title: Posttransplant Lymphoproliferative Disorder After Clinical Islet Transplantation: Report of the First Two Cases.
abstract: We report the first two cases of posttransplant lymphoproliferative disorder (PTLD) in recipients of islet transplants worldwide. First, a 44-year-old recipient of three islet infusions developed PTLD 80 months after his initial transplantation, presenting with abdominal pain and diffuse terminal ileum thickening on imaging. He was treated with surgical excision, reduction of immunosuppression, and rituximab. Seven months later, he developed central nervous system PTLD, presenting with vertigo and diplopia; immunosuppression was discontinued, resulting in graft loss, and he was given high-dose methotrexate and underwent consolidative autologous stem cell transplantation. He remains in remission 37 months after the initial diagnosis. Second, a 58-year-old female recipient of two islet infusions developed PTLD 24 months after initial islet infusion, presenting with pancytopenia secondary to extensive bone marrow involvement. Immunosuppression was discontinued, resulting in graft loss, and she received rituximab and chemotherapy, achieving complete remission. Both patients were monomorphic B cell PTLD subtype by histology and negative for Epstein-Barr virus in tissue or blood. These cases document the first occurrences of this rare complication in islet transplantation, likely secondary to prolonged, intensive immunosuppression, and highlight the varying clinical manifestations of PTLD. Further studies are needed to determine incidence rate and risk factors in islet transplantation.
authors.affiliation: Division of Clinical Hematology, Department of Medicine, University of Alberta, Edmonton, Canada.
authors.forename: A
authors.initials: A
authors.lastname: Peters
authors.affiliation: Clinical Islet Transplant Program, University of Alberta, Edmonton, Canada.
authors.forename: T
authors.initials: T
authors.lastname: Olateju
authors.affiliation: Department of Laboratory Medicine and Pathology, University of Alberta, Edmonton, Canada.
authors.forename: J
authors.initials: J
authors.lastname: Deschenes
authors.affiliation: Clinical Islet Transplant Program, University of Alberta, Edmonton, Canada.
authors.forename: S H
authors.initials: SH
authors.lastname: Shankarnarayan
authors.affiliation: Department of Oncology, University of Alberta, Edmonton, Canada.
authors.forename: N
authors.initials: N
authors.lastname: Chua
authors.affiliation: Clinical Islet Transplant Program, University of Alberta, Edmonton, Canada.
authors.forename: A M J
authors.initials: AMJ
authors.lastname: Shapiro
authors.affiliation: Clinical Islet Transplant Program, University of Alberta, Edmonton, Canada.|Division of Endocrinology, Department of Medicine, University of Alberta, Edmonton, Canada.
authors.forename: P
authors.initials: P
authors.lastname: Senior
mesh_terms: D000328:Adult; D003922:Diabetes Mellitus, Type 1 / Q000601:surgery*; D005260:Female; D006084:Graft Rejection / Q000188:drug therapy / Q000209:etiology*; D006085:Graft Survival / Q000187:drug effects; D006801:Humans; D007166:Immunosuppressive Agents / Q000627:therapeutic use; D016381:Islets of Langerhans Transplantation / Q000009:adverse effects*; D008232:Lymphoproliferative Disorders / Q000188:drug therapy / Q000209:etiology*; D008297:Male; D008875:Middle Aged; D011183:Postoperative Complications / Q000188:drug therapy / Q000209:etiology*; D011379:Prognosis; D012307:Risk Factors
chemical_list: D007166:Immunosuppressive Agents
keywords: cancer/malignancy/neoplasia; clinical research/practice; complication; diabetes: type 1; hematology/oncology; islet transplantation; malignant; pathology/histopathology; posttransplant lymphoproliferative disorder (PTLD)