A tale of two sisters with hypertrophic cardiomyopathy and recurrent embolism: When is the optimal timing of the intervention for left atrial appendage?

Mai Kimura; Takashi Kohno; Shinji Makino; Shigeo Okuda; Kan Nawata; Ryo Yanagisawa; Hidenori Kojima; Takahiko Nishiyama; Yoshiyasu Aizawa; Shinsuke Yuasa; Mitsushige Murata; Yuichiro Maekawa; Kazuma Okamoto; Hideyuki Shimizu; Keiichi Fukuda

doi:10.1016/j.hrtlng.2018.08.010

A tale of two sisters with hypertrophic cardiomyopathy and recurrent embolism: When is the optimal timing of the intervention for left atrial appendage?

Mai Kimura, Takashi Kohno, Shinji Makino, Shigeo Okuda, Kan Nawata, Ryo Yanagisawa, Hidenori Kojima, Takahiko Nishiyama, Yoshiyasu Aizawa, Shinsuke Yuasa, Mitsushige Murata, Yuichiro Maekawa, Kazuma Okamoto, Hideyuki Shimizu, Keiichi Fukuda

研究成果: Article › 査読

抄録

Hypertrophic cardiomyopathy (HCM) is an extremely heterogeneous genetic disease that affects the left ventricle (LV) and has a varied clinical course and phenotypic expression. Here, we report a case of two sisters with HCM who developed a massive refractory left atrial appendage (LAA) thrombus and recurrent embolism. The older sister, who was at a high surgical risk due to progressive LV systolic dysfunction with an ejection fraction of 19%, underwent LAA plication in combination with implantation of an LV assist device after progression to treatment-refractory heart failure at the age of 49. The younger sister underwent surgical thrombectomy, LAA plication, and Maze surgery before deterioration of heart failure at the age of 47. She was free from embolism and atrial fibrillation for 2years after surgery. Individualized therapeutic approaches targeting the LAA at a relatively early stage are required in the subgroups of HCM patients with left atrial dysfunction.

本文言語	English
ページ（範囲）	198-200
ページ数	3
ジャーナル	Heart and Lung
巻	48
号	3
DOI	https://doi.org/10.1016/j.hrtlng.2018.08.010
出版ステータス	Published - 2019 5月 1

ASJC Scopus subject areas

呼吸器内科
集中医療医学
循環器および心血管医学

文献へのアクセス

10.1016/j.hrtlng.2018.08.010

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引用スタイル

Kimura, M., Kohno, T., Makino, S., Okuda, S., Nawata, K., Yanagisawa, R., Kojima, H., Nishiyama, T., Aizawa, Y., Yuasa, S., Murata, M., Maekawa, Y., Okamoto, K., Shimizu, H., & Fukuda, K. (2019). A tale of two sisters with hypertrophic cardiomyopathy and recurrent embolism: When is the optimal timing of the intervention for left atrial appendage? Heart and Lung, 48(3), 198-200. https://doi.org/10.1016/j.hrtlng.2018.08.010

Kimura, M, Kohno, T, Makino, S, Okuda, S, Nawata, K, Yanagisawa, R, Kojima, H, Nishiyama, T, Aizawa, Y, Yuasa, S, Murata, M, Maekawa, Y, Okamoto, K, Shimizu, H & Fukuda, K 2019, 'A tale of two sisters with hypertrophic cardiomyopathy and recurrent embolism: When is the optimal timing of the intervention for left atrial appendage?', Heart and Lung, vol. 48, no. 3, pp. 198-200. https://doi.org/10.1016/j.hrtlng.2018.08.010

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AU - Kojima, Hidenori

AU - Nishiyama, Takahiko

AU - Aizawa, Yoshiyasu

AU - Yuasa, Shinsuke

AU - Murata, Mitsushige

AU - Maekawa, Yuichiro

AU - Okamoto, Kazuma

AU - Shimizu, Hideyuki

AU - Fukuda, Keiichi

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N2 - Hypertrophic cardiomyopathy (HCM) is an extremely heterogeneous genetic disease that affects the left ventricle (LV) and has a varied clinical course and phenotypic expression. Here, we report a case of two sisters with HCM who developed a massive refractory left atrial appendage (LAA) thrombus and recurrent embolism. The older sister, who was at a high surgical risk due to progressive LV systolic dysfunction with an ejection fraction of 19%, underwent LAA plication in combination with implantation of an LV assist device after progression to treatment-refractory heart failure at the age of 49. The younger sister underwent surgical thrombectomy, LAA plication, and Maze surgery before deterioration of heart failure at the age of 47. She was free from embolism and atrial fibrillation for 2years after surgery. Individualized therapeutic approaches targeting the LAA at a relatively early stage are required in the subgroups of HCM patients with left atrial dysfunction.

AB - Hypertrophic cardiomyopathy (HCM) is an extremely heterogeneous genetic disease that affects the left ventricle (LV) and has a varied clinical course and phenotypic expression. Here, we report a case of two sisters with HCM who developed a massive refractory left atrial appendage (LAA) thrombus and recurrent embolism. The older sister, who was at a high surgical risk due to progressive LV systolic dysfunction with an ejection fraction of 19%, underwent LAA plication in combination with implantation of an LV assist device after progression to treatment-refractory heart failure at the age of 49. The younger sister underwent surgical thrombectomy, LAA plication, and Maze surgery before deterioration of heart failure at the age of 47. She was free from embolism and atrial fibrillation for 2years after surgery. Individualized therapeutic approaches targeting the LAA at a relatively early stage are required in the subgroups of HCM patients with left atrial dysfunction.

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