Clinical and oncological outcomes in single-stage versus staged surgery for pediatric craniopharyngiomas: a multicenter retrospective study
Purpose Craniopharyngiomas (CPGs) are aggressive brain tumors responsible of severe morbidity in children. The best treatment strategies are under debate. Our study evaluates surgical, pituitary, and hypothalamic outcomes of a tailored staged-surgical approach compared to a single-stage radical appr...
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creator | Agresta, G. Campione, A. Veiceschi, P. Gallo, D. Agosti, E. Massimi, L. Piatelli, G. Consales, A. Linsler, S. Oertel, J. Pozzi, F. Tanda, M. L. Castelnuovo, P. Locatelli, D. |
description | Purpose
Craniopharyngiomas (CPGs) are aggressive brain tumors responsible of severe morbidity in children. The best treatment strategies are under debate. Our study evaluates surgical, pituitary, and hypothalamic outcomes of a tailored staged-surgical approach compared to a single-stage radical approach in children with CPGs.
Methods
Multicenter retrospective study enrolling 96 children treated for CPGs in the period 2010–2022. The surgical management was selected after a multidisciplinary evaluation. Primary endpoint includes the inter-group comparison of preservation/improvement of hypothalamic–pituitary function, the extent of resection, and progression-free survival (PFS). Secondary endpoints include overall survival (OS), morbidity, and quality of life (QoL).
Results
Gross Total Resection (GTR) was reached in 46.1% of cases in the single-stage surgery group (82 patients, age at surgery 9 ± 4.7 years) and 33.3% after the last operation in the staged surgery group (14 patients age 7.64 ± 4.57 years at first surgery and 9.36 ± 4.7 years at the last surgery). The PFS was significantly higher in patients addressed to staged- compared to single-stage surgery (93.75% vs 70.7% at 5 years, respectively,
p
= 0.03). The recurrence rate was slightly higher in the single-stage surgery group. No significant differences emerged in the endocrinological, visual, hypothalamic outcome, OS, and QoL comparing the two groups.
Conclusions
In pediatric CPGs’ surgical radicality and timing of intervention should be tailored considering both anatomical extension and hypothalamic–pituitary function. In selected patients, a staged approach offers a safer and more effective disease control, preserving psychophysical development. |
doi_str_mv | 10.1007/s40618-022-01993-2 |
format | Article |
fullrecord | <record><control><sourceid>proquest_cross</sourceid><recordid>TN_cdi_proquest_miscellaneous_2758109903</recordid><sourceformat>XML</sourceformat><sourcesystem>PC</sourcesystem><sourcerecordid>2823643972</sourcerecordid><originalsourceid>FETCH-LOGICAL-c375t-8df2696a249695233443067f7554d0c6125fe8e4d9546e84dcac640e8692abed3</originalsourceid><addsrcrecordid>eNp9kU1vFSEUhomxsbX6B1wYEjduxjLAMODO3PiVNOmmrgmFMyPNDIx8NLm_oX9aem_9iAtXcMJzXjg8CL3qybuekPEicyJ62RFKO9IrxTr6BJ31IyWdZFI8_Wt_ip7nfEsIG5kcn6FTJoaBUMHP0P1u8cFbs2ATHI7BxiXOhzrWYuMKGfuAsw_zAl0uZgZ8BynXjA-Fw7mmGdIeTzHhDZw3JXmLbTLBx-27Sfsw-7ia_B4bvNaleAuhQMIJSop5A1v8HbSw6vYv0MlklgwvH9dz9O3Tx-vdl-7y6vPX3YfLzrJxKJ10ExVKGMqVUANljHNGxDiNw8AdsaKnwwQSuFMDFyC5s8YKTkAKRc0NOHaO3h5ztxR_VMhFrz5bWBYTINas6TjInihFWEPf_IPexppCe52mkjLBmRppo-iRsm2knGDSW_Jrm133RD-o0kdVuqnSB1X6oen1Y3S9WcH9bvnlpgHsCOR2FNon_7n7P7E_AbKcoXI</addsrcrecordid><sourcetype>Aggregation Database</sourcetype><iscdi>true</iscdi><recordtype>article</recordtype><pqid>2823643972</pqid></control><display><type>article</type><title>Clinical and oncological outcomes in single-stage versus staged surgery for pediatric craniopharyngiomas: a multicenter retrospective study</title><source>MEDLINE</source><source>SpringerLink Journals</source><creator>Agresta, G. ; Campione, A. ; Veiceschi, P. ; Gallo, D. ; Agosti, E. ; Massimi, L. ; Piatelli, G. ; Consales, A. ; Linsler, S. ; Oertel, J. ; Pozzi, F. ; Tanda, M. L. ; Castelnuovo, P. ; Locatelli, D.</creator><creatorcontrib>Agresta, G. ; Campione, A. ; Veiceschi, P. ; Gallo, D. ; Agosti, E. ; Massimi, L. ; Piatelli, G. ; Consales, A. ; Linsler, S. ; Oertel, J. ; Pozzi, F. ; Tanda, M. L. ; Castelnuovo, P. ; Locatelli, D.</creatorcontrib><description>Purpose
Craniopharyngiomas (CPGs) are aggressive brain tumors responsible of severe morbidity in children. The best treatment strategies are under debate. Our study evaluates surgical, pituitary, and hypothalamic outcomes of a tailored staged-surgical approach compared to a single-stage radical approach in children with CPGs.
Methods
Multicenter retrospective study enrolling 96 children treated for CPGs in the period 2010–2022. The surgical management was selected after a multidisciplinary evaluation. Primary endpoint includes the inter-group comparison of preservation/improvement of hypothalamic–pituitary function, the extent of resection, and progression-free survival (PFS). Secondary endpoints include overall survival (OS), morbidity, and quality of life (QoL).
Results
Gross Total Resection (GTR) was reached in 46.1% of cases in the single-stage surgery group (82 patients, age at surgery 9 ± 4.7 years) and 33.3% after the last operation in the staged surgery group (14 patients age 7.64 ± 4.57 years at first surgery and 9.36 ± 4.7 years at the last surgery). The PFS was significantly higher in patients addressed to staged- compared to single-stage surgery (93.75% vs 70.7% at 5 years, respectively,
p
= 0.03). The recurrence rate was slightly higher in the single-stage surgery group. No significant differences emerged in the endocrinological, visual, hypothalamic outcome, OS, and QoL comparing the two groups.
Conclusions
In pediatric CPGs’ surgical radicality and timing of intervention should be tailored considering both anatomical extension and hypothalamic–pituitary function. In selected patients, a staged approach offers a safer and more effective disease control, preserving psychophysical development.</description><identifier>ISSN: 1720-8386</identifier><identifier>ISSN: 0391-4097</identifier><identifier>EISSN: 1720-8386</identifier><identifier>DOI: 10.1007/s40618-022-01993-2</identifier><identifier>PMID: 36550264</identifier><language>eng</language><publisher>Cham: Springer International Publishing</publisher><subject>Brain tumors ; Child ; Child, Preschool ; Children ; Craniopharyngioma - pathology ; Craniopharyngioma - surgery ; Disease control ; Endocrinology ; Humans ; Hypothalamus ; Internal Medicine ; Medicine ; Medicine & Public Health ; Metabolic Diseases ; Morbidity ; Neoplasm Recurrence, Local - pathology ; Original Article ; Patients ; Pediatrics ; Pituitary ; Pituitary Diseases ; Pituitary Neoplasms - pathology ; Pituitary Neoplasms - surgery ; Psychophysics ; Quality of Life ; Retrospective Studies ; Surgery ; Survival ; Treatment Outcome</subject><ispartof>Journal of endocrinological investigation, 2023-06, Vol.46 (6), p.1219-1232</ispartof><rights>The Author(s), under exclusive licence to Italian Society of Endocrinology (SIE) 2022. Springer Nature or its licensor (e.g. a society or other partner) holds exclusive rights to this article under a publishing agreement with the author(s) or other rightsholder(s); author self-archiving of the accepted manuscript version of this article is solely governed by the terms of such publishing agreement and applicable law.</rights><rights>2022. The Author(s), under exclusive licence to Italian Society of Endocrinology (SIE).</rights><lds50>peer_reviewed</lds50><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c375t-8df2696a249695233443067f7554d0c6125fe8e4d9546e84dcac640e8692abed3</citedby><cites>FETCH-LOGICAL-c375t-8df2696a249695233443067f7554d0c6125fe8e4d9546e84dcac640e8692abed3</cites><orcidid>0000-0002-3715-1318</orcidid></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><linktopdf>$$Uhttps://link.springer.com/content/pdf/10.1007/s40618-022-01993-2$$EPDF$$P50$$Gspringer$$H</linktopdf><linktohtml>$$Uhttps://link.springer.com/10.1007/s40618-022-01993-2$$EHTML$$P50$$Gspringer$$H</linktohtml><link.rule.ids>314,780,784,27924,27925,41488,42557,51319</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/36550264$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Agresta, G.</creatorcontrib><creatorcontrib>Campione, A.</creatorcontrib><creatorcontrib>Veiceschi, P.</creatorcontrib><creatorcontrib>Gallo, D.</creatorcontrib><creatorcontrib>Agosti, E.</creatorcontrib><creatorcontrib>Massimi, L.</creatorcontrib><creatorcontrib>Piatelli, G.</creatorcontrib><creatorcontrib>Consales, A.</creatorcontrib><creatorcontrib>Linsler, S.</creatorcontrib><creatorcontrib>Oertel, J.</creatorcontrib><creatorcontrib>Pozzi, F.</creatorcontrib><creatorcontrib>Tanda, M. L.</creatorcontrib><creatorcontrib>Castelnuovo, P.</creatorcontrib><creatorcontrib>Locatelli, D.</creatorcontrib><title>Clinical and oncological outcomes in single-stage versus staged surgery for pediatric craniopharyngiomas: a multicenter retrospective study</title><title>Journal of endocrinological investigation</title><addtitle>J Endocrinol Invest</addtitle><addtitle>J Endocrinol Invest</addtitle><description>Purpose
Craniopharyngiomas (CPGs) are aggressive brain tumors responsible of severe morbidity in children. The best treatment strategies are under debate. Our study evaluates surgical, pituitary, and hypothalamic outcomes of a tailored staged-surgical approach compared to a single-stage radical approach in children with CPGs.
Methods
Multicenter retrospective study enrolling 96 children treated for CPGs in the period 2010–2022. The surgical management was selected after a multidisciplinary evaluation. Primary endpoint includes the inter-group comparison of preservation/improvement of hypothalamic–pituitary function, the extent of resection, and progression-free survival (PFS). Secondary endpoints include overall survival (OS), morbidity, and quality of life (QoL).
Results
Gross Total Resection (GTR) was reached in 46.1% of cases in the single-stage surgery group (82 patients, age at surgery 9 ± 4.7 years) and 33.3% after the last operation in the staged surgery group (14 patients age 7.64 ± 4.57 years at first surgery and 9.36 ± 4.7 years at the last surgery). The PFS was significantly higher in patients addressed to staged- compared to single-stage surgery (93.75% vs 70.7% at 5 years, respectively,
p
= 0.03). The recurrence rate was slightly higher in the single-stage surgery group. No significant differences emerged in the endocrinological, visual, hypothalamic outcome, OS, and QoL comparing the two groups.
Conclusions
In pediatric CPGs’ surgical radicality and timing of intervention should be tailored considering both anatomical extension and hypothalamic–pituitary function. In selected patients, a staged approach offers a safer and more effective disease control, preserving psychophysical development.</description><subject>Brain tumors</subject><subject>Child</subject><subject>Child, Preschool</subject><subject>Children</subject><subject>Craniopharyngioma - pathology</subject><subject>Craniopharyngioma - surgery</subject><subject>Disease control</subject><subject>Endocrinology</subject><subject>Humans</subject><subject>Hypothalamus</subject><subject>Internal Medicine</subject><subject>Medicine</subject><subject>Medicine & Public Health</subject><subject>Metabolic Diseases</subject><subject>Morbidity</subject><subject>Neoplasm Recurrence, Local - pathology</subject><subject>Original Article</subject><subject>Patients</subject><subject>Pediatrics</subject><subject>Pituitary</subject><subject>Pituitary Diseases</subject><subject>Pituitary Neoplasms - pathology</subject><subject>Pituitary Neoplasms - surgery</subject><subject>Psychophysics</subject><subject>Quality of Life</subject><subject>Retrospective Studies</subject><subject>Surgery</subject><subject>Survival</subject><subject>Treatment Outcome</subject><issn>1720-8386</issn><issn>0391-4097</issn><issn>1720-8386</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2023</creationdate><recordtype>article</recordtype><sourceid>EIF</sourceid><recordid>eNp9kU1vFSEUhomxsbX6B1wYEjduxjLAMODO3PiVNOmmrgmFMyPNDIx8NLm_oX9aem_9iAtXcMJzXjg8CL3qybuekPEicyJ62RFKO9IrxTr6BJ31IyWdZFI8_Wt_ip7nfEsIG5kcn6FTJoaBUMHP0P1u8cFbs2ATHI7BxiXOhzrWYuMKGfuAsw_zAl0uZgZ8BynXjA-Fw7mmGdIeTzHhDZw3JXmLbTLBx-27Sfsw-7ia_B4bvNaleAuhQMIJSop5A1v8HbSw6vYv0MlklgwvH9dz9O3Tx-vdl-7y6vPX3YfLzrJxKJ10ExVKGMqVUANljHNGxDiNw8AdsaKnwwQSuFMDFyC5s8YKTkAKRc0NOHaO3h5ztxR_VMhFrz5bWBYTINas6TjInihFWEPf_IPexppCe52mkjLBmRppo-iRsm2knGDSW_Jrm133RD-o0kdVuqnSB1X6oen1Y3S9WcH9bvnlpgHsCOR2FNon_7n7P7E_AbKcoXI</recordid><startdate>20230601</startdate><enddate>20230601</enddate><creator>Agresta, G.</creator><creator>Campione, A.</creator><creator>Veiceschi, P.</creator><creator>Gallo, D.</creator><creator>Agosti, E.</creator><creator>Massimi, L.</creator><creator>Piatelli, G.</creator><creator>Consales, A.</creator><creator>Linsler, S.</creator><creator>Oertel, J.</creator><creator>Pozzi, F.</creator><creator>Tanda, M. L.</creator><creator>Castelnuovo, P.</creator><creator>Locatelli, D.</creator><general>Springer International Publishing</general><general>Springer Nature B.V</general><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>7X8</scope><orcidid>https://orcid.org/0000-0002-3715-1318</orcidid></search><sort><creationdate>20230601</creationdate><title>Clinical and oncological outcomes in single-stage versus staged surgery for pediatric craniopharyngiomas: a multicenter retrospective study</title><author>Agresta, G. ; Campione, A. ; Veiceschi, P. ; Gallo, D. ; Agosti, E. ; Massimi, L. ; Piatelli, G. ; Consales, A. ; Linsler, S. ; Oertel, J. ; Pozzi, F. ; Tanda, M. L. ; Castelnuovo, P. ; Locatelli, D.</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c375t-8df2696a249695233443067f7554d0c6125fe8e4d9546e84dcac640e8692abed3</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2023</creationdate><topic>Brain tumors</topic><topic>Child</topic><topic>Child, Preschool</topic><topic>Children</topic><topic>Craniopharyngioma - pathology</topic><topic>Craniopharyngioma - surgery</topic><topic>Disease control</topic><topic>Endocrinology</topic><topic>Humans</topic><topic>Hypothalamus</topic><topic>Internal Medicine</topic><topic>Medicine</topic><topic>Medicine & Public Health</topic><topic>Metabolic Diseases</topic><topic>Morbidity</topic><topic>Neoplasm Recurrence, Local - pathology</topic><topic>Original Article</topic><topic>Patients</topic><topic>Pediatrics</topic><topic>Pituitary</topic><topic>Pituitary Diseases</topic><topic>Pituitary Neoplasms - pathology</topic><topic>Pituitary Neoplasms - surgery</topic><topic>Psychophysics</topic><topic>Quality of Life</topic><topic>Retrospective Studies</topic><topic>Surgery</topic><topic>Survival</topic><topic>Treatment Outcome</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Agresta, G.</creatorcontrib><creatorcontrib>Campione, A.</creatorcontrib><creatorcontrib>Veiceschi, P.</creatorcontrib><creatorcontrib>Gallo, D.</creatorcontrib><creatorcontrib>Agosti, E.</creatorcontrib><creatorcontrib>Massimi, L.</creatorcontrib><creatorcontrib>Piatelli, G.</creatorcontrib><creatorcontrib>Consales, A.</creatorcontrib><creatorcontrib>Linsler, S.</creatorcontrib><creatorcontrib>Oertel, J.</creatorcontrib><creatorcontrib>Pozzi, F.</creatorcontrib><creatorcontrib>Tanda, M. L.</creatorcontrib><creatorcontrib>Castelnuovo, P.</creatorcontrib><creatorcontrib>Locatelli, D.</creatorcontrib><collection>Medline</collection><collection>MEDLINE</collection><collection>MEDLINE (Ovid)</collection><collection>MEDLINE</collection><collection>MEDLINE</collection><collection>PubMed</collection><collection>CrossRef</collection><collection>MEDLINE - Academic</collection><jtitle>Journal of endocrinological investigation</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Agresta, G.</au><au>Campione, A.</au><au>Veiceschi, P.</au><au>Gallo, D.</au><au>Agosti, E.</au><au>Massimi, L.</au><au>Piatelli, G.</au><au>Consales, A.</au><au>Linsler, S.</au><au>Oertel, J.</au><au>Pozzi, F.</au><au>Tanda, M. L.</au><au>Castelnuovo, P.</au><au>Locatelli, D.</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Clinical and oncological outcomes in single-stage versus staged surgery for pediatric craniopharyngiomas: a multicenter retrospective study</atitle><jtitle>Journal of endocrinological investigation</jtitle><stitle>J Endocrinol Invest</stitle><addtitle>J Endocrinol Invest</addtitle><date>2023-06-01</date><risdate>2023</risdate><volume>46</volume><issue>6</issue><spage>1219</spage><epage>1232</epage><pages>1219-1232</pages><issn>1720-8386</issn><issn>0391-4097</issn><eissn>1720-8386</eissn><abstract>Purpose
Craniopharyngiomas (CPGs) are aggressive brain tumors responsible of severe morbidity in children. The best treatment strategies are under debate. Our study evaluates surgical, pituitary, and hypothalamic outcomes of a tailored staged-surgical approach compared to a single-stage radical approach in children with CPGs.
Methods
Multicenter retrospective study enrolling 96 children treated for CPGs in the period 2010–2022. The surgical management was selected after a multidisciplinary evaluation. Primary endpoint includes the inter-group comparison of preservation/improvement of hypothalamic–pituitary function, the extent of resection, and progression-free survival (PFS). Secondary endpoints include overall survival (OS), morbidity, and quality of life (QoL).
Results
Gross Total Resection (GTR) was reached in 46.1% of cases in the single-stage surgery group (82 patients, age at surgery 9 ± 4.7 years) and 33.3% after the last operation in the staged surgery group (14 patients age 7.64 ± 4.57 years at first surgery and 9.36 ± 4.7 years at the last surgery). The PFS was significantly higher in patients addressed to staged- compared to single-stage surgery (93.75% vs 70.7% at 5 years, respectively,
p
= 0.03). The recurrence rate was slightly higher in the single-stage surgery group. No significant differences emerged in the endocrinological, visual, hypothalamic outcome, OS, and QoL comparing the two groups.
Conclusions
In pediatric CPGs’ surgical radicality and timing of intervention should be tailored considering both anatomical extension and hypothalamic–pituitary function. In selected patients, a staged approach offers a safer and more effective disease control, preserving psychophysical development.</abstract><cop>Cham</cop><pub>Springer International Publishing</pub><pmid>36550264</pmid><doi>10.1007/s40618-022-01993-2</doi><tpages>14</tpages><orcidid>https://orcid.org/0000-0002-3715-1318</orcidid></addata></record> |
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subjects | Brain tumors Child Child, Preschool Children Craniopharyngioma - pathology Craniopharyngioma - surgery Disease control Endocrinology Humans Hypothalamus Internal Medicine Medicine Medicine & Public Health Metabolic Diseases Morbidity Neoplasm Recurrence, Local - pathology Original Article Patients Pediatrics Pituitary Pituitary Diseases Pituitary Neoplasms - pathology Pituitary Neoplasms - surgery Psychophysics Quality of Life Retrospective Studies Surgery Survival Treatment Outcome |
title | Clinical and oncological outcomes in single-stage versus staged surgery for pediatric craniopharyngiomas: a multicenter retrospective study |
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