Successful treatment of childhood intramedullary spinal cord astrocytomas with irinotecan and cisplatin

Abstract

Childhood spinal cord astrocytomas are rare diseases, and their management is controversial. We report here our successful experience using irinotecan and cisplatin in three consecutive infants with progressing intramedullary astrocytomas. The first patient was a 16-month-old girl who presented with a grade III intramedullary astrocytoma that rapidly progressed after surgery and adjuvant chemotherapy. Weekly irinotecan (50 mg/m(2)) and cisplatin (30 mg/m(2)) for four consecutive weeks (one cycle) for a total of four cycles (I/C regimen) was used in order to avoid or delay radiotherapy. Radiological complete remission was achieved 10 months after completion of therapy, and 3.5 years after diagnosis the patient remains disease free. The second patient was a 19-month-old boy with a C3-T4 grade II intramedullary astrocytoma who received up-front vincristine and carboplatin for two months but remained clinically symptomatic. A followup MRI showed a larger tumor, and the patient was switched to the I/C regimen. A marked clinical improvement occurred after the first cycle, and MRI showed a very good partial remission at the end of therapy. At 16 months after diagnosis, the patient remains disease free. The third patient was a 10-month-old girl with a C2-T3 grade II intramedullary astrocytoma. She presented with severe pain that became steroid dependent during the month she was treated with the vincristine-carboplatin regimen. When she was switched to the I/C regimen, the clinical symptoms responded within days. MRI at the end of therapy showed a significant reduction in tumor size, and one year after diagnosis the patient remains symptom free. Using this I/C regimen for childhood intramedullary astrocytoma, we obtained remarkable clinicoradiological responses while avoiding the use of radiotherapy.

Pediatric spinal cord astrocytomas: a retrospective study of 348 patients from the SEER database

“…11 Additionally, various chemotherapy regimens have been proposed over the years, mostly drawing from studies of intracranial gliomas; however, due to small sample sizes, there has been no agreed-upon chemotherapy protocol. 20 Patients who were documented in this study early on certainly may have been treated differently. However, we saw no difference in survival between the 3 decades during which these data were collected.…”

Temozolomide for recurrent low‐grade spinal cord gliomas in adults

“…In addition from surgery and radiotherapy, to our knowledge there are few data regarding the utility of chemotherapy in the treatment of spinal cord gliomas. [11][12][13][14][15][16] Trials of chemotherapy for recurrent spinal cord gliomas are challenging because these tumors are infrequent (10-fold less common than brain tumors), and consequently have not been considered by brain tumor consortia. Therefore, the literature regarding chemotherapy of spinal cord tumors is comprised of case reports that provide limited practical guidance.…”

“…Most often, these case reports, similar to the current study, use chemotherapy regimens used for intracranial gliomas. [11][12][13][14][15][16] Increasingly, stereotactic reradiation is administered for spinal cord tumors through the CyberKnife radiotherapy system, a therapy used in 2 patients in this series. To our knowledge, the role of reradiation of spinal cord tumors is unclear because there are no prospective trials that address this issue.…”

“…4 Less clear is the utility of chemotherapy in the management of intramedullary spinal cord gliomas, although anecdotal evidence exists for use at the time of tumor recurrence. [11][12][13][14][15][16] The majority of reports using chemotherapy in this context have used nitrosourea-based chemotherapy. Temozolomide (TMZ), an oral methylating chemotherapy, has profoundly altered the management of adult intracranial gliomas, and is now considered standard therapy for adults with newly diagnosed glioblastoma.…”

Local non-viral gene delivery of apoptin delays the onset of paresis in an experimental model of intramedullary spinal cord tumor

“…Although some studies report benefits from the use of radiation therapy as part of the therapeutic regimen, [8][9][10][11][12] concerns remain about the effect of radiation on children. Moreover, chemotherapeutic management of IMSCT has its supporters, [13][14][15][16][17] but not enough studies have been done to support a field-wide consensus.…”