Isolated cerebral manifestation of Epstein–Barr virus‐associated post‐transplant lymphoproliferative disorder after allogeneic hematopoietic stem cell transplantation: a case of clinical and diagnostic challenges

Kittan, Nicolai A.; Beier, Fabian; Kurz, Katharina; Niller, Hans Helmut; Egger, L.; Jilg, Wolfgang; Andreesen, Reinhard; Holler, Ernst

doi:10.1111/j.1399-3062.2011.00621.x

Cited by 25 publications

(26 citation statements)

References 41 publications

(61 reference statements)

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“…EBV-DNA loads of blood are acted as the main basis for diagnosis, preemptive therapy and therapeutic evaluation, but our study and others cases reports [12,13] showed that special EBV-associated disease in CNS and pulmonary had the discrepancy in EBV-DNA loads; sometimes repeated testing with real-time PCR in peripheral blood failed to show any evidence of EBV reactivation, and the change of EBV-DNA loads of blood was inconsistent with disease development. But frequent quantitative monitoring of EBV viral load in high-risk patients is still important to prevent occurrence of EBV-PTLD [14].…”

contrasting

confidence: 55%

Epstein - Barr virus - associated Diseases in Allogeneic Hematopoietic Stem Cell Transplantation

Liu

2012

J Hematol Oncol

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contrasting

confidence: 55%

Epstein - Barr virus - associated Diseases in Allogeneic Hematopoietic Stem Cell Transplantation

Liu

2012

J Hematol Oncol

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“…In immunocompromised individuals, herpesvirus-associated CNS diseases, such as encephalitis/myelitis and lymphoproliferative diseases, are representative of acute complications [12-14]. Since specific therapy is limited to only several viral agents, accurate diagnosis and early therapy reduces the extent of permanent injury in survivors and positively influences survival rate [15].…”

Section: Introductionmentioning

confidence: 99%

Herpesvirus-Associated Central Nervous System Diseases after Allogeneic Hematopoietic Stem Cell Transplantation

Huang

Jiang

et al. 2013

PLoS ONE

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Herpesvirus infections of the central nervous system (CNS) are associated with encephalitis/myelitis and lymphoproliferative diseases in immunocompromised individuals. As of now, data of herpesvirus-associated CNS diseases in transplant recipients is limited. Hence, in this prospective study, we investigated the incidence of herpesvirus-associated CNS diseases and explored the diagnosis of these diseases in 281 allogeneic hematopoietic stem cell transplantation (allo-HSCT) recipients. Herpesvirus-DNA and cerebrospinal fluid (CSF) cells were sampled from 58 recipients with herpesvirus-associated diseases or with unexplainable CNS manifestations. Results showed that 23 patients were diagnosed as herpesvirus-associated CNS diseases, including 15 Epstein-Barr virus (EBV)-associated diseases (4 encephalitis and 11 lymphoproliferative diseases), 5 herpes simplex virus type 1 encephalitis, 2 cytomegalovirus encephalitis/myelitis and 1 varicella zoster virus encephalitis. The median time of diseases onset was 65 (range 22-542) days post-transplantation. The 3-year cumulative incidence of herpesvirus-associated encephalitis/myelitis and post-transplant lymphoproliferative disorder (PTLD) was 6.3% ±1.9% and 4.1% ±1.2%, respectively. Of the evaluable cases, CSF cells mainly consisted of CD19+CD20+ B cells (7/11) and had clonal rearrangement of immunoglobulin genes (3/11) in patients with CNS-PTLD. On the contrary, in patients with encephalitis/myelitis, CSF cells were comprised of different cell populations and none of the gene rearrangement was detected. Herpesvirus-associated CNS diseases are common in the early stages of allo-HSCT, wherein EBV is the most frequent causative virus. The immunophenotypic and clonal analysis of CSF cells might be helpful in the differential diagnosis between encephalitis and lymphoproliferative diseases.

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“…Five previous case reports have described a negative EBV PCR of the peripheral blood in the presence of an EBV‐positive PTLD isolated to the CNS [10–14]. See Table 1.…”

Section: Discussionmentioning

confidence: 99%

Epstein-Barr virus-positive post-transplant lymphoproliferative disorder of the central nervous system, after renal transplantation with a discrepancy in viral load between peripheral blood and cerebrospinal fluid

Boersma

Zanden²,

Laverman³

et al. 2012

Transplant International

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Summary A 43‐year‐old female developed an Epstein–Barr virus (EBV)‐positive post‐transplant lymphoproliferative disorder (PTLD) in the central nervous system (CNS), 14 years after renal transplantation. One year prior to presentation, the patients’ treatment regimen was altered from cyclosporine, azathioprine, and prednisone to mycophenolate mofetil and prednisone. Magnetic resonance imaging of the brain revealed lesions suspect for malignant lymphoma. The EBV real‐time polymerase chain reaction (PCR) on peripheral blood was negative, but highly positive on cerebrospinal fluid. EBV‐positive PTLD was confirmed using histological analysis of cerebral biopsies. Despite tapering of immune suppressive medication and treatment with rituximab and chemotherapy, the patient deceased 50 days after presentation. This case illustrates that vigilance is required when presented with a negative EBV PCR result on peripheral blood when PTLD of the CNS is suspected. This late presentation suggests a relation to the switch in immunosuppressive regimen 1 year earlier.

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Isolated cerebral manifestation of Epstein–Barr virus‐associated post‐transplant lymphoproliferative disorder after allogeneic hematopoietic stem cell transplantation: a case of clinical and diagnostic challenges

Cited by 25 publications

References 41 publications

Epstein - Barr virus - associated Diseases in Allogeneic Hematopoietic Stem Cell Transplantation

Epstein - Barr virus - associated Diseases in Allogeneic Hematopoietic Stem Cell Transplantation

Herpesvirus-Associated Central Nervous System Diseases after Allogeneic Hematopoietic Stem Cell Transplantation

Epstein-Barr virus-positive post-transplant lymphoproliferative disorder of the central nervous system, after renal transplantation with a discrepancy in viral load between peripheral blood and cerebrospinal fluid

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