Congenital absence of aortic valve (AAV) is a rare cardiac anomaly associated with high mortality. We present 2 prenatally diagnosed cases of AAV. In both cases, fetal echocardiography showed no aortic valve tissue and free aortic regurgitation. At 24 weeks' gestation, case 1 showed a hypoplastic hypocontractile left ventricle and mitral atresia, but did not develop hydrops and was born at term. Bilateral pulmonary arterial banding was performed with continuous infusion of prostaglandin E1 at 5 days of age followed by Norwood and bidirectional Glenn procedures at 3 months of age. The hypoplastic non-compliant left ventricle and mitral atresia might have limited aortic regurgitation. Case 2 showed hydrops at 23 weeks' gestation. An enlarged hypocontractile left ventricle and massive mitral insufficiency were noted. The regurgitant flow was directed to the right atrium through a foramen ovale. This peculiar hemodynamic established a malignant circuit which was arbitrarily called ‘inverse circulatory shunt'. Intrauterine death occurred at 28 weeks' gestation. An autopsy showed absent aortic valve leaflets but three tiny membranous remnants. Hemodynamic evaluation by fetal echocardiography should include the documentation of the presence of and assessment of the grade of aortic regurgitation, mitral regurgitation and blood flow through a foramen ovale as well as left ventricular function.
Spontaneous dehiscence of the aortic wall at the aortic commissure is not recognized as one of the usual pathological causes of aortic regurgitation. We describe the case of a 56-year-old man with hypertension, who experienced acutely progressive congestive heart failure due to massive aortic regurgitation. Local layer dehiscence around the commissure was noted with partial detachment of the commissure resulting in the loss of commissural support with secondary rupture of a non-coronary cusp, which led to massive aortic regurgitation.
We replaced the aortic root in a 43-year-old woman with Takayasu's aortitis associated with prosthetic aortic valve detachment. The patient's aortic valve had been replaced when she was 31 years old with a mechanical prosthesis to treat aortic regurgitation. Though C-reactive protein was kept almost normal with prednisolone, complete atrioventricular block suddenly appeared 12 years after the first operation. After the implantation of an artificial pacemaker, we closely followed up aortic root status. Aortography and echocardiography showed that the valve moved up and down, probably due to enlargement of the sinuses of Valsalva, without perivalvular leakage. We removed the prosthetic aortic valve, which was partially detached from the aortic valve ring at the right- and non-coronary cusps and successfully replaced the aortic root with a mechanical prosthesis inserted into a 26 mm woven graft. Although the postoperative course was uneventful, we closely continue to observe the case and to administer of antiinflammatory medication.
Osteochondroma is the most common benign bone tumor, which can sometimes cause vascular complications. Here we report two rare cases (a 48-year-old woman and a 28-yearold woman) presenting with pain and a pulsatile mass in the popliteal region. Computed tomography revealed pseudoaneurysm in the popliteal artery, which was closely associated with a protrusion of a femoral osteochondroma. Surgical repairs were performed, and the patients remained asymptomatic during follow-up. Therefore, considering the potential risk of vascular complications, close observation is mandatory in patients with femoral osteochondroma.
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