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Paradoxical Kinesia Induced by Nightmare: Unique Case Report and Insights regarding the Neural Mechanism Based on Human and Rat Studies. 梦魇诱发的反常运动:独特的病例报告以及基于人类和大鼠研究的神经机制启示。
IF 0.6 Q4 CLINICAL NEUROLOGY Pub Date : 2024-05-30 eCollection Date: 2024-01-01 DOI: 10.1159/000539548
Jorge Gelvane Tostes, Mateus Dal Fabbro, David J Pedrosa, Norberto Cysne Coimbra, Rainer Schwarting, Liana Melo-Thomas

Introduction: Bradykinesia, characterized by slowed movement, stands out as a primary symptom observed in individuals with Parkinson's disease (PD). Nonetheless, there are instances where PD patients exhibit sudden and effective movements despite the presence of bradykinesia. This phenomenon, referred to as paradoxical kinesia, has remained a subject of interest for neuroscientists, who have struggled to unravel its underlying neural mechanisms for decades.

Case presentation: We describe a patient who is suffering from advanced PD. The patient has severe motor limitations, including difficulty rising from bed and walking, as well as cognitive decline and visual impairment. However, an interesting occurrence took place during a nightmare episode. Surprisingly, the patient was able to get out of bed and quickly run away from the perceived threat within the nightmare, without any assistance.

Conclusion: This report presents the first documented case of paradoxical kinesia induced by nightmares in a patient with PD. This phenomenon raises questions about the neurological mechanisms involved, which are still not fully understood. Based on existing research conducted on both animal and human subjects, we propose that after processing the emotion of fear, the brain aversive system activates motor outputs to generate appropriate behavior. Thus, the brain aversive system converts the emotion of fear into action through projections from the inferior colliculus to motor-related areas such as the mesencephalic locomotor region, pontine nuclei, and substantia nigra.

简介运动迟缓是帕金森病(PD)患者的主要症状。然而,在某些情况下,帕金森病患者尽管存在运动迟缓,但仍会表现出突然而有效的运动。这种现象被称为悖论性运动障碍,一直是神经科学家感兴趣的课题,几十年来他们一直在努力揭示其潜在的神经机制:我们描述了一名患有晚期帕金森病的患者。病例介绍:我们描述了一名患有晚期帕金森病的患者,患者的运动能力严重受限,包括起床和行走困难,以及认知能力下降和视力受损。然而,在一次噩梦中发生了一件有趣的事情。令人惊讶的是,患者能够下床并在没有任何帮助的情况下迅速逃离噩梦中的威胁:本报告首次记录了一名帕金森病患者因噩梦而诱发的反常运动症状。这一现象引起了人们对相关神经机制的疑问,而人们对这一机制的理解尚不充分。基于现有的动物和人类研究,我们提出,在处理恐惧情绪后,大脑厌恶系统会激活运动输出以产生适当的行为。因此,大脑厌恶系统通过下丘向间脑运动区、桥脑核和黑质等运动相关区域的投射,将恐惧情绪转化为行动。
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引用次数: 0
Case Report: Treatment of Akinetic Mutism after Unilateral Anterior Cerebral Artery Infarction with Methylphenidate and Levodopa/Benserazide. 病例报告:用哌醋甲酯和左旋多巴/苄丝肼治疗单侧大脑前动脉梗塞后的动眼神经障碍。
IF 0.6 Q4 CLINICAL NEUROLOGY Pub Date : 2024-05-28 eCollection Date: 2024-01-01 DOI: 10.1159/000539033
Julia Zibold, Moritz Luigi Schmidbauer, Johannes Wischmann, Konstantinos Dimitriadis

Introduction: Akinetic mutism is a severe state of impaired volition that can result from a stroke. Its therapeutic evidence relies on single case reports that used atomoxetine or levodopa with variable latency and efficacy.

Case presentation: We present the case of a 54-year-old woman who developed akinetic mutism after infarction in the territory of the right anterior cerebral artery, successfully treated with methylphenidate and levodopa/benserazide. Clinical examination showed a patient lacking any spontaneous speech and movement while opening her eyes and fixating. Suspecting akinetic mutism after a comprehensive diagnostic work-up, we started an individual therapy attempt with methylphenidate 10 mg and levodopa/benserazide 100/25 mg twice daily. Both drugs affect the dopaminergic and noradrenergic transmission in the frontal-subcortical circuit, compromised in akinetic mutism. We saw rapid and sustained improvement in her volitional actions, devoid of side effects. Finally, the patient was actively communicating and moving her limbs.

Conclusion: We discuss the patient's favorable clinical course in response to the synergistic combination of methylphenidate and levodopa/benserazide, emerging as a promising treatment strategy, and provide a brief literature review of treatment options in akinetic mutism following stroke.

导言激越性缄默症是一种严重的意志障碍,可由中风引起。其治疗证据依赖于使用阿托西汀或左旋多巴的单个病例报告,其潜伏期和疗效各不相同:我们介绍了一例 54 岁女性患者的病例,她在右侧大脑前动脉区域发生脑梗塞后出现运动性缄默症,使用哌醋甲酯和左旋多巴/苄丝肼治疗后获得成功。临床检查显示,患者在睁眼和凝视时没有任何自发言语和动作。经过全面诊断后,我们怀疑患者患有动眼神经缄默症,于是开始尝试使用哌醋甲酯 10 毫克和左旋多巴/苄丝肼 100/25 毫克进行个体治疗,每天两次。这两种药物都会影响多巴胺能和去甲肾上腺素能在额叶-皮层下回路的传递,而动情性缄默症则会受到这种影响。我们发现她的意志行动得到了迅速而持续的改善,而且没有副作用。最后,患者开始积极交流并活动四肢:我们讨论了该患者对哌醋甲酯和左旋多巴/苯塞拉嗪的协同组合疗法的良好临床疗效(这是一种很有前景的治疗策略),并对中风后运动性缄默症的治疗方案进行了简要的文献综述。
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引用次数: 0
Acute Leriche Syndrome Mimicking Spinal Cord Infarction: A Case Report. 模仿脊髓梗死的急性莱里切综合征:病例报告
IF 0.6 Q4 CLINICAL NEUROLOGY Pub Date : 2024-05-24 eCollection Date: 2024-01-01 DOI: 10.1159/000539456
Takamichi Kanbayashi, Sonoko Tanaka, Kiyoshi Matsukura, Masahiro Sonoo, Shunsuke Kobayashi

Introduction: Acute Leriche syndrome is a rare but potentially life-threatening condition. Pain, pallor, and coldness of the lower extremities serve as clues for suspecting Leriche syndrome. However, the absence of these findings may pose a diagnostic challenge.

Case presentation: An 83-year-old man presented at our emergency department with a complaint of sudden-onset paraparesis. Initially, spinal cord infarction was suspected due to clinical course and neurological findings, but thoracolumbar MRI showed normal findings. On admission, symptoms associated with aortoiliac occlusion were not present, except for muscle atrophy in the thigh. CT angiography revealed aortoiliac occlusion, leading to a diagnosis of Leriche syndrome.

Conclusion: Leriche syndrome should be considered as a potential differential diagnosis in patients with acute paraparesis. Muscle atrophy of the lower limbs disproportionate to the clinical course may be the clue for suspecting acute Leriche syndrome with symptoms related to atherosclerotic occlusion which are inconspicuous.

简介急性莱里什综合征是一种罕见但可能危及生命的疾病。下肢疼痛、苍白和冰冷是怀疑莱里什综合征的线索。然而,如果没有这些发现,可能会给诊断带来困难:一名 83 岁的男性因突发偏瘫到我院急诊科就诊。根据临床表现和神经系统检查结果,起初怀疑是脊髓梗死,但胸腰椎磁共振检查结果显示正常。入院时,除了大腿肌肉萎缩外,没有出现与主动脉髂骨闭塞相关的症状。CT 血管造影显示主动脉髂骨闭塞,因此诊断为勒里什综合征:结论:Leriche 综合征应被视为急性截瘫患者的潜在鉴别诊断。下肢肌肉萎缩与临床病程不相称,可能是怀疑急性莱里什综合征的线索,其症状与动脉粥样硬化闭塞有关,但并不明显。
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引用次数: 0
Unilateral Anterior Spinal Artery Syndrome following Spinal Anesthesia for Cesarian Section: A Case Report. 剖腹产脊髓麻醉后单侧脊髓前动脉综合征:病例报告。
IF 0.6 Q4 CLINICAL NEUROLOGY Pub Date : 2024-05-22 eCollection Date: 2024-01-01 DOI: 10.1159/000539405
Matthew J Kraus, Joseph Nguyen

Introduction: Spinal cord infarction is a rare but serious neurologic complication of spinal anesthesia. Direct vessel injury, intra-arterial anesthetic injection, and anesthetic-induced local hypotension are potential mechanisms of infarction during this procedure. The proximity of the artery of Adamkiewicz to the spinal levels used for spinal anesthesia may also play a role. This case of unilateral anterior spinal artery syndrome highlights the potential for an atypical pattern of injury and deficits due to the complexity of the spinal cord's anterior circulation.

Case presentation: We present a 38-year-old female patient who presented with left lower extremity weakness, loss of temperature sensation, and urinary retention following spinal anesthesia for cesarian section. Magnetic resonance imaging of the spine demonstrated T2 hyperintensities in the left central spinal cord from T8 to the conus medullaris. A diagnosis of spinal cord infarction was made after lumbar puncture testing showed no evidence of inflammatory myelitis. The patient was treated with steroids empirically until lumbar puncture testing showed no inflammation. The patient was discharged on daily aspirin with persistent left lower extremity weakness and loss of temperature sensation. A plan for outpatient physical therapy was made for rehabilitation.

Conclusion: Awareness of the potential for spinal cord infarction secondary to spinal anesthesia must increase among anesthesiologists, obstetricians, and neurologists. The risk of systemic hypotension during and after spinal anesthesia is important to recognize for both primary and secondary prevention of this complication. The hyperacute onset of myelopathic symptoms should point neurologists to investigate an ischemic etiology in the proper clinical context.

简介脊髓梗死是脊髓麻醉的一种罕见但严重的神经系统并发症。直接血管损伤、动脉内麻醉剂注射和麻醉剂引起的局部低血压是脊髓梗死的潜在机制。亚当凯维奇动脉与用于脊髓麻醉的脊柱水平很近也可能是原因之一。本例单侧脊髓前动脉综合征突出表明,由于脊髓前循环的复杂性,可能会出现非典型的损伤和缺损模式:我们为您介绍一位 38 岁的女性患者,她在接受剖腹产脊髓麻醉后出现左下肢无力、体温感觉丧失和尿潴留。脊柱磁共振成像显示,从 T8 到延髓锥体的左侧中央脊髓 T2 高密度。腰椎穿刺检查未发现炎性脊髓炎的证据,因此诊断为脊髓梗死。患者接受了经验性类固醇治疗,直到腰椎穿刺检查显示没有炎症。患者出院时每天服用阿司匹林,但左下肢持续无力,体温感觉减退。为患者制定了门诊理疗康复计划:结论:麻醉师、产科医生和神经科医生必须提高对脊髓麻醉继发脊髓梗死可能性的认识。脊髓麻醉期间和麻醉后出现全身性低血压的风险必须得到认识,以便对这种并发症进行一级和二级预防。脊髓病症状的急性发作应促使神经科医生在适当的临床背景下调查缺血性病因。
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引用次数: 0
No Efficacy with Noninvasive Brain Stimulation for Painful Legs and Moving Toes: A Case Report. 无创脑电刺激治疗疼痛腿和移动脚趾无效:病例报告。
IF 0.7 Q4 CLINICAL NEUROLOGY Pub Date : 2024-02-28 eCollection Date: 2024-01-01 DOI: 10.1159/000536467
Anna Brück, Jaakko Pullinen, Janne Nummelin, Saara Lehto, Juho Joutsa

Introduction: Painful legs and moving toes (PLMT) is a rare neurological disorder characterized by neuropathic pain and involuntary movements in the lower limbs. The pathophysiological mechanisms are unclear, but central mechanisms might be involved, suggesting that noninvasive brain stimulation might be helpful. Thus far, no reports have been published on noninvasive brain stimulation to treat PLMT.

Case presentation: A 70-year-old female had a 1-year history of PLMT. After several unsuccessful medical attempts, the patient received repetitive transcranial magnetic stimulation and transcranial direct current stimulation to alleviate the pain and involuntary movements with no benefit.

Conclusion: This is the first report on noninvasive brain stimulation in a PLMT patient. Although ineffective in our patient, noninvasive brain stimulation should be further studied in this often difficult to treat and debilitating syndrome.

简介疼痛性腿和移动脚趾(PLMT)是一种罕见的神经系统疾病,其特征是神经性疼痛和下肢不自主运动。其病理生理机制尚不清楚,但可能涉及中枢机制,这表明非侵入性脑部刺激可能会有所帮助。迄今为止,还没有关于无创脑部刺激治疗 PLMT 的报道:一名 70 岁的女性有 1 年的 PLMT 病史。在多次尝试药物治疗未果后,患者接受了重复经颅磁刺激和经颅直流电刺激来缓解疼痛和不自主运动,但没有任何效果:这是第一份关于对 PLMT 患者进行无创脑部刺激的报告。尽管对我们的患者无效,但对于这种通常难以治疗且使人衰弱的综合征,应进一步研究无创脑部刺激疗法。
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引用次数: 0
Repetitive Transcranial Magnetic Stimulation for Major Depressive Disorder in Huntington Disease Patient with Improvement in Neuropsychiatric and Movement Symptoms: A Case Report. 重复经颅磁刺激治疗亨廷顿病患者的重度抑郁症并改善其神经精神和运动症状:病例报告。
IF 0.7 Q4 CLINICAL NEUROLOGY Pub Date : 2024-02-14 eCollection Date: 2024-01-01 DOI: 10.1159/000537750
Cheyenne Rahn, Kris Peterson, Elizabeth Lamb

Introduction: Huntington disease (HD) is a progressive disorder characterized by significant neurodegeneration that results in severe neuropsychiatric symptoms and disordered movement. Repetitive transcranial magnetic stimulation (rTMS) is a noninvasive treatment that has been used in major depressive disorder (MDD) with great success.

Case presentation: We present a case of a patient with newly diagnosed HD, persistent MDD with suicidal ideation, and generalized anxiety disorder who was treated with rTMS and had sustained significant improvement of her mood disorder with additional improvement of her movement disorder.

Conclusion: This result brings into question the use of rTMS to treat MDD and chorea in patients with HD, especially early in its course.

简介亨廷顿病(Huntington disease,HD)是一种进行性疾病,其特征是显著的神经变性,导致严重的神经精神症状和运动障碍。重复经颅磁刺激(rTMS)是一种非侵入性治疗方法,已成功用于重度抑郁症(MDD)的治疗:我们介绍了一例新诊断为 HD、有自杀倾向的持续性 MDD 和广泛性焦虑症的患者,她接受了经颅磁刺激治疗,情绪障碍得到了持续显著的改善,运动障碍也得到了改善:结论:这一结果对使用经颅磁刺激治疗 HD 患者的 MDD 和舞蹈症提出了质疑,尤其是在病程早期。
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引用次数: 0
Disseminated Aspergillosis with Mediastinal Invasion Causing Fatal Stroke in an Immunocompetent Young Man. 一名免疫功能正常的年轻男子因弥散性曲霉菌病并发纵隔侵犯而导致致命中风
IF 0.7 Q4 CLINICAL NEUROLOGY Pub Date : 2024-02-05 eCollection Date: 2024-01-01 DOI: 10.1159/000536594
Robert Yarham, Rudy Goh, Eamon P Raith, Krishnaswamy Sundararajan, Timothy Kleinig

Introduction: Aspergillus flavus is a common cause of aspergillosis.

Case presentation: A previously fit and well, immunocompetent 27-year-old male living in Australia developed disseminated A. flavus complex infection with mediastinal and cardiac invasion, superior vena cava obstruction and stroke, with fatal haemorrhagic transformation.

Conclusion: Aspergillus Flavus is a rare but important cause of serious disease in the immunocompetent.

简介:黄曲霉菌是曲霉菌病的常见病因:黄曲霉菌是曲霉菌病的常见病因:病例介绍:一名居住在澳大利亚、身体健康、免疫功能正常的27岁男性发生了黄曲霉菌复合体播散性感染,并伴有纵隔和心脏受侵、上腔静脉阻塞和中风,并出现致命的出血性转变:结论:黄曲霉菌是导致免疫力低下者严重疾病的罕见但重要的病因。
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引用次数: 0
Migraine Symptoms Induced by an Auricular Piercing in a 27-Year-Old Female: A Case Report. 一名 27 岁女性因耳廓穿刺引发偏头痛症状:病例报告。
IF 0.7 Q4 CLINICAL NEUROLOGY Pub Date : 2024-01-31 eCollection Date: 2024-01-01 DOI: 10.1159/000536132
Sophia Uddin, Jordan Terry

Introduction: Migraines are common and debilitating, and have high direct and indirect costs. They can be difficult to treat, and many patients make use of alternative medicine techniques. One of these is acupuncture applied to locations on the auricle thought to modulate migraine symptoms. Some patients obtain piercings in these locations in hopes of relieving their symptoms; however, the literature does not address the possibility of migraine symptoms being worsened or even induced by such piercings.

Case presentation: We present a case of a 27-year-old female with a history of transient hemiplegia without headaches who developed headaches, visual disturbances, and nausea after a piercing of the inferior crus of her left antihelix (known as a rook piercing). No abnormalities were found on workup, and symptoms were treated with supportive care. After removing the piercing 9 months later, the patient's symptoms resolved.

Conclusion: The mechanism linking the piercing with the migraine symptoms is unclear, but may involve modulation of trigeminal or vagal pain pathways, as both of these cranial nerves innervate this area of the auricle. Regardless, in patients presenting with migraine symptoms, history and physical exam should not overlook piercings as potential contributory factors.

导言:偏头痛是一种常见的使人衰弱的疾病,其直接和间接成本都很高。偏头痛很难治疗,许多患者会使用替代医学技术。其中之一就是在耳廓上被认为能调节偏头痛症状的位置进行针灸。有些患者在这些位置穿孔,希望能缓解症状;然而,文献并未提及穿孔会加重甚至诱发偏头痛症状的可能性:我们为您介绍一例病例:一名 27 岁女性,曾有一过性偏瘫病史,无头痛,在左侧反螺旋下皱襞穿刺(称为 "大鹏穿刺")后出现头痛、视觉障碍和恶心。检查未发现异常,患者接受了支持性治疗。9 个月后取出穿孔,患者症状缓解:穿孔与偏头痛症状的关联机制尚不清楚,但可能涉及三叉神经或迷走神经疼痛通路的调节,因为这两种颅神经都支配耳廓的这一区域。无论如何,对于出现偏头痛症状的患者,病史和体格检查都不应忽视穿孔这一潜在诱因。
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引用次数: 0
A First Case of Acute Flaccid Myelitis Related to Enterovirus D68 in Belgium: Case Report. 比利时首例与肠道病毒 D68 相关的急性弛缓性脊髓炎病例:病例报告。
IF 0.7 Q4 CLINICAL NEUROLOGY Pub Date : 2024-01-31 eCollection Date: 2024-01-01 DOI: 10.1159/000535316
Marine Rodesch, Claudine Sculier, Valentina Lolli, Gauthier Remiche, Iris Delpire, Christophe Fricx, Françoise Vermeulen, Florence Christiaens

Introduction: We describe the first case of acute flaccid myelitis (AFM) related to enterovirus D68 (EV-D68) infection in Belgium. The clinical and radiological presentation of AFM associated with EV-D68 although well described currently remains a challenging diagnosis. Through this interesting clinical case, we aimed to review the differential diagnosis of acute flaccid palsy in a child and discuss the specific point of interest related to AFM.

Case presentation: We present the case of a 4-year-old girl with a torticollis associated with an acute palsy of the right upper limb. The magnetic resonance imaging revealed an increased T2 signal intensity of the entire central gray matter of the cervical cord with involvement of the posterior brainstem. A polymerase chain reaction (PCR) conducted on a nasopharyngeal swab was found positive for EV-D68. The definition of AFM proposed by the Center for Disease Control and Prevention (CDC) is an acute-onset flaccid weakness of one or more limbs in the absence of a clear alternative diagnosis and the radiological evidence of gray matter involvement on an MRI picture, and our case fits these two criteria. A prompt and detailed workup is required to distinguish this emergent disease from other forms of acute flaccid palsy. The functional prognosis of AFM is poor, and there are no evidence-based treatment guidelines so far.

Conclusion: AFM is an emerging pathology that requires the attention of pediatricians to quickly rule out differential diagnoses and adequately manage the patient. Further research is needed to optimize treatments, improve outcomes, and provide scientifically based guidelines.

导言:我们描述了比利时首例与肠道病毒 D68(EV-D68)感染相关的急性弛缓性脊髓炎(AFM)病例。与 EV-D68 相关的急性弛缓性脊髓炎(AFM)的临床和放射学表现虽然已被详细描述,但目前仍是一个具有挑战性的诊断。通过这个有趣的临床病例,我们旨在回顾儿童急性弛缓性麻痹的鉴别诊断,并讨论与 AFM 相关的特殊关注点:本病例是一名 4 岁女孩的病例,她患有右上肢急性麻痹伴肢体扭转。磁共振成像显示,整个颈髓中央灰质的 T2 信号强度增高,脑干后部受累。对鼻咽拭子进行的聚合酶链反应(PCR)发现 EV-D68 呈阳性。美国疾病控制和预防中心(CDC)提出的 AFM 定义是:在没有明确替代诊断的情况下,急性发作的一侧或多侧肢体弛缓性无力,以及核磁共振成像图片上灰质受累的放射学证据,而我们的病例符合这两个标准。我们的病例符合这两个标准。要将这种突发疾病与其他形式的急性弛缓性麻痹区分开来,需要进行及时而详细的检查。急性弛缓性麻痹的功能性预后较差,目前尚无循证治疗指南:结论:急性弛缓性麻痹是一种新出现的病理现象,需要儿科医生的关注,以快速排除鉴别诊断并对患者进行适当的管理。需要进一步开展研究,以优化治疗、改善疗效并提供科学的指导原则。
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引用次数: 0
Highly Active Relapsing-Remitting Multiple Sclerosis with Neurofibromatosis Type 1: Radiological Aspects and Therapeutic Challenges - Case Report. 神经纤维瘤病 1 型高活性复发性多发性硬化症:放射学方面和治疗挑战--病例报告。
IF 0.7 Q4 CLINICAL NEUROLOGY Pub Date : 2024-01-24 eCollection Date: 2024-01-01 DOI: 10.1159/000536463
Marios Lemonaris, Kleopas A Kleopa

Introduction: Multiple sclerosis (MS) is an autoimmune neurodegenerative disease which can rarely co-exist with neurofibromatosis 1 (NF1), a neurocutaneous inherited disorder that predisposes to oncogenesis. Patients who suffer from both conditions can be challenging cases for clinicians, as clinical symptoms and radiological findings may overlap, while MS immune-modifying treatments could further increase the risk of oncogenesis.

Case presentation: In this study, we describe the case of a 27-year-old woman who presented with signs and symptoms of optic neuritis and was then diagnosed with both MS and NF1. As the patient continued to experience MS relapses despite initial interferon-beta treatment, she was subsequently switched to natalizumab and responded well.

Conclusion: This case illustrates how MRI lesion differentiation with the co-existence of MS and NF1 can be difficult due to overlaps in lesion characteristics, while treatment decisions can be challenging mainly due to scarce data on the oncogenic risk of MS immunomodulary therapies. Therefore, clinicians need to balance out the risk of malignancy development with the risk of progressive neurological disability when treating such patients.

简介:多发性硬化症(MS)是一种自身免疫性神经退行性疾病:多发性硬化症(MS)是一种自身免疫性神经退行性疾病,很少与神经纤维瘤病 1(NF1)同时存在,NF1 是一种易导致肿瘤发生的神经皮肤遗传性疾病。同时患有这两种疾病的患者对临床医生来说可能是具有挑战性的病例,因为临床症状和放射学检查结果可能会重叠,而 MS 免疫调节治疗可能会进一步增加肿瘤发生的风险:在本研究中,我们描述了一名 27 岁女性的病例,她出现了视神经炎的体征和症状,随后被诊断同时患有多发性硬化症和 NF1。由于患者在最初接受干扰素-β治疗后仍有多发性硬化症复发,因此她后来转用了纳他珠单抗,并取得了良好的疗效:本病例说明,由于病变特征的重叠,MS 和 NF1 并存时 MRI 病变的鉴别可能会很困难,而治疗决策可能会具有挑战性,这主要是由于有关 MS 免疫调节疗法致癌风险的数据很少。因此,临床医生在治疗这类患者时,需要权衡恶性肿瘤发展的风险和进行性神经残疾的风险。
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引用次数: 0
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Case Reports in Neurology
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