Kaneko Mitsunobu, Kasao Masashi, Obikane Hiyo, Ueda Kazutaka
Department of Cardiology, Tokyo Metropolitan Police Hospital, 4-22-1 Nakano, Nakano, Tokyo 164-8541, Japan.
Department of Pathology, Tokyo Metropolitan Police Hospital, 4-22-1 Nakano, Nakano, Tokyo 164-8541, Japan.
Eur Heart J Case Rep. 2024 Nov 14;8(12):ytae608. doi: 10.1093/ehjcr/ytae608. eCollection 2024 Dec.
Drug-induced phospholipidosis (DIPL) is an acquired lysosomal storage disorder characterized by the accumulation of lamellar bodies and phospholipids, typically associated with the use of cationic amphiphilic drugs (CADs). Over 200 marketed CADs, including widely prescribed β-blockers, have the potential to induce phospholipid deposition in various organs. In rare cases, DIPL may lead to secondary cardiomyopathy.
We report the case of a 70-year-old man with a history of hypertension, permanent atrial fibrillation, and Stanford type B aortic dissection. The patient presented with a 2-week history of worsening dyspnoea. Examination revealed cardiomegaly, elevated B-type natriuretic peptide, and left ventricular dysfunction with an ejection fraction of 24%. Despite intensive medical treatment, the patient developed severe pulmonary congestion and died on Day 35. Post-mortem examination revealed vacuolar degeneration and lamellar body accumulation in the myocardium, consistent with DIPL. The most likely causal agent was bisoprolol, one of the patient's prescribed CADs.
While β-blockers are commonly used for the treatment of hypertension and heart failure, their potential to induce phospholipid deposition in the heart is rare but significant. This case underscores the need for awareness of DIPL as a potential adverse effect, especially in patients receiving CADs.
药物性磷脂沉积症（DIPL）是一种获得性溶酶体贮积症，其特征为板层小体和磷脂的蓄积，通常与阳离子两亲性药物（CADs）的使用有关。超过200种已上市的CADs，包括广泛处方的β受体阻滞剂，都有可能在各个器官中诱导磷脂沉积。在罕见情况下，DIPL可能导致继发性心肌病。
我们报告了一名70岁男性的病例，他有高血压、永久性心房颤动和B型主动脉夹层病史。患者出现进行性加重的呼吸困难2周。检查发现心脏扩大、B型利钠肽升高以及左心室功能障碍，射血分数为24%。尽管进行了强化治疗，患者仍出现严重的肺淤血，并于第35天死亡。尸检显示心肌有空泡变性和板层小体积聚，符合DIPL。最可能的致病因素是比索洛尔，它是患者所服用的CADs之一。
虽然β受体阻滞剂常用于治疗高血压和心力衰竭，但其在心脏中诱导磷脂沉积的可能性虽罕见但很显著。该病例强调了认识到DIPL作为一种潜在不良反应的必要性，尤其是在接受CADs治疗的患者中。
