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

Anthony P.Y. Liu, Camden Hastings, Shengjie Wu, Johnnie K. Bass, Andrew M. Heitzer, Jason Ashford, Robert Vestal, Mary E. Hoehn, Yahya Ghazwani, Sahaja Acharya, Heather M. Conklin, Frederick Boop, Thomas E. Merchant, Amar Gajjar, Ibrahim Qaddoumi

Research output: Contribution to journalArticle

Abstract

Background: 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.

Original languageEnglish (US)
Pages (from-to)1163-1175
Number of pages13
JournalCancer
Volume125
Issue number7
DOIs
StatePublished - Apr 1 2019

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Glioma
Health
Neoplasms
Therapeutics
Morbidity
Cerebrovascular Disorders
Second Primary Neoplasms
Vision Disorders
Hearing Loss
Brain Stem
Disease-Free Survival
Disease Progression
Seizures
Chronic Disease
Radiotherapy
Survival Rate
Retrospective Studies
Obesity
Radiation
Drug Therapy

All Science Journal Classification (ASJC) codes

  • Oncology
  • Cancer Research

Cite this

Liu, A. P. Y., Hastings, C., Wu, S., Bass, J. K., Heitzer, A. M., Ashford, J., ... Qaddoumi, I. (2019). Treatment burden and long-term health deficits of patients with low-grade gliomas or glioneuronal tumors diagnosed during the first year of life. Cancer, 125(7), 1163-1175. https://doi.org/10.1002/cncr.31918

Treatment burden and long-term health deficits of patients with low-grade gliomas or glioneuronal tumors diagnosed during the first year of life. / Liu, Anthony P.Y.; Hastings, Camden; Wu, Shengjie; Bass, Johnnie K.; Heitzer, Andrew M.; Ashford, Jason; Vestal, Robert; Hoehn, Mary E.; Ghazwani, Yahya; Acharya, Sahaja; Conklin, Heather M.; Boop, Frederick; Merchant, Thomas E.; Gajjar, Amar; Qaddoumi, Ibrahim.

In: Cancer, Vol. 125, No. 7, 01.04.2019, p. 1163-1175.

Research output: Contribution to journalArticle

Liu, APY, Hastings, C, Wu, S, Bass, JK, Heitzer, AM, Ashford, J, Vestal, R, Hoehn, ME, Ghazwani, Y, Acharya, S, Conklin, HM, Boop, F, Merchant, TE, Gajjar, A & Qaddoumi, I 2019, 'Treatment burden and long-term health deficits of patients with low-grade gliomas or glioneuronal tumors diagnosed during the first year of life', Cancer, vol. 125, no. 7, pp. 1163-1175. https://doi.org/10.1002/cncr.31918
Liu, Anthony P.Y. ; Hastings, Camden ; Wu, Shengjie ; Bass, Johnnie K. ; Heitzer, Andrew M. ; Ashford, Jason ; Vestal, Robert ; Hoehn, Mary E. ; Ghazwani, Yahya ; Acharya, Sahaja ; Conklin, Heather M. ; Boop, Frederick ; Merchant, Thomas E. ; Gajjar, Amar ; Qaddoumi, Ibrahim. / Treatment burden and long-term health deficits of patients with low-grade gliomas or glioneuronal tumors diagnosed during the first year of life. In: Cancer. 2019 ; Vol. 125, No. 7. pp. 1163-1175.
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abstract = "Background: 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.",
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T1 - Treatment burden and long-term health deficits of patients with low-grade gliomas or glioneuronal tumors diagnosed during the first year of life

AU - Liu, Anthony P.Y.

AU - Hastings, Camden

AU - Wu, Shengjie

AU - Bass, Johnnie K.

AU - Heitzer, Andrew M.

AU - Ashford, Jason

AU - Vestal, Robert

AU - Hoehn, Mary E.

AU - Ghazwani, Yahya

AU - Acharya, Sahaja

AU - Conklin, Heather M.

AU - Boop, Frederick

AU - Merchant, Thomas E.

AU - Gajjar, Amar

AU - Qaddoumi, Ibrahim

PY - 2019/4/1

Y1 - 2019/4/1

N2 - Background: 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.

AB - Background: 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.

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