Performance of PML diagnostic criteria in natalizumab-associated PML: data from the Dutch-Belgian cohort

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Abstract

OBJECTIVE: To test the current progressive multifocal leukoencephalopathy (PML) diagnostic criteria by applying them to patients previously diagnosed with natalizumab (NTZ)-associated PML in a real-world clinical setting.

METHODS: Patients from the Dutch-Belgian NTZ-PML cohort (n=28) were reviewed at the time of first diagnostic work-up and during follow-up, using the PML diagnostic criteria as proposed in a consensus statement from the American Academy of Neurology.

RESULTS: At first diagnostic work-up, 18 patients (64.3%) met the criteria for high diagnostic certainty for PML ('definite PML' or 'probable PML'). During follow-up, this increased to 20 patients (71.4%) as JC virus DNA was detected in cerebrospinal fluid of two additional patients. Nonetheless, 28.6% of patients were still classified as 'possible PML' or 'not PML' (6 (21.5%) and 2 (7.1%) patients, respectively) despite a very high suspicion for PML based on lesion evolution and signs of PML-immune reconstitution inflammatory syndrome on MRI, and development of compatible symptoms.

CONCLUSIONS: The current case definition of PML has low sensitivity for diagnosis of NTZ-PML in a real-world clinical setting in which MRI is frequently used for PML screening. This may delay diagnosis and appropriate management of PML, and may complicate a valid estimation of PML incidence during NTZ therapy.

Original languageEnglish
Pages (from-to)44-46
JournalJournal of Neurology, Neurosurgery and Psychiatry
Volume90
Issue number1
DOIs
Publication statusPublished - Jan 2019

Cite this

@article{57e6441b1efe40eebe830f6b4cc6a422,
title = "Performance of PML diagnostic criteria in natalizumab-associated PML: data from the Dutch-Belgian cohort",
abstract = "OBJECTIVE: To test the current progressive multifocal leukoencephalopathy (PML) diagnostic criteria by applying them to patients previously diagnosed with natalizumab (NTZ)-associated PML in a real-world clinical setting.METHODS: Patients from the Dutch-Belgian NTZ-PML cohort (n=28) were reviewed at the time of first diagnostic work-up and during follow-up, using the PML diagnostic criteria as proposed in a consensus statement from the American Academy of Neurology.RESULTS: At first diagnostic work-up, 18 patients (64.3{\%}) met the criteria for high diagnostic certainty for PML ('definite PML' or 'probable PML'). During follow-up, this increased to 20 patients (71.4{\%}) as JC virus DNA was detected in cerebrospinal fluid of two additional patients. Nonetheless, 28.6{\%} of patients were still classified as 'possible PML' or 'not PML' (6 (21.5{\%}) and 2 (7.1{\%}) patients, respectively) despite a very high suspicion for PML based on lesion evolution and signs of PML-immune reconstitution inflammatory syndrome on MRI, and development of compatible symptoms.CONCLUSIONS: The current case definition of PML has low sensitivity for diagnosis of NTZ-PML in a real-world clinical setting in which MRI is frequently used for PML screening. This may delay diagnosis and appropriate management of PML, and may complicate a valid estimation of PML incidence during NTZ therapy.",
author = "Wijburg, {Martijn T} and Clemens Warnke and Frederik Barkhof and Uitdehaag, {Bernard M J} and Joep Killestein and Wattjes, {Mike P}",
note = "{\circledC} Author(s) (or their employer(s)) 2018. No commercial re-use. See rights and permissions. Published by BMJ.",
year = "2019",
month = "1",
doi = "10.1136/jnnp-2018-318261",
language = "English",
volume = "90",
pages = "44--46",
journal = "Journal of Neurology, Neurosurgery and Psychiatry",
issn = "0022-3050",
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TY - JOUR

T1 - Performance of PML diagnostic criteria in natalizumab-associated PML

T2 - data from the Dutch-Belgian cohort

AU - Wijburg, Martijn T

AU - Warnke, Clemens

AU - Barkhof, Frederik

AU - Uitdehaag, Bernard M J

AU - Killestein, Joep

AU - Wattjes, Mike P

N1 - © Author(s) (or their employer(s)) 2018. No commercial re-use. See rights and permissions. Published by BMJ.

PY - 2019/1

Y1 - 2019/1

N2 - OBJECTIVE: To test the current progressive multifocal leukoencephalopathy (PML) diagnostic criteria by applying them to patients previously diagnosed with natalizumab (NTZ)-associated PML in a real-world clinical setting.METHODS: Patients from the Dutch-Belgian NTZ-PML cohort (n=28) were reviewed at the time of first diagnostic work-up and during follow-up, using the PML diagnostic criteria as proposed in a consensus statement from the American Academy of Neurology.RESULTS: At first diagnostic work-up, 18 patients (64.3%) met the criteria for high diagnostic certainty for PML ('definite PML' or 'probable PML'). During follow-up, this increased to 20 patients (71.4%) as JC virus DNA was detected in cerebrospinal fluid of two additional patients. Nonetheless, 28.6% of patients were still classified as 'possible PML' or 'not PML' (6 (21.5%) and 2 (7.1%) patients, respectively) despite a very high suspicion for PML based on lesion evolution and signs of PML-immune reconstitution inflammatory syndrome on MRI, and development of compatible symptoms.CONCLUSIONS: The current case definition of PML has low sensitivity for diagnosis of NTZ-PML in a real-world clinical setting in which MRI is frequently used for PML screening. This may delay diagnosis and appropriate management of PML, and may complicate a valid estimation of PML incidence during NTZ therapy.

AB - OBJECTIVE: To test the current progressive multifocal leukoencephalopathy (PML) diagnostic criteria by applying them to patients previously diagnosed with natalizumab (NTZ)-associated PML in a real-world clinical setting.METHODS: Patients from the Dutch-Belgian NTZ-PML cohort (n=28) were reviewed at the time of first diagnostic work-up and during follow-up, using the PML diagnostic criteria as proposed in a consensus statement from the American Academy of Neurology.RESULTS: At first diagnostic work-up, 18 patients (64.3%) met the criteria for high diagnostic certainty for PML ('definite PML' or 'probable PML'). During follow-up, this increased to 20 patients (71.4%) as JC virus DNA was detected in cerebrospinal fluid of two additional patients. Nonetheless, 28.6% of patients were still classified as 'possible PML' or 'not PML' (6 (21.5%) and 2 (7.1%) patients, respectively) despite a very high suspicion for PML based on lesion evolution and signs of PML-immune reconstitution inflammatory syndrome on MRI, and development of compatible symptoms.CONCLUSIONS: The current case definition of PML has low sensitivity for diagnosis of NTZ-PML in a real-world clinical setting in which MRI is frequently used for PML screening. This may delay diagnosis and appropriate management of PML, and may complicate a valid estimation of PML incidence during NTZ therapy.

U2 - 10.1136/jnnp-2018-318261

DO - 10.1136/jnnp-2018-318261

M3 - Article

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SP - 44

EP - 46

JO - Journal of Neurology, Neurosurgery and Psychiatry

JF - Journal of Neurology, Neurosurgery and Psychiatry

SN - 0022-3050

IS - 1

ER -