Risk factors for relapse and guidance for follow-up of extracranial immature teratomas in children: A systematic review

Abstract

Objective

This systematic review was performed to identify risk factors for recurrence of extracranial immature teratomas (ITs) and to provide recommendations for post-operative follow-up.

Methods

The search focused on risk factors for recurrent disease and/or follow-up management of extracranial IT in the paediatric population. The initial search, conducted across three databases, yielded 2131 articles. Ultimately six articles were selected for inclusion in this review, comprising a total of 379 patients.

Results

The six studies included were heterogeneous, comprising both retrospective and prospective designs, and were conducted across various countries and continents. The majority of patients were diagnosed with an ovarian IT and were thus female. In addition, the majority of ITs were classified as grade 3 and/or stage 1. Although, the treatment approach was predominantly surgical resection, five articles incorporated adjuvant chemotherapy for more advanced ITs. A total of 29 recurrences were documented between two- and 36-months post-treatment, with the highest incidence of recurrence occurring within the first year after treatment. Higher stage (stage II-III), higher grade (grade 3) and incomplete surgery were identified as risk factors for extracranial IT recurrence. The overall survival rates were excellent in all studies. Based on the findings our recommended follow-up guidelines are differentiated according to patients at risk and IT localisation.

Conclusions

This review offers insight into the risk factors associated with recurrence and provides recommendations for follow-up in paediatric extracranial IT patients. Further research is required to explore the identified associations for recurrence, with the aim of providing higher-level evidence that will further enhance the recommended follow-up regimen.