Japanese

Title	レポート全身型重症筋無力症寛解期に免疫チェックポイント阻害薬投与後に筋無力症クリーゼ・筋炎・胃腸炎・肝炎を併発した1例
Subtitle	治療経験
Authors	佐竹紅音*, 森嶋悠人**, 栗田尚史**, 土屋舞**, 羽田貴礼**, 新藤和雅**, 小宮山泰之***, 平山和義****, 佐藤葉子*****, 瀧山嘉久*
Authors (kana)
Organization	*笛吹中央病院神経内科, **山梨大学医学部付属病院神経内科, ***山梨大学医学部付属病院消化器内科, ****山梨大学医学部付属病院消化器外科, *****山梨PET画像診断クリニック
Journal	神経治療学
Volume	41
Number	1
Page	73-77
Year/Month	2024 /
Article	報告
Publisher	日本神経治療学会
Abstract	〔要約〕 患者は48歳, 女性. 既往歴, 胸腺腫. 2002年に眼瞼下垂と上肢の筋力低下が出現し当科受診した. 抗アセチルコリン受容体抗体(抗ACh-R抗体)陽性, エドロホニウム試験陽性であり重症筋無力症(myasthenia graves:MG)の診断で, 副腎皮質ステロイドパルス療法, 血漿交換を施行した. その後は少量の免疫抑制剤の内服で寛解状態を維持していた. 2021年10月, 横行結腸癌stage IVbと診断され, 外科的治療後に免疫チェックポイント阻害薬(immune-checkpoint inhibitors:ICIs)の2剤併用療法を受けた. その後, 血清CK値上昇, 全身の筋力低下, 眼瞼下垂, 嚥下機能悪化を認め, 免疫関連副作用(immune-related adverse events:irAE)による筋炎とMGのクリーゼ合併と診断され, その後, 呼吸障害が出現し, 人工呼吸器管理となった. Dexamethasoneの増量, 免疫グロブリン大量療法, 血漿交換を行うも効果はなかった. その後, irAE腸炎・肝炎の合併を来し, 第94病日に永眠した. MG治療中の患者では寛解期であってもICIsを使用する場合は多彩なirAEを合併する可能性があり, 注意が必要と思われた.
Practice	臨床医学：内科系
Keywords	myasthenia gravis, myasthenic crisis, myositis, immune-checkpoint inhibitor during, immune-related adverse event

English

Title	A case of myasthenic crisis, myositis, gastroenteritis, and hepatitis after administration of an immune-checkpoint inhibitor during remission of myasthenia gravis
Subtitle
Authors	Akane SATAKE*, Yuto MORISHIMA**, Takafumi KURITA**, Mai TSUCHIYA**, Takanori HATA**, Kazumasa SHINDO**, Yasuyuki KOMIYAMA***, Kazuyoshi HIRAYAMA****, Yoko SATOH*****, Yoshihisa TAKIYAMA*
Authors (kana)
Organization	*Department of Neurology, Fuefuki Central Hospital, **Department of Neurology, Hospital University of Yamanashi, ***Department of Gastroenterology, Hospital University of Yamanashi, ****Department of Gastroenterological Surgery, Hospital University of Yamanashi, *****Yamanashi PET Imaging Clinic
Journal	Neurological Therapeutics
Volume	41
Number	1
Page	73-77
Year/Month	2024 /
Article	Report
Publisher	Japanese Society of Neurological Therapeutics
Abstract	In 2010, a 48-year-old woman underwent extended thymectomy and radiation therapy for treatment of thymoma. In 2011, she came to our department because of ptosis and muscle weakness in her upper limbs and was diagnosed with generalized myasthenia gravis (MG) on the basis of an anti-acetylcholine receptor antibody level of 85 nmol/L. She underwent corticosteroid pulse therapy and plasma exchange, and her MG symptoms gradually improved. Subsequently, she was in remission for over 10 years under treatment with small amounts of dexamethasone and tacrolimus. In October 2021, she received combination anti-programmed cell death protein-1/CTLA-4 antibody therapy after surgical treatment for stage IV transverse colon cancer at our Department of Surgery. Then, she presented with generalized muscle weakness with elevated serum creatinine kinase, ptosis, and worsening of swallowing function and was diagnosed with myositis and MG crisis as an immune-related adverse event (irAE). The dexamethasone dosage was increased, and intravenous immunoglobulin was administered ; however, respiratory failure progressed, and she was placed on an artificial ventilator. On day 12 after the start of symptoms, hematemesis occurred, and upper endoscopy led to a diagnosis of gastroenteritis as an irAE. Thereafter, liver enzymes rapidly increased, and because no other diseases were involved, a diagnosis of hepatitis by an irAE was made. Plasma exchange therapy was performed, but the patient did not respond to treatment, and oral mycophenolate mofetil for hepatitis was not effective. She subsequently developed pneumonia due to cytomegalovirus and died on day 94 after the start of symptoms. This case highlights that physicians must be aware that patients with MG may experience a variety of irAEs when treated with immune-checkpoint inhibitors, even when they are in remission.
Practice	Clinical internal medicine
Keywords	myasthenia gravis, myasthenic crisis, myositis, immune-checkpoint inhibitor during, immune-related adverse event

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