A Case of Epstein-Barr Virus–Associated Leiomyosarcoma Concurrently With Posttransplant Lymphoproliferative Disorders After Renal Transplantation

Abstract

Epstein-Barr virus (EBV) infection might induce not only posttransplantation lymphoproliferative disorder (PTLD) but also leiomyosarcoma. We report a case of EBV-associated leiomyosarcoma concurrently with PTLD after renal transplantation. The patient was a 30-year-old woman who underwent living donor kidney transplantation at 27 years of age. Preoperative EBV viral capsid antibody immunoglobulin M, immunoglobulin G (IgG), and EBV nuclear antigen IgG were negative. Multiple lung and liver tumors were detected 1.5 years after transplantation. She was diagnosed with PTLD after tumor biopsy. Her EBV DNA was 110 copies/mL detected by real-time polymerase chain reaction when PTLD was diagnosed. She received dose reduction of immunosuppressive therapy and several chemotherapies. Because her hepatic lesion was still progressive while pulmonary lesion was reduced, a liver tumor biopsy was performed, but the biopsy specimens were necrotic. A left lateral segmentectomy was performed as a third biopsy for treatment-resistant hepatic lesion 2.5 years after her first PTLD diagnosis. Pathologically, she was diagnosed with EBV-associated leiomyosarcoma. She was treated with sirolimus, but died 7 months after the operation. This is the first case of the coincidence of leiomyosarcoma associated with EBV and PTLD. This case was exceedingly rare; however, we must consider the coincidence of leiomyosarcoma associated with EBV and PTLD when encountering treatment-resistant PTLD.

Recent Advances in the Role of Microorganisms in Cancer Incidence: Mechanisms and Health Precautions

“…This encodes all of the enzymes involved in the replication of its DNA and in nucleotide biosynthesis 60 . It has been linked to a number of malignancies, such as Hodgkin's disease 61 , B-and T-cell lymphomas 62 , leiomyosarcomas 63 , post-transplant lymphoproliferative disease 64 , and nasopharyngeal carcinomas 65 . Among all of these cancers, the frequency of Burkitt's lymphoma, leiomyosarcomas and posttransplant lymphoproliferative disease have dramatically increased in the past few years especially in patients who suffer from immunodeficiency, revealing the role of immunosurveillance in the prevention and suppression of malignant transformation 66 .…”

Epstein–Barrvirus‐associated leiomyosarcoma of the larynx in an adult patient with human immunodeficiency virus infection: Case report and review of the literature

“…Several cases of EBV‐related LMS in immunosuppressed patients have been reported 25,29,39,56–98 . LMS in immunosuppressed patients after organ transplantation and in patients with human immunodeficiency virus (HIV) is associated with EBV.…”

“…LMS in immunosuppressed patients after organ transplantation and in patients with human immunodeficiency virus (HIV) is associated with EBV. EBV‐associated LMS is common in children with HIV, and uncommon in adult patients with HIV 25,29,39,56–98 . Marioni et al 19 reported LLMS in a patient with two kidney transplants.…”

“…Several cases of EBV‐related LMS in immunosuppressed patients have been reported. 25 , 29 , 39 , 56 , 57 , 58 , 59 , 60 , 61 , 62 , 63 , 64 , 65 , 66 , 67 , 68 , 69 , 70 , 71 , 72 , 73 , 74 , 75 , 76 , 77 , 78 , 79 , 80 , 81 , 82 , 83 , 84 , 85 , 86 , 87 , 88 , 89 , 90 , 91 , 92 , 93 , 94 , 95 , 96 , 97 , 98 LMS in immunosuppressed patients after organ transplantation and in patients with human immunodeficiency virus (HIV) is associated with EBV. EBV‐associated LMS is common in children with HIV, and uncommon in adult patients with HIV.…”

“…EBV‐associated LMS is common in children with HIV, and uncommon in adult patients with HIV. 25 , 29 , 39 , 56 , 57 , 58 , 59 , 60 , 61 , 62 , 63 , 64 , 65 , 66 , 67 , 68 , 69 , 70 , 71 , 72 , 73 , 74 , 75 , 76 , 77 , 78 , 79 , 80 , 81 , 82 , 83 , 84 , 85 , 86 , 87 , 88 , 89 , 90 , 91 , 92 , 93 , 94 , 95 , 96 , 97 , 98 Marioni et al 19 reported LLMS in a patient with two kidney transplants. They detected EBV in LMS tissues but not neoplastic tissues.…”