Recurrent epileptic seizures with multifocal brain MRI lesions after paternal lymphocyte immunization: A causal relationship to multiple sclerosis?
Recurrent epileptic seizures with multifocal brain MRI lesions after paternal lymphocyte immunization: A causal relationship to multiple sclerosis?
复制标题
父亲淋巴细胞免疫后反复癫痫发作伴多灶性脑部 MRI 病变：与多发性硬化症的因果关系？
DOI：
10.1111/j.1440-1789.2010.01162.x
复制
发表时间：
2011-02
期刊：
影响因子：
2.3
通讯作者：
Zhou Dong
中科院分区：
文献类型：
--
作者：
Chen Lei;Zhou Qiao;Kinoshita Masako;Liu Ling;Zhou Dong
父亲淋巴细胞免疫(PLI)是一种广泛应用于同种异体免疫介导的复发性自然流产(RSA)的治疗方法，但报道的不良反应很少。我们报告一例癫痫患者在PLI后不久出现多灶性复发性脑部病变，并经抗癫痫药物和类固醇治疗后得到良好控制。我们诊断此病例为多发性硬化症(MS)，这可能是一种与PLI相关的罕见副作用。一名患有RSA的35岁女性接受了标准疗程(四次)的PLI治疗。治疗结束时，她出现部分性癫痫和继发性全身性癫痫发作，EEG显示额颞区两侧有尖锐波。发作间期一般体格检查、神经功能检查、心电图、脑脊液检查正常。患者既往无感染、发热、头痛或脑膜炎征象。MRI显示多灶性T2高信号和T1低信号结节状病变，皮质和脑室周围区域强化，主要位于顶叶左侧。在推测为脑部病变和症状性癫痫的情况下，她接受了拉莫三嗪75 mg/天的治疗。半年后，她反复发作(持续1天)。一般的身体和神经检查仍然正常。脑电波与前一次相似。脑脊液呈阳性寡克隆条带(OB)，其他均正常。头颅MRI发现额颞叶白质和皮质的多发性结节状病变增多，而顶叶皮质的病变减少。胸部-腹部CT和脑磁共振血管成像均正常。免疫标志物(IgA/M/G/E、C3/4、类风湿因子、抗核抗体、抗中性粒细胞胞浆抗体、抗史密斯抗体(SM)、抗干燥综合征A抗体(SSA)、抗磷脂抗体等)、肿瘤和梅毒标志物、HIV和肝炎病毒抗体、寄生虫和TORCH(弓形虫病、其他、风疹、巨细胞病毒和单纯疱疹)均为阴性。在她知情同意西中国医院伦理委员会批准的方案后，对左侧额叶病变进行脑活组织检查(组织学测试如图1所示)。根据临床表现和MRI、脑脊液OB检查和组织学检查证实的脑部病变在时间和空间上的多发性，当考虑持续1天以上的群发性癫痫发作时，根据McDonald诊断标准诊断为MS。她接受负荷量的甲基强的松龙静脉注射(1.0g/d)，连续5天，然后口服泼尼松(开始30 mg/d，4个月后逐渐减少)和拉莫三嗪(100 mg/d)。之后，她只出现了几次癫痫发作。随访MRI显示脑室周围残留典型的MS斑块，未见新的病变。患者在PLI后发展为多发性硬化症，首发症状为癫痫。尽管King等人。报道1例PLI诱发癫痫，该患者在PLI前已诊断为多发性硬化症。此外，本病例中仅表现为癫痫发作的患者在MS中非常罕见。该患者可能患有多相播散性脑脊髓炎(MDEM)，其特征是急性播散性脑脊髓炎(ADEM)复发，而这些复发被认为是同一急性单相过程的一部分。最近提出的MS和ADEM的鉴别诊断标准表明，ADEM对应于至少以下两种情况的患者：(1)包括癫痫在内的MS的临床不典型症状；(2)脑脊液中没有OB；(3)灰质受累。本患者首次出现上述(1)和(3)项症状，并复发，症状与最初的症状相似。此病例的活检材料显示血管周围脱髓鞘，也是ADEM的特征。然而，根据2010年11月9日在线发表的机制，我们认为MS更有可能是因为缺乏急性颅内炎症、OB阳性和存在脑室周围病变的临床表现，这些都是MS的典型特征。神经病理学2011年；31，98-99 DOI：10.1111/j.1440-1789.2010.01162.x
Paternal lymphocyte immunization (PLI) is a widely used treatment for alloimmune-mediated recurrent spontaneous abortion (RSA) with rare adverse effects reported. We report a case in which the patient suffered from epilepsy with multifocal recurrent brain lesions shortly after PLI, and was controlled well by antiepileptic drugs and steroids. We diagnosed this case as multiple sclerosis (MS) which may be a rare side effect associated with PLI. A 35-year-old woman with RSA received a standard course (four times) of PLI treatment. At the end of treatment, she had a cluster of partial seizures and secondarily generalized seizures with sharp waves on both sides of the fronto-temporal areas on EEG. General physical and neurological examinations in interictal periods, ECG and CSF were normal. The patient showed no sign of preceding infection,fever,headache or meningism.Brain MRI showed multifocal T2-hyperintensity and T1-hypointensity lesions in nodular shapes with enhancement in cortical and periventricular areas,mainly on the left side of the parietal lobe.She was treated with Lamotrigine 75 mg/day under presumptive diagnosis of a brain lesion and symptomatic epilepsy. A half year later, she had a cluster of recurrent seizures (duration > 1 day). General physical and neurological examinations were still normal. EEG was similar to the previous one. CSF showed affirmative oligoclonal bands (OB) but other aspects were all normal. Brain MRI discovered increase of multiple nodular lesions in the white matter and cortex of the fronto-temporal lobe; however, lesions in the parietal lobe cortex decreased. Breast– abdomen CT and brain magnetic resonance angiography were normal. Immunology markers (IgA/M/G/E, C3/4, rheumatoid factor, antinuclear antibodies, antineutrophil cytoplasmic antibodies, antiSmith antibody (SM), antiSjogren syndrome A antibody (SSA), antiphospholipid antibody, etc.), tumor and syphilis markers, HIV and hepatitis virus antibody, parasite and TORCH (toxoplasmosis, others, , rubella, cytomegalovirus and herpes simplex) of blood were all negative. A brain biopsy was performed on the left frontal lobe lesions after she gave an informed consent to the protocol approved by the Ethics Committee of West China Hospital (histology test shown in Fig. 1). Based on the brain lesions multiple in time and space by clinical manifestations and MRI, affirmative OB in CSF and histology findings, the patient was diagnosed as MS according to McDonald diagnosis criteria, when we consider a cluster of seizures lasting for more than 1 day in duration. She received a loading dose of intravenous methylprednisolone (1.0 g/day) for 5 days, followed by oral prednisone (beginning with 30 mg/day, tapered off over 4 months) and Lamotrigine (100 mg/day). Afterwards, she developed only a few seizures. The follow-up MRI showed residual typical MS plaques in the periventricular region without new lesions. The patient developed MS after PLI with the first manifestation of seizures. Although King et al. reported a case of epilepsy induced by PLI, that patient was already diagnosed with MS before PLI. Furthermore, patients manifesting with only epileptic seizures as in this case, is quite rare in MS. There is a possibility that this patient may have multiphasic disseminated encephalomyelitis (MDEM), which is characterized by relapses of acute disseminated encephalomyelitis (ADEM) when these relapses are thought to represent part of the same acute monophasic process. Recently proposed diagnostic criteria for differentiation between MS and ADEM indicate that ADEM corresponds to patients with at least two of the following: (1) clinical atypical symptoms of MS including seizures; (2) absence of OB in the cerebrospinal fluid; (3) gray matter involvement. The present patient fulfilled (1) and (3) of the above at the first presentation, and relapsed with similar symptoms to the initial incident. Biopsied materials of this case showed perivascular demyelinations that were also characteristic of ADEM. However, we consider MS is more likely because of lack of clinical manifestations of acute intracranial inflammation, positive OB, and presence of periventricular lesions which are typical of MS. According to the mechanism of Published online 9 November 2010. Neuropathology 2011; 31, 98–99 doi:10.1111/j.1440-1789.2010.01162.x
登录
查看更多内容
DOI：
10.1016/b978-0-12-813014-8.00016-0
发表时间：
2019
期刊：
Handbook of Sleep Disorders in Medical Conditions
影响因子：
--
作者：
C. Veauthier;F. Paul
通讯作者：
C. Veauthier;F. Paul
影响因子：
3.3
作者：
Gout, O
通讯作者：
Gout, O
DOI：
10.1007/978-981-32-9636-7_14
发表时间：
2019-01-01
期刊：
MYELIN: BASIC AND CLINICAL ADVANCES
影响因子：
--
作者：
Yamasaki, Ryo;Kira, Jun-ichi
通讯作者：
Kira, Jun-ichi
影响因子：
9.9
作者：
W. Tourtellotte;A. Potvin;J. Fleming;Kolar Murthy;J. Levy;K. Syndulko;J. Potvin
通讯作者：
W. Tourtellotte;A. Potvin;J. Fleming;Kolar Murthy;J. Levy;K. Syndulko;J. Potvin
DOI：
10.1016/b978-0-444-53485-9.00020-9
发表时间：
2016-01-01
期刊：
NEUROIMAGING, PT I
影响因子：
--
作者：
Filippi, Massimo;Preziosa, Paolo;Rocca, Maria A.
通讯作者：
Rocca, Maria A.