A case of relapsing spinal atypical teratoid/rhabdoid tumor (AT/RT) responding to vinorelbine, cyclophosphamide, and celecoxib

Gotti, Giacomo; Biassoni, Veronica; Schiavello, Elisabetta; Spreafico, Filippo; Antonelli, Manila; Calareso, Giuseppina; Pecori, Emilia; Gandola, Lorenza; Massimino, Maura

doi:10.1007/s00381-015-2755-x

Cited by 16 publications

(16 citation statements)

References 11 publications

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“…Furthermore, it has been shown that infected cells can bioactivate CPA, and the active chemotherapeutic metabolites can diffuse and kill non-infected bystander tumor cells 26, 28, 29. Because CPA is commonly used in chemotherapy regimens to treat both medulloblastoma and AT/RT, we postulated that studies investigating its inclusion with rRp450 in the treatment regimen were rational and may yield similar improvements 30, 31, 32, 33, 34. To evaluate the sensitivity to CPA in the context of rRp450 infection, we infected medulloblastoma and AT/RT cells with rRp450 in the presence or absence of CPA.…”

Section: Discussionmentioning

confidence: 99%

Oncolytic Herpes Virus rRp450 Shows Efficacy in Orthotopic Xenograft Group 3/4 Medulloblastomas and Atypical Teratoid/Rhabdoid Tumors

Studebaker

Hutzen

Pierson

et al. 2017

Molecular Therapy - Oncolytics

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Pediatric brain tumors including medulloblastoma and atypical teratoid/rhabdoid tumor are associated with significant mortality and treatment-associated morbidity. While medulloblastoma tumors within molecular subgroups 3 and 4 have a propensity to metastasize, atypical teratoid/rhabdoid tumors frequently afflict a very young patient population. Adjuvant treatment options for children suffering with these tumors are not only sub-optimal but also associated with many neurocognitive obstacles. A potentially novel treatment approach is oncolytic virotherapy, a developing therapeutic platform currently in early-phase clinical trials for pediatric brain tumors and recently US Food and Drug Administration (FDA)-approved to treat melanoma in adults. We evaluated the therapeutic potential of the clinically available oncolytic herpes simplex vector rRp450 in cell lines derived from medulloblastoma and atypical teratoid/rhabdoid tumor. Cells of both tumor types were supportive of virus replication and virus-mediated cytotoxicity. Orthotopic xenograft models of medulloblastoma and atypical teratoid/rhabdoid tumors displayed significantly prolonged survival following a single, stereotactic intratumoral injection of rRp450. Furthermore, addition of the chemotherapeutic prodrug cyclophosphamide (CPA) enhanced rRp450’s in vivo efficacy. In conclusion, oncolytic herpes viruses with the ability to bioactivate the prodrug CPA within the tumor microenvironment warrant further investigation as a potential therapy for pediatric brain tumors.

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Section: Discussionmentioning

confidence: 99%

Oncolytic Herpes Virus rRp450 Shows Efficacy in Orthotopic Xenograft Group 3/4 Medulloblastomas and Atypical Teratoid/Rhabdoid Tumors

Studebaker

Hutzen

Pierson

et al. 2017

Molecular Therapy - Oncolytics

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“…[ 7 10 11 12 ] Better prognoses correlate with older age, localized disease, and adequate resection. [ 5 ]…”

Section: Discussionmentioning

confidence: 99%

“…Findings within the spinal cord are rare. In particular, there have been only five prior cases of primary spinal AT/RT in adult patients[ 3 5 6 12 14 ] [ Table 1 ]. Patients typically present with axial neck or back pain and radiculopathy; only one exhibited cauda equina syndrome requiring acute surgical resection and decompression.…”

Section: Discussionmentioning

confidence: 99%

“…Immediate multimodal treatment has been advocated, including gross tumor resection, followed by high-dose chemotherapy with autologous hematopoietic stem cells rescue and radiotherapy. [ 5 11 ] Metronomic chemotherapy has also been utilized in pediatric patients with recurrent and refractory solid tumors (e.g., treatment with drugs for an extended period, targeting tumor vasculature instead of tumor cells). [ 1 4 ] Gotti et al .…”

Section: Discussionmentioning

confidence: 99%

“…Atypical teratoid/rhabdoid tumor (AT/RT) is an aggressive neoplasm that constitutes approximately 6% of pediatric central nervous system (CNS) tumors. [ 5 9 ] Primary spinal AT/RT, especially within an adult patient, is rare. [ 3 5 6 12 14 ]…”

Section: Introductionmentioning

confidence: 99%

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Primary atypical teratoid/rhabdoid tumor of the spine in an adult patient

Patel

Nguyen

et al. 2016

Surg Neurol Int

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Background:Atypical teratoid/rhabdoid tumor (AT/RT) is an aggressive neoplasm of the central nervous system that generally arises intracranially in patients under 2 years of age. Primary spinal AT/RT in an adult is rare.Case Description:A 23-year-old female presented with left lower extremity sciatica attributed to a magnetic resonance imaging (MRI)-documented intradural mass between L2 and L4. The lesion was biopsied (was unresectable) and treated with high-dose chemotherapy (methotrexate, vincristine, cyclophosphamide, etoposide, and cisplatin) with autologous hematopoietic stem cells rescue, followed by 2 months of radiation therapy (36 Gy to craniospinal axis, 20 Gy to lumbar region) with concurrent temozolomide; the latter was discontinued after 3 weeks due to myelosuppression. Tumor relapsed 1 year later at C7–T1 level. She was started on oral metronomic therapy, and bevacizumab was added 2 months later. Three months later, a cervical MRI showed progression of the tumor, along with new lesions in the thoracic/lumbar spine plus intracranial punctate nodular tumors. Following resection of the C7/T1 lesion, she was started on palliative alisertib; a month later, a cranial computed tomography showed progression of her disease with hydrocephalus. Treatment was discontinued, and she expired 12 months after initial diagnosis.Conclusion:Primary spinal AT/RT in the adult patient is rare. The pathology is associated with early recurrence and a poor prognosis. Although potential benefits of metronomic chemotherapy and alisertib have been reported, the patient in this study did not favorably respond to these modalities.

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Clinical evidence for a biological effect of epigenetically active decitabine in relapsed or progressive rhabdoid tumors

Steinbügl

Nemes

Johann

et al. 2021

Pediatric Blood & Cancer

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A case of relapsing spinal atypical teratoid/rhabdoid tumor (AT/RT) responding to vinorelbine, cyclophosphamide, and celecoxib

Abstract: The peculiarity of this case report is the clinical-radiological response to a metronomic therapy in a case of early-relapsing spinal AT/RT despite previous maximal surgery, chemotherapy, and radiotherapy.

Cited by 16 publications

References 11 publications

Oncolytic Herpes Virus rRp450 Shows Efficacy in Orthotopic Xenograft Group 3/4 Medulloblastomas and Atypical Teratoid/Rhabdoid Tumors

Oncolytic Herpes Virus rRp450 Shows Efficacy in Orthotopic Xenograft Group 3/4 Medulloblastomas and Atypical Teratoid/Rhabdoid Tumors

Primary atypical teratoid/rhabdoid tumor of the spine in an adult patient

Clinical evidence for a biological effect of epigenetically active decitabine in relapsed or progressive rhabdoid tumors

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