Expression of Wilms tumor 1 gene in a variety of pediatric tumors

Oue, Takaharu; Uehara, Shuichiro; Yamanaka, Hiroaki; Takama, Yuichi; Oji, Yusuke; Fukuzawa, Masahiro

doi:10.1016/j.jpedsurg.2011.09.004

Cited by 19 publications

(16 citation statements)

References 15 publications

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“…For example, many anticancer agents appear to be more active after allogeneic bone marrow transplantation (alloBMT) than pretransplant, including tyrosine kinase inhibitors in Philadelphia chromosome–positive leukemias [4,5], azaciditine for acute myeloid leukemia and myelodysplastic syndrome [6,7], rituximab in lymphomas [8,9], and, perhaps most impressively, tyrosine kinase inhibitors for FMS-like tyrosine kinase receptor-3 gene– mutated leukemias [10–12]. Of particular interest, metastatic tumors such as neuroblastoma, rhabdomyosarcoma, and Ewing sarcoma often present with bone marrow disease and may express targetable antigens such as WT-1, PRAME, and survivin [13–18]. Such antigen expression may be targetable by an unedited immunologic response and therefore provide the context to restudy alloBMT in solid tumors.…”

Section: Introductionmentioning

confidence: 99%

Reduced-Intensity Haploidentical Bone Marrow Transplantation with Post-Transplant Cyclophosphamide for Solid Tumors in Pediatric and Young Adult Patients

Llosa

Cooke

Chen

et al. 2017

Biology of Blood and Marrow Transplantation

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High-risk, recurrent, or refractory solid tumors in pediatric, adolescent, and young adult (AYA) patients have an extremely poor prognosis despite current intensive treatment regimens. We piloted an allogeneic bone marrow transplant platform using reduced-intensity conditioning (RIC) and partially HLA-mismatched (haploidentical) related donors for this population of pediatric and AYA solid tumor patients. Sixteen patients received fludarabine, cyclophosphamide, melphalan, and low-dose total body irradiation RIC haploidentical BMT (haploBMT) followed by post-transplantation cyclophosphamide (PTCy), mycophenolate mofetil, and sirolimus. All assessable patients were full donor chimeras on day 30 with a median neutrophil recovery of 19 days and platelet recovery of 21 days. One patient (7%) exhibited secondary graft failure associated with concomitant infection. The median follow-up time was 15 months. Overall survival was 88%, 56%, and 21% at 6, 12, and 24 months, respectively. Median survival from transplant date was 14 months with a median progression-free survival 7 months. We observed limited graft-versus-host disease in 3 patients and nonrelapse mortality in 1 patient. We demonstrated that RIC haploBMT with PTCy is feasible and has acceptable toxicities in patients with incurable pediatric and AYA solid tumors; thus, this approach serves as a platform for post-transplant strategies to prevent relapse and optimize progression-free survival.

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Section: Introductionmentioning

confidence: 99%

Reduced-Intensity Haploidentical Bone Marrow Transplantation with Post-Transplant Cyclophosphamide for Solid Tumors in Pediatric and Young Adult Patients

Llosa

Cooke

Chen

et al. 2017

Biology of Blood and Marrow Transplantation

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“…64 The Wilms tumor antigen (WT1) is expressed on many pediatric solid malignancies including nephroblastoma (Wilms tumor), neuroblastoma and RMS with. 65 WT1 was ranked as the most promising tumor antigen by the NCI in 2009. 66 It has been targeted in multiple trials in patients with leukemia.…”

Section: Anticancer Vaccinesmentioning

confidence: 99%

“…67 WT1 may prove to be a useful target for pediatric solid tumors. 65 As in adult cancers, the number of vaccine trials continues to grow with much anticipation of its role in solid tumor therapy.…”

Section: Anticancer Vaccinesmentioning

confidence: 99%

Targeted immunotherapy for pediatric solid tumors

Kopp

Katsanis

2015

OncoImmunology

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“…Several research groups have noticed a difference in clinical behavior between microcystic and macrocystic lesions (6). Wilms Tumor 1 (WT1) is a transcription factor that is activated in some human neoplasias, including Wilms Tumor (7,8). WT1 may be linked to the uncontrolled epithelial-to-mesenchymal transition observed in a number of developmental abnormalities and diseases (9).…”

Section: Wilms Tumor 1 Protein Is Not Expressed In Oral Lymphangiomasmentioning

confidence: 99%

Wilms tumor 1 protein is not expressed in oral lymphangiomas

et al. 2012

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Lymphangiomas are benign hamartomatous lesions of lymphatic vessels. Wilms Tumor 1 (WT1) is a transcription factor that is activated in some human neoplasias. WT1 protein expression is observed in endothelial cells during angiogenesis and is a useful marker to distinguish between vascular proliferations and vascular malformations. The purpose of the present study is to report a case series of oral lymphangiomas together with an immunohistochemical investigation of WT1. Seventeen cases of oral lymphangioma were retrieved and reviewed. Immunohistochemical analysis of WT1 protein was performed and pyogenic granuloma samples were used as positive controls. The male/female ratio was 1.125 and most of the lesions occurred in young subjects. While pyogenic granuloma showed positive staining for WT1, the endothelial cells lining the thin-walled dilated lymphatic vessels of lymphangiomas were negative for this protein. The findings strengthen the idea that oral lymphangioma is a vascular malformation characterized by lymphatic dilatation without significant endothelial proliferation.

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Expression of Wilms tumor 1 gene in a variety of pediatric tumors

Cited by 19 publications

References 15 publications

Reduced-Intensity Haploidentical Bone Marrow Transplantation with Post-Transplant Cyclophosphamide for Solid Tumors in Pediatric and Young Adult Patients

Reduced-Intensity Haploidentical Bone Marrow Transplantation with Post-Transplant Cyclophosphamide for Solid Tumors in Pediatric and Young Adult Patients

Targeted immunotherapy for pediatric solid tumors

Wilms tumor 1 protein is not expressed in oral lymphangiomas

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