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Clinical Experience with Dimethyl Fumarate and Natalizumab in Pregnant Women with Multiple Sclerosis: A Four-Patient Case Series. 多发性硬化症孕妇使用富马酸二甲酯和纳他珠单抗的临床经验:四例患者病例系列。
IF 0.9 Q4 CLINICAL NEUROLOGY Pub Date : 2024-07-16 eCollection Date: 2024-01-01 DOI: 10.1155/2024/7808140
Satoshi Saito, Ryotaro Ikeguchi, Kazuo Kitagawa, Yuko Shimizu

Interferon β and glatiramer acetate are the disease-modifying drugs (DMDs) considered relatively safe for use in pregnant women with multiple sclerosis (MS); however, the safety profile of dimethyl fumarate (DMF) and natalizumab (NTZ) in this population remains inconclusive. Here, we present four cases of pregnant women with MS who were treated with DMF and NTZ (n = 2 patients, each) during their pregnancy and discuss our observations with the use of these drugs and the clinical courses of the patients. We retrospectively examined relapse of MS during pregnancy and after delivery; duration of exposure to DMDs; maternal, fetal, and neonatal adverse events; breastfeeding; and timing of resumption of DMDs. The two patients treated with DMF discontinued treatment 5 or 6 weeks after the discovery of pregnancy. DMF was resumed 1 week postpartum, and mixed breastfeeding was initiated. Brain magnetic resonance imaging in one patient 9 months after delivery revealed a new lesion; however, it was not classified as a clinical relapse. In two patients treated with NTZ, the dosing interval was extended to 6 weeks after the discovery of pregnancy. One patient discontinued NTZ at 30 weeks and the other at 25 weeks of gestation, as a slight restriction in fetal growth was observed owing to hyperemesis gravidarum. Both patients opted for formula feeding, and no relapse was observed within 1 year postpartum. Additionally, no abnormalities were observed in any of the patients during the perinatal period, and their development was normal. Investigation of drug safety in pregnant and parturient women primarily relies on registries, postmarketing surveillance, and case reports due to ethical limitations on conducting randomized controlled trials. Our findings demonstrated that DMF and NTZ were not contraindicated during pregnancy or the perinatal period in women with MS; nevertheless, vigilant monitoring is essential to ensure the safety of these drugs.

多发性硬化症(MS)孕妇使用β干扰素和醋酸格拉替雷被认为是相对安全的疾病修饰药物(DMD);然而,富马酸二甲酯(DMF)和纳他珠单抗(NTZ)在这一人群中的安全性仍无定论。在此,我们介绍了四例在怀孕期间接受 DMF 和 NTZ 治疗的多发性硬化症孕妇(每例 2 人),并讨论了我们对这些药物的使用和患者临床病程的观察结果。我们回顾性地检查了多发性硬化症在妊娠期间和分娩后的复发情况;接触 DMDs 的持续时间;母体、胎儿和新生儿不良事件;母乳喂养;以及恢复使用 DMDs 的时间。两名接受DMF治疗的患者在发现怀孕5周或6周后停止了治疗。产后 1 周恢复使用 DMF,并开始混合母乳喂养。一名患者在产后 9 个月进行脑磁共振成像检查时发现了新的病灶,但未被归类为临床复发。在两名接受 NTZ 治疗的患者中,用药间隔延长至发现怀孕后 6 周。一名患者在妊娠 30 周时停用了 NTZ,另一名患者在妊娠 25 周时停用了 NTZ,原因是妊娠剧吐导致胎儿生长受到轻微限制。这两名患者都选择了配方奶喂养,产后一年内均未发现复发。此外,围产期也未发现异常,发育正常。由于开展随机对照试验存在伦理限制,对孕妇和产妇用药安全性的调查主要依赖于登记、上市后监测和病例报告。我们的研究结果表明,多发性硬化症妇女在妊娠期或围产期并不禁用DMF和NTZ;然而,警惕性监测对于确保这些药物的安全性至关重要。
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引用次数: 0
A Severe Alzheimer's Disease Patient Improved by Intravenous Mesenchymal Stem Cell Transplant. 静脉间充质干细胞移植改善了一名重度阿尔茨海默病患者的病情。
IF 0.9 Q4 CLINICAL NEUROLOGY Pub Date : 2024-07-15 eCollection Date: 2024-01-01 DOI: 10.1155/2024/8353492
Takahiro Honda Pazili

Alzheimer's disease (AD) is a progressive neurological disorder and is the most common form of dementia. The terminal stage of AD is characterized by severe cognitive and substantial functional decline, requiring extensive assistance with daily activities. As effective therapies at this stage are not fully available, development of therapeutics that can recover any symptoms would be important to improve the quality of life. Recently, stem cell therapy has gathered a lot of attention in several neurological diseases, including AD. Here, we report an AD patient at the terminal stage whose symptoms were improved by the intravenous administration of ex vivo-expanded bone marrow-derived mesenchymal stem cells (MSC). The case is a 61-year-old woman with severe Alzheimer's disease who had been admitted to the special nursing home. She could neither walk nor sit up independently. She also did neither smile nor gaze properly when talked to. Rigidity including neck motion was observed. She was on dysphagia diets. We cultured her bone-marrow-derived MSCs and intravenously administered 1,5 × 108 cells. After the treatment, smile loss, eye movement dysfunction, and neck immobility were improved. This is the first case report that showed the therapeutic effects of MSCs on terminal symptoms of AD.

阿尔茨海默病(AD)是一种进行性神经系统疾病,也是最常见的痴呆症。阿尔茨海默氏症晚期的特征是认知能力和功能严重衰退,日常活动需要大量协助。由于在这一阶段还没有完全有效的疗法,因此开发能够恢复任何症状的疗法对于改善生活质量非常重要。最近,干细胞疗法在包括AD在内的多种神经系统疾病中备受关注。在此,我们报告了一名处于晚期的AD患者,通过静脉注射活体扩增的骨髓间充质干细胞(MSC),其症状得到了改善。该病例是一名患有严重阿尔茨海默氏症的61岁女性,曾入住特殊疗养院。她既不能独立行走,也不能独立坐起。与她交谈时,她既不会微笑,也不会正常注视。她的颈部活动僵硬。她正在接受吞咽困难饮食治疗。我们培养了她的骨髓间充质干细胞,并静脉注射了 1,5 × 108 个细胞。治疗后,她的微笑消失、眼球运动障碍和颈部活动障碍均得到了改善。这是首例显示间充质干细胞对 AD 终末症状有治疗作用的病例报告。
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引用次数: 0
Impact of Focal Muscle Vibration on Flaccid Upper Limb Motor Paralysis following Acute Brain Disease: A Case Study. 病灶肌肉振动对急性脑病后弛缓性上肢运动麻痹的影响:病例研究
IF 0.9 Q4 CLINICAL NEUROLOGY Pub Date : 2024-06-25 eCollection Date: 2024-01-01 DOI: 10.1155/2024/2469074
Hirotaka Saito, Haruka Kobayashi, Kodai Oba, Yosuke Hamaya

Focal muscle vibration (FMV) is increasingly being recognized as a rehabilitative therapy for enhancing motor function in central nervous system (CNS) diseases, particularly in patients with fine motor control deficits stemming from CNS damage. Brain lesions from these diseases disrupt the motor networks, necessitating novel rehabilitation strategies. By applying vibrations to muscles, FMV stimulates sensory fibers to induce cortical activity and kinesthetic illusions. While initial studies have highlighted FMV's role in reducing spasticity, recent evidence points to its potential in treating motor paralysis. However, prior research has been limited by the lack of acute-phase studies and a focus on patients with minimal muscle contraction capability. This report aimed to explore FMV's efficacy on upper limb motor function in patients with flaccid motor paralysis immediately after acute CNS diseases. We report the case of a septuagenarian male with a brain abscess in the right parietal lobe, leading to flaccid motor paralysis. Rehabilitation included 28 sessions of occupational and physical therapy that incorporated FMV. Significant improvements were observed in upper extremity function, with moderate to very large effect sizes, while lower limb function showed lesser improvement without adverse effects. This case suggests the utility of FMV in enhancing upper-limb motor function after acute CNS injuries, potentially serving as a supplementary therapy for spontaneous recovery. This report contributes to emerging evidence on FMV's benefits in acute flaccid motor paralysis, expanding the documented therapeutic scope.

病灶肌肉振动(FMV)越来越被视为一种康复疗法,可增强中枢神经系统(CNS)疾病患者的运动功能,尤其是中枢神经系统受损导致精细运动控制功能障碍的患者。这些疾病引起的脑损伤会破坏运动网络,因此需要新的康复策略。通过对肌肉施加振动,调频伏特可刺激感觉纤维,诱发大脑皮层活动和运动错觉。虽然最初的研究强调了调频伏特在减轻痉挛方面的作用,但最近的证据表明它在治疗运动性瘫痪方面具有潜力。然而,之前的研究由于缺乏急性期研究和只关注肌肉收缩能力极弱的患者而受到限制。本报告旨在探讨 FMV 对急性中枢神经系统疾病后即刻出现弛缓性运动麻痹的患者上肢运动功能的疗效。我们报告了一例七旬男性患者的病例,该患者因右侧顶叶脑脓肿而导致弛缓性运动麻痹。康复治疗包括 28 个疗程的职业疗法和物理疗法,其中纳入了调频变压器。患者的上肢功能得到了明显改善,疗效达到中等至非常大,而下肢功能改善较小,但无不良反应。该病例表明,调强核磁共振可用于增强急性中枢神经系统损伤后的上肢运动功能,有可能成为自发恢复的辅助疗法。本报告为 FMV 在急性弛缓性运动麻痹中的益处提供了新的证据,扩大了有据可查的治疗范围。
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引用次数: 0
Efgartigimod as Rescue Medication in a Patient with Therapy-Refractory Myasthenic Crisis. 将依夫加替莫德作为治疗难治性肌无力危象患者的救治药物
IF 0.9 Q4 CLINICAL NEUROLOGY Pub Date : 2024-06-14 eCollection Date: 2024-01-01 DOI: 10.1155/2024/9455237
Omar Alhaj Omar, Norma J Diel, Stefan T Gerner, Anna Mück, Hagen B Huttner, Heidrun H Krämer-Best

Myasthenic crises (MC) are potentially life-threatening acute exacerbations of myasthenia gravis (MG) characterized by profound muscle weakness, bulbar symptoms, and potential for respiratory failure. Intravenous immunoglobulins (IVIG) and plasma exchange (PLEX) are conventional treatments for myasthenic exacerbations. Recently, new therapeutic options for generalized acetylcholine-receptor antibody positive (AchR+) MG were approved as an add-on therapy. They mainly consist of complement C5 inhibitors such as eculizumab and ravulizumab and neonatal Fc receptor antagonists such as efgartigimod with the approval of more options pending, e.g., zilucoplan and rozanolixizumab. More therapeutic options are in the pipeline. Although the data show a quick and reliable treatment response, these medications have not been studied for the therapy of myasthenic crisis. We present the case of a 57-year-old male with his first episode of generalized myasthenia gravis (MG) and positive acetylcholine-receptor antibodies (AchR+) who was transferred to our neurological intensive care unit with worsening generalized weakness, dysphagia, and respiratory distress. The crisis was triggered by pneumonia due to dysphagia. He was diagnosed with myasthenic crisis and treated with intravenous pyridostigmine, plasmapheresis (PLEX), and continued prednisone. Initial improvement was followed by deterioration, requiring readmission and additional PLEX. After a further decline, efgartigimod was administered, leading to significant improvement within 48 hours, as evidenced by reduced MG-ADL and QMG scores. The patient continued to improve and was stable enough for transfer to a rehabilitation facility. This case illustrates the potential of efgartigimod as a novel treatment for refractory myasthenic crises.

肌无力危象(MC)是一种可能危及生命的重症肌无力症(MG)急性加重期,其特点是肌肉极度无力、出现球部症状并可能导致呼吸衰竭。静脉注射免疫球蛋白(IVIG)和血浆置换(PLEX)是治疗肌无力加重的传统方法。最近,针对全身乙酰胆碱受体抗体阳性(AchR+)的 MG 的新疗法被批准作为附加疗法。它们主要包括补体 C5 抑制剂(如 eculizumab 和 ravulizumab)和新生儿 Fc 受体拮抗剂(如 efgartigimod),还有更多治疗方案(如 zilucoplan 和 rozanolixizumab)有待批准。更多的治疗方案正在审批中。尽管数据显示治疗反应迅速可靠,但这些药物尚未用于肌无力危象的治疗研究。我们报告了一例 57 岁男性患者的病例,他是第一次全身性肌无力(MG)发作,乙酰胆碱受体抗体(AchR+)阳性,因全身乏力、吞咽困难和呼吸窘迫恶化而转入我们的神经重症监护病房。由于吞咽困难,肺炎引发了危机。他被诊断为肌无力危象,接受了静脉注射吡啶斯的明、血浆置换术(PLEX)和持续泼尼松治疗。最初病情有所好转,但随后又出现恶化,需要再次入院并追加 PLEX。病情进一步恶化后,患者接受了依加替莫德治疗,48 小时内病情明显好转,MG-ADL 和 QMG 评分均有所下降。患者病情持续好转,稳定到足以转入康复机构。本病例说明了依加替莫德作为一种新型疗法治疗难治性肌无力危象的潜力。
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引用次数: 0
Chronic Inflammatory Demyelinating Polyradiculoneuropathy with Diplopia Caused by an Alternative Coronavirus Disease 2019 Vaccine. 一种替代性冠状病毒病2019疫苗引发的伴有复视的慢性炎症性脱髓鞘多发性神经病
IF 0.9 Q4 CLINICAL NEUROLOGY Pub Date : 2024-06-11 eCollection Date: 2024-01-01 DOI: 10.1155/2024/8584482
Satoshi Saito, Mutsumi Iijima, Misa Seki, Ayato Shimomura, Kazuo Kitagawa

The etiology of chronic inflammatory demyelinating polyradiculoneuropathy (CIDP) remains elusive and is believed to involve multiple contributing factors. There have been cases linking CIDP to the coronavirus disease 2019 (COVID-19) mRNA vaccine. However, there are no documented instances following alternative vaccines. We report a case of a 48-year-old woman, previously vaccinated with Pfizer-BioNTech's COVID-19 vaccine (BNT162b2), who subsequently received the Moderna mRNA-1273 vaccine. Within 2 days postvaccination, she developed diplopia and numbness in the lower limbs' distal extremities. Cerebrospinal fluid analysis exhibited protein-cell dissociation, while F-wave studies showed demyelinating activity in the bilateral tibial nerves. Given the disease's progressive nature, the patient was presumed to have CIDP and commenced steroid pulse therapy and intravenous immunoglobulin therapy. The onset of CIDP may be associated with variations in mRNA sequences and vaccine constituents.

慢性炎症性脱髓鞘多发性神经病(CIDP)的病因仍然难以捉摸,据信涉及多种诱因。有病例表明,CIDP 与冠状病毒病 2019(COVID-19)mRNA 疫苗有关。然而,目前还没有接种其他疫苗的病例记录。我们报告了一例 48 岁女性的病例,她之前接种了辉瑞生物技术公司的 COVID-19 疫苗 (BNT162b2),之后又接种了 Moderna mRNA-1273 疫苗。接种后 2 天内,她出现了复视和下肢远端麻木。脑脊液分析显示蛋白细胞离解,F 波研究显示双侧胫神经有脱髓鞘活动。考虑到疾病的进行性,患者被推测为患有脊髓灰质炎,并开始接受类固醇脉冲治疗和静脉注射免疫球蛋白治疗。CIDP 的发病可能与 mRNA 序列和疫苗成分的变化有关。
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引用次数: 0
Syncope vs. Seizure: Ictal Bradycardia and Ictal Asystole. 晕厥与癫痫发作:心动过缓和心动过速。
IF 0.9 Q4 CLINICAL NEUROLOGY Pub Date : 2024-05-06 eCollection Date: 2024-01-01 DOI: 10.1155/2024/1299282
Sumika Ouchida, Kaitlyn Parratt, Armin Nikpour, Greg Fairbrother

Background: Ictal arrhythmia is a rare condition that causes arrhythmic manifestations induced by epileptic seizures, including asystole or bradycardia. Ictal asystole (IA) is a very rare condition found in patients undergoing video-encephalography (EEG) monitoring. It is often related to temporal lobe epilepsy and can cause syncope, which can lead to injury or even death. Case Presentation. Two patients with epilepsy showed symptoms of syncope. Both patients underwent 4-day ambulatory EEG tests and were diagnosed with IA. Following the tests, the patients were implanted with a permanent pacemaker, and one of them underwent a temporal lobectomy. As a result of these procedures, the patients experienced a reduction in episodes of symptomatic syncope.

Conclusion: Patients with ictal asystole and symptomatic ictal bradycardia are at increased risk of falls due to seizures. Although there are no specific guidelines for managing this condition, antiseizure medications, epilepsy surgery, and cardiac pacemaker implantation have been effective treatments.

背景:异位性心律失常是一种罕见的疾病,由癫痫发作诱发心律失常表现,包括间歇或心动过缓。在接受视频脑电图(EEG)监测的患者中,极位性心律失常(IA)是一种非常罕见的病症。它通常与颞叶癫痫有关,可引起晕厥,导致受伤甚至死亡。病例介绍。两名癫痫患者出现晕厥症状。两名患者均接受了为期 4 天的动态脑电图检测,并被诊断为癫痫综合征。检查后,患者被植入了永久起搏器,其中一人还接受了颞叶切除术。通过这些治疗,患者症状性晕厥的发作次数有所减少:结论:发作性心搏骤停和症状性发作性心动过缓患者因癫痫发作而跌倒的风险增加。虽然目前还没有管理这种情况的具体指南,但抗癫痫药物、癫痫手术和心脏起搏器植入术都是有效的治疗方法。
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引用次数: 0
Novel SLC18A2 Variant in Infantile Dystonia-Parkinsonism Type 2. 婴儿肌张力障碍-帕金森氏症 2 型中的新型 SLC18A2 变体
IF 0.9 Q4 CLINICAL NEUROLOGY Pub Date : 2024-04-30 eCollection Date: 2024-01-01 DOI: 10.1155/2024/4767647
Sakari Kaasalainen, Harri Arikka, Mika H Martikainen, Valtteri Kaasinen

Infantile dystonia-parkinsonism type 2 (PKDYS2) is a rare inherited autosomal recessive movement disorder with onset in infancy. The disease is associated with a mutation in the solute carrier family 18 member A2 gene (SLC18A2). There are reports of trials with dopaminergic drugs and the condition of patients given levodopa almost always worsens and dopamine agonists give varying degrees of benefit to some. Here, we report a PKDYS2 patient with a new variant in the SLC18A2 gene who underwent multiple trials of pharmacotherapy. The abnormalities in development and neurological examination of the case were first noted at the age of 2 months, and after a series of treatment attempts (e.g., with antiepileptics) and diagnostic procedures, the diagnosis of PKDYS2 was determined when whole exome sequencing (WES) at age 6, revealed a homozygous pathologic variant NM_003054.4:c.1107dup, p.(Val370Serfs91) in the SLC18A2 gene. The patient then received treatment with multiple dopaminergic drugs (e.g., levodopa, pramipexole, and methylphenidate). The patient with PKDYS2 harbored a new variant in SLC18A2. The phenotype of the patient resembles that of some previously reported patients with PKDYS2. The patient received minor benefits from certain dopaminergic drugs, such as pramipexole, but side effects led to the discontinuation of tested medications.

婴儿肌张力障碍-帕金森氏症 2 型(PKDYS2)是一种罕见的常染色体隐性遗传运动障碍疾病,在婴儿期发病。该病与溶质运载家族 18 成员 A2 基因(SLC18A2)的突变有关。有报告称,在使用多巴胺能药物的试验中,服用左旋多巴的患者病情几乎总是恶化,而多巴胺激动剂则会给一些患者带来不同程度的益处。在此,我们报告了一名患有 SLC18A2 基因新变异的 PKDYS2 患者,该患者接受了多种药物治疗试验。该病例在 2 个月大时首次发现发育异常和神经系统检查异常,经过一系列治疗尝试(如使用抗癫痫药)和诊断程序后,在 6 岁时进行的全外显子组测序(WES)发现 SLC18A2 基因存在同卵病理变异 NM_003054.4:c.1107dup,p.(Val370Serfs∗91),从而确定了 PKDYS2 的诊断。患者随后接受了多种多巴胺能药物(如左旋多巴、普拉克索和哌醋甲酯)的治疗。PKDYS2患者携带SLC18A2的一个新变体。该患者的表型与之前报道的一些 PKDYS2 患者相似。该患者从某些多巴胺能药物(如普拉克索)中略有获益,但副作用导致其停药。
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引用次数: 0
A Case of Symptomatic Intraluminal Internal Carotid Artery Thrombus in a Patient with Essential Thrombocythemia Surgically Treated by CEA. 原发性血小板增多症患者经CEA手术治疗并发腔内症状性颈内动脉血栓1例。
IF 0.9 Q4 CLINICAL NEUROLOGY Pub Date : 2023-11-24 eCollection Date: 2023-01-01 DOI: 10.1155/2023/9152009
Satoshi Takahashi, Masahiro Katsumata, Hirotsugu Nogawa, Kento Takahara, Jin Nakahara, Masahiro Toda

We report a patient with a symptomatic intraluminal internal carotid artery thrombus clinically revealed by cerebral infarction. In the preoperative evaluation, it was revealed that essential thrombocythemia existed in the background. Therefore, medical treatment with antithrombotic agents in conjunction with hydroxycarbamide for essential thrombocythemia was initiated, but the thrombus was not dissolved by three weeks. At this time, the patient underwent carotid endarterectomy, which removed the thrombus completely with its adjacent plaque without any perioperative stroke. The possibility of essential thrombocythemia may also be kept in mind when an increased platelet count is observed in patients with internal carotid artery thrombus. It is a reasonable option to precede medical treatment, including anticoagulant therapy, by setting the time limit for surgical intervention in such a disease state.

我们报告一个病人有症状的腔内颈动脉血栓临床表现为脑梗死。在术前评估中,发现背景中存在原发性血小板增多症。因此,开始使用抗血栓药物联合羟基脲治疗原发性血小板增多症,但血栓在三周后仍未溶解。此时,患者行颈动脉内膜切除术,将血栓及其邻近斑块完全清除,无围手术期卒中。当观察到颈内动脉血栓患者血小板计数增加时,也要注意原发性血小板增多症的可能性。在这种疾病状态下,通过设定手术干预的时间限制,在药物治疗(包括抗凝治疗)之前是一个合理的选择。
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引用次数: 0
Penetrating Head Injury by a Hit of Rake in a Child: A Case Report and Literature Review. 儿童耙击致头部穿透伤1例报告及文献复习。
IF 0.9 Q4 CLINICAL NEUROLOGY Pub Date : 2023-11-18 eCollection Date: 2023-01-01 DOI: 10.1155/2023/9921985
Issa Ibrahim Assoumane, Nicaise Kpègnon Agada, Rabiou Maman Sani, Aminath Kélani

Background: A penetrating head injury (PHI) refers to a situation where a projectile has breached the cranium but does not exit it. It constitutes about 0.4% of all head injuries. Several nonmissile materials inserting the skull have been reported. But to our knowledge, never before has any case of PHI caused by a hit of rake been reported. We report a first case of PHI caused by a rake in a child; then, we relate our experience with its management and discuss the relevant literature. Cases Description. A 5-year-old boy has been admitted with a rake embedded in his head. That occurred during a violent play with a neighbor. At presentation, the child was alert; there was no neurological deficit. The rake was embedded in the parietal regions on each side of the midline. The head Computed Tomography (CT) scan performed showed a biparietal hyperdensity from either side of the midline with a metal artifact. In the operating room, after a transversal incision joining the 2 tips of the object, we performed successively bone flaps; object extraction; debridement; duraplasty; and closing. The outcome was uneventful.

Conclusion: This is the first case of PHI by a rake. The surgical management constitutes the main challenging point.

背景:穿透性头部损伤(PHI)是指抛射物穿透颅骨但没有离开的情况。它约占所有头部伤害的0.4%。一些非导弹材料插入颅骨已被报道。但据我们所知,以前从未有过因击打耙子而导致PHI的报道。我们报告了第一例PHI引起的耙在一个孩子;在此基础上,结合自身的管理经验,对相关文献进行了讨论。案例描述。一名5岁男孩被送进医院,他的头上插着一把耙子。那是在和邻居暴力玩耍时发生的。在陈述时,孩子是警觉的;没有神经缺陷。耙嵌入在中线两侧的顶叶区域。头部计算机断层扫描(CT)显示中线两侧双顶叶高密度伴金属伪影。在手术室中,我们在横切口连接物体的两个尖端后,连续进行骨瓣;对象提取;清创术;duraplasty;和关闭。结果平安无事。结论:本病例为第一例由耙子引起的PHI。手术处理是主要的挑战点。
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引用次数: 0
Generalized Extension of Referred Trigeminal Pain due to Greater Occipital Nerve Entrapment 枕大神经卡压引起的牵涉性三叉神经痛的广泛性延伸
Q4 CLINICAL NEUROLOGY Pub Date : 2023-11-11 DOI: 10.1155/2023/1099222
Jung-woo Hyung, Byung-chul Son
We report a very rare case of referred pain caused by greater occipital nerve (GON) entrapment, inducing spontaneous pain in the whole body as well as in the trigeminal nerve region of the face and head. It has already been reported that entrapment of the GON can induce referred pain in the ipsilateral limb as well as the ipsilateral hemiface. A 42-year-old female patient presented with chronic pain in her gums, jaw angle, submandibular region, retro-auricular suboccipital, and temporo-occipital vertex that had been ongoing for four years. As the patient’s head pain and facial pain became severe, severe spontaneous pain occurred in the arm, waist, and both lower extremities. This patient’s pain in the occipital and neck, spontaneous pain in the face, jaw, and whole body improved with decompression of the GON. Anatomical basis of pain referral to the facial trigeminal area caused by chronic GON entrapment is convergence of nociceptive inflow from high cervical C1–C3 structures and trigeminal orofacial area in the dorsal horn of the cervical spinal cord from the C2 segment up to the medullary dorsal horn (MDH). The major afferent contribution among the suboccipital and high cervical structure is mediated by spinal root C2 that is peripherally represented by the GON. Chronic noxious input from GON entrapment can cause sensitization and hypersensitivity in second order neurons in the trigeminocervical complex (TCC) and MDH in the caudal trigeminal nucleus and high cervical cord. Generalized extension of referred pain due to GON entrapment is thought to involve two possible pathophysiologies. One is the possibility that generalized pain is caused by sensitization of third-order nociceptive neurons in the thalamus. Another speculation is that spontaneous pain may occur throughout the body due to dysfunction of the descending brain stem pain-modulating pathway by sensitization and hyperexcitation of the MDH and trigeminal brainstem sensory nuclear complex (TBSNC).
我们报告一个非常罕见的病例牵涉疼痛引起的大枕神经(GON)卡压,诱发自发性疼痛在全身以及在三叉神经区域的脸和头部。已经有报道称,下丘脑的压迫可引起同侧肢体和同侧半侧的牵涉性疼痛。女,42岁,以牙龈、颌角、下颌下、耳后枕下、颞枕顶慢性疼痛4年为主诉。随着患者头部疼痛和面部疼痛加重,手臂、腰部和双下肢出现严重的自发性疼痛。该患者的枕部和颈部疼痛,面部、下颌和全身自发性疼痛随着颏部减压得到改善。慢性神经激素压迫引起的疼痛转介到面三叉神经区的解剖学基础是颈脊髓背角的高颈C1-C3结构和三叉神经口面区从C2节向上至髓质背角(MDH)的伤害性流入汇合。枕下和高颈结构的主要传入神经是由脊柱根C2介导的,其周围以神经根为代表。来自神经神经毒素包埋的慢性有害输入可引起三叉神经颈复合体(TCC)二级神经元的致敏和超敏,以及三叉神经尾核和高颈髓的MDH。由于神经甾体夹闭引起的牵涉性疼痛的广泛性延伸被认为涉及两种可能的病理生理。一种可能性是,全身性疼痛是由丘脑中第三级伤害性神经元的敏化引起的。另一种推测是自发性疼痛可能发生在全身,这是由于下行脑干疼痛调节通路的功能障碍,这是由MDH和三叉脑干感觉核复合体(TBSNC)的致敏和过度兴奋引起的。
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Case Reports in Neurological Medicine
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