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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Veröffentlicht in:Journal of endocrinological investigation 2023-06, Vol.46 (6), p.1219-1232
Hauptverfasser: 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.
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container_end_page 1232
container_issue 6
container_start_page 1219
container_title Journal of endocrinological investigation
container_volume 46
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
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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. 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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. 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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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