Reactive Hyperemia-Peripheral Arterial Tonometry (RH-PAT)により血管内皮機能障害が示唆された冠攣縮性狭心症の10歳男児例

Sho Ikeda; 池田翔; Taku Ishii; 石井卓; Susumu Hosokawa; 細川奨; Tomohiro Nomura; 野村知弘; Ayako Nagashima; 長島彩子; Tomohiro Watanabe; 渡邉友博; Shozaburo Doi; 土井庄三郎

doi:10.9794/jspccs.37.220

Reactive Hyperemia-Peripheral Arterial Tonometry (RH-PAT)により血管内皮機能障害が示唆された冠攣縮性狭心症の10歳男児例A Case of Vascular Endothelial Dysfunction Detected Using Reactive Hyperemia-Peripheral Arterial Tonometry Contributed to the Development of Coronary Spastic Angina in a 10-Year-Old Boy

池田翔¹，石井卓²，細川奨²，野村知弘³，長島彩子²，渡邉友博²，土井庄三郎⁴Sho Ikeda¹, Taku Ishii², Susumu Hosokawa², Tomohiro Nomura³, Ayako Nagashima², Tomohiro Watanabe², Shozaburo Doi⁴

¹ 総合病院　土浦協同病院　小児科Department of Pediatrics, General Hospital Tsuchiura Kyodo Hospital ◇ Ibaraki, Japan

² 東京医科歯科大学医学部附属病院　小児科Department of Pediatrics, Tokyo Medical and Dental University Faculty of Medicine ◇ Tokyo, Japan

³ 川口市立医療センター　小児科Department of Pediatrics, Kawaguchi Municipal Medical Center ◇ Saitama, Japan

⁴ 独立行政法人　国立病院機構　災害医療センター　小児科Department of Pediatrics, National Hospital Organization Disaster Medical Center ◇ Tokyo, Japan

受付日：2020年9月13日Received: September 13, 2020

受理日：2021年4月11日Accepted: April 11, 2021

発行日：2021年11月1日Published: November 1, 2021

冠攣縮性狭心症（Coronary Spastic Angina: CSA）を小児期に発症することは稀であり，その病態も不明な部分が多い．症例は10歳男児で，以前より非運動時に15～30分ほどの胸痛を認めていた．就寝中に突然絞扼感を伴う左前胸部痛が出現し，30～40分で自然軽快した．心電図で広範な誘導におけるSTの上昇を認め，心筋逸脱酵素も有意に上昇していた．心エコーで急性心筋炎や冠動脈の器質的疾患を疑う所見を認めず，CSAを疑い硝酸薬を開始した後は症状再発なく経過した．アセチルコリン負荷試験では冠動脈3枝のびまん性の攣縮とV4–6でのST上昇を認めた．CSAと診断してカルシウム拮抗薬の内服を開始し，以後，狭心症状はみられていない．退院後に施行したReactive Hyperemia-Peripheral Arterial Tonometryでは血管内皮機能の指標である反応性血管指数が1.17と正常下限（1.67）を大きく下回っていた．基礎病態として全身性の血管内皮機能障害の存在が示唆され，小児期発症CSAの病態に関与する可能性があると考えられた．

The pathophysiology of coronary spastic angina (CSA), which rarely develops in childhood, remains unknown. A 10-year-old boy with a previous history of non-sustained chest pain at rest was admitted to the hospital because of a strong chest pain continuing for 30–40 minutes with a feeling of chest tightness during sleeping. Electrocardiography revealed an ST elevation in a wide range of leads, and myocardial enzyme levels were elevated. Echocardiography revealed no findings to suggest acute myocarditis or structural disease of the coronary artery. We made a diagnosis of CSA and started nitroglycerin therapy, with no pain recurrence. An acetylcholine provocation test induced diffuse spasm in all of the three coronary artery branches and ST elevation in V4–V6. We diagnosed the patient with CSA and started administration of a calcium channel blocker. No CSA recurrence was observed after the treatment. The reactive hyperemia index, which represents vascular endothelial function, was 1.17, as shown by reactive hyperemia-peripheral arterial tonometry. This value was far below the normal value (≥1.67). We suggest that systemic endothelial dysfunction might contribute to the development of CSA in childhood.

Key words: coronary spastic angina; pediatric; microvascular endothelial dysfunction; genetic factor; Reactive hyperemia-peripheral arterial tonometry

日本小児循環器学会雑誌 Pediatric Cardiology and Cardiac Surgery

症例報告Case Report