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Article: Treatment burden and long‐term health deficits of patients with low‐grade gliomas or glioneuronal tumors diagnosed during the first year of life

TitleTreatment burden and long‐term health deficits of patients with low‐grade gliomas or glioneuronal tumors diagnosed during the first year of life
Authors
Keywordsinfant
low‐grade glioma
morbidities
prognostic factors
treatment burden
Issue Date2019
PublisherJohn Wiley & Sons, Inc. The Journal's web site is located at http://onlinelibrary.wiley.com/journal/10.1002/(ISSN)1097-0142
Citation
Cancer, 2019, v. 125 n. 7, p. 1163-1175 How to Cite?
AbstractBackground: Low‐grade gliomas (LGGs) and low‐grade glioneuronal tumors (LGGNTs) diagnosed during the first year of life carry unique clinical characteristics and challenges in management. However, data on the treatment burden, outcomes, and morbidities are lacking. Methods: A retrospective study of LGGs and LGGNTs diagnosed in patients younger than 12 months at St. Jude Children’s Research Hospital (1986‐2015) was conducted. Results: For the 51 patients (including 31 males), the mean age at diagnosis was 6.47 months (range, 0.17‐11.76 months), and the mean follow‐up period was 11.8 years (range, 0.21‐29.19 years). Tumor locations were hypothalamic/optic pathway (61%), hemispheric (12%), brainstem (12%), cerebellar (8%), and spinal (8%). There were 41 patients with histological diagnoses: 28 had World Health Organization grade 1 tumors, 6 had grade 2 tumors, and 7 had an LGG/LGGNT not definitively graded. Forty‐one patients required an active intervention at diagnosis. Throughout their treatment course, 41 patients eventually underwent tumor‐directed surgeries (median, 2 surgeries; range, 1‐6), 39 received chemotherapy (median, 2 regimens; range, 1‐13), and 21 received radiotherapy. Forty patients experienced disease progression (median, 2 progressions; range, 1‐18). Ten patients died of progression (n = 5), malignant transformation (n = 2), a second cancer (n = 2), or a shunt infection (n = 1). The 10‐year overall survival, progression‐free survival, and radiation‐free survival rates were 85% ± 5.3%, 16.9% ± 5.3%, and 51.2% ± 7.5%, respectively. Forty‐nine patients experienced health deficits (eg, endocrinopathies, obesity, seizures, visual/hearing impairments, neurocognitive impairments, and cerebrovascular disease). Predictors of progression and toxicities were defined. Conclusions: Infantile LGG/LGGNT is a chronic, progressive disease universally associated with long‐term morbidities and requires multidisciplinary intervention.
Persistent Identifierhttp://hdl.handle.net/10722/288134
ISSN
2020 Impact Factor: 6.86
2020 SCImago Journal Rankings: 3.052
PubMed Central ID
ISI Accession Number ID

 

DC FieldValueLanguage
dc.contributor.authorLiu, APY-
dc.contributor.authorHastings, C-
dc.contributor.authorWu, S-
dc.contributor.authorBass, JK-
dc.contributor.authorHeitzer, AM-
dc.contributor.authorAshford, J-
dc.contributor.authorVestal, R-
dc.contributor.authorHoehn, ME-
dc.contributor.authorGhazwani, Y-
dc.contributor.authorAcharya, S-
dc.contributor.authorConklin, HM-
dc.contributor.authorBoop, F-
dc.contributor.authorMerchant, TE-
dc.contributor.authorGajjar, A-
dc.contributor.authorQaddoumi, I-
dc.date.accessioned2020-10-05T12:08:22Z-
dc.date.available2020-10-05T12:08:22Z-
dc.date.issued2019-
dc.identifier.citationCancer, 2019, v. 125 n. 7, p. 1163-1175-
dc.identifier.issn0008-543X-
dc.identifier.urihttp://hdl.handle.net/10722/288134-
dc.description.abstractBackground: Low‐grade gliomas (LGGs) and low‐grade glioneuronal tumors (LGGNTs) diagnosed during the first year of life carry unique clinical characteristics and challenges in management. However, data on the treatment burden, outcomes, and morbidities are lacking. Methods: A retrospective study of LGGs and LGGNTs diagnosed in patients younger than 12 months at St. Jude Children’s Research Hospital (1986‐2015) was conducted. Results: For the 51 patients (including 31 males), the mean age at diagnosis was 6.47 months (range, 0.17‐11.76 months), and the mean follow‐up period was 11.8 years (range, 0.21‐29.19 years). Tumor locations were hypothalamic/optic pathway (61%), hemispheric (12%), brainstem (12%), cerebellar (8%), and spinal (8%). There were 41 patients with histological diagnoses: 28 had World Health Organization grade 1 tumors, 6 had grade 2 tumors, and 7 had an LGG/LGGNT not definitively graded. Forty‐one patients required an active intervention at diagnosis. Throughout their treatment course, 41 patients eventually underwent tumor‐directed surgeries (median, 2 surgeries; range, 1‐6), 39 received chemotherapy (median, 2 regimens; range, 1‐13), and 21 received radiotherapy. Forty patients experienced disease progression (median, 2 progressions; range, 1‐18). Ten patients died of progression (n = 5), malignant transformation (n = 2), a second cancer (n = 2), or a shunt infection (n = 1). The 10‐year overall survival, progression‐free survival, and radiation‐free survival rates were 85% ± 5.3%, 16.9% ± 5.3%, and 51.2% ± 7.5%, respectively. Forty‐nine patients experienced health deficits (eg, endocrinopathies, obesity, seizures, visual/hearing impairments, neurocognitive impairments, and cerebrovascular disease). Predictors of progression and toxicities were defined. Conclusions: Infantile LGG/LGGNT is a chronic, progressive disease universally associated with long‐term morbidities and requires multidisciplinary intervention.-
dc.languageeng-
dc.publisherJohn Wiley & Sons, Inc. The Journal's web site is located at http://onlinelibrary.wiley.com/journal/10.1002/(ISSN)1097-0142-
dc.relation.ispartofCancer-
dc.rightsPreprint This is the pre-peer reviewed version of the following article: [FULL CITE], which has been published in final form at [Link to final article using the DOI]. This article may be used for non-commercial purposes in accordance with Wiley Terms and Conditions for Use of Self-Archived Versions. Postprint This is the peer reviewed version of the following article: [FULL CITE], which has been published in final form at [Link to final article using the DOI]. This article may be used for non-commercial purposes in accordance with Wiley Terms and Conditions for Use of Self-Archived Versions.-
dc.subjectinfant-
dc.subjectlow‐grade glioma-
dc.subjectmorbidities-
dc.subjectprognostic factors-
dc.subjecttreatment burden-
dc.titleTreatment burden and long‐term health deficits of patients with low‐grade gliomas or glioneuronal tumors diagnosed during the first year of life-
dc.typeArticle-
dc.identifier.emailLiu, APY: apyliu@hku.hk-
dc.identifier.authorityLiu, APY=rp01357-
dc.description.naturelink_to_OA_fulltext-
dc.identifier.doi10.1002/cncr.31918-
dc.identifier.pmid30620400-
dc.identifier.pmcidPMC6420385-
dc.identifier.scopuseid_2-s2.0-85059701898-
dc.identifier.hkuros315669-
dc.identifier.volume125-
dc.identifier.issue7-
dc.identifier.spage1163-
dc.identifier.epage1175-
dc.identifier.isiWOS:000461867800018-
dc.publisher.placeUnited States-
dc.identifier.issnl0008-543X-

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