Accelerated Atherogenicity in Tangier Disease: A Case Accompanied by Extensive Atherosclerotic Lesions, Leriche Syndrome and Bleeding Tendency, and Review of the Literature
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- Muratsu Jun
- Department of Cardiovascular Medicine, Sumitomo Hospital
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- Koseki Masahiro
- Department of Cardiovascular Medicine, Osaka University Graduate School of Medicine Department of Health Care Center, Osaka University
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- Masuda Daisaku
- Department of Cardiovascular Medicine, Osaka University Graduate School of Medicine
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- Yasuga Yuji
- Department of Cardiovascular Medicine, Sumitomo Hospital
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- Tomoyama Satoki
- Department of Cardiovascular Medicine, Sumitomo Hospital
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- Ataka Keiji
- Department of Cardiovascular Surgery, Sumitomo Hospital
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- Yagi Yoshiki
- Department of Cardiovascular Medicine, Nissay Hospital
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- Nakagawa Atsushi
- Department of Cardiovascular Medicine, Nissay Hospital
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- Hamada Hidehumi
- Department of Cardiovascular Medicine, Nissay Hospital
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- Fujita Shigeki
- Department of Pathology, Sumitomo Hospital
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- Hattori Hiroaki
- Research Department, R&D Center, BML
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- Ohama Tohru
- Department of Cardiovascular Medicine, Osaka University Graduate School of Medicine
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- Nishida Makoto
- Department of Cardiovascular Medicine, Osaka University Graduate School of Medicine Department of Health Care Center, Osaka University
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- Hiraoka Hisatoyo
- Department of Cardiovascular Medicine, Sumitomo Hospital
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- Matsuzawa Yuji
- Department of Cardiovascular Medicine, Sumitomo Hospital
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- Yamashita Shizuya
- Department of Cardiovascular Medicine, Osaka University Graduate School of Medicine Department of Cardiovascular Medicine, Rinku General Medical Center
書誌事項
- タイトル別名
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- Accelerated Atherogenicity in Tangier Disease
- A Case Accompanied by Extensive Atherosclerotic Lesions, Leriche Syndrome and Bleeding Tendency, and Review of the Literature
抄録
<p>We report a case of Tangier disease with Leriche syndrome and bleeding tendency. In this male patient, nasal hemorrhage had been observed frequently throughout childhood. At 46 years old, he experienced effort angina, and coronary angiography demonstrated 75% stenosis in the right coronary artery. Orange-colored tonsils, mild hepatosplenomegaly and very low levels of serum high-density lipoprotein cholesterol (HDL-C) were observed, and the patient was diagnosed with Tangier disease. At 52 years old, effort angina recurred. Coronary angiography revealed 75% stenosis of the left main trunk, left anterior descending, and right coronary arteries. Stenosis of the brachiocephalic and right common iliac arteries was also recorded. Stents were implanted, and coronary artery bypass surgery was performed. At 53 years old, 15 months after surgery, the patient reported intermittent claudication, coldness of feet, and impotence. Aortic angiography showed progression of the stenosis at the bifurcation of the common iliac artery. The patient was diagnosed with Leriche syndrome, and aorta–left external iliac artery graft bypass surgery was performed. After surgery, oozing from subcutaneous tissue and leaking from the anastomotic region were observed. Additional analysis revealed two single-nucleotide polymorphisms (V825I and N935T) in the ATP-binding cassette transporter A1 (ABCA1) gene, and accumulation of small dense low-density lipoprotein together with low levels of HDL-C. In Tangier disease, HDL-C is markedly decreased because of ABCA1 deficiency. However, this is the first reported case to exhibit extensive atherosclerosis and bleeding tendency. This patient had atypical extensive and multiple atherosclerotic lesions, accompanied by Leriche syndrome and uncontrollable bleeding.</p>
収録刊行物
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- Ｊｏｕｒｎａｌ ｏｆ Ａｔｈｅｒｏｓｃｌｅｒｏｓｉｓ ａｎｄ Ｔｈｒｏｍｂｏｓｉｓ
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Ｊｏｕｒｎａｌ ｏｆ Ａｔｈｅｒｏｓｃｌｅｒｏｓｉｓ ａｎｄ Ｔｈｒｏｍｂｏｓｉｓ 25 (10), 1076-1085, 2018-10-01
一般社団法人 日本動脈硬化学会
