Hyperthermic intraperitoneal chemotherapy for recurrent pediatric genitourinary rhabdomyosarcoma: Single center experience of first 3 cases
Patricio C. Gargollo, MD, Nitin Abrol, MBBS, Travis E. Grotz, MD, Nadia Laack, MD, Candace F. Granberg, MD.
Mayo Clinic, Rochester, MN, USA.
BACKGROUND: Rhabdomyosarcoma (RMS) is the most common soft tissue sarcoma in children. Despite multimodal treatment approaches, approximately quarter of patients present with failure. Children with recurrent intraperitoneal RMS have very poor prognosis. There are limited guidelines for the management of recurrent RMS. Hyperthermic Intraperitoneal Chemotherapy (HIPEC) with cytoreductive surgery (CRS) has an established role in adult intraperitoneal carcinomatosis. We present our experience of using HIPEC for recurrent pediatric intraperitoneal rhabdomyosarcoma.
METHODS: We offered HIPEC to cases with relapsed RMS only if complete cytoreduction seemed feasible. The data on disease related (stage at presentation; previous chemotherapy, surgeries, or radiation therapy) and patient related (age, gender) variables were prospectively recorded. Three dimensional modelling was done to help with surgical planning. HIPEC was performed by open technique using cisplatin (100 mg/m2) alone or in combination with doxorubicin (15 mg/m2). All patients also received systemic thiosulfate. All patients were on regular follow up after discharge from the hospital.
RESULTS: Three boys with median age of 5.75 years (range: 4.1 - 6.8 years) received HIPEC with cytoreduction surgery. Two boys had stage 3 group 3 embryonal RMS while one had undifferentiated variant at the time of initial presentation. All were treated elsewhere for primary disease and were referred to our center for relapse after treatment. Median time for relapse was 16 months (range: 4 - 18 months) from the time of first diagnosis. Complete cytoreduction was possible in all cases. One case required excision of iliac vessels en block with the tumor followed by cryopreserved interposition grafting and intraoperative radiation therapy for local control. All patients had uneventful course in the hospital and were discharged to home within 2 weeks. One patient developed non-occlusive thrombus in the iliac vein graft and other developed mild right side hydronephrosis. All patients are disease free on follow up (2-8 months).
CONCLUSIONS: HIPEC with complete cytoreductive surgery is a safe treatment option for children with relapsed intraperitoneal rhabdomyosarcoma. However, long term efficacy of this procedure is unknown at present. Well-designed multicenter trial enrolling large number cases is required to study its efficacy.
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