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Recurrent Enteritis Associated with Epstein-Barr Virus-positive CD4+ T-cell Lymphoproliferative Disorder after Autologous Stem Cell Transplantation.

We encountered a patient with multiple myeloma treated with autologous hematopoietic stem cell transplantation (HSCT) who developed repeated episodes of enteritis but regressed spontaneously. An endoscopic examination revealed no abnormalities, but biopsy specimens showed massive infiltration of CD4+ and Epstein-Barr encoding region (EBER+) abnormal lymphocytes in which a high copy number of Epstein Barr virus (EBV) genomes was detected by quantitative polymerase chain reaction (qPCR). EBV infection was exclusively detected in CD4+ T-cells, leading to a diagnosis of EBV-positive CD4+ T-cell ly

18F-FDG PET for Detection of Primary Tracheobronchial T-/Natural Killer-Cell-Derived Posttransplant Lymphoproliferative Disorder After Lung Transplantation.

Posttransplant lymphoproliferative disorder (PTLD) is a rare, but potentially devastating complication of solid organ transplantation. Epstein-Barr virus-associated PTLDs of T-cell origin are exceedingly rare. We present the case of a 20-year-old man with extranasal T-/natural killer-cell-derived PTLD after lung transplantation who underwent an F-FDG PET/CT scan for staging of disease. The F-FDG-PET scan demonstrated an unusual pattern of lymphomatous involvement limited to the recipients tracheobronchial tree without concomitant involvement of the donor's bronchi. This case highlights the pot

A unique case of an indolent myometrial T-cell lymphoproliferative disorder with phenotypic features resembling uterine CD8+ resident memory T cells.

OBJECTIVE: Indolent extranodal T-cell lymphoproliferative disorders have recently been described as new entities in the gastrointestinal tract and acral sites displaying clonal T-cell receptor (TCR) rearrangement and nonactivated cytotoxic CD8+ T-cell phenotypes. METHODS/RESULTS: We report a unique case of an atypical myometrial T-cell lymphoproliferation in a 39-year-old multiparous woman, which shares many of the features mentioned above: CD8+/TIA1+/granzyme B- phenotype, clonal TCR rearrangement and indolent course. CONCLUSION(S): We hypothesize that it might derive from a subset of uteri…

Epstein-Barr virus-related post-transplant lymphoproliferative disorder occurring after bone marrow transplantation for aplastic anemia in Down's syndrome.

It is well established that Down's syndrome exhibits a predisposition to development of leukemia, however, association between aplastic anemia and Down's syndrome is exceptional. Herein, we describe a case of aplastic anemia occurring in Down's syndrome following post-transplant lymphoproliferative disorder (PTLD) after bone marrow transplantation (BMT). A 27-year-old Japanese male with Down's syndrome presented with a headache. Laboratory tests revealed severe pancytopenia, and bone marrow biopsy demonstrated hypocellular bone marrow with decrease of trilineage cells, which led to a diagnosis

Epstein-Barr virus-associated lymphoproliferative disorder developed following autologous peripheral blood stem cell transplantation for relapsing Hodgkin's lymphoma.

Post-transplant lymphoproliferative disorders (PTLDs) are lymphoid or plasmacytic proliferations that develop as a consequence of immunosuppression in a recipient of a solid organ, bone marrow or stem cell allograft. The development of PTLDs is usually associated with Epstein-Barr virus (EBV) and the disorder is also termed EBV-associated lymphoproliferative disorder (LPD). The development of PTLD is a rare complication in autologous bone marrow/peripheral blood stem cell transplantation. In the present study, we report a case of EBV-associated LPD which developed following autologous peripher…

Collision tumors with synchronous presentation of breast carcinoma and lymphoproliferative disorders in the axillary nodes of patients with newly diagnosed breast cancer: a case series.

Although occurrence of a second tumor is a well-recognized phenomenon in patients with a treated malignancy, simultaneous presentation with a second primary malignancy ("collision tumor") is rare in patients with breast cancer. We report a series of 7 patients who were undergoing axillary nodal staging of a newly diagnosed breast cancer. Six patients were found to have collision tumors in their axillary lymph nodes consisting of metastatic breast cancer and a previously unknown lymphoproliferative disorder. In 1 additional patient, a lymph node biopsy of a palpable axillary node resulted in th

EBV-positive extranodal marginal zone lymphoma of mucosa-associated lymphoid tissue in the posttransplant setting: a distinct type of posttransplant lymphoproliferative disorder?

The 2008 World Health Organization Classification of Tumours of Haematopoietic and Lymphoid Tissues defines monomorphic posttransplant lymphoproliferative disorders (M-PTLDs) as lymphoid or plasmacytic proliferations that fulfill the criteria for one of the B-cell or T/NK-cell neoplasms recognized in immunocompetent patients. However, indolent B-cell lymphomas, such as extranodal marginal zone lymphoma of mucosa-associated lymphoid tissue (MALT lymphoma), are specifically excluded from this category. In this study, we describe the clinicopathologic features of 4 posttransplant lymphoma-like pr…

Hodgkin lymphoma post-transplant lymphoproliferative disorder following pediatric renal transplant: serial imaging with F-18 FDG PET/CT.

Post-transplant lymphoproliferative disorder (PTLD) occurs in 1.2% of pediatric renal transplant patients, and is frequently Epstein-Barr Virus mediated. Hodgkin Lymphoma PTLD is the rarest of the 4 types of PTLDs recognized by the World Health Organization, with an incidence of <4% of all PTLD patients. It has a distinct clinical course and treatment from all other types of PTLD. This is a case of a 16-year-old girl who had a renal transplant in 2000 due to Moya Moya disease. Her first F-18 FDG PET/CT done in 2006 showed mildly FDG-avid mediastinal adenopathy (histologically nonspecific react

Constitutive activation of Jak/STAT proteins in Epstein-Barr virus-infected B-cell lines from patients with posttransplant lymphoproliferative disorder.

BACKGROUND: Posttransplant lymphoproliferative disease (PTLD) is a major complication after bone marrow and solid organ transplantation. The disease encompasses a spectrum of abnormal, Epstein-Barr virus (EBV)-associated B-cell proliferations. We have previously shown that EBV-infected, spontaneous lymphoblastoid cell lines (SLCL) derived from PTLD patients require autocrine interleukin (IL)-10 to proliferate. To determine if cytokine signal transduction is involved in the autonomous growth of the SLCL, the activation states of the Jak/STAT signaling pathway proteins were analyzed in three dif

T gamma/delta hepatosplenic lymphoma in a heart transplant patient after an Epstein-Barr virus positive lymphoproliferative disorder: a case report.

BACKGROUND: An unusual case of a peripheral T-cell lymphoma of T gamma/delta hepatosplenic type (Tgamma/deltaHSL) that arose in a child 5 years after she received a heart transplant and 9 months after she developed Epstein-Barr virus (EBV) positive, B-cell lymphoid hyperplasia involving the tonsils is presented. The majority of the reported cases of Tgamma/deltaHSL have been described in young adult men without antecedent immunodeficiency; several well documented cases of Tgamma/deltaHSL in the posttransplant setting have been described previously, but none has been described in a child (or an

Epstein-Barr virus-related posttransplantation lymphoproliferative disorder involving pancreas allografts: histological differential diagnosis from acute allograft rejection.

The clinical and pathological features of acute pancreas allograft rejection and involvement of the graft by posttransplantation lymphoproliferative disorders (PTLD) overlap. Because the treatment is diametrically opposite in these two types of lesions, an accurate diagnosis is essential. The histological features in pancreas allograft needle biopsy specimens (n=7) and pancreatectomies (n=4) from four patients with Epstein-Barr virus (EBV)-related PTLD were compared with the material from 14 patients who did not develop PTLD after 12 to 58 months of follow-up and whose biopsy specimens (n=10) …

Lymphoproliferative disorder of granular lymphocytes: nine cases including one with features of CD56 (NKH1)-positive aggressive natural killer cell lymphoma.

Twelve cases of large granular lymphocytosis were examined by flow cytometry and Southern blot analysis to correlate immunophenotypic and molecular genetic markers with clinical features. Nine cases fulfilled clinical criteria for the lymphoproliferative disorder of granular lymphocytes, and eight of these demonstrated the molecular features of T-cell-type lymphoproliferative disorder of granular lymphocytes including surface expression of CD3, expression of one or more natural killer (NK)-cell antigens (CD11b, CD16, or CD57), and clonal rearrangement of both the T-cell receptor beta- and gamm

Vitamin C Conditioning Generates Tumor-Targeting CAR T Cells with Superior Cytotoxicity and Fitness in a Posttransplant Lymphoproliferative Disorder Tumor Model.

Chimeric antigen receptor (CAR) T-cell therapy has shown efficacy in hematologic malignancies but faces challenges in solid tumors and virus-associated malignancies such as posttransplant lymphoproliferative disorder (PTLD). Various strategies, including optimization of receptor design, genetic modifications addressing immunomodulatory mechanisms, and refining the manufacturing process, have been explored to overcome limited in vivo persistence and tumor infiltration, antigen escape, and the immunosuppressive tumor microenvironment. This study investigated the effect of vitamin C (vitC) condit

A 21-year-old man with anemia and gastrointestinal bleeding caused by jejunal EBV-associated T/natural killer-cell lymphoproliferative disorder.

We report a 21-year-old man who developed abdominal pain, bleeding, and anemia caused by a rare Epstein-Barr virus-related T/NK-cell lymphoproliferative disorder in the small intestine. Initial endoscopy detected ulcers in the duodenum and jejunum, but the cause was unclear from the clinical and imaging findings alone. Biopsy later showed abnormal lymphoid cell growth, and further testing, including immunohistochemistry and EBER staining, confirmed EBV-associated disease limited to the gastrointestinal tract. The patient began combination chemotherapy and remains under follow-up. This case hig

Iatrogenic immunodeficiency-associated polymorphic lymphoproliferative disorder resulting in complete remission after tacrolimus withdrawal in systemic lupus erythematosus: a case report.

A 27-year-old woman was diagnosed with systemic lupus erythematosus and successfully achieved remission. She remained in long-term remission without relapse whilst receiving prednisolone, tacrolimus, and hydroxychloroquine. At the age of 42, after receiving an influenza vaccination, she developed multiple, progressively enlarging, painless cervical lymph nodes and was referred to our hospital. She presented with fever and cervical lymphadenopathy. Laboratory examination revealed an elevated sIL-2 receptor of 3300 U/mL. Computed tomography demonstrated lymphadenopathy in the left posterior cerv

[Small intestinal perforation manifesting as post-transplant lymphoproliferative disorder occurring 23 years after living lung transplantation: a case report].

A Japanese female patient underwent a living lung transplant at age 29 and has been on immunomodulatory drug therapy since then. At age 52, she presented with sudden hematochezia. Despite abdominal computed tomography scan, esophagogastroduodenoscopy, and colonoscopy, no definitive source of bleeding was identified. Considering the possibility of small bowel bleeding, video capsule endoscopy was performed, which revealed a suspected ulcerative lesion in the jejunum. Subsequent per-oral double-balloon endoscopy confirmed the presence of an ulcerative lesion in the jejunum. A potential pathologi

Monomorphic T-cell post-transplant lymphoproliferative disorder with features of HHV8-negative primary effusion lymphoma: an autopsy case and review of the literature.

A 67-year-old man underwent renal transplantation in his twenties. He developed refractory pleural effusion, with many large lymphocytes with severe atypia and mitosis in the effusion, indicating malignant lymphoma. He finally died of respiratory failure. An autopsy revealed atypical lymphocytes positive for CD3, CD4, and CD30 and negative for CD8, CD20, PAX5, human herpesvirus (HHV) 8, and Epstein-Barr virus-encoded small RNAs by immunohistochemistry and in situ hybridization. Atypical lymphocytes also had T-cell receptor gene rearrangements Jβ2, Jγ2, and Jδ1 and chromosomal aberrations der(8

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… lymphoproliferative disorders. … ) Post-transplant lymphoproliferative disorders occurring after renal transplantation in adults: report of 230 cases from the French registry. …

Post-transplant lymphoproliferative disorder associated Epstein-Barr virus DNAemia after liver transplantation in children: Experience from single center.

The most prevalent malignancy that complicates both adult and pediatric solid organ transplantation is post-transplant lymphoproliferative disorder (PTLD). This study aimed to analyze the clinical and pathological characteristics, treatments, and outcomes of Epstein-Barr virus (EBV) DNAemia and PTLD in pediatric liver transplant recipients. A retrospective chart review was performed on 112 patients less than 18 years of age who underwent isolated orthotopic liver transplantation (OLT) between 2010 and 2022 at Ege University Children's Hospital. Data gathered for 1-year post-OLT included age at

Primary CNS EBV-positive post-transplant lymphoproliferative disorder with polymorphic and classic Hodgkin lymphoma features: A case report and literature review.

Background  Antero-laterally located meningiomas of the foramen magnum (FM) pose significant surgical resection challenges. The effect of FM shape on surgical resection of FM meningiomas has not been previously studied. The present study investigates how FM shape effects the extent of tumor resection and complication rates in antero-lateral FM meningiomas. Materials and Methods  This retrospective study included 16 consecutive patients with antero-lateral FM meningiomas operated on by a single surgeon. FMs were classified as ovoid ( n  = 8) and nonovoid ( n  = 8) using radiographic evaluation.

Role of brentuximab vedotin plus sirolimus in the treatment of classical Hodgkin lymphoma type post-transplant lymphoproliferative disorder: a case-based review.

Post-transplant lymphoproliferative disorder (PTLD) is a common secondary malignancy after transplantation, which has been recognized as a life-threatening complication. Hodgkin lymphoma (HL)-type PTLD is the rarest of four subtypes of PTLD, which has no treatment guideline due to its rarity. HL-type PTLD includes classical HL-type PTLD (cHL-PTLD) and HL-like PTLD. In our study, we reported the case of successful treatment using brentuximab vedotin (BV) plus sirolimus for a patient with classical HL-type PTLD in detail. Lymph node biopsy showed a picture of classical HL with mixed cellularity