Gonadal dysgenesis is associated with worse outcomes in patients with ovarian nondysgerminomatous tumors: A report of the Children's Oncology Group AGCT 0132 study
Purpose In this report, we characterize the timing and behavior of malignant ovarian germ cell tumors (GCTs) in pediatric patients with dysgenetic gonads compared to those with normal gonadal development. Patients and methods Patients from the Children's Oncology Group AGCT0132 with malignant o...
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creator | Dicken, Bryan J. Billmire, Deborah F. Krailo, Mark Xia, Caihong Shaikh, Furqan Cullen, John W. Olson, Thomas A. Pashankar, Farzana Malogolowkin, Marcio H. Amatruda, James F. Rescorla, Frederick J. Egler, Rachel A. Ross, Jonathan H. Rodriguez‐Galindo, Carlos Frazier, A. Lindsay |
description | Purpose
In this report, we characterize the timing and behavior of malignant ovarian germ cell tumors (GCTs) in pediatric patients with dysgenetic gonads compared to those with normal gonadal development.
Patients and methods
Patients from the Children's Oncology Group AGCT0132 with malignant ovarian GCTs were included. Within this population, we sought to identify patients with gonadoblastoma, streak ovaries, or other evidence of gonadal dysgenesis (GD). Patients with malignant GCTs containing one or more of the following histologies—yolk sac tumor, embryonal carcinoma, or choriocarcinoma—were included. Patients were compared with respect to event‐free survival (EFS) and overall survival (OS).
Results
Nine patients with GD, including seven with gonadoblastoma (mean age, 9.3 years), were compared to 100 non‐GD patients (mean age, 12.1 years). The estimated 3‐year EFS for patients with GD was 66.7% (95% CI 28.2–87.8%) and for non‐GD patients was 88.8% (95% CI 80.2–93.8%). The estimated 3‐year OS for patients with GD was 87.5% (95% CI 38.7–98.1%) and for non‐GD patients was 97.6% (95% CI of 90.6–99.4%).
Conclusion
Patients presenting with nongerminomatous malignant ovarian GCTs in the context of GD have a higher rate of events and death than counterparts with normal gonads. These findings emphasize the importance of noting a contralateral streak ovary or gonadoblastoma at histology for any ovarian GCT and support the recommendation for early bilateral gonadectomy in patients known to have GD with Y chromosome material. In contrast to those with pure dysgerminoma, these patients may represent a high‐risk group that requires a more aggressive chemotherapy regimen. |
doi_str_mv | 10.1002/pbc.26913 |
format | Article |
fullrecord | <record><control><sourceid>proquest_pubme</sourceid><recordid>TN_cdi_pubmedcentral_primary_oai_pubmedcentral_nih_gov_6219870</recordid><sourceformat>XML</sourceformat><sourcesystem>PC</sourcesystem><sourcerecordid>2006812460</sourcerecordid><originalsourceid>FETCH-LOGICAL-c4433-52b398b7aad342f512d31ed3a2fb631fe2a5f6132ae7ddbeda32bc7e563e68683</originalsourceid><addsrcrecordid>eNp1kd9r1TAUx4sobk4f_Ack4MP04W750aStD8K16HUwmA_zOaTN6b0ZbVKTdJf-PftHzdZ5UWEQSOB88uGc882ytwSfEYzp-di0Z1RUhD3LjgnP-YpjUjw_vHF1lL0K4SahAvPyZXZEK1oKzvlxdrdxVmnVIz2HLVgIJqB0VAiuNSqCRnsTd2jvfADkpti6ARJh0aiiARvDUne3yhtlkXX2QeQHY92gopsCitOQfn9Ca-RhdD4i16G4A1TvTK892NOArmzrered0ca7aUTrTX2NMGEUhTjp-XX2olN9gDeP90n289vX6_r76vJqc1GvL1dtnjO24rRhVdkUSmmW044TqhkBzRTtGsFIB1TxTiSrgkLrBrRitGkL4IKBKEXJTrLPi3ecmgF0m8bzqpejN4Pys3TKyH8r1uzk1t1KQUlVFjgJPjwKvPs1QYhyMKGFvlcW0iZkomiZM8FpQt__h964yds0nqQYi5LQXNwLPy5U610IHrpDMwTL--hlil4-RJ_Yd393fyD_ZJ2A8wXYmx7mp03yx5d6Uf4GN5a79g</addsrcrecordid><sourcetype>Open Access Repository</sourcetype><iscdi>true</iscdi><recordtype>article</recordtype><pqid>2006812460</pqid></control><display><type>article</type><title>Gonadal dysgenesis is associated with worse outcomes in patients with ovarian nondysgerminomatous tumors: A report of the Children's Oncology Group AGCT 0132 study</title><source>Access via Wiley Online Library</source><creator>Dicken, Bryan J. ; Billmire, Deborah F. ; Krailo, Mark ; Xia, Caihong ; Shaikh, Furqan ; Cullen, John W. ; Olson, Thomas A. ; Pashankar, Farzana ; Malogolowkin, Marcio H. ; Amatruda, James F. ; Rescorla, Frederick J. ; Egler, Rachel A. ; Ross, Jonathan H. ; Rodriguez‐Galindo, Carlos ; Frazier, A. Lindsay</creator><creatorcontrib>Dicken, Bryan J. ; Billmire, Deborah F. ; Krailo, Mark ; Xia, Caihong ; Shaikh, Furqan ; Cullen, John W. ; Olson, Thomas A. ; Pashankar, Farzana ; Malogolowkin, Marcio H. ; Amatruda, James F. ; Rescorla, Frederick J. ; Egler, Rachel A. ; Ross, Jonathan H. ; Rodriguez‐Galindo, Carlos ; Frazier, A. Lindsay</creatorcontrib><description>Purpose
In this report, we characterize the timing and behavior of malignant ovarian germ cell tumors (GCTs) in pediatric patients with dysgenetic gonads compared to those with normal gonadal development.
Patients and methods
Patients from the Children's Oncology Group AGCT0132 with malignant ovarian GCTs were included. Within this population, we sought to identify patients with gonadoblastoma, streak ovaries, or other evidence of gonadal dysgenesis (GD). Patients with malignant GCTs containing one or more of the following histologies—yolk sac tumor, embryonal carcinoma, or choriocarcinoma—were included. Patients were compared with respect to event‐free survival (EFS) and overall survival (OS).
Results
Nine patients with GD, including seven with gonadoblastoma (mean age, 9.3 years), were compared to 100 non‐GD patients (mean age, 12.1 years). The estimated 3‐year EFS for patients with GD was 66.7% (95% CI 28.2–87.8%) and for non‐GD patients was 88.8% (95% CI 80.2–93.8%). The estimated 3‐year OS for patients with GD was 87.5% (95% CI 38.7–98.1%) and for non‐GD patients was 97.6% (95% CI of 90.6–99.4%).
Conclusion
Patients presenting with nongerminomatous malignant ovarian GCTs in the context of GD have a higher rate of events and death than counterparts with normal gonads. These findings emphasize the importance of noting a contralateral streak ovary or gonadoblastoma at histology for any ovarian GCT and support the recommendation for early bilateral gonadectomy in patients known to have GD with Y chromosome material. In contrast to those with pure dysgerminoma, these patients may represent a high‐risk group that requires a more aggressive chemotherapy regimen.</description><identifier>ISSN: 1545-5009</identifier><identifier>EISSN: 1545-5017</identifier><identifier>DOI: 10.1002/pbc.26913</identifier><identifier>PMID: 29286555</identifier><language>eng</language><publisher>United States: Wiley Subscription Services, Inc</publisher><subject>Chemotherapy ; Children ; Choriocarcinoma ; Gonadal dysgenesis ; Gonadectomy ; Gonads ; Hematology ; Histology ; malignant ovarian germ cell tumor ; Oncology ; Ovaries ; pediatric outcome ; Pediatrics ; Tumors ; Y chromosomes ; Yolk ; Yolk sac</subject><ispartof>Pediatric blood & cancer, 2018-04, Vol.65 (4), p.n/a</ispartof><rights>2017 Wiley Periodicals, Inc.</rights><rights>2018 Wiley Periodicals, Inc.</rights><lds50>peer_reviewed</lds50><oa>free_for_read</oa><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c4433-52b398b7aad342f512d31ed3a2fb631fe2a5f6132ae7ddbeda32bc7e563e68683</citedby><cites>FETCH-LOGICAL-c4433-52b398b7aad342f512d31ed3a2fb631fe2a5f6132ae7ddbeda32bc7e563e68683</cites><orcidid>0000-0001-5058-5973</orcidid></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><linktopdf>$$Uhttps://onlinelibrary.wiley.com/doi/pdf/10.1002%2Fpbc.26913$$EPDF$$P50$$Gwiley$$H</linktopdf><linktohtml>$$Uhttps://onlinelibrary.wiley.com/doi/full/10.1002%2Fpbc.26913$$EHTML$$P50$$Gwiley$$H</linktohtml><link.rule.ids>230,314,780,784,885,1417,27924,27925,45574,45575</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/29286555$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Dicken, Bryan J.</creatorcontrib><creatorcontrib>Billmire, Deborah F.</creatorcontrib><creatorcontrib>Krailo, Mark</creatorcontrib><creatorcontrib>Xia, Caihong</creatorcontrib><creatorcontrib>Shaikh, Furqan</creatorcontrib><creatorcontrib>Cullen, John W.</creatorcontrib><creatorcontrib>Olson, Thomas A.</creatorcontrib><creatorcontrib>Pashankar, Farzana</creatorcontrib><creatorcontrib>Malogolowkin, Marcio H.</creatorcontrib><creatorcontrib>Amatruda, James F.</creatorcontrib><creatorcontrib>Rescorla, Frederick J.</creatorcontrib><creatorcontrib>Egler, Rachel A.</creatorcontrib><creatorcontrib>Ross, Jonathan H.</creatorcontrib><creatorcontrib>Rodriguez‐Galindo, Carlos</creatorcontrib><creatorcontrib>Frazier, A. Lindsay</creatorcontrib><title>Gonadal dysgenesis is associated with worse outcomes in patients with ovarian nondysgerminomatous tumors: A report of the Children's Oncology Group AGCT 0132 study</title><title>Pediatric blood & cancer</title><addtitle>Pediatr Blood Cancer</addtitle><description>Purpose
In this report, we characterize the timing and behavior of malignant ovarian germ cell tumors (GCTs) in pediatric patients with dysgenetic gonads compared to those with normal gonadal development.
Patients and methods
Patients from the Children's Oncology Group AGCT0132 with malignant ovarian GCTs were included. Within this population, we sought to identify patients with gonadoblastoma, streak ovaries, or other evidence of gonadal dysgenesis (GD). Patients with malignant GCTs containing one or more of the following histologies—yolk sac tumor, embryonal carcinoma, or choriocarcinoma—were included. Patients were compared with respect to event‐free survival (EFS) and overall survival (OS).
Results
Nine patients with GD, including seven with gonadoblastoma (mean age, 9.3 years), were compared to 100 non‐GD patients (mean age, 12.1 years). The estimated 3‐year EFS for patients with GD was 66.7% (95% CI 28.2–87.8%) and for non‐GD patients was 88.8% (95% CI 80.2–93.8%). The estimated 3‐year OS for patients with GD was 87.5% (95% CI 38.7–98.1%) and for non‐GD patients was 97.6% (95% CI of 90.6–99.4%).
Conclusion
Patients presenting with nongerminomatous malignant ovarian GCTs in the context of GD have a higher rate of events and death than counterparts with normal gonads. These findings emphasize the importance of noting a contralateral streak ovary or gonadoblastoma at histology for any ovarian GCT and support the recommendation for early bilateral gonadectomy in patients known to have GD with Y chromosome material. In contrast to those with pure dysgerminoma, these patients may represent a high‐risk group that requires a more aggressive chemotherapy regimen.</description><subject>Chemotherapy</subject><subject>Children</subject><subject>Choriocarcinoma</subject><subject>Gonadal dysgenesis</subject><subject>Gonadectomy</subject><subject>Gonads</subject><subject>Hematology</subject><subject>Histology</subject><subject>malignant ovarian germ cell tumor</subject><subject>Oncology</subject><subject>Ovaries</subject><subject>pediatric outcome</subject><subject>Pediatrics</subject><subject>Tumors</subject><subject>Y chromosomes</subject><subject>Yolk</subject><subject>Yolk sac</subject><issn>1545-5009</issn><issn>1545-5017</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2018</creationdate><recordtype>article</recordtype><recordid>eNp1kd9r1TAUx4sobk4f_Ack4MP04W750aStD8K16HUwmA_zOaTN6b0ZbVKTdJf-PftHzdZ5UWEQSOB88uGc882ytwSfEYzp-di0Z1RUhD3LjgnP-YpjUjw_vHF1lL0K4SahAvPyZXZEK1oKzvlxdrdxVmnVIz2HLVgIJqB0VAiuNSqCRnsTd2jvfADkpti6ARJh0aiiARvDUne3yhtlkXX2QeQHY92gopsCitOQfn9Ca-RhdD4i16G4A1TvTK892NOArmzrered0ca7aUTrTX2NMGEUhTjp-XX2olN9gDeP90n289vX6_r76vJqc1GvL1dtnjO24rRhVdkUSmmW044TqhkBzRTtGsFIB1TxTiSrgkLrBrRitGkL4IKBKEXJTrLPi3ecmgF0m8bzqpejN4Pys3TKyH8r1uzk1t1KQUlVFjgJPjwKvPs1QYhyMKGFvlcW0iZkomiZM8FpQt__h964yds0nqQYi5LQXNwLPy5U610IHrpDMwTL--hlil4-RJ_Yd393fyD_ZJ2A8wXYmx7mp03yx5d6Uf4GN5a79g</recordid><startdate>201804</startdate><enddate>201804</enddate><creator>Dicken, Bryan J.</creator><creator>Billmire, Deborah F.</creator><creator>Krailo, Mark</creator><creator>Xia, Caihong</creator><creator>Shaikh, Furqan</creator><creator>Cullen, John W.</creator><creator>Olson, Thomas A.</creator><creator>Pashankar, Farzana</creator><creator>Malogolowkin, Marcio H.</creator><creator>Amatruda, James F.</creator><creator>Rescorla, Frederick J.</creator><creator>Egler, Rachel A.</creator><creator>Ross, Jonathan H.</creator><creator>Rodriguez‐Galindo, Carlos</creator><creator>Frazier, A. Lindsay</creator><general>Wiley Subscription Services, Inc</general><scope>NPM</scope><scope>AAYXX</scope><scope>CITATION</scope><scope>7T5</scope><scope>7TK</scope><scope>7TO</scope><scope>8FD</scope><scope>FR3</scope><scope>H94</scope><scope>K9.</scope><scope>P64</scope><scope>RC3</scope><scope>7X8</scope><scope>5PM</scope><orcidid>https://orcid.org/0000-0001-5058-5973</orcidid></search><sort><creationdate>201804</creationdate><title>Gonadal dysgenesis is associated with worse outcomes in patients with ovarian nondysgerminomatous tumors: A report of the Children's Oncology Group AGCT 0132 study</title><author>Dicken, Bryan J. ; Billmire, Deborah F. ; Krailo, Mark ; Xia, Caihong ; Shaikh, Furqan ; Cullen, John W. ; Olson, Thomas A. ; Pashankar, Farzana ; Malogolowkin, Marcio H. ; Amatruda, James F. ; Rescorla, Frederick J. ; Egler, Rachel A. ; Ross, Jonathan H. ; Rodriguez‐Galindo, Carlos ; Frazier, A. Lindsay</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c4433-52b398b7aad342f512d31ed3a2fb631fe2a5f6132ae7ddbeda32bc7e563e68683</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2018</creationdate><topic>Chemotherapy</topic><topic>Children</topic><topic>Choriocarcinoma</topic><topic>Gonadal dysgenesis</topic><topic>Gonadectomy</topic><topic>Gonads</topic><topic>Hematology</topic><topic>Histology</topic><topic>malignant ovarian germ cell tumor</topic><topic>Oncology</topic><topic>Ovaries</topic><topic>pediatric outcome</topic><topic>Pediatrics</topic><topic>Tumors</topic><topic>Y chromosomes</topic><topic>Yolk</topic><topic>Yolk sac</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Dicken, Bryan J.</creatorcontrib><creatorcontrib>Billmire, Deborah F.</creatorcontrib><creatorcontrib>Krailo, Mark</creatorcontrib><creatorcontrib>Xia, Caihong</creatorcontrib><creatorcontrib>Shaikh, Furqan</creatorcontrib><creatorcontrib>Cullen, John W.</creatorcontrib><creatorcontrib>Olson, Thomas A.</creatorcontrib><creatorcontrib>Pashankar, Farzana</creatorcontrib><creatorcontrib>Malogolowkin, Marcio H.</creatorcontrib><creatorcontrib>Amatruda, James F.</creatorcontrib><creatorcontrib>Rescorla, Frederick J.</creatorcontrib><creatorcontrib>Egler, Rachel A.</creatorcontrib><creatorcontrib>Ross, Jonathan H.</creatorcontrib><creatorcontrib>Rodriguez‐Galindo, Carlos</creatorcontrib><creatorcontrib>Frazier, A. Lindsay</creatorcontrib><collection>PubMed</collection><collection>CrossRef</collection><collection>Immunology Abstracts</collection><collection>Neurosciences Abstracts</collection><collection>Oncogenes and Growth Factors Abstracts</collection><collection>Technology Research Database</collection><collection>Engineering Research Database</collection><collection>AIDS and Cancer Research Abstracts</collection><collection>ProQuest Health & Medical Complete (Alumni)</collection><collection>Biotechnology and BioEngineering Abstracts</collection><collection>Genetics Abstracts</collection><collection>MEDLINE - Academic</collection><collection>PubMed Central (Full Participant titles)</collection><jtitle>Pediatric blood & cancer</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Dicken, Bryan J.</au><au>Billmire, Deborah F.</au><au>Krailo, Mark</au><au>Xia, Caihong</au><au>Shaikh, Furqan</au><au>Cullen, John W.</au><au>Olson, Thomas A.</au><au>Pashankar, Farzana</au><au>Malogolowkin, Marcio H.</au><au>Amatruda, James F.</au><au>Rescorla, Frederick J.</au><au>Egler, Rachel A.</au><au>Ross, Jonathan H.</au><au>Rodriguez‐Galindo, Carlos</au><au>Frazier, A. Lindsay</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Gonadal dysgenesis is associated with worse outcomes in patients with ovarian nondysgerminomatous tumors: A report of the Children's Oncology Group AGCT 0132 study</atitle><jtitle>Pediatric blood & cancer</jtitle><addtitle>Pediatr Blood Cancer</addtitle><date>2018-04</date><risdate>2018</risdate><volume>65</volume><issue>4</issue><epage>n/a</epage><issn>1545-5009</issn><eissn>1545-5017</eissn><abstract>Purpose
In this report, we characterize the timing and behavior of malignant ovarian germ cell tumors (GCTs) in pediatric patients with dysgenetic gonads compared to those with normal gonadal development.
Patients and methods
Patients from the Children's Oncology Group AGCT0132 with malignant ovarian GCTs were included. Within this population, we sought to identify patients with gonadoblastoma, streak ovaries, or other evidence of gonadal dysgenesis (GD). Patients with malignant GCTs containing one or more of the following histologies—yolk sac tumor, embryonal carcinoma, or choriocarcinoma—were included. Patients were compared with respect to event‐free survival (EFS) and overall survival (OS).
Results
Nine patients with GD, including seven with gonadoblastoma (mean age, 9.3 years), were compared to 100 non‐GD patients (mean age, 12.1 years). The estimated 3‐year EFS for patients with GD was 66.7% (95% CI 28.2–87.8%) and for non‐GD patients was 88.8% (95% CI 80.2–93.8%). The estimated 3‐year OS for patients with GD was 87.5% (95% CI 38.7–98.1%) and for non‐GD patients was 97.6% (95% CI of 90.6–99.4%).
Conclusion
Patients presenting with nongerminomatous malignant ovarian GCTs in the context of GD have a higher rate of events and death than counterparts with normal gonads. These findings emphasize the importance of noting a contralateral streak ovary or gonadoblastoma at histology for any ovarian GCT and support the recommendation for early bilateral gonadectomy in patients known to have GD with Y chromosome material. In contrast to those with pure dysgerminoma, these patients may represent a high‐risk group that requires a more aggressive chemotherapy regimen.</abstract><cop>United States</cop><pub>Wiley Subscription Services, Inc</pub><pmid>29286555</pmid><doi>10.1002/pbc.26913</doi><tpages>6</tpages><orcidid>https://orcid.org/0000-0001-5058-5973</orcidid><oa>free_for_read</oa></addata></record> |
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subjects | Chemotherapy Children Choriocarcinoma Gonadal dysgenesis Gonadectomy Gonads Hematology Histology malignant ovarian germ cell tumor Oncology Ovaries pediatric outcome Pediatrics Tumors Y chromosomes Yolk Yolk sac |
title | Gonadal dysgenesis is associated with worse outcomes in patients with ovarian nondysgerminomatous tumors: A report of the Children's Oncology Group AGCT 0132 study |
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