Department of General Medicine, Saga University Hospital, Saga, Japan.
Department of Internal Medicine, Nagahama City Kohoku Hospital, Nagahama, Shiga, Japan.
Am J Case Rep. 2023 Mar 11;24:e938654. doi: 10.12659/AJCR.938654.
BACKGROUND Sporadic Creutzfeldt-Jakob disease (sCJD) is a fatal disease caused by the change of prion protein (PrP). Affected patients present with rapidly progressive cognitive dysfunction, myoclonus, or akinetic mutism. Diagnosing the Heidenhain variant of sCJD, which initially causes various visual symptoms, can be particularly difficult. CASE REPORT A 72-year-old woman presented with a 2- to 3-month history of photophobia, blurring vision in both eyes. Seven days previously, she showed visual impairment of 20/2000 in both eyes. Left homonymous hemianopia and restricted downward movement of the left eye were observed with an intact pupillary light reflex and normal fundoscopy. On admission, her visual acuity was light perception. Cranial magnetic resonance imaging revealed no abnormality, and electroencephalography showed no periodic synchronous discharges. Cerebrospinal fluid examination on the sixth hospital day revealed tau and 14-3-3 protein with a positive result of real-time quaking-induced conversion. She thereafter developed myoclonus and akinetic mutism and died. Autopsy revealed thinning and spongiform change of the cerebral cortex of the right occipital lobe. Immunostaining showed synaptic-type deposits of abnormal PrP and hypertrophic astrocytes. Consequently, she was diagnosed with the Heidenhain variant of sCJD with both methionine/methionine type 1 and type 2 cortical form based on the western blot of cerebral tissue and PrP gene codon 129 polymorphism. CONCLUSIONS When a patient presents with various progressive visual symptoms, even without typical findings of electroencephalography or cranial magnetic resonance imaging, it is essential to suspect the Heidenhain variant of sCJD and perform appropriate cerebrospinal fluid tests.
散发性克雅氏病(sCJD)是一种由朊病毒蛋白(PrP)改变引起的致命疾病。受影响的患者表现出快速进行性认知功能障碍、肌阵挛或无动性缄默。诊断最初引起各种视觉症状的海登海因变异型 sCJD 可能特别困难。
一名 72 岁女性因畏光、双眼视力模糊就诊,病史为 2-3 个月。7 天前,她出现双眼视力障碍,视力为 20/2000。左眼同向偏盲和左眼运动受限,瞳孔光反射正常,眼底检查正常。入院时,她的视力为光感。颅脑磁共振成像未见异常,脑电图无周期性同步放电。入院第 6 天行脑脊液检查,发现 tau 和 14-3-3 蛋白,实时震颤诱导转化呈阳性结果。此后,她出现肌阵挛和无动性缄默,并死亡。尸检显示右侧枕叶大脑皮质变薄和海绵状改变。免疫染色显示异常 PrP 的突触型沉积和肥大星形胶质细胞。因此,根据脑组织的 Western blot 和 PrP 基因密码子 129 多态性,她被诊断为海登海因变异型 sCJD,具有蛋氨酸/蛋氨酸 1 型和 2 型皮质形式。
当患者出现各种进行性视觉症状,即使脑电图或颅脑磁共振成像无典型发现时,必须怀疑海登海因变异型 sCJD,并进行适当的脑脊液检查。