Hashimoto Hirotsugu, Kurata Atsushi, Mizuno Hideaki, Nashiro Tamaki, Hangaishi Akira, Kuroda Masahiko, Usuki Kensuke, Horiuchi Hajime
Department of Diagnostic Pathology, NTT Medical Center TokyoTokyo, Japan; Department of Molecular Pathology, Tokyo Medical UniversityTokyo, Japan.
Department of Molecular Pathology, Tokyo Medical University Tokyo, Japan.
Int J Clin Exp Pathol. 2015 Nov 1;8(11):15391-5. eCollection 2015.
Systemic amyloidosis is characterized by amyloid deposition throughout the body and subsequent dysfunction of various organs. Although pulmonary amyloidosis does occur, pulmonary hypertension (PH) caused by amyloidosis is extremely rare. In most of these cases, amyloid deposition occurred diffusely in alveolar septa, indicating that PH was due to lung disease and/or hypoxia. On the other hand, the mechanism of PH due to amyloid deposition in the pulmonary arteries has never been demonstrated. Here, we report the first case of PH due to amyloid deposition in pulmonary elastic arteries and muscular artery, which was complicated by multiple myeloma (MM). In the autopsy specimen of the patient, amyloid deposition was found mainly in the pulmonary arterial media, along with intimal thickening with luminal narrowing. PH thus appeared to be caused by marked decrease of pulmonary elasticity due to the amyloid deposition in the arterial media that resulted in stasis of the blood flow and subsequent luminal narrowing. Our present data demonstrates a new concept of PH caused by amyloidosis, namely, pulmonary arterial hypertension due to amyloidosis.
系统性淀粉样变性的特征是淀粉样蛋白在全身沉积以及随后各器官功能障碍。虽然肺淀粉样变性确实会发生,但由淀粉样变性引起的肺动脉高压(PH)极为罕见。在大多数这些病例中,淀粉样蛋白沉积在肺泡隔中弥漫性发生,表明PH是由肺部疾病和/或缺氧所致。另一方面,肺动脉中淀粉样蛋白沉积导致PH的机制从未得到证实。在此,我们报告首例因淀粉样蛋白沉积于肺弹性动脉和肌性动脉而导致的PH病例,该病例合并多发性骨髓瘤(MM)。在该患者的尸检标本中,淀粉样蛋白沉积主要见于肺动脉中膜,同时伴有内膜增厚和管腔狭窄。因此,PH似乎是由于动脉中膜淀粉样蛋白沉积导致肺弹性显著降低,进而引起血流淤滞和随后的管腔狭窄所致。我们目前的数据证明了淀粉样变性导致PH的一个新概念,即淀粉样变性所致肺动脉高压。