Case Reports in Neurological Medicine最新文献

{"title":"Diabetic Striatopathy: Case Report and Possible New Actors.","authors":"Chiara Mozzini,&nbsp;Raffaele Ghirardi,&nbsp;Mauro Pagani","doi":"10.1155/2022/4176419","DOIUrl":"https://doi.org/10.1155/2022/4176419","url":null,"abstract":"<p><p>Diabetic striatopathy is a very rare neurological complication of diabetes. We report the case of an 86-year-old woman with poorly controlled type 2 diabetes admitted to the internal medicine ward for sudden onset of altered sensorium and severe bilateral choreiform and ballistic movements. The precise pathophysiology of this condition is not well understood. Our communication aims to remind clinicians to consider the possibility of diabetic striatopathy when poor-controlled diabetic patients have sudden-onset choreiform and ballistic movements. Moreover, this case suggests the possibility that oxidative and endoplasmic reticulum stress may be involved in this process.</p>","PeriodicalId":9615,"journal":{"name":"Case Reports in Neurological Medicine","volume":"2022 ","pages":"4176419"},"PeriodicalIF":0.9,"publicationDate":"2022-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9792229/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"10509861","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Ipsilateral Limb Extension of Referred Trigeminal Facial Pain due to Greater Occipital Nerve Entrapment: A Case Report.","authors":"Byung-Chul Son,&nbsp;Changik Lee","doi":"10.1155/2022/9381881","DOIUrl":"https://doi.org/10.1155/2022/9381881","url":null,"abstract":"<p><p>We report a very rare case of referred pain associated with entrapment of the greater occipital nerve (GON) occurring not only in the ipsilateral hemiface but also in the ipsilateral limb. There is an extensive convergence of cutaneous, tooth pulp, visceral, neck, and muscle afferents onto nociceptive and nonnociceptive neurons in the trigeminal nucleus caudalis (medullary dorsal horn). In addition, nociceptive input from trigeminal, meningeal afferents projects into trigeminal nucleus caudalis and dorsal horn of C1 and C2. Together, they form a functional unit, the trigeminocervical complex (TCC). The nociceptive inflow from suboccipital and high cervical structures is mediated with small-diameter afferent fibers in the upper cervical roots terminating in the dorsal horn of the cervical cord extending from the C2 segment up to the medullary dorsal horn. The major afferent contribution is mediated by the spinal root C2 that is peripherally represented by the greater occipital nerve (GON). Convergence of afferent signals from the trigeminal nerve and the GON onto the TCC is regarded as an anatomical basis of pain referral in craniofacial pain and primary headache syndrome. Ipsilateral limb pain occurs long before the onset of the referred facial pain. The subsequent severe hemifacial pain suggested GON entrapment. The occipital nerve block provided temporary relief from facial and extremity pain. Imaging studies found a benign osteoma in the ipsilateral suboccipital bone, but no direct contact with GON was identified. During GON decompression, severe entrapment of the GON was observed by the tendinous aponeurotic edge of the trapezius muscle, but the osteoma had no contact with the nerve. Following GON decompression, the referred trigeminal and extremity pain completely disappeared. The pain referral from GON entrapment seems to be attributed to the sensitization and hypersensitivity of the trigeminocervical complex (TCC). The clinical manifestations of TCC hypersensitivity induced by chronic entrapment of GONs are diverse when considering the occurrence of extremity pain as well as facial pain.</p>","PeriodicalId":9615,"journal":{"name":"Case Reports in Neurological Medicine","volume":"2022 ","pages":"9381881"},"PeriodicalIF":0.9,"publicationDate":"2022-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9734007/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"10681860","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"A Case of Carbamazepine-Induced Aggravation of Self-Limited Epilepsy with Centrotemporal Spikes Epilepsy and Valproate-Induced Hyperammonemic Encephalopathy in a Child with Heterozygous Gene Variant of Carbomoyl Phosphatase Synthetase Deficiency.","authors":"Imalke Kankananarachchi,&nbsp;Eresha Jasinge,&nbsp;Gemunu Hewawitharana","doi":"10.1155/2021/2362679","DOIUrl":"https://doi.org/10.1155/2021/2362679","url":null,"abstract":"<p><p>Antiepileptics drugs are the mainstay of the management of epilepsy in children. Sodium valproate (VPA) and carbamazepine (CBZ) are widely used medications in childhood epilepsy. Hyperammonemia has been described as a known side effect of valproate therapy. It is known that VPA-associated HA is common among patients who hold genetic mutations of the carbomoyl phosphatase synthase 1 gene (CPS1). Aggravation of self-limited epilepsy with centrotemporal spikes (SLECTS) is a rare side effect of CBZ. Here, we present a child who had CBZ-induced aggravation of rolandic epilepsy and VPA-induced HA encephalopathy in the background of an unrecognised heterozygous gene variant of CPS1. An 8-year-old boy with SLECTS presented with a history of abnormal behaviours and drowsiness. He was apparently well until six years when he developed seizures in favour of rolandic epilepsy. His electroencephalogram (EEG) showed bilateral predominantly on the right-sided central-temporal spikes and waves. The diagnosis of SLECTS was made, and he was commenced on CBZ. Though he showed some improvement at the beginning, his seizure frequency increased when the dose of CBZ was increased. His repeat EEG showed electrical status in slow-wave sleep, and CBZ was stopped. Subsequently, he was started on VPA, and with that, he developed features of encephalopathy. He had elevated serum ammonia with normal liver functions. VPA was stopped with the suspicion of VPA-induced hyperammonemia. Tandem mass spectrometry did not show significant abnormality in the amino acid profile. Specific genetic analysis revealed a c.2756 C > T.p (Ser919Leu) heterozygote genetic mutation of the CSP 1 gene. This is a classic example where side effects of treatment determine the choice of antiepileptics drugs (AEDs) in childhood epilepsy. It is essential to keep in mind that SLECTS can be aggravated with certain AEDs, and VPA-induced HA in the absence of live failure could be due to underlying inherited metabolic disorders.</p>","PeriodicalId":9615,"journal":{"name":"Case Reports in Neurological Medicine","volume":" ","pages":"2362679"},"PeriodicalIF":0.9,"publicationDate":"2021-12-31","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8741391/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"39802853","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Muscle-Specific Tyrosine Kinase-Associated Myasthenia Gravis: A Neuromuscular Surprise.","authors":"Hassam Ali,&nbsp;Rahul Pamarthy,&nbsp;Nayab Ahsan,&nbsp;WashmaAwan,&nbsp;Shiza Sarfraz","doi":"10.1155/2021/1326442","DOIUrl":"https://doi.org/10.1155/2021/1326442","url":null,"abstract":"<p><p>Myasthenia gravis is a neuromuscular autoimmune disease that results in skeletal muscle weakness that worsens after periods of activity and improves after rest. Myasthenia gravis means \"grave (serious), muscle weakness.\" Although not completely curable, it can be managed well with a relatively high quality of life and expectancy. In myasthenia gravis, antibodies against the acetylcholine receptors at the neuromuscular junction interfere with regular muscular contraction. Although most commonly caused by antibodies to the acetylcholine receptor, antibodies against MuSK (muscle-specific kinase) protein can also weaken transmission at the neuromuscular junction. Muscle-specific tyrosine kinase myasthenia gravis (MuSK-Ab MG) is a rare subtype of myasthenia gravis with distinct pathogenesis and unique clinical features. Diagnosis can be challenging due to its atypical presentation as compared to seropositive myasthenia gravis. It responds inconsistently to steroids, but plasma exchange and immunosuppressive therapies have shown promising results. We report a case of a 49-year-old female who presented with acute hypoxic respiratory failure. Our patient experienced progressive, undiagnosed MuSK-Ab MG for years without a diagnosis.</p>","PeriodicalId":9615,"journal":{"name":"Case Reports in Neurological Medicine","volume":" ","pages":"1326442"},"PeriodicalIF":0.9,"publicationDate":"2021-12-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8727168/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"39668881","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Spontaneous Recovery of Penetrating Cervical Spinal Cord Injury with Physiotherapeutic Treatment: Case Report and Review of the Literature.","authors":"Yao Christian Hugues Dokponou,&nbsp;Mamoune El Mostarchid,&nbsp;Housni Abderrahmane,&nbsp;Niamien Patrice Koffi,&nbsp;Miloudi Gazzaz,&nbsp;Brahim El Mostarchid","doi":"10.1155/2021/3741461","DOIUrl":"https://doi.org/10.1155/2021/3741461","url":null,"abstract":"<p><p>Stab wounds to the cervical spine are less common than injuries from road accidents, sports injuries, and falls. The presence of vital, vascular, neural, respiratory, and digestive structures in the neck region mean that this kind of spinal injury is generally critical, and its management is a challenge. We report a unique case of a previously healthy 17-year-old adolescent admitted for quadriplegia secondary to a stab wound to the cervical spine at the C4C5 level. There was no surgical indication. The patient underwent physiotherapy. He showed spontaneous neurological improvement two weeks later and was able to sit on his own and to walk about three months of physical rehabilitation.</p>","PeriodicalId":9615,"journal":{"name":"Case Reports in Neurological Medicine","volume":" ","pages":"3741461"},"PeriodicalIF":0.9,"publicationDate":"2021-12-26","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8720595/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"39788034","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Vasculitic Tibial Mononeuropathy Associated with Inherited Immune Dysregulation: A Review of Tibial Mononeuropathies with Electrodiagnostic Considerations.","authors":"James Liu,&nbsp;Yue Ding,&nbsp;Sandra Camelo-Piragua,&nbsp;James Richardson","doi":"10.1155/2021/7161757","DOIUrl":"https://doi.org/10.1155/2021/7161757","url":null,"abstract":"<p><p>Compressive tibial mononeuropathies are uncommon and can be caused by conditions including posterior compartment syndrome, soleal sling syndrome, and tarsal tunnel syndrome. Therefore, it is critical to consider noncompressive etiologies when a tibial mononeuropathy is suspected. This is a patient with a history of rare inherited immune dysregulation that presented to the electrodiagnostic laboratory with severe neuropathic pain in the right foot associated with plantarflexion weakness, concerning for a tibial mononeuropathy. However, the patient's clinical presentation and results on electrodiagnostic testing were not consistent with any of the above entities. Therefore, noncompressive etiologies of tibial mononeuropathies such as vasculitis had to be considered. The patient subsequently underwent sural nerve biopsy which confirmed small-vessel vasculitis as the cause of the tibial mononeuropathy. She was then started on appropriate immunosuppressive treatment which resulted in significant pain relief and was discharged home. This case highlights the importance of considering noncompressive causes of tibial nerve injury. Compressive and vasculitic tibial mononeuropathies along with their electrodiagnostic considerations are reviewed. Furthermore, this case highlights the critical role of the electromyographer and ability to maximize the impact on patient care through a solid foundation in anatomy, pathophysiology, and electrodiagnosis blended with clinical acumen.</p>","PeriodicalId":9615,"journal":{"name":"Case Reports in Neurological Medicine","volume":" ","pages":"7161757"},"PeriodicalIF":0.9,"publicationDate":"2021-11-12","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8604612/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"39897529","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Multiple Cranial Nerve Palsies in a Pediatric Case of Lemierre's Syndrome due to <i>Streptococcus viridans</i>.","authors":"Samantha Novotny,&nbsp;Kenneth Serrano,&nbsp;Danielle Bazer,&nbsp;Louis Manganas","doi":"10.1155/2021/4455789","DOIUrl":"https://doi.org/10.1155/2021/4455789","url":null,"abstract":"<p><strong>Background: </strong>Lemierre's syndrome is a rare condition of internal jugular vein thrombosis following oropharyngeal infection. While it usually results from <i>Fusobacterium necrophorum</i> infection, atypical cases associated with other pathogens have been reported.</p><p><strong>Objective: </strong>To describe a unique case of pediatric Lemierre's syndrome with <i>Streptococcus viridans</i> infection resulting in cavernous sinus thrombosis and oculomotor, trochlear, and abducens nerve palsies. <i>Case Report</i>. A 14-year-old female initially presented after six days of fever, myalgias, and sore throat and was admitted for hyperbilirubinemia and acute kidney injury. She developed a fixed, dilated pupil with complete ophthalmoplegia, ptosis, and severe pain. Imaging revealed retromandibular space abscess, external and internal jugular vein thrombosis, cavernous sinus thrombosis, internal carotid artery stenosis, pulmonary embolism, and bilateral pneumonia. She was diagnosed with Lemierre's syndrome with cultures positive for <i>Streptococcus viridans</i> and treated with a combination of antibiotics and anticoagulation. <i>Conclusion and Relevance</i>. Both antibiotics and anticoagulation were effective management for this Lemierre's syndrome patient with cavernous sinus thrombosis. Early diagnosis and treatment of Lemierre's syndrome is essential. A multidisciplinary treatment team is beneficial for managing the sequelae of this condition.</p>","PeriodicalId":9615,"journal":{"name":"Case Reports in Neurological Medicine","volume":" ","pages":"4455789"},"PeriodicalIF":0.9,"publicationDate":"2021-10-26","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8564190/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"39686873","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Paraplegia after Hypotension with Pneumothorax Episode during Thoracic Spine Tumor Surgery.","authors":"Zhao-Quan Liu,&nbsp;Jen-Hsien Liao,&nbsp;Chih-Ju Chang","doi":"10.1155/2021/2232769","DOIUrl":"https://doi.org/10.1155/2021/2232769","url":null,"abstract":"<p><p>Paraplegia after spine surgery is a catastrophic complication. Here, we present a patient who, following laminectomy and fusion for decompression of metastatic tumor, developed paraplegia. We tried to find out the possible reason for the paraplegia. Due to prolonged hypotension during operation and new onset of pneumothorax, we think that intraoperative prolonged hypotension leads to the spinal cord ischemia which may cause neurological deterioration of paraplegia. Maintaining hemodynamic stability during spinal surgery is very important.</p>","PeriodicalId":9615,"journal":{"name":"Case Reports in Neurological Medicine","volume":" ","pages":"2232769"},"PeriodicalIF":0.9,"publicationDate":"2021-10-23","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8557050/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"39849159","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Paradoxical and Retrograde Air Embolism from Pressurized Peripheral Bolus.","authors":"Joshua Santucci,&nbsp;Naresh Mullaguri,&nbsp;Anusha Battineni,&nbsp;Raviteja R Guddeti,&nbsp;Christopher R Newey","doi":"10.1155/2021/1063264","DOIUrl":"https://doi.org/10.1155/2021/1063264","url":null,"abstract":"<p><strong>Introduction: </strong>Cerebral air embolism is a rare, yet serious neurological occurrence with unclear incidence and prevalence. Here, we present a case of fatal cerebral arterial and venous cerebral gas embolism in a patient with infective endocarditis and known large right-to-left shunt and severe tricuspid regurgitation following pressurized fluid bolus administration. <i>Case Presentation</i>. A 32-year-old female was admitted to the medical intensive care unit from a long-term acute care facility with acute on chronic respiratory failure. Her medical history was significant for intravenous heroin and cocaine abuse, methicillin-sensitive <i>Staphylococcus aureus</i> tricuspid valve infective endocarditis on vancomycin, patent foramen ovale, septic pulmonary embolism with cavitation, tracheostomy with chronic ventilator dependence, multifocal cerebral infarction, hepatitis C, nephrolithiasis, anxiety, and depression. After intravenous fluid administration, she became unresponsive with roving gaze, sluggish pupils, and hypotensive requiring vasopressors. CT of the brain showed diffuse arterial and venous cerebral air embolism secondary to accidental air administration from fluid bolus. Magnetic resonance imaging of the brain showed diffuse global anoxic injury and flattening of the globe at the optic nerve insertion. Given poor prognosis, her family chose comfort measures and she died.</p><p><strong>Conclusions: </strong>Fatal cerebral air embolism can occur through peripheral intravenous routes when the lines are inadequately primed and fluids administered with pressure. Caution must be exercised in patients with right-to-left shunting as air may gain access to systemic circulation.</p>","PeriodicalId":9615,"journal":{"name":"Case Reports in Neurological Medicine","volume":" ","pages":"1063264"},"PeriodicalIF":0.9,"publicationDate":"2021-10-05","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8510818/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"39519511","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Familial Multiple Sclerosis in a Mother and Son Pair: A Sri Lankan and a South Asian First.","authors":"Ishani Rajapakshe,&nbsp;Bimsara Senanayake","doi":"10.1155/2021/1172870","DOIUrl":"https://doi.org/10.1155/2021/1172870","url":null,"abstract":"<p><p>Multiple sclerosis (MS) is an immune-mediated demyelinating disorder involving the central nervous system (CNS). It is common amongst young females. Although the exact cause of MS is yet unknown, viral infections such as EBV, environmental factors, and autoimmune and genetic mechanisms involving HLA-DRB1 loci are implicated. Familial MS is reported from some geographic locations and ethnic groups but is thought to be rare in Asia. In this paper, we present both a Sri Lankan mother and her son, with clinically definite MS conforming to McDonald's 2017 clinical and MAGNIMS 2016 radiological criteria. Both had oligoclonal bands in their CSF (OCB-IEF) with no serum bands indicating intrathecal production and were negative for AQP4 and MOG IgG serology. Familial MS is more common among siblings, with sister-sister relationship having the highest rate. The lowest relation was amongst father-son and mother-son pairs. Amongst siblings, the risk of MS is between 3.5% and 4.7%. Inherited factors rather than common environmental exposure influence susceptibility in such cases. To the best of our knowledge, MS occurring in a mother-son pair has not been reported before either from Sri Lanka or South Asia.</p>","PeriodicalId":9615,"journal":{"name":"Case Reports in Neurological Medicine","volume":" ","pages":"1172870"},"PeriodicalIF":0.9,"publicationDate":"2021-09-23","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8486554/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"39482683","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}