Jain Sakshi, Sirekulam Vaishnavi, Kinthada SudhaRani, Patel Rashi Bharat, Naik Nishthaben, Jain Shikha, Khan Tanzina, Gill Harmeet, Patel Neel, Nanjundappa Athmananda, Vala Lovekumar, Siripuram Chandu
Geriatrics, Hackensack University Medical Center, Hackensack, USA.
Medicine, Vijayanagar Institute of Medical Sciences, Ballari, IND.
Cureus. 2024 May 12;16(5):e60165. doi: 10.7759/cureus.60165. eCollection 2024 May.
Cerebral autosomal dominant arteriopathy with subcortical infarcts and leukoencephalopathy (CADASIL) is an uncommon genetic disorder that affects small blood vessels in the brain. It leads to neurological symptoms, disability-adjusted life years, and difficult emotional and physical situations for patients and their families. As unusual brain symptoms appear, it becomes important to understand the different clinical manifestations of CADASIL. Our case report and review examine several cases to demonstrate different presentations and management strategies of CADASIL. A 52-year-old male with a family history of strokes at a young age from his father and paternal grandfather presented to a neurology clinic for left facial droop and drooling. Brain magnetic resonance imaging showed extensive periventricular and subcortical white matter disease, including the external capsule and subcortical white matter of the temporal lobe. Findings were suggestive of small vessel vasculopathy. A cerebral angiogram showed that all large extra- and intracranial vessels were patent without evidence of aneurysm formation. There was no obvious evidence of beading of the distal intracranial vessels. Cerebrospinal fluid studies were normal. The NOTCH3 mutation was sent to test for CADASIL, which came back positive. The patient was started on aspirin (81 mg) and atorvastatin (20 mg) daily. The patient was counseled on the possibility of having an ischemic or hemorrhagic stroke. Aspirin and atorvastatin were continued, a neuropsychological evaluation was ordered, and CADASIL genetic counseling and testing were offered to him and his children. Over several years, patients developed several strokes and seizures due to infarcts. He also developed intraparenchymal hemorrhage complicated by dysphagia, requiring a feeding tube. Due to his severe physical debility, he was discharged to a nursing home for rehabilitation, where he did not improve with therapy and remained bedbound. He was discharged and sent home with his family. CADASIL can present as a diagnostic challenge due to its common presentation with migraines, transient ischemic attacks, and strokes, with or without risk factors. This unique presentation of CADASIL with facial palsy highlights the importance of emerging atypical presentations and the need for a detailed history of neuroimaging, family history, and personal history of neurovascular events. By accurately diagnosing the condition, patients and families can be counseled on the disease course and genetics. Management requires a multidisciplinary approach with neurology, genetic counseling, physical therapy, psychology, and psychiatry if depression or anxiety is present, with the aim of improving the patient's quality of life.
伴有皮质下梗死和白质脑病的常染色体显性遗传性脑动脉病(CADASIL)是一种罕见的遗传性疾病,会影响脑部的小血管。它会导致神经症状、伤残调整生命年,并给患者及其家人带来情感和身体上的困境。随着不寻常的脑部症状出现,了解CADASIL的不同临床表现变得很重要。我们的病例报告和综述研究了几例病例,以展示CADASIL的不同表现和管理策略。一名52岁男性,其父亲和祖父在年轻时有中风家族史,因左侧面部下垂和流口水就诊于神经科诊所。脑部磁共振成像显示广泛的脑室周围和皮质下白质病变,包括外囊和颞叶皮质下白质。检查结果提示小血管血管病变。脑血管造影显示所有大的颅外和颅内血管通畅,无动脉瘤形成迹象。颅内远端血管无明显串珠样改变证据。脑脊液检查正常。进行了NOTCH3突变检测以诊断CADASIL,结果呈阳性。患者开始每天服用阿司匹林(81毫克)和阿托伐他汀(20毫克)。告知患者有发生缺血性或出血性中风的可能性。继续服用阿司匹林和阿托伐他汀,安排了神经心理学评估,并为患者及其子女提供了CADASIL遗传咨询和检测。几年间,患者因梗死多次发生中风和癫痫。他还发生了脑实质内出血并伴有吞咽困难,需要插入喂食管。由于他身体极度虚弱,出院后被送往疗养院康复,但治疗后并无改善,仍卧床不起。他出院后与家人回家。CADASIL因其常与偏头痛、短暂性脑缺血发作和中风共同出现,无论有无危险因素,都可能构成诊断挑战。CADASIL这种伴有面瘫的独特表现凸显了新出现的非典型表现的重要性,以及详细了解神经影像学病史、家族史和神经血管事件个人史的必要性。通过准确诊断病情,可以为患者及其家人提供关于疾病病程和遗传学的咨询。管理需要多学科方法,包括神经科、遗传咨询、物理治疗、心理学,如果存在抑郁或焦虑还需要精神科,目的是提高患者的生活质量。