Nolte K B
Office of the Medical Investigator, University of New Mexico School of Medicine, Albuquerque 87131-5091, USA.
Hum Pathol. 2000 Aug;31(8):1002-4. doi: 10.1053/hupa.2000.0311002.
A 26-year-old woman collapsed and died suddenly while dancing. Autopsy findings included the cutaneous lesions of pseudoxanthoma elasticum (PXE), a rare genetic disease with autosomal dominant and recessive inheritance patterns. Pathologic findings of PXE (degenerated elastic fibers) were seen in the stenotic epicardial coronary arteries, the intramyocardial arterioles, the subendocardium, the mitral valve, and the blood vessels of other viscera. The mitral valve was slightly myxoid. Intramyocardial arteriolar involvement has not been previously described in PXE. The other cardiac findings have only been described in a few cases. Although mitral valve prolapse in PXE has been shown echocardiographically, it is unclear whether or not the mitral valve findings in this case represent the substrate for this condition. It is important that autopsy pathologists search carefully for the pathognomonic skin lesions of PXE in cases of sudden death associated with coronary disease, mitral valve prolapse, or endocardial lesions. Recognition of this disease is essential for proper genetic counseling of surviving family members.
一名26岁女性在跳舞时突然晕倒并死亡。尸检结果包括弹性假黄瘤(PXE)的皮肤病变,这是一种罕见的具有常染色体显性和隐性遗传模式的遗传病。在狭窄的心外膜冠状动脉、心肌内小动脉、心内膜下、二尖瓣以及其他内脏血管中可见PXE的病理表现(弹性纤维退变)。二尖瓣略显黏液样。心肌内小动脉受累在PXE中此前未曾有过描述。其他心脏表现仅在少数病例中有过描述。尽管通过超声心动图已显示PXE中有二尖瓣脱垂,但尚不清楚该病例中的二尖瓣表现是否为此种情况的基础。对于与冠心病、二尖瓣脱垂或心内膜病变相关的猝死病例,尸检病理学家仔细查找PXE的特征性皮肤病变非常重要。识别这种疾病对于对幸存家庭成员进行正确的遗传咨询至关重要。