EBV-positive post-transplant lymphoproliferative disorder presenting as primary diffuse large B-cell lymphoma of the central nervous system

Xu, Huiping; Rewerska, Julia; Aardsma, Nathan; Slavin, Konstantin; Vályi-Nagy, Tibor; Ni, Hongyu

doi:10.5114/fn.2017.70487

Cited by 8 publications

(7 citation statements)

References 14 publications

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“…PTLD can affect any organ transplant patient on immunosuppression, and it can occur following a solid organ transplant such as heart, lung, liver, kidney, and pancreas as well as, following a hematopoietic cell transplant [ 8 ]. The incidence of PTLD in kidney transplant recipients is lower compared to other solid organs; this could be attributed to the choice of immunosuppression regimen, which tends to be less aggressive than for other solid organ transplants [ 9 ]. Furthermore, studies have investigated the choice of immunosuppressant and its relationship to the incidence of PTLD in solid organ transplant recipients, but no clear trends have emerged to show a correlation between specific medications and the disease [ 4 , 10 ].…”

Section: Discussionmentioning

confidence: 99%

“…One case series of 84 cases between 1997 and 2010 reported a median overall survival of 17 months, which is significantly lower than the median overall survival of 31.5 months reported in a cohort of 107 patients between 1970 and 2003, in spite of the fact that the majority of these patients were in the pre-rituximab era [ 4 , 8 ]. More recent data is showing improvement with a median overall survival of four years that could be attributed to a better understanding of immunosuppression, use of rituximab, advances in radiation techniques, and improved supportive care [ 4 , 9 ].…”

Section: Discussionmentioning

confidence: 99%

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Successful Treatment of Central Nervous System Post-Transplant Lymphoproliferative Disease With a Reduced Dose of High-Dose Methotrexate

Albusoul¹,

Abu-Hashyeh²,

Donthireddy³

2022

Cureus

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Post-transplant lymphoproliferative disease (PTLD) is a complication of solid organ and hematopoietic stem cell transplantation that occurs as a result of immunosuppression. PTLD isolated to the central nervous system (CNS) is a rare disease and it presents with nonspecific signs and symptoms. Optimal therapy guidelines have not yet been established for CNS PTLD. Here, we report a case of successful treatment of CNS PTLD in an adult female following two subsequent kidney transplants. Initial management was with immunosuppression reduction and a trial of rituximab. There were concerns regarding using methotrexate (MTX) given the patient's fragile transplant status. Magnetic resonance imaging of the brain following four cycles of rituximab revealed the progression of the disease. Subsequently, high-dose MTX (HD-MTX) was considered within the constraints of potential kidney toxicities given her transplant status and chronic kidney disease. Potential toxicities from other therapies, such as brain radiation, also factored into the final decision. The patient was treated with one cycle of a combination of rituximab and HD-MTX 1 g/m 2 . The patient tolerated HD-MTX and did not have evidence of renal toxicity in laboratory studies. Following that, she was started on a reduced dose of HD-MTX at 2 g/m 2 every two weeks instead of the higher MTX dose range of 3.5 to 8 g/m 2 , which was a shared decision with the patient and nephrology after weighing the risk of kidney dysfunction with the possibility of a less than optimal response with regards to her lymphoma. She was followed with a magnetic resonance imaging of the brain, which demonstrated a complete response after four cycles. Further consolidation treatments with HD-MTX 2 g/m 2 every four weeks were administered to complete one year of treatment. Following the completion of chemotherapy, the patient was able to achieve and maintain a complete response without affecting her kidney function. She continues to do well one year following treatment. This case highlights the significance of tailoring therapy to each individual based on their comorbidities and clinical response, as well as the possible merit in exploring the use of a reduced dose of HD-MTX in the treatment of CNS PTLD in patients at high risk for renal toxicity.

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Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Successful Treatment of Central Nervous System Post-Transplant Lymphoproliferative Disease With a Reduced Dose of High-Dose Methotrexate

Albusoul¹,

Abu-Hashyeh²,

Donthireddy³

2022

Cureus

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“…Two independent researchers screened titles and abstracts of published manuscripts available on PubMed ( https://pubmed.ncbi.nlm.nih.gov/ ) from the establishment of the database until December 2023. Inclusion criteria required manuscripts to be published in English, meet the definition of PCNS-PTLD ( 2 , 4 ), involve patients who underwent kidney or HSCT before the onset of clinical symptoms, and qualify as case reports or case series. The search strategy involved combinations of the following terms: “primary central nervous system post-transplant lymphoproliferative disorder”, “PCNS-PTLD”, “kidney transplant”, “HSCT”, “renal transplant”, and “hematopoietic stem cell transplantation”.…”

Section: Methodsmentioning

confidence: 99%

“…Post-transplant lymphoproliferative disorder (PTLD) refers to a group of lymphoproliferative disorders that occur in recipients of solid organ transplantation (SOT) or hematopoietic stem cell transplantation (HSCT) under pharmacologic immunosuppression ( 1 ). Although about 7%–15% of all PTLD patients involve the central nervous system (CNS), primary involvement of the nervous system is quite rare ( 2 , 3 ). Primary CNS post-transplant lymphoproliferative disorder (PCNS-PTLD) is defined as a lymphoproliferative disorder limited to the CNS occurring after SOT or HSCT, with no evidence of systemic PTLD ( 2 , 4 ).…”

Section: Introductionmentioning

confidence: 99%

“…Although about 7%–15% of all PTLD patients involve the central nervous system (CNS), primary involvement of the nervous system is quite rare ( 2 , 3 ). Primary CNS post-transplant lymphoproliferative disorder (PCNS-PTLD) is defined as a lymphoproliferative disorder limited to the CNS occurring after SOT or HSCT, with no evidence of systemic PTLD ( 2 , 4 ). The incidence rate of PCNS-PTLD is approximately 11.5 per 100,000 person-years ( 3 ).…”

Section: Introductionmentioning

confidence: 99%

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A disease warranting attention from neurosurgeons: primary central nervous system post-transplant lymphoproliferative disorder

Jin,

Lu,

Yan

et al. 2024

Front. Neurol.

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BackgroundPrimary central nervous system post-transplant lymphoproliferative disorder (PCNS-PTLD) is a rare condition, posing diagnostic and treatment challenges, with histological biopsy essential for diagnosis. Standardized treatment protocols are lacking. This disease requires urgent attention due to the increasing number of organ transplant surgeries and the use of immunosuppressive agents.MethodsFrom 2020 to 2023, our center diagnosed five patients with PCNS-PTLD. We reviewed their clinical records and conducted a comprehensive analysis of 22 literatures on PCNS-PTLD cases following renal transplantation or allogeneic hematopoietic stem cell transplantation (HSCT).ResultsFour patients had previously received a kidney transplant, one had undergone allogeneic HSCT. The median time from the last transplant surgery to the diagnosis of PCNS-PTLD differs between kidney transplant (21.5 years) and allogeneic HSCT (9 months). Common symptoms included motor weakness (n = 4), headache (n = 2), confusion (n = 2), and nausea (n = 2), with ring-enhancing (n = 5), typically solitary (n = 3) and supratentorial (n = 3) lesions on imaging. Diagnosis involved robot-assisted stereotactic brain biopsy (n = 4) or craniotomy (n = 1), all showing Epstein-Barr virus and CD20 positivity. Most cases (n = 4) were monomorphic diffuse large B-cell lymphoma. Treatment included rituximab (n = 3), surgical resection (n = 2), zanubrutinib (n = 1), whole-brain radiation (n = 1), and methotrexate (n = 1). At the last follow-up, the median duration of follow-up for all patients was 19 months. During this time, 3 patients had died and 2 patients were still alive.ConclusionIn patients with a history of kidney transplantation or allogeneic HSCT who are on long-term immunosuppressive therapy, any neurological symptoms, particularly the presence of supratentorial ring-enhancing masses in the brain on imaging, whether solitary or multiple, should raise high suspicion for this disease, warranting a timely brain biopsy. Additionally, we found that besides reducing immunosuppressants, zanubrutinib may be a potential, safe, and effective treatment for this condition. Moreover, post-surgical administration of rituximab in conjunction with whole-brain radiotherapy also appears to be a potentially safe and effective approach.

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CNS Infections in Immunoincompetent Patients

et al. 2019

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EBV-positive post-transplant lymphoproliferative disorder presenting as primary diffuse large B-cell lymphoma of the central nervous system

Cited by 8 publications

References 14 publications

Successful Treatment of Central Nervous System Post-Transplant Lymphoproliferative Disease With a Reduced Dose of High-Dose Methotrexate

Successful Treatment of Central Nervous System Post-Transplant Lymphoproliferative Disease With a Reduced Dose of High-Dose Methotrexate

A disease warranting attention from neurosurgeons: primary central nervous system post-transplant lymphoproliferative disorder

CNS Infections in Immunoincompetent Patients

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