Nouduri Sirisha, Padmanabhan Rajiv, Hicks Richard, Abbott Mary-Alice, O'Brien Dennis, Schlaug Gottfried
Department of Neurology, Baystate Medical Center, University of Massachusetts Chan Medical School-Baystate, Springfield, MA, United States.
Department of Radiology, Baystate Medical Center, University of Massachusetts Chan Medical School-Baystate, Springfield, MA, United States.
Front Neurol. 2022 Dec 14;13:1043695. doi: 10.3389/fneur.2022.1043695. eCollection 2022.
Mitochondrial encephalomyopathy, lactic acidosis, stroke-like episodes, and other features (short stature, headaches, seizures, and sensorineural hearing loss) constitute characteristics of MELAS syndrome. MELAS is a rare condition due to mutations in maternally inherited mitochondrial DNA with levels of heteroplasmy possibly related to late adulthood presentation. A previously reported MELAS case coexisted with presumed Antiphospholipid Antibody Syndrome (APLAS), but the connection between MELAS and a potential APLAS is unclear. A 29-year-old woman presented with mild right-sided sensorimotor symptoms and mixed aphasia in November 2021. She presented again in May 2022 for unrelenting headaches and was found to have a new right hemisphere syndrome with mild left-sided sensorimotor symptoms, hemineglect, and anosognosia. Characteristic lab and imaging studies were obtained. During the first presentation (October 2021), the discovery of anticardiolipin IgM antibodies (aCL) (and their replication 3 months later) led to a diagnosis of APLAS, and Warfarin was initiated. During the second admission (May 2022), a new stroke-like lesion on the right hemisphere with characteristic features not suggestive of ischemia was detected, which led to a diagnosis of MELAS (m3243A > G mutation). Although MELAS and APLAS could co-exist, alternatively, it is possible that antiphospholipid antibodies might be generated when the strongly anionic Cardiolipin-Hydroperoxide from the inner mitochondrial membrane is exposed to immune component cells upon cell lysis. Thus, the presence of aCL in patients with stroke-like lesions might masquerade as an APLAS, but should probably be questioned if only aCL are repeatedly found and imaging findings are not characteristic for ischemic lesions.
线粒体脑肌病、乳酸酸中毒、卒中样发作及其他特征(身材矮小、头痛、癫痫发作和感音神经性听力损失)构成了MELAS综合征的特点。MELAS是一种罕见疾病,由母系遗传的线粒体DNA突变引起,异质性水平可能与成年后期发病有关。先前报道的一例MELAS病例与疑似抗磷脂抗体综合征(APLAS)共存,但MELAS与潜在APLAS之间的联系尚不清楚。一名29岁女性于2021年11月出现轻度右侧感觉运动症状和混合性失语。她于2022年5月因持续性头痛再次就诊,被发现患有新的右半球综合征,伴有轻度左侧感觉运动症状、偏侧忽视和疾病感缺失。进行了特征性的实验室和影像学检查。在首次就诊时(2021年10月),抗心磷脂IgM抗体(aCL)的发现(3个月后复查结果一致)导致APLAS的诊断,并开始使用华法林治疗。在第二次入院时(2022年5月),检测到右半球有一个具有特征性表现、不提示缺血的新的卒中样病变,这导致了MELAS(m3243A>G突变)的诊断。虽然MELAS和APLAS可能共存,但也有可能是当线粒体内膜上带强负电荷的心磷脂过氧化氢在细胞裂解时暴露于免疫细胞成分时产生了抗磷脂抗体。因此,有卒中样病变的患者中aCL的存在可能会伪装成APLAS,但如果只是反复发现aCL且影像学表现不符合缺血性病变的特征,则可能需要对此提出质疑。