Isaacs' syndrome associated with myasthenia gravis, showing remission after cytoreductive surgery of pleural recurrence of thymoma.
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.
Fukushima
Kazuhiro
K
Department of Internal Medicine (Neurology and Rheumatology), Shinshu University School of Medicine, Matsumoto 390-8621, Japan.
Sato
Toshio
T
Mitsuhashi
Shigeaki
S
Kaneko
Kazuma
K
Yazaki
Masahide
M
Matsuda
Masayuki
M
Hashimoto
Takao
T
Hamanaka
Kazutoshi
K
Yoshida
Kazuo
K
Ikeda
Shu-ichi
S
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