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- Title
Intracranial growing teratoma syndrome (iGTS): an international case series and review of the literature.
- Authors
Michaiel, George; Strother, Douglas; Gottardo, Nicholas; Bartels, Ute; Coltin, Hallie; Hukin, Juliette; Wilson, Beverly; Zelcer, Shayna; Hansford, Jordan R.; Hassall, Timothy; AbdelBaki, Mohamed S.; Cole, Kristina A.; Hoffman, Lindsey; Smiley, Natasha P.; Smith, Amy; Vinitsky, Anna; Vitanza, Nicholas A.; Wright, Avery; Yeo, Kee K.; Chow, Lionel M. L.
- Abstract
Purpose: Intracranial growing teratoma syndrome (iGTS) is a rare phenomenon of paradoxical growth of a germ cell tumor (GCT) during treatment despite normalization of tumor markers. We sought to evaluate the frequency, clinical characteristics and outcome of iGTS in Western countries. Methods: Pediatric patients from 22 North American and Australian institutions diagnosed with iGTS between 2000 and 2017 were retrospectively evaluated. Results: From a total of 777 cases of central nervous system (CNS) GCT, 39 cases of iGTS were identified for an overall frequency of 5%. Pineal region was a more frequent location for iGTS as compared to cases of GCT without iGTS (p < 0.00001). In patients with an initial tissue diagnosis of GCT, immature teratoma was present in 50%. Serum AFP or ßhCG was detectable in 87% of patients (median values 66 ng/mL and 44 IU/L, respectively). iGTS occurred at a median of 2 months (range 0.5–32) from diagnosis, in the majority of patients. All patients underwent surgical resection, leading to gross total resection in 79%. Following surgery, all patients resumed adjuvant therapy or post treatment follow-up for GCT. At a median follow-up of 5.3 years (range 0.2–11.8), 37 (95%) of patients are alive, including 5 with stable residual mass. Conclusion: iGTS occurs in 5% of patients with GCT in Western countries. Tumors of the pineal region and GCT containing immature teratoma appear to be associated with a higher risk of developing iGTS. Complete surgical resection is the mainstay of treatment. Overall survival of patients developing iGTS remains favorable.
- Subjects
GERM cell tumors; SURGICAL excision; LITERATURE reviews; CENTRAL nervous system; TUMOR markers; IMMUNE reconstitution inflammatory syndrome
- Publication
Journal of Neuro-Oncology, 2020, Vol 147, Issue 3, p721
- ISSN
0167-594X
- Publication type
Article
- DOI
10.1007/s11060-020-03486-9