Progressive multifocal leukoencephalopathy after stem cell transplantation, unsuccessfully treated with cidofovir

Osorio, Sonia; Cámara, Rafael de la; Golbano, N; Martí, Edelmira; Fedele, Cesare Giovanni; Nieto, Santiago; Manzanares, Rafael; Fernández-Rañada, J M

doi:10.1038/sj.bmt.1703704

Cited by 40 publications

(25 citation statements)

References 19 publications

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“…There are eight reported cases of PML after autologous HCT [17][18][19][20][21][22][23] (Table 2). The median age of autograft recipients was 44 years (range: 31-60).…”

Section: Discussionmentioning

confidence: 99%

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Two cases of progressive multifocal leukoencephalopathy after allogeneic hematopoietic cell transplantation and a review of the literature

Kharfan‐Dabaja

Ayala

Greene

et al. 2006

Bone Marrow Transplant

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Progressive multifocal leukoencephalopathy (PML) is a rare subacute demyelinating disorder of the central nervous system (CNS) caused by the DNA JC human polyomavirus. In immunocompromised hosts, PML is caused by reactivation of a latent infection rather than de novo primary exposure. PML in the setting of hematopoietic cell transplantation (HCT) is exceedingly rare. PML should be considered in the differential diagnosis of HCT recipients, autologous or allogeneic, presenting with worsening of neurological symptoms, especially associated with post-transplant neurodegenerative findings. Although DNA polymerase chain reaction (PCR) of the cerebrospinal fluid (CSF) has emerged as a promising tool for detecting JC virus, a negative result does not rule out PML. Brain biopsy remains the most reliable and accurate method for diagnosing JC virus-associated PML. Presently, there is no universally effective antiviral therapy against JC virus and outcome is fatal in the majority of cases. We hereby describe two cases of PML developing after allogeneic HCT and provide a comprehensive review of the literature.

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“…There are eight reported cases of PML after autologous HCT [17][18][19][20][21][22][23] (Table 2). The median age of autograft recipients was 44 years (range: 31-60).…”

Section: Discussionmentioning

confidence: 99%

“…Cidofovir was administered as monotherapy to a patient with a CD4 count of 535/mm 3 without clinical benefit. 18 Neurological complications are not considered among the most important causes of morbidity and mortality following HCT. Nonetheless, Gallardo et al 24 demonstrated a 37% incidence of neurologic symptoms in allograft recipients.…”

Section: Discussionmentioning

confidence: 99%

Two cases of progressive multifocal leukoencephalopathy after allogeneic hematopoietic cell transplantation and a review of the literature

Kharfan‐Dabaja

Ayala

Greene

et al. 2006

Bone Marrow Transplant

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“…Another explanation could be that the association between Hodgkin disease and PML is already well known and therefore not worth reporting. In contrast, after 1990, its appears that LPDs-associated PML is largely restricted to two groups of severely T-cell immunosuppressed patients: B-CLL patients who had received purine analogues [30,33,[35][36][37][39][40][41][42][46][47][48] and heavily pretreated patients with aggressive non-Hodgkin lymphoma after HDT/HSCT [29,31,34,[43][44][45]49].…”

Section: Discussionmentioning

confidence: 99%

“…One patient who survived was treated with intravenous immunoglobulins [33]. Cidofovir has been used in 6 cases without improvement [35,41,[43][44][45]48]. This is not surprising because most experts consider that this agent plays no role in the treatment of established PML [66].…”

Section: Discussionmentioning

confidence: 99%

Changes in the natural history of progressive multifocal leukoencephalopathy in HIV-negative lymphoproliferative disorders: Impact of novel therapies

et al. 2005

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The aims of this study were to evaluate the clinical characteristics of HIV-negative patients affected by lymphoproliferative disorders (LPD) who developed progressive multifocal leukoencephalopathy (PML), to delineate the risk factors, and to analyze whether the new antineoplastic therapies are changing the natural history of this infectious disease. We retrospectively analyzed 46 cases with confirmed LPD-associated PML published from 1958 to 2004. Patients were stratified according to two different time periods: group A included patients diagnosed before 1989, and group B included patients diagnosed since 1990, after introduction of purine analogues. Group A patients (n = 22) had received alkylating agents and/or radiotherapy, and the majority (63.6%) had advanced Hodgkin disease. At univariate analysis, uncontrolled Hodgkin disease was the only risk factor for PML. In group B patients (n = 24), the most frequent treatments received were purine analogues (58.3%) and high-dose therapy with hematopoietic stem cell transplantation (33.3%; HDT/HSCT). B-cell chronic lymphocytic leukemia (45.8%) and aggressive non-Hodgkin lymphoma (24.9%) were the most frequent underlying LPDs. Patients treated with purine analogues were more likely to have active LPD, lower CD4 cell counts, and to be older and male than were HSCT recipients. The median interval from purine analogues or HDT/HSCT to PML was 11 months. In HDT/HSCT recipients, this interval was delayed for 10 months when peri-transplantation rituximab was used. Univariate analysis identified age >55 years, male sex, and CD4 cell counts £0.2 · 10 9 /L as risk factors for PML in patients treated with purine analogues. Mortality rates were 95.4% (group A patients), 90% (purine analogues), and 62.5% (HDT/HSCT recipients). At univariate analysis, the only factor that significantly correlated with recovery from infection was female sex. Our findings indicate (1) the possible reduction in reported cases associated with Hodgkin disease and the increasing number of published cases associated with the new antineoplastic therapies (purine analogues and HDT/ HSCT); (2) among patients treated with purine analogues, PML is more common in male patients with CD4 cell counts £0.2 · 10 9 /L; (3) the use of rituximab after HDT/ HSCT seems to delay the onset of PML; and (4) the prognosis is slightly better in transplant recipients. Am.

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“…The patient required long-lasting steroid treatment and although bronchiolytis evolved with success, he died 9 months later because of PML due to JC virus, unresponsive to treatment with cidofovir. 5 All patients were in remission when these complications were diagnosed.…”

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confidence: 99%

Unexpected immune complications in patients with non-Hodgkin's lymphoma receiving CD34-selected autologous peripheral stem cell transplantation

Arranz

Cannata

2005

Bone Marrow Transplant

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Progressive multifocal leukoencephalopathy after stem cell transplantation, unsuccessfully treated with cidofovir

Cited by 40 publications

References 19 publications

Two cases of progressive multifocal leukoencephalopathy after allogeneic hematopoietic cell transplantation and a review of the literature

Two cases of progressive multifocal leukoencephalopathy after allogeneic hematopoietic cell transplantation and a review of the literature

Changes in the natural history of progressive multifocal leukoencephalopathy in HIV-negative lymphoproliferative disorders: Impact of novel therapies

Unexpected immune complications in patients with non-Hodgkin's lymphoma receiving CD34-selected autologous peripheral stem cell transplantation

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