title: Isaacs' syndrome associated with myasthenia gravis, showing remission after cytoreductive surgery of pleural recurrence of thymoma.
abstract: We report a patient with Isaacs' syndrome associated with myasthenia gravis and pleural recurrence of thymoma, who showed severe limb pain attributed to hyperexcitability of sensory nerves. Myokymia and severe pain were successfully treated with cytoreductive surgery and intraoperative hyperthermic intrathoracic perfusion chemotherapy, but neither pharmacotherapy nor plasma exchange showed obvious clinical effects. Pleural thymoma in our patient may have caused Isaacs' syndrome, probably by unconfirmed humoral immune mechanisms. Cytoreductive treatment for recurrent thymoma should be actively considered as a potent therapeutic option in refractory patients with disabling neuromyotonia symptoms.
authors.affiliation: Department of Internal Medicine (Neurology and Rheumatology), Shinshu University School of Medicine, Matsumoto 390-8621, Japan.
authors.forename: Kazuhiro
authors.initials: K
authors.lastname: Fukushima
authors.forename: Toshio
authors.initials: T
authors.lastname: Sato
authors.forename: Shigeaki
authors.initials: S
authors.lastname: Mitsuhashi
authors.forename: Kazuma
authors.initials: K
authors.lastname: Kaneko
authors.forename: Masahide
authors.initials: M
authors.lastname: Yazaki
authors.forename: Masayuki
authors.initials: M
authors.lastname: Matsuda
authors.forename: Takao
authors.initials: T
authors.lastname: Hashimoto
authors.forename: Kazutoshi
authors.initials: K
authors.lastname: Hamanaka
authors.forename: Kazuo
authors.initials: K
authors.lastname: Yoshida
authors.forename: Shu-ichi
authors.initials: S
authors.lastname: Ikeda
mesh_terms: D000328:Adult; D004576:Electromyography; D006801:Humans; D020386:Isaacs Syndrome / Q000209:etiology* / Q000503:physiopathology / Q000628:therapy*; D008297:Male; D018482:Muscle, Skeletal / Q000503:physiopathology; D009157:Myasthenia Gravis / Q000150:complications* / Q000276:immunology / Q000503:physiopathology; D009364:Neoplasm Recurrence, Local / Q000150:complications / Q000473:pathology / Q000601:surgery*; D012074:Remission Induction; D013945:Thymoma / Q000150:complications / Q000473:pathology / Q000601:surgery*; D013953:Thymus Neoplasms / Q000150:complications / Q000473:pathology / Q000601:surgery*; D014057:Tomography, X-Ray Computed