PMID- 30077976
OWN - NLM
STAT- MEDLINE
DCOM- 20181211
LR  - 20200804
IS  - 1757-790X (Electronic)
IS  - 1757-790X (Linking)
VI  - 2018
DP  - 2018 Aug 4
TI  - Sensory ganglionopathy associated with drug-induced hypersensitivity syndrome 
      caused by mexiletine.
LID - bcr-2017-222540 [pii]
LID - 10.1136/bcr-2017-222540 [doi]
LID - bcr2017222540
AB  - Although various causes are reported for sensory ganglionopathy, drug-induced 
      hypersensitivity syndrome (DIHS) has not been considered a possibility. We 
      describe a 70-year-old woman, previously administered mexiletine hydrochloride 
      for 4 weeks, who presented with systemic oedematous erythema and subacute 
      progressive gait disturbance. Evaluation revealed lymphadenopathy with atypical 
      lymphocytosis and eosinophilia, and human herpesvirus 6 (HHV-6) reactivation. 
      Neurological examination indicated the almost complete loss of joint positional 
      sense in her extremities; her tendon reflex was lost and there was marked 
      pseudoathetosis and Romberg's sign. Skin biopsy revealed spongiosis with 
      lymphocyte infiltration. Based on these findings, we diagnosed acute sensory 
      ganglionopathy secondary to DIHS. Although her DIHS-induced symptoms subsided 
      after methylprednisolone treatment, partial remission of sensory ganglionopathy 
      occurred, even after subsequent intravenous immunoglobulin therapy. This case 
      suggests the possibility that reactivation of HHV-6 may be involved in the 
      pathomechanism of sensory ganglionopathy.
CI  - (c) BMJ Publishing Group Limited 2018. No commercial re-use. See rights and 
      permissions. Published by BMJ.
FAU - Yokote, Akira
AU  - Yokote A
AD  - Department of Neurology, Fukuoka University School of Medicine, Fukuoka, Japan.
AD  - Department of Neurology, Utano National Hospital, National Hospital Organization, 
      Kyoto, Japan.
FAU - Tomita, Satoshi
AU  - Tomita S
AD  - Department of Neurology, Utano National Hospital, National Hospital Organization, 
      Kyoto, Japan.
FAU - Sawada, Hideyuki
AU  - Sawada H
AD  - Department of Neurology, Utano National Hospital, National Hospital Organization, 
      Kyoto, Japan.
LA  - eng
PT  - Case Reports
PT  - Journal Article
DEP - 20180804
PL  - England
TA  - BMJ Case Rep
JT  - BMJ case reports
JID - 101526291
RN  - 0 (Anti-Arrhythmia Agents)
RN  - 0 (Glucocorticoids)
RN  - 0 (Immunoglobulins, Intravenous)
RN  - 1U511HHV4Z (Mexiletine)
RN  - X4W7ZR7023 (Methylprednisolone)
SB  - IM
MH  - Aged
MH  - Anti-Arrhythmia Agents/*adverse effects
MH  - Drug Hypersensitivity Syndrome/*complications/therapy
MH  - Female
MH  - Ganglia, Sensory/*pathology
MH  - Glucocorticoids/therapeutic use
MH  - Humans
MH  - Immunoglobulins, Intravenous/therapeutic use
MH  - Methylprednisolone/therapeutic use
MH  - Mexiletine/*adverse effects
MH  - Peripheral Nervous System Diseases/*chemically induced/therapy
MH  - Skin/pathology
PMC - PMC6078255
OTO - NOTNLM
OT  - peripheral nerve disease
OT  - unwanted effects / adverse reactions
COIS- Competing interests: HS received a research grant from the National Hospital 
      Organization, Tokyo, Japan.
EDAT- 2018/08/06 06:00
MHDA- 2018/12/12 06:00
PMCR- 2020/08/04
CRDT- 2018/08/06 06:00
PHST- 2018/08/06 06:00 [entrez]
PHST- 2018/08/06 06:00 [pubmed]
PHST- 2018/12/12 06:00 [medline]
PHST- 2020/08/04 00:00 [pmc-release]
AID - bcr-2017-222540 [pii]
AID - 10.1136/bcr-2017-222540 [doi]
PST - epublish
SO  - BMJ Case Rep. 2018 Aug 4;2018:bcr2017222540. doi: 10.1136/bcr-2017-222540.