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Clinical and neuropsychological outcome of pediatric non-midline central nervous system germinoma treated with chemotherapy and reduced dose/volume irradiation: The Children's Hospital Los Angeles experience.
Pediatr Blood Cancer 2019; :e27983PB

Abstract

BACKGROUND

Germ cell tumors (GCT) arising from non-midline structures (basal ganglia, thalamus, and posterior fossa) are rare. Although patients with midline (pineal and suprasellar) germinoma have excellent survival with chemotherapy and whole ventricular irradiation (WVI), germinoma in non-midline locations have traditionally been treated with craniospinal irradiation (CSI) or whole brain irradiation (WBI) to achieve similar outcomes. However, CSI and WBI are associated with significant long-term neuropsychological sequelae.

METHODS

We describe the clinical and neuropsychological outcomes of patients with non-midline germinoma treated at the Children's Hospital Los Angeles between 1990 and 2015.

RESULTS

Nine patients had basal ganglia/thalamic germinoma and one patient had a cerebellar primary. Eight patients received chemotherapy followed by reduced dose/volume irradiation, whereas two patients received chemotherapy alone as upfront therapy. One patient in the chemotherapy alone group relapsed after 4.3 years and was salvaged with CSI plus boost. The overall survival for the entire cohort was 100% at a median follow-up of 8.5 years. Neuropsychological data were available for six patients at a median of five months (baseline) and 4.2 years (follow-up) post-diagnosis. At four-year follow-up, data available revealed intact overall cognitive ability, verbal memory, and executive functioning, but persistent deficits in fine motor function. Comparison of baseline to follow-up suggests a downward trend in working memory, planning/problem-solving, verbal memory, and visuospatial integration.

CONCLUSION

Chemotherapy followed by reduced dose/volume of irradiation is an effective strategy resulting in long-term survival in patients with non-midline germinoma. Neuropsychological data suggest relatively minimal morbidity over time.

Authors+Show Affiliations

Children's Center for Cancer and Blood Diseases, Division of Hematology, Oncology and Blood and Marrow Transplantation, Children's Hospital Los Angeles, Los Angeles, California. Dana-Farber/Boston Children's Cancer and Blood Disorders Center, Boston, Massachusetts.Children's Center for Cancer and Blood Diseases, Division of Hematology, Oncology and Blood and Marrow Transplantation, Children's Hospital Los Angeles, Los Angeles, California.Children's Center for Cancer and Blood Diseases, Division of Hematology, Oncology and Blood and Marrow Transplantation, Children's Hospital Los Angeles, Los Angeles, California.Children's Center for Cancer and Blood Diseases, Division of Hematology, Oncology and Blood and Marrow Transplantation, Children's Hospital Los Angeles, Los Angeles, California. University of Southern California, Department of Radiation Oncology, Los Angeles, California.Children's Center for Cancer and Blood Diseases, Division of Hematology, Oncology and Blood and Marrow Transplantation, Children's Hospital Los Angeles, Los Angeles, California.The Neuro-oncology Program, Division of Hematology, Oncology and BMT, Nationwide Children's Hospital and The Ohio State University, Columbus, Ohio.Children's Center for Cancer and Blood Diseases, Division of Hematology, Oncology and Blood and Marrow Transplantation, Children's Hospital Los Angeles, Los Angeles, California. The Alabama Center for Childhood Cancer and Blood Disorders at Children's of Alabama, University of Alabama at Birmingham (UAB), Birmingham, Alabama.

Pub Type(s)

Journal Article

Language

eng

PubMed ID

31502379

Citation

Yeo, Kee Kiat, et al. "Clinical and Neuropsychological Outcome of Pediatric Non-midline Central Nervous System Germinoma Treated With Chemotherapy and Reduced Dose/volume Irradiation: the Children's Hospital Los Angeles Experience." Pediatric Blood & Cancer, 2019, pp. e27983.
Yeo KK, Kayser K, Margol AS, et al. Clinical and neuropsychological outcome of pediatric non-midline central nervous system germinoma treated with chemotherapy and reduced dose/volume irradiation: The Children's Hospital Los Angeles experience. Pediatr Blood Cancer. 2019.
Yeo, K. K., Kayser, K., Margol, A. S., Wong, K. K., Robison, N., Finlay, J., & Dhall, G. (2019). Clinical and neuropsychological outcome of pediatric non-midline central nervous system germinoma treated with chemotherapy and reduced dose/volume irradiation: The Children's Hospital Los Angeles experience. Pediatric Blood & Cancer, pp. e27983. doi:10.1002/pbc.27983.
Yeo KK, et al. Clinical and Neuropsychological Outcome of Pediatric Non-midline Central Nervous System Germinoma Treated With Chemotherapy and Reduced Dose/volume Irradiation: the Children's Hospital Los Angeles Experience. Pediatr Blood Cancer. 2019 Sep 10;e27983. PubMed PMID: 31502379.
* Article titles in AMA citation format should be in sentence-case
TY - JOUR T1 - Clinical and neuropsychological outcome of pediatric non-midline central nervous system germinoma treated with chemotherapy and reduced dose/volume irradiation: The Children's Hospital Los Angeles experience. AU - Yeo,Kee Kiat, AU - Kayser,Kimberly, AU - Margol,Ashley S, AU - Wong,Kenneth K, AU - Robison,Nathan, AU - Finlay,Jonathan, AU - Dhall,Girish, Y1 - 2019/09/10/ PY - 2019/06/05/received PY - 2019/08/19/revised PY - 2019/08/20/accepted PY - 2019/9/11/entrez KW - basal ganglia KW - central nervous system KW - germinoma KW - neuropsychological test KW - radiation KW - thalamus SP - e27983 EP - e27983 JF - Pediatric blood & cancer JO - Pediatr Blood Cancer N2 - BACKGROUND: Germ cell tumors (GCT) arising from non-midline structures (basal ganglia, thalamus, and posterior fossa) are rare. Although patients with midline (pineal and suprasellar) germinoma have excellent survival with chemotherapy and whole ventricular irradiation (WVI), germinoma in non-midline locations have traditionally been treated with craniospinal irradiation (CSI) or whole brain irradiation (WBI) to achieve similar outcomes. However, CSI and WBI are associated with significant long-term neuropsychological sequelae. METHODS: We describe the clinical and neuropsychological outcomes of patients with non-midline germinoma treated at the Children's Hospital Los Angeles between 1990 and 2015. RESULTS: Nine patients had basal ganglia/thalamic germinoma and one patient had a cerebellar primary. Eight patients received chemotherapy followed by reduced dose/volume irradiation, whereas two patients received chemotherapy alone as upfront therapy. One patient in the chemotherapy alone group relapsed after 4.3 years and was salvaged with CSI plus boost. The overall survival for the entire cohort was 100% at a median follow-up of 8.5 years. Neuropsychological data were available for six patients at a median of five months (baseline) and 4.2 years (follow-up) post-diagnosis. At four-year follow-up, data available revealed intact overall cognitive ability, verbal memory, and executive functioning, but persistent deficits in fine motor function. Comparison of baseline to follow-up suggests a downward trend in working memory, planning/problem-solving, verbal memory, and visuospatial integration. CONCLUSION: Chemotherapy followed by reduced dose/volume of irradiation is an effective strategy resulting in long-term survival in patients with non-midline germinoma. Neuropsychological data suggest relatively minimal morbidity over time. SN - 1545-5017 UR - https://www.unboundmedicine.com/medline/citation/31502379/Clinical_and_neuropsychological_outcome_of_pediatric_non-midline_central_nervous_system_germinoma_treated_with_chemotherapy_and_reduced_dose/volume_irradiation:_The_Children's_Hospital_Los_Angeles_experience L2 - https://doi.org/10.1002/pbc.27983 DB - PRIME DP - Unbound Medicine ER -