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CASE REPORT
Sensory ganglionopathy associated with drug-induced hypersensitivity syndrome caused by mexiletine
  1. Akira Yokote1,2,
  2. Satoshi Tomita2,
  3. Hideyuki Sawada2
  1. 1 Department of Neurology, Fukuoka University School of Medicine, Fukuoka, Japan
  2. 2 Department of Neurology, Utano National Hospital, National Hospital Organization, Kyoto, Japan
  1. Correspondence to Dr Hideyuki Sawada, sawada.unh{at}gmail.com

Summary

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.

  • peripheral nerve disease
  • unwanted effects / adverse reactions
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Footnotes

  • Contributors AY evaluated the patient, analysed and interpreted the data, reviewed the literature and was the primary author of the manuscript. ST was the primary physician for the patient and contacted the patient for consent. HS was involved in the planning and guidance of the written manuscript. AY, ST and HS were equally involved in medical management of the patient. All authors read and approved the final manuscript.

  • Funding The authors have not declared a specific grant for this research from any funding agency in the public, commercial or not-for-profit sectors.

  • Competing interests HS received a research grant from the National Hospital Organization, Tokyo, Japan.

  • Patient consent Obtained.

  • Provenance and peer review Not commissioned; externally peer reviewed.

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