ABSTRACT
Malignant vascular tumors are exceedingly rare in childhood. Generally, their prognosis is dependent from a microscopically complete surgical resection. We observed the case of a 4-year-old boy affected by malignant endovascular papillary angioendothelioma, a rare vascular tumor of intermediate malignancy, involving all his left leg and partially the pelvis. Its very large size hampered any surgical intervention. However, we could demonstrate high sensitivity of the tumor to lyposomal anthracyclines and the child was eventually cured by the intra-arterial administration of carboplatin and doxorubicin coupled with local hyperthermia. This experience probably represents the first step toward an effective treatment of malignant vascular tumors in infancy.
Subject(s)
Antineoplastic Combined Chemotherapy Protocols/therapeutic use , Carcinoma, Papillary/therapy , Doxorubicin/analogs & derivatives , Hemangioendothelioma/therapy , Hyperthermia, Induced , Polyethylene Glycols/therapeutic use , Vascular Neoplasms/therapy , Carboplatin/administration & dosage , Child, Preschool , Combined Modality Therapy , Doxorubicin/administration & dosage , Doxorubicin/therapeutic use , Humans , Infusions, Intra-Arterial , MaleABSTRACT
Progressive or recurrent high-grade gliomas are characterized by a very poor prognosis, and the relevance of second-line chemotherapy is still unassessed. Although it has been reported that liposomal anthracyclines and carboplatin show some activity in these patients, their association has never been investigated. We have treated six children with recurrent high-grade glioma after surgery, radiotherapy and chemotherapy, and one child with progressive teratoid/rhabdoid tumor with the combination of liposomal daunorubicin and carboplatin plus etoposide. Five out of seven children showed a major response and the 29 month progression-free survival was 38%. The above regimen was feasible and children showed only little and transient hematological toxicity. In our opinion, these results justify further investigation of the above combination chemotherapy for recurrent or progressive malignant brain tumors in children.
Subject(s)
Antibiotics, Antineoplastic/administration & dosage , Antineoplastic Agents, Phytogenic/administration & dosage , Antineoplastic Agents/administration & dosage , Brain Neoplasms/drug therapy , Carboplatin/administration & dosage , Daunorubicin/administration & dosage , Etoposide/administration & dosage , Antibiotics, Antineoplastic/adverse effects , Antineoplastic Agents/adverse effects , Antineoplastic Agents, Phytogenic/adverse effects , Brain Neoplasms/diagnosis , Carboplatin/adverse effects , Child , Child, Preschool , Daunorubicin/adverse effects , Etoposide/adverse effects , Female , Humans , Liposomes , Magnetic Resonance Imaging , Male , Survival Analysis , Treatment OutcomeABSTRACT
This report refers to a 3-month-old male, with a residual choroid plexus carcinoma following partial resection, who was successfully treated with sequential chemotherapy without any postoperative radiation therapy. Along with carboplatin, we also used doxorubicin and methotrexate, hypothesizing that, given the patient's age, the blood-brain barrier should not hamper drug delivery to the tumor. According to this hypothesis, the treatment achieved complete remission of the disease, which lasts 27 months after the diagnosis. This result deserves further studies to assess the possible curative role of chemotherapy in very young patients suffering from choroid plexus carcinoma.
