Acute heart failure in a patient with Osler's disease presenting with circumferential left ventricular wall thickening during the perinatal period: a case report.

BACKGROUND: Osler's disease (hereditary haemorrhagic telangiectasia, HHT) is associated with telangiectasia and arteriovenous malformations, but reports of myocardial involvement are rare.

CASE SUMMARY: A 37-year-old woman with a history of pulmonary arteriovenous fistula and paradoxical embolism due to Osler's disease with endoglin mutation (HHT1) became pregnant despite high-risk considerations and delivered by cesarean section. On postpartum day 23, she developed dyspnoea and was diagnosed with acute heart failure. Echocardiography showed decreased cardiac output and circumferential left ventricular wall thickening compared with normal findings before delivery. Myocardial biopsy revealed no inflammatory infiltration but showed interstitial enlargement and red blood cell leakage, suggestive of myocardial haemorrhage. Coronary angiography demonstrated contrast retention in distal coronary arteries. Treatment with diuretics led to clinical improvement, and cardiac function returned to normal by day 78 postpartum.

DISCUSSION: This is a rare case of acute heart failure in Osler's disease during the perinatal period, associated with transient circumferential left ventricular wall thickening. The findings suggest that abnormal myocardial microvasculature in Osler's disease may predispose to interstitial haemorrhage under perinatal haemodynamic stress and anticoagulant therapy.