Is surgical resection and observation sufficient for stage I and II sacrococcygeal germ cell tumors? A case series and review

Background Sacrococcygeal teratoma (SCT) is the most common germ cell tumor (GCT) of infancy. Up to 35% of infants may have malignant elements. The standard of care for SCT with malignant elements (SCT‐ME) has been surgery and chemotherapy. However, cases where low‐stage SCT‐ME have been successfull...

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Veröffentlicht in:Pediatric blood & cancer 2017-05, Vol.64 (5), p.np-n/a
Hauptverfasser: Egler, Rachel A., Gosiengfiao, Yasmin, Russell, Heidi, Wickiser, Jonathan E., Frazier, A. Lindsay
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creator Egler, Rachel A.
Gosiengfiao, Yasmin
Russell, Heidi
Wickiser, Jonathan E.
Frazier, A. Lindsay
description Background Sacrococcygeal teratoma (SCT) is the most common germ cell tumor (GCT) of infancy. Up to 35% of infants may have malignant elements. The standard of care for SCT with malignant elements (SCT‐ME) has been surgery and chemotherapy. However, cases where low‐stage SCT‐ME have been successfully observed following resection have been reported. Procedure To better understand the outcomes of low‐stage SCT‐ME that do not receive chemotherapy, we reviewed SCT pathology reports from five children's hospitals from 1999 to 2009. Information regarding staging workup, tumor markers, treatment, and outcome was collected for patients with stage I or II SCT‐ME. An English language literature review was also performed. Results Seventy‐four SCT were identified: 51 stage I and 23 stage II; 13 (18%) were SCT‐ME: 5 stage I and 8 stage II; four stage I and four stage II tumors were not treated with chemotherapy. No stage I tumors recurred; all of the stage II tumors recurred and were successfully salvaged, two had no ME at recurrence. We identified another 10 stage I SCT‐ME in the literature managed with active surveillance—two recurred and were successfully treated with surgery and chemotherapy. Conclusions Overall, of the 14 cases of stage I SCT‐ME, 12 survived with no recurrence and the two who did recur were successfully treated with platinum‐based chemotherapy (EFS = 86%, overall survival [OS] = 100%); this suggests that patients with stage I SCT‐ME could be observed after surgery and treated only upon recurrence. Stage II SCT‐ME require further study in a clinical trial setting.
doi_str_mv 10.1002/pbc.26311
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A case series and review</title><source>MEDLINE</source><source>Wiley Online Library Journals Frontfile Complete</source><creator>Egler, Rachel A. ; Gosiengfiao, Yasmin ; Russell, Heidi ; Wickiser, Jonathan E. ; Frazier, A. Lindsay</creator><creatorcontrib>Egler, Rachel A. ; Gosiengfiao, Yasmin ; Russell, Heidi ; Wickiser, Jonathan E. ; Frazier, A. Lindsay</creatorcontrib><description>Background Sacrococcygeal teratoma (SCT) is the most common germ cell tumor (GCT) of infancy. Up to 35% of infants may have malignant elements. The standard of care for SCT with malignant elements (SCT‐ME) has been surgery and chemotherapy. However, cases where low‐stage SCT‐ME have been successfully observed following resection have been reported. Procedure To better understand the outcomes of low‐stage SCT‐ME that do not receive chemotherapy, we reviewed SCT pathology reports from five children's hospitals from 1999 to 2009. Information regarding staging workup, tumor markers, treatment, and outcome was collected for patients with stage I or II SCT‐ME. An English language literature review was also performed. Results Seventy‐four SCT were identified: 51 stage I and 23 stage II; 13 (18%) were SCT‐ME: 5 stage I and 8 stage II; four stage I and four stage II tumors were not treated with chemotherapy. No stage I tumors recurred; all of the stage II tumors recurred and were successfully salvaged, two had no ME at recurrence. We identified another 10 stage I SCT‐ME in the literature managed with active surveillance—two recurred and were successfully treated with surgery and chemotherapy. Conclusions Overall, of the 14 cases of stage I SCT‐ME, 12 survived with no recurrence and the two who did recur were successfully treated with platinum‐based chemotherapy (EFS = 86%, overall survival [OS] = 100%); this suggests that patients with stage I SCT‐ME could be observed after surgery and treated only upon recurrence. Stage II SCT‐ME require further study in a clinical trial setting.</description><identifier>ISSN: 1545-5009</identifier><identifier>EISSN: 1545-5017</identifier><identifier>DOI: 10.1002/pbc.26311</identifier><identifier>PMID: 27786428</identifier><language>eng</language><publisher>United States: Wiley Subscription Services, Inc</publisher><subject>Antineoplastic Combined Chemotherapy Protocols - therapeutic use ; Biomarkers, Tumor ; Chemotherapy ; Children ; Combined Modality Therapy ; Disease-Free Survival ; English language ; germ cell tumor ; Hematology ; Hospitals ; Humans ; Infant ; Infant, Newborn ; Infants ; Language ; Literature reviews ; Neoplasm Recurrence, Local - drug therapy ; Neoplasm Recurrence, Local - pathology ; Neoplasm Staging ; Neoplasms, Germ Cell and Embryonal - drug therapy ; Neoplasms, Germ Cell and Embryonal - pathology ; Neoplasms, Germ Cell and Embryonal - surgery ; Oncology ; Pathology ; Pediatrics ; Platinum ; Retrospective Studies ; Sacrococcygeal Region - pathology ; Sacrococcygeal Region - surgery ; sacrococcygeal teratoma ; Surgery ; surveillance ; Survival ; Teratoma ; Teratoma - pathology ; Tumor markers ; Tumors</subject><ispartof>Pediatric blood &amp; cancer, 2017-05, Vol.64 (5), p.np-n/a</ispartof><rights>2016 Wiley Periodicals, Inc.</rights><rights>2017 Wiley Periodicals, Inc.</rights><lds50>peer_reviewed</lds50><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c4141-d2921b43784bba0ce1321c0c2b95dffe461ed6252eee9a9bc5ec4a38ba32ee293</citedby><cites>FETCH-LOGICAL-c4141-d2921b43784bba0ce1321c0c2b95dffe461ed6252eee9a9bc5ec4a38ba32ee293</cites></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.26311$$EPDF$$P50$$Gwiley$$H</linktopdf><linktohtml>$$Uhttps://onlinelibrary.wiley.com/doi/full/10.1002%2Fpbc.26311$$EHTML$$P50$$Gwiley$$H</linktohtml><link.rule.ids>314,776,780,1411,27901,27902,45550,45551</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/27786428$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Egler, Rachel A.</creatorcontrib><creatorcontrib>Gosiengfiao, Yasmin</creatorcontrib><creatorcontrib>Russell, Heidi</creatorcontrib><creatorcontrib>Wickiser, Jonathan E.</creatorcontrib><creatorcontrib>Frazier, A. Lindsay</creatorcontrib><title>Is surgical resection and observation sufficient for stage I and II sacrococcygeal germ cell tumors? A case series and review</title><title>Pediatric blood &amp; cancer</title><addtitle>Pediatr Blood Cancer</addtitle><description>Background Sacrococcygeal teratoma (SCT) is the most common germ cell tumor (GCT) of infancy. Up to 35% of infants may have malignant elements. The standard of care for SCT with malignant elements (SCT‐ME) has been surgery and chemotherapy. However, cases where low‐stage SCT‐ME have been successfully observed following resection have been reported. Procedure To better understand the outcomes of low‐stage SCT‐ME that do not receive chemotherapy, we reviewed SCT pathology reports from five children's hospitals from 1999 to 2009. Information regarding staging workup, tumor markers, treatment, and outcome was collected for patients with stage I or II SCT‐ME. An English language literature review was also performed. Results Seventy‐four SCT were identified: 51 stage I and 23 stage II; 13 (18%) were SCT‐ME: 5 stage I and 8 stage II; four stage I and four stage II tumors were not treated with chemotherapy. No stage I tumors recurred; all of the stage II tumors recurred and were successfully salvaged, two had no ME at recurrence. We identified another 10 stage I SCT‐ME in the literature managed with active surveillance—two recurred and were successfully treated with surgery and chemotherapy. Conclusions Overall, of the 14 cases of stage I SCT‐ME, 12 survived with no recurrence and the two who did recur were successfully treated with platinum‐based chemotherapy (EFS = 86%, overall survival [OS] = 100%); this suggests that patients with stage I SCT‐ME could be observed after surgery and treated only upon recurrence. 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Lindsay</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Is surgical resection and observation sufficient for stage I and II sacrococcygeal germ cell tumors? A case series and review</atitle><jtitle>Pediatric blood &amp; cancer</jtitle><addtitle>Pediatr Blood Cancer</addtitle><date>2017-05</date><risdate>2017</risdate><volume>64</volume><issue>5</issue><spage>np</spage><epage>n/a</epage><pages>np-n/a</pages><issn>1545-5009</issn><eissn>1545-5017</eissn><abstract>Background Sacrococcygeal teratoma (SCT) is the most common germ cell tumor (GCT) of infancy. Up to 35% of infants may have malignant elements. The standard of care for SCT with malignant elements (SCT‐ME) has been surgery and chemotherapy. However, cases where low‐stage SCT‐ME have been successfully observed following resection have been reported. Procedure To better understand the outcomes of low‐stage SCT‐ME that do not receive chemotherapy, we reviewed SCT pathology reports from five children's hospitals from 1999 to 2009. Information regarding staging workup, tumor markers, treatment, and outcome was collected for patients with stage I or II SCT‐ME. An English language literature review was also performed. Results Seventy‐four SCT were identified: 51 stage I and 23 stage II; 13 (18%) were SCT‐ME: 5 stage I and 8 stage II; four stage I and four stage II tumors were not treated with chemotherapy. No stage I tumors recurred; all of the stage II tumors recurred and were successfully salvaged, two had no ME at recurrence. We identified another 10 stage I SCT‐ME in the literature managed with active surveillance—two recurred and were successfully treated with surgery and chemotherapy. Conclusions Overall, of the 14 cases of stage I SCT‐ME, 12 survived with no recurrence and the two who did recur were successfully treated with platinum‐based chemotherapy (EFS = 86%, overall survival [OS] = 100%); this suggests that patients with stage I SCT‐ME could be observed after surgery and treated only upon recurrence. Stage II SCT‐ME require further study in a clinical trial setting.</abstract><cop>United States</cop><pub>Wiley Subscription Services, Inc</pub><pmid>27786428</pmid><doi>10.1002/pbc.26311</doi><tpages>5</tpages></addata></record>
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subjects Antineoplastic Combined Chemotherapy Protocols - therapeutic use
Biomarkers, Tumor
Chemotherapy
Children
Combined Modality Therapy
Disease-Free Survival
English language
germ cell tumor
Hematology
Hospitals
Humans
Infant
Infant, Newborn
Infants
Language
Literature reviews
Neoplasm Recurrence, Local - drug therapy
Neoplasm Recurrence, Local - pathology
Neoplasm Staging
Neoplasms, Germ Cell and Embryonal - drug therapy
Neoplasms, Germ Cell and Embryonal - pathology
Neoplasms, Germ Cell and Embryonal - surgery
Oncology
Pathology
Pediatrics
Platinum
Retrospective Studies
Sacrococcygeal Region - pathology
Sacrococcygeal Region - surgery
sacrococcygeal teratoma
Surgery
surveillance
Survival
Teratoma
Teratoma - pathology
Tumor markers
Tumors
title Is surgical resection and observation sufficient for stage I and II sacrococcygeal germ cell tumors? A case series and review
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