chicago Chicago Illinois"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Joel M.","family":"Reid","sequence":"additional","affiliation":[{"name":"Mayo Clinic Comprehensive Cancer Center Rochester Minnesota"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"David","family":"Hall","sequence":"additional","affiliation":[{"name":"Children's Oncology Group Monrovia California"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Donald A.","family":"Barkauskas","sequence":"additional","affiliation":[{"name":"Keck School of Medicine, University of Southern California Los Angeles California"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Stephen D.","family":"Voss","sequence":"additional","affiliation":[{"name":"Dana\u2010Farber Cancer Institute Boston Massachusetts"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Steve Y.","family":"Cho","sequence":"additional","affiliation":[{"name":"University of Wisconsin Hospital and Clinics Madison Wisconsin"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Stacey L.","family":"Berg","sequence":"additional","affiliation":[{"name":"Dan L. Duncan Comprehensive Cancer Center Baylor College of Medicine Houston Texas"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jeffrey S.","family":"Dome","sequence":"additional","affiliation":[{"name":"Children's National Medical Center Washington DC"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Elizabeth","family":"Fox","sequence":"additional","affiliation":[{"name":"Children's Hospital of Philadelphia Philadelphia Pennsylvania"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Brenda J.","family":"Weigel","sequence":"additional","affiliation":[{"name":"University of Minnesota Medical Center Minneapolis 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