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  • Title: Markedly improved survival in malignant sacro-coccygeal teratomas--16 years, experience.
    Author: Misra D, Pritchard J, Drake DP, Kiely EM, Spitz L.
    Journal: Eur J Pediatr Surg; 1997 Jun; 7(3):152-5. PubMed ID: 9241501.
    Abstract:
    AIM OF STUDY: To see the impact of cisplatin- and carboplatin-based protocols on survival in malignant sacrococcygeal teratomas (SCT). METHODS: Twenty infants and children with malignant SCT were treated at the Hospital for Sick Children, Great Ormond Street, London, over a 16-year period from 1979 to 1994. There were 5 males and 15 females, age ranged from 1 day to 3.5 years (mean 18 months). RESULTS: Twelve patients (60%) had distant metastasis: 9 lung metastasis, 2 liver involvement, 2 bony metastases, 1 cerebral metastasis and 1 bilateral inguinal lymph node deposits. From 1982 to 1986, patients received the BEP chemotherapy protocol which included cis-platinum, bleomycin and etoposide (VP 16). After 1986, cis-platinum was replaced with carboplatin in the new JEB protocol. Patient 1 did not receive any chemotherapy, Patients 2-4 received varying protocols (2 deaths), Patients 5-8 received the BEP regime (1 death) and Patients 9-20 received JEB (1 death). The first three deaths were due to uncontrolled local disease and/or metastasis, while the latter death was due to bleomycin toxicity. Overall, 9 of 12 (75%) patients with distant metastasis survived as opposed to 7 of 8 (88%) patients with localised disease. Of the 12 patients who received the JEB protocol, 11 (92%) survived, including 7 patients with metastatic disease and 2 with local recurrence. Seven patients (35%) had relapse while on treatment or follow-up, 4 of these are disease-free with further therapy. In 6 of these children, serum AFP rose before there was clinical or radiological evidence of relapse. In 2 other patients, further chemotherapy was recommenced solely on the basis of rising serum AFP, these patients did not subsequently develop overt metastasis. CONCLUSIONS: We conclude that the treatment of choice for malignant SCT is the JEB regime, to be given for 4 courses or to be continued for 2 courses beyond documented Complete Response (CR). Excision of the primary tumour and coccyx should be done in all cases even if a CR has been documented. Metastases not responding to chemotherapy would need appropriate surgery, radiotherapy is hardly ever needed. An overall cure rate exceeding 90% can be expected.
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