Astara Kyriaki, Stenos Christos, Kalafatakis Konstantinos, Lypiridou Maria, Stouraitis Georgios
Department of Neurology, 417 Army Share Fund Hospital, Athens, GRC.
Department of Neurology, Errikos Ntynan Hospital Center, Athens, GRC.
Cureus. 2024 Dec 23;16(12):e76261. doi: 10.7759/cureus.76261. eCollection 2024 Dec.
Posterior reversible encephalopathy syndrome (PRES) is a clinical manifestation of various underlying causes, characterized by the combination of clinical and imaging findings associated with the posterior cerebral areas and relating to arterial hypertension and endothelial dysfunction. No association was made so far between PRES and McCune-Albright syndrome (MAS), a rare genetic disorder resulting in fibrous dysplasia. A 33-year-old female with MAS was presented to the emergency department of the 417 Army Share Fund Hospital in Athens (Greece) after seizure activity with two episodes of ocular upward deviation and transient facial palsy, each lasting a few minutes, followed by a postictal phase. On arrival, vital sign measurements revealed sinus tachycardia and elevated systolic and diastolic blood pressure (177/102 mm Hg). A neurologic examination demonstrated irritability and confusion, complete blindness from the left eye, and progressive visual loss from the right due to compression of optic nerves by fibro-osseous tissue bilaterally. Neuroimaging revealed the symmetrical presence of signs of vasogenic edema and dysfunction of the posterior parts of the cerebral hemispheres and, in conjunction with the clinical manifestations, advocated the diagnosis of PRES. The patient was treated with systematic administration of anticonvulsants, antihypertensive agents, and nebivolol. Laboratory examination indicated the presence of Cushing syndrome. The patient was discharged afebrile, hemodynamically stable, and clinically improved. MAS covers a spectrum of endocrine dysregulation, resulting in clinical manifestations of high variability. Even in cases of mild hypercortisolemia, it is vital to raise the clinical suspicion of CS, as its reverberations may occur with abrupt onset, like PRES.
后部可逆性脑病综合征(PRES)是多种潜在病因的临床表现,其特征是与大脑后部区域相关的临床和影像学表现相结合,且与动脉高血压和内皮功能障碍有关。到目前为止,尚未发现PRES与McCune - Albright综合征(MAS)之间存在关联,MAS是一种导致纤维性发育异常的罕见遗传疾病。一名患有MAS的33岁女性因癫痫发作后出现两次眼球上翻和短暂性面瘫,每次持续几分钟,随后进入发作后期,被送往雅典(希腊)417陆军共享基金医院急诊科。到达时,生命体征测量显示窦性心动过速,收缩压和舒张压升高(177/102 mmHg)。神经系统检查发现患者烦躁不安、意识模糊,左眼完全失明,右眼因双侧纤维骨性组织对视神经的压迫而逐渐视力丧失。神经影像学检查显示大脑半球后部存在对称性血管源性水肿和功能障碍迹象,结合临床表现,支持PRES的诊断。患者接受了抗惊厥药、降压药和奈必洛尔的系统治疗。实验室检查表明存在库欣综合征。患者出院时无发热,血流动力学稳定,临床症状改善。MAS涵盖一系列内分泌失调,导致临床表现高度可变。即使在轻度皮质醇增多症的情况下,提高对库欣综合征(CS)的临床怀疑也至关重要,因为其影响可能像PRES一样突然发作。