A case series and literature review on 98 pediatric patients of germ cell tumor developing growing teratoma syndrome

Author:

Hsieh Ming‐Yun123,Chen Hsin‐Hung24,Lee Chih‐Ying256,Hung Giun‐Yi256ORCID,Chang Tsung‐Yen7,Chen Shih‐Hsiang7,Lai Jin‐Yao8,Jaing Tang‐Her7,Cheng Chao‐Neng9,Chen Jiann‐Shiuh9,Tsai Hsin‐Lin210,Yu Ting‐Yen11,Hou Ming‐Hsin25,Ho Cheng‐Yin25,Yen Hsiu‐Ju256ORCID

Affiliation:

1. Department of Pediatrics Kaohsiung Veterans General Hospital Kaohsiung Taiwan

2. School of Medicine, National Yang‐Ming Chiao‐Tung University Hsinchu Taiwan

3. Department of Biological Sciences National Sun Yat‐Sen University Kaohsiung Taiwan

4. Division of Pediatric Neurosurgery Neurological Institute, Taipei Veterans General Hospital Taipei Taiwan

5. Department of Pediatrics Taipei Veterans General Hospital Taipei Taiwan

6. Department of Life Science National Taiwan Normal University Taipei Taiwan

7. Department of Pediatrics Chang Gung Memorial Hospital, School of Medicine, Chang Gung University Taoyuan Taiwan

8. Department of Pediatric Surgery Chang Gung Memorial Hospital, School of Medicine, Chang Gung University Taoyuan Taiwan

9. Department of Pediatrics National Cheng Kung University Hospital, College of Medicine, National Cheng Kung University Tainan Taiwan

10. Division of Pediatric Surgery, Department of Surgery Taipei Veterans General Hospital Taipei Taiwan

11. Department of Pediatrics Far Eastern Memorial Hospital New Taipei City Taiwan

Abstract

AbstractIntroductionMalignant germ cell tumors (MGCTs) can develop either extracranially or intracranially. Growing teratoma syndrome (GTS) may develop in these patients following chemotherapy. Reports on the clinical characteristics and outcomes of GTS in children with MGCTs are limited.MethodsWe retrospectively collected the data, including the clinical characteristics and outcomes of five patients in our series and 93 pediatric patients selected through a literature review of MGCTs. This study aimed to analyze survival outcomes and risk factors for subsequent events in pediatric patients with MGCTs developing GTS.ResultsThe sex ratio was 1.09 (male/female). In total, 52 patients (53.1%) had intracranial MGCTs. Compared with patients with extracranial GCTs, those with intracranial GCTs were younger, predominantly boys, had shorter intervals between MGCT and GTS, and had GTS mostly occurring over the initial site (all p < 0.001). Ninety‐five patients (96.9%) were alive. However, GTS recurrence (n = 14), GTS progression (n = 9), and MGCT recurrence (n = 19) caused a substantial decrease in event‐free survival (EFS). Multivariate analyses showed that the only significant risk factors for these events were incomplete GTS resection and different locations of GCT and GTS. Patients without any risk had a 5‐year EFS of 78.8% ± 7.8%, whereas those with either risk had 41.7% ± 10.2% (p < 0.001).ConclusionFor patients with high‐risk features, every effort should be made to closely monitor, completely remove, and pathologically prove any newly developed mass to guide relevant treatment. Further studies incorporating the risk factors into treatment strategies may be required to optimize adjuvant therapy.

Funder

Ministry of Health and Welfare

Taipei Veterans General Hospital

Publisher

Wiley

Subject

Cancer Research,Radiology, Nuclear Medicine and imaging,Oncology

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