PMID- 35093766
OWN - NLM
STAT- MEDLINE
DCOM- 20220414
LR  - 20220414
IS  - 1872-6968 (Electronic)
IS  - 0303-8467 (Linking)
VI  - 214
DP  - 2022 Mar
TI  - Tofacitinib-induced Progressive Multifocal Leukoencephalopathy with Immune 
      Reconstitution Inflammatory Syndrome.
PG  - 107143
LID - S0303-8467(22)00024-5 [pii]
LID - 10.1016/j.clineuro.2022.107143 [doi]
AB  - BACKGROUND: Progressive multifocal leukoencephalopathy (PML) with subsequent 
      immune reconstitution inflammatory syndrome (IRIS) is a rare disease associated 
      with compromised immune systems. It has never been described in a patient taking 
      tofacitinib. CASE PRESENTATION: A 49-year-old woman with history of systemic 
      lupus erythematous treated with tofacitinib presented with several weeks of 
      intermittent fevers and altered mental status. MRI revealed multifocal 
      T2-weighted FLAIR hyperintensities in the subcortical white matter, including the 
      subcortical U-fibers, without mass effect or contrast enhancement, compatible 
      with PML. Tofacitinib was stopped and the patient's symptoms initially improved. 
      However, the patient presented again less than a week after discharge with three 
      days of left arm weakness, left facial droop, dysarthria, and one day of 
      confusion. Repeat MRI demonstrated interval progression in T2/FLAIR 
      hyperintensities with development of patchy gadolinium enhancement on T1-post 
      contrasted sequences, consistent with development of IRIS in the setting of 
      tofacitinib cessation. DISCUSSION: This is the first case describing PML-IRIS in 
      the setting of administration and subsequent cessation of tofacitinib.
CI  - Published by Elsevier B.V.
FAU - Yun, Johanna
AU  - Yun J
AD  - Emory University School of Medicine, Department of Neurology, Atlanta, GA, United 
      States. Electronic address: jyun33@emory.edu.
FAU - Osehobo, Ehizele
AU  - Osehobo E
AD  - Emory University School of Medicine, Department of Neurology, Atlanta, GA, United 
      States.
FAU - Lawson, Eric C
AU  - Lawson EC
AD  - Emory University School of Medicine, Department of Neurology, Atlanta, GA, United 
      States.
FAU - Harrison, Taylor
AU  - Harrison T
AD  - Emory University School of Medicine, Department of Neurology, Atlanta, GA, United 
      States.
FAU - Harrison, Amy
AU  - Harrison A
AD  - Emory University School of Medicine, Department of Neurology, Atlanta, GA, United 
      States.
LA  - eng
PT  - Case Reports
PT  - Journal Article
DEP - 20220124
PL  - Netherlands
TA  - Clin Neurol Neurosurg
JT  - Clinical neurology and neurosurgery
JID - 7502039
RN  - 0 (Contrast Media)
RN  - 0 (Piperidines)
RN  - 0 (Pyrimidines)
RN  - 87LA6FU830 (tofacitinib)
RN  - AU0V1LM3JT (Gadolinium)
SB  - IM
MH  - Contrast Media/adverse effects
MH  - Female
MH  - Gadolinium/adverse effects
MH  - Humans
MH  - *Immune Reconstitution Inflammatory Syndrome/drug therapy
MH  - *JC Virus
MH  - *Leukoencephalopathy, Progressive Multifocal/chemically 
      induced/complications/diagnostic imaging
MH  - Middle Aged
MH  - Piperidines
MH  - Pyrimidines
OTO - NOTNLM
OT  - Case report
OT  - JC virus
OT  - PML-IRIS
OT  - Tofacitinib
EDAT- 2022/01/31 06:00
MHDA- 2022/04/15 06:00
CRDT- 2022/01/30 20:33
PHST- 2021/08/02 00:00 [received]
PHST- 2021/12/27 00:00 [revised]
PHST- 2022/01/16 00:00 [accepted]
PHST- 2022/01/31 06:00 [pubmed]
PHST- 2022/04/15 06:00 [medline]
PHST- 2022/01/30 20:33 [entrez]
AID - S0303-8467(22)00024-5 [pii]
AID - 10.1016/j.clineuro.2022.107143 [doi]
PST - ppublish
SO  - Clin Neurol Neurosurg. 2022 Mar;214:107143. doi: 10.1016/j.clineuro.2022.107143. 
      Epub 2022 Jan 24.