Progressive multifocal leukoencephalopathy presenting with acute sensorineural hearing loss in an intestinal transplant recipient

Lippa, Andrew M.; Ocwieja, Karen E.; Iglesias, Julie; Fawaz, Rima; Elisofon, Scott A.; Lee, Christine; Sharma, Tanvi

doi:10.1111/tid.13304

Cited by 4 publications

(4 citation statements)

References 51 publications

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“…In total, there have been 14 reported cases of PML post SOT or haematopoietic stem cell transplant (HSCT) in whom mirtazapine was used as adjunctive therapy and which are summarised in table 2 13 15–25. The reported time post-transplantation ranged from a few months to almost 20 years post-transplantation, with a mortality rate of 42.8% and reported survival of 10.7 months.…”

Section: Discussionmentioning

confidence: 99%

Progressive multifocal leukoencephalopathy 10 years following transplant: 5HT receptor antagonism as an adjunct to immune reconstitution

Chandler

Isbel

2022

BMJ Case Rep

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We report a case of a patient presenting with subacute neurological symptoms 10 years postkidney transplant. Cognitive deficits included acalculia and left upper limb dysesthesia, progressing to hemiplegic upper motor neuron weakness. Investigations included an MRI with multiple FLAIR hyperintensities, while a lumbar puncture was sterile with negative flow cytometry. Ultimately, PCR testing for John Cunningham virus was positive on cerebrospinal fluid. The diagnosis of progressive multifocal leukoencephalopathy (PML) was confirmed on the basis of the above.Initially, the patient was managed with withdrawal of immunosuppressants and close observation. Mirtazapine was commenced based on case reports of successful use in non-transplant patients; the patient’s recovery was temporally related to withdrawal of immunosuppression and increasing mirtazapine dosage. The patient is currently maintained on prednisolone and mirtazapine with stable graft function and improved mobility and cognitive function.

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Section: Discussionmentioning

confidence: 99%

Progressive multifocal leukoencephalopathy 10 years following transplant: 5HT receptor antagonism as an adjunct to immune reconstitution

Chandler

Isbel

2022

BMJ Case Rep

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“…The patient continually deteriorated after the withdrawal of prior immunosuppression therapy and subsequent treatment with mirtazapine, maraviroc, and steroids. The patient died of PML five months after his initial clinical presentation [52]. Ahmadinejad et al described PML that developed in a 41-year-old female after liver transplantation [53].…”

Section: Transplant Patientsmentioning

confidence: 99%

“…Lippa et al reported PML presenting with acute sensorineural hearing loss in an intestinal transplant recipient (Figure 2). The study involved a 20-year-old male that clinically presented with rapidly progressive sensorineural hearing loss 3.5 years after intestinal transplant surgery [52]. The MRI of the brain was consistent with inflammation and/or demyelination.…”

Section: Transplant Patientsmentioning

confidence: 99%

JC Polyomavirus and Transplantation: Implications for Virus Reactivation after Immunosuppression in Transplant Patients and the Occurrence of PML Disease

Hildreth

Alcendor

2021

Transplantology

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The JC polyomavirus (JCPyV/JCV) is a member of the Polyomaviridae family and is ubiquitious in the general population, infecting 50–80% of individuals globally. A primary infection with JCV usally results in an asymptomatic, persistent infection that establishes latency in the renourinary tract. Reactivation from latency via iatrogenic immununosuppression for allograft transplantation may result in organ pathology and a potential life-threatening neuropathological disease in the form of progressive multifocal leukoencephalopathy (PML). Currently, no treatment exists for PML, a rare complication that occurs after transplantation, with an incidence of 1.24 per 1000 persons a year among solid organ transplant patients. PML is also observed in HIV patients who are immununosuppressed and are not receiving antiretroviral therapy, as well as individuals treated with biologics to suppress chronic inflammatory responses due to multiple sclerosis, Crohn’s disease, non-Hodgkin’s lymphoma, rheumatoid arthritis, and other autoimmune-mediated hematological disorders. Here, we describe the proposed mechanisms of JCV reactivation as it relates to iatrogenic immunosuppression for graft survival and the treatment of proinflammatory disease, such as biologics, proposed trafficking of JCV from the renourinary tract, JCV central nervous system dissemination and the pathology of PML in immunosuppressed patients, and potential novel therapeutics for PML disease.

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“…Progressive multifocal leukoencephalopathy (PML) is a rare syndrome of CNS demyelination caused by the reactivation of John Cunningham (JC) virus. Lippa et al reviewed recent existing case reports and systematic reviews published on posttransplant PML [80 ▪ ]. In a systematic review by Mateen et al [81], 83% of cases were diagnosed within the first 5 years post-SOT, but the range varied.…”

Section: Progressive Multifocal Leukoencephalopathymentioning

confidence: 99%

Central nervous system infections after solid organ transplantation

Puthanveedu

Lum

2021

Current Opinion in Infectious Diseases

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Purpose of review Significant advances to our understanding of several neuroinfectious complications after a solid organ transplant (SOT) have occurred in the last few years. Here, we review the central nervous system (CNS) infections that are relevant to SOT via a syndromic approach with a particular emphasis on recent updates in the field. Recent findings A few key studies have advanced our understanding of the epidemiology and clinical characteristics of several CNS infections in SOT recipients. Risk factors for poor prognosis and protective effects of standard posttransplant prophylactic strategies have been better elucidated. Newer diagnostic modalities which have broad clinical applications like metagenomic next-generation sequencing, as well as those that help us better understand esoteric concepts of disease pathogenesis have been studied. Finally, several studies have provided newer insights into the treatment of these diseases. Summary Recent findings reflect the steady progress in our understanding of CNS infections post SOT. They provide several avenues for improvement in the prevention, early recognition, and therapeutic outcomes of these diseases.

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Progressive multifocal leukoencephalopathy presenting with acute sensorineural hearing loss in an intestinal transplant recipient

Cited by 4 publications

References 51 publications

Progressive multifocal leukoencephalopathy 10 years following transplant: 5HT receptor antagonism as an adjunct to immune reconstitution

Progressive multifocal leukoencephalopathy 10 years following transplant: 5HT receptor antagonism as an adjunct to immune reconstitution

JC Polyomavirus and Transplantation: Implications for Virus Reactivation after Immunosuppression in Transplant Patients and the Occurrence of PML Disease

Central nervous system infections after solid organ transplantation

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