Response of metastatic recurrent neuroblastoma to nitisinone: A modulator of tyrosine metabolism
Nitisinone blocks the tyrosine pathway and may be effective in treating neuroblastoma. A 33‐month‐old male with heavily treated metastatic, recurrent, N‐MYC amplified neuroblastoma received nitsinone (0.8 mg/kg/day escalated to 5.0 mg/kg/day). Dramatic tumor regression and resolution of pain without...
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| Veröffentlicht in: | Pediatric Blood & Cancer 2006-04, Vol.46 (4), p.517-520 |
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| creator | Kobrinsky, Nathan L. Sjolander, Diane E. |
| description | Nitisinone blocks the tyrosine pathway and may be effective in treating neuroblastoma. A 33‐month‐old male with heavily treated metastatic, recurrent, N‐MYC amplified neuroblastoma received nitsinone (0.8 mg/kg/day escalated to 5.0 mg/kg/day). Dramatic tumor regression and resolution of pain without toxicity were observed. At 10 weeks, the tumor progressed. Nitisinone, low dose cyclophosphamide and doxorubicin subsequently produced a very good partial response. At 18 months the disease progressed. The child succumbed 21 months after starting nitisinone. Nitisinone produced an increase in tyrosine and catecholamine metabolite (HVA, VMA, and metanephrines) levels. Nitisinone may be a promising agent in metastatic neuroblastoma. Pediatr Blood Cancer 2006, 46:517–520. © 2005 Wiley‐Liss, Inc. |
| doi_str_mv | 10.1002/pbc.20420 |
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A 33‐month‐old male with heavily treated metastatic, recurrent, N‐MYC amplified neuroblastoma received nitsinone (0.8 mg/kg/day escalated to 5.0 mg/kg/day). Dramatic tumor regression and resolution of pain without toxicity were observed. At 10 weeks, the tumor progressed. Nitisinone, low dose cyclophosphamide and doxorubicin subsequently produced a very good partial response. At 18 months the disease progressed. The child succumbed 21 months after starting nitisinone. Nitisinone produced an increase in tyrosine and catecholamine metabolite (HVA, VMA, and metanephrines) levels. Nitisinone may be a promising agent in metastatic neuroblastoma. 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Blood Cancer</addtitle><description>Nitisinone blocks the tyrosine pathway and may be effective in treating neuroblastoma. A 33‐month‐old male with heavily treated metastatic, recurrent, N‐MYC amplified neuroblastoma received nitsinone (0.8 mg/kg/day escalated to 5.0 mg/kg/day). Dramatic tumor regression and resolution of pain without toxicity were observed. At 10 weeks, the tumor progressed. Nitisinone, low dose cyclophosphamide and doxorubicin subsequently produced a very good partial response. At 18 months the disease progressed. The child succumbed 21 months after starting nitisinone. Nitisinone produced an increase in tyrosine and catecholamine metabolite (HVA, VMA, and metanephrines) levels. Nitisinone may be a promising agent in metastatic neuroblastoma. Pediatr Blood Cancer 2006, 46:517–520. © 2005 Wiley‐Liss, Inc.</description><subject>Antineoplastic Agents - adverse effects</subject><subject>Antineoplastic Agents - therapeutic use</subject><subject>Antineoplastic Combined Chemotherapy Protocols - therapeutic use</subject><subject>Biological and medical sciences</subject><subject>Child, Preschool</subject><subject>Combined Modality Therapy</subject><subject>Cyclohexanones - adverse effects</subject><subject>Cyclohexanones - therapeutic use</subject><subject>Disease Progression</subject><subject>Dose-Response Relationship, Drug</subject><subject>Drug Administration Schedule</subject><subject>Enzyme Inhibitors - adverse effects</subject><subject>Enzyme Inhibitors - therapeutic use</subject><subject>Fatal Outcome</subject><subject>General aspects</subject><subject>Humans</subject><subject>HVA</subject><subject>Male</subject><subject>Medical sciences</subject><subject>metanephrine</subject><subject>Neoplasm Recurrence, Local - drug therapy</subject><subject>neuroblastoma</subject><subject>Neuroblastoma - drug therapy</subject><subject>Neuroblastoma - secondary</subject><subject>nitisinone</subject><subject>Nitrobenzoates - adverse effects</subject><subject>Nitrobenzoates - therapeutic use</subject><subject>Remission Induction</subject><subject>Treatment Outcome</subject><subject>Tumors</subject><subject>tyrosine</subject><subject>Tyrosine - drug effects</subject><subject>Tyrosine - metabolism</subject><subject>VMA</subject><issn>1545-5009</issn><issn>1545-5017</issn><issn>1096-911X</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2006</creationdate><recordtype>article</recordtype><sourceid>EIF</sourceid><recordid>eNp1kE1vFDEMhiNERUvhwB9Ac6ESh2mdZDIf3MoKlkrVghCox-DJOFJgZrIkGZX996TdpT1hybIlP35tvYy94nDOAcTFtjfnAioBT9gJV5UqFfDm6UMP3TF7HuPPjNag2mfsmKuugkaoE_bjK8WtnyMV3hYTJYwJkzNFILOEQHMqZlqC78c88BMWyRezSy662c_0rrgsJj8sIyYf7gTSLvg8onul3o8uTi_YkcUx0stDPWXfP374tvpUXn9eX60ur0tTCQGllQI7tBzQdt0wYEuDgCGHtbWqlTRd1wqsULQ9r9AASCUMl0KKhnegennKzva62-B_LxSTnlw0NI44k1-iFlBzLmWdwbd70ORfYyCrt8FNGHaag76zU2c79b2dmX19EF36iYZH8uBfBt4cAIwGRxtwNi4-co1qaw4ycxd77taNtPv_Rf3l_erf6XK_4WKiPw8bGH7pupGN0jebdc7NenMjQYP8CzCum_w</recordid><startdate>200604</startdate><enddate>200604</enddate><creator>Kobrinsky, Nathan L.</creator><creator>Sjolander, Diane E.</creator><general>Wiley Subscription Services, Inc., A Wiley Company</general><general>Wiley</general><scope>BSCLL</scope><scope>IQODW</scope><scope>CGR</scope><scope>CUY</scope><scope>CVF</scope><scope>ECM</scope><scope>EIF</scope><scope>NPM</scope><scope>AAYXX</scope><scope>CITATION</scope><scope>7TK</scope></search><sort><creationdate>200604</creationdate><title>Response of metastatic recurrent neuroblastoma to nitisinone: A modulator of tyrosine metabolism</title><author>Kobrinsky, Nathan L. ; 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Blood Cancer</addtitle><date>2006-04</date><risdate>2006</risdate><volume>46</volume><issue>4</issue><spage>517</spage><epage>520</epage><pages>517-520</pages><issn>1545-5009</issn><eissn>1545-5017</eissn><eissn>1096-911X</eissn><abstract>Nitisinone blocks the tyrosine pathway and may be effective in treating neuroblastoma. A 33‐month‐old male with heavily treated metastatic, recurrent, N‐MYC amplified neuroblastoma received nitsinone (0.8 mg/kg/day escalated to 5.0 mg/kg/day). Dramatic tumor regression and resolution of pain without toxicity were observed. At 10 weeks, the tumor progressed. Nitisinone, low dose cyclophosphamide and doxorubicin subsequently produced a very good partial response. At 18 months the disease progressed. The child succumbed 21 months after starting nitisinone. Nitisinone produced an increase in tyrosine and catecholamine metabolite (HVA, VMA, and metanephrines) levels. Nitisinone may be a promising agent in metastatic neuroblastoma. 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| subjects | Antineoplastic Agents - adverse effects Antineoplastic Agents - therapeutic use Antineoplastic Combined Chemotherapy Protocols - therapeutic use Biological and medical sciences Child, Preschool Combined Modality Therapy Cyclohexanones - adverse effects Cyclohexanones - therapeutic use Disease Progression Dose-Response Relationship, Drug Drug Administration Schedule Enzyme Inhibitors - adverse effects Enzyme Inhibitors - therapeutic use Fatal Outcome General aspects Humans HVA Male Medical sciences metanephrine Neoplasm Recurrence, Local - drug therapy neuroblastoma Neuroblastoma - drug therapy Neuroblastoma - secondary nitisinone Nitrobenzoates - adverse effects Nitrobenzoates - therapeutic use Remission Induction Treatment Outcome Tumors tyrosine Tyrosine - drug effects Tyrosine - metabolism VMA |
| title | Response of metastatic recurrent neuroblastoma to nitisinone: A modulator of tyrosine metabolism |
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