Pub Date : 2024-11-29eCollection Date: 2024-01-01DOI: 10.1159/000528310
Hasanain A Al-Khalidi, Hayder K Hassoun, Zahra Aljid, Zuhair Allebban
Introduction: Neurological complication due to coronavirus disease 2019 (COVID-19) is accumulating and compressive myelopathy due to spinal subdural hematoma (SSDH) is rarely reported in association with COVID-19.
Case presentation: A 55-year-old male was presented with sudden onset of areflexic paraparesis, urinary retention, loss of all sensations below twelve spinal thoracic segments, and severe back pain. This condition necessitated an immediate order of a spinal cord MRI followed by an urgent surgery, which was crucial to save the spinal cord. COVID-19 was confirmed by a positive reverse-transcription-polymerase chain reaction and spinal MRI showed SSDH.
Conclusion: For a patient who presents with acute onset of severe back pain and myelopathy without a history of trauma, SSDH should be suspected. Additionally, coagulopathy associated with COVID-19 infection should increase the suspicion of SSDH which needs immediate surgical treatment to save the spinal cord.
{"title":"COVID-19-Related Spinal Subdural Hematoma Presented with Acute Compressive Myelopathy with a Review of the Literature.","authors":"Hasanain A Al-Khalidi, Hayder K Hassoun, Zahra Aljid, Zuhair Allebban","doi":"10.1159/000528310","DOIUrl":"https://doi.org/10.1159/000528310","url":null,"abstract":"<p><strong>Introduction: </strong>Neurological complication due to coronavirus disease 2019 (COVID-19) is accumulating and compressive myelopathy due to spinal subdural hematoma (SSDH) is rarely reported in association with COVID-19.</p><p><strong>Case presentation: </strong>A 55-year-old male was presented with sudden onset of areflexic paraparesis, urinary retention, loss of all sensations below twelve spinal thoracic segments, and severe back pain. This condition necessitated an immediate order of a spinal cord MRI followed by an urgent surgery, which was crucial to save the spinal cord. COVID-19 was confirmed by a positive reverse-transcription-polymerase chain reaction and spinal MRI showed SSDH.</p><p><strong>Conclusion: </strong>For a patient who presents with acute onset of severe back pain and myelopathy without a history of trauma, SSDH should be suspected. Additionally, coagulopathy associated with COVID-19 infection should increase the suspicion of SSDH which needs immediate surgical treatment to save the spinal cord.</p>","PeriodicalId":9639,"journal":{"name":"Case Reports in Neurology","volume":"16 1","pages":"294-303"},"PeriodicalIF":0.6,"publicationDate":"2024-11-29","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11606623/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142766538","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}
Introduction: Herpes simplex virus type 1 (HSV-1) is the leading cause of sporadic fatal encephalitis, typically presenting with temporal lobe abnormalities. It usually manifests as fever, headache, seizure, altered sensorium, and focal neurological deficit. Hyperphagia as a sole complication of HSV-1 encephalitis is a rare presentation.
Case presentation: We report a 25-year-old woman with a 10-day history of fever, headache, and vomiting, progressing to confusion, visual hallucinations, and drowsiness. She had a history of meningoencephalitis at age 8 and well-controlled focal seizures. Upon admission, magnetic resonance imaging showed T2/fluid-attenuated inversion recovery hyperintensities in both temporal lobes with diffusion restriction. Electroencephalography indicated generalized slowing and cerebrospinal fluid (CSF) analysis revealed lymphocytic pleocytosis with elevated protein levels. Viral encephalitis was suspected, and intravenous acyclovir was initiated. CSF polymerase chain reaction (PCR) confirmed HSV-1. With treatment, she gradually improved but developed hyperphagia during hospital stay. Hyperphagia, a rare complication of herpes simplex virus (HSV) encephalitis, is a part of Kluver-Bucy syndrome typically associated with other cognitive dysfunctions. Despite early treatment, voracious appetite remained partially, emphasizing the need for rapid diagnosis and treatment to prevent severe outcomes.
Conclusion: The case highlights that acute onset hyperphagia can be an isolated complication of HSV encephalitis, requiring tailored therapeutic strategies. Follow-up showed significant weight gain with partial improvement in hyperphagia, underscoring the challenges in managing this condition.
{"title":"Virus-Induced Voracity: Uncovering Hyperphagia Post-Herpes Simplex Virus Type 1.","authors":"Arpan Mitra, Nayana Bhuyan, Ankur Vivek, Akansha Jain, Vijaya Nath Mishra, Abhishek Pathak","doi":"10.1159/000541698","DOIUrl":"https://doi.org/10.1159/000541698","url":null,"abstract":"<p><strong>Introduction: </strong>Herpes simplex virus type 1 (HSV-1) is the leading cause of sporadic fatal encephalitis, typically presenting with temporal lobe abnormalities. It usually manifests as fever, headache, seizure, altered sensorium, and focal neurological deficit. Hyperphagia as a sole complication of HSV-1 encephalitis is a rare presentation.</p><p><strong>Case presentation: </strong>We report a 25-year-old woman with a 10-day history of fever, headache, and vomiting, progressing to confusion, visual hallucinations, and drowsiness. She had a history of meningoencephalitis at age 8 and well-controlled focal seizures. Upon admission, magnetic resonance imaging showed T2/fluid-attenuated inversion recovery hyperintensities in both temporal lobes with diffusion restriction. Electroencephalography indicated generalized slowing and cerebrospinal fluid (CSF) analysis revealed lymphocytic pleocytosis with elevated protein levels. Viral encephalitis was suspected, and intravenous acyclovir was initiated. CSF polymerase chain reaction (PCR) confirmed HSV-1. With treatment, she gradually improved but developed hyperphagia during hospital stay. Hyperphagia, a rare complication of herpes simplex virus (HSV) encephalitis, is a part of Kluver-Bucy syndrome typically associated with other cognitive dysfunctions. Despite early treatment, voracious appetite remained partially, emphasizing the need for rapid diagnosis and treatment to prevent severe outcomes.</p><p><strong>Conclusion: </strong>The case highlights that acute onset hyperphagia can be an isolated complication of HSV encephalitis, requiring tailored therapeutic strategies. Follow-up showed significant weight gain with partial improvement in hyperphagia, underscoring the challenges in managing this condition.</p>","PeriodicalId":9639,"journal":{"name":"Case Reports in Neurology","volume":"16 1","pages":"262-268"},"PeriodicalIF":0.6,"publicationDate":"2024-10-09","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11521529/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142544016","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}
Pub Date : 2024-10-04eCollection Date: 2024-01-01DOI: 10.1159/000541631
Adrienne Elisabeth van der Hoeven, Josephus Johannes Kerremans, Tjerk Joppe Lagrand
Introduction: Capnocytophaga canimorsus is a Gram-negative bacterium found in the oral flora of dogs and cats, transmitted to humans through bites, licks, or scratches. Infections can lead to severe manifestations, including meningitis, particularly in immunocompromised individuals.
Case presentation: A 46-year-old immunocompetent man presented with somnolence, headache, and fever after being licked by his dog. Neurological examination revealed signs of meningeal irritation, and cerebrospinal fluid analysis showed an elevated white cell count and protein levels consistent with bacterial meningitis. Treatment followed Dutch guidelines with amoxicillin, ceftriaxone, and dexamethasone, resulting in rapid clinical improvement. Microbiological confirmation of C. canimorsus followed later. The patient was treated with antibiotics for the duration of 1 week and remained symptom-free after being discharged.
Conclusion: C. canimorsus meningitis, although rare, poses diagnostic challenges due to its variable presentation and slow growth in culture. Empirical therapy guided by susceptibility testing contributes to favorable outcomes. This case underscores the importance of considering a C. canimorsus infection in patients with animal exposure and of taking diagnostic findings, precedent, and clinical response into account when determining the treatment duration.
{"title":"A Rare Case of <i>Capnocytophaga canimorsus</i> Meningitis in an Immunocompetent Patient: A Successful Outcome with a Short Course of Antibiotics.","authors":"Adrienne Elisabeth van der Hoeven, Josephus Johannes Kerremans, Tjerk Joppe Lagrand","doi":"10.1159/000541631","DOIUrl":"https://doi.org/10.1159/000541631","url":null,"abstract":"<p><strong>Introduction: </strong><i>Capnocytophaga canimorsus</i> is a Gram-negative bacterium found in the oral flora of dogs and cats, transmitted to humans through bites, licks, or scratches. Infections can lead to severe manifestations, including meningitis, particularly in immunocompromised individuals.</p><p><strong>Case presentation: </strong>A 46-year-old immunocompetent man presented with somnolence, headache, and fever after being licked by his dog. Neurological examination revealed signs of meningeal irritation, and cerebrospinal fluid analysis showed an elevated white cell count and protein levels consistent with bacterial meningitis. Treatment followed Dutch guidelines with amoxicillin, ceftriaxone, and dexamethasone, resulting in rapid clinical improvement. Microbiological confirmation of <i>C</i>. <i>canimorsus</i> followed later. The patient was treated with antibiotics for the duration of 1 week and remained symptom-free after being discharged.</p><p><strong>Conclusion: </strong><i>C. canimorsus</i> meningitis, although rare, poses diagnostic challenges due to its variable presentation and slow growth in culture. Empirical therapy guided by susceptibility testing contributes to favorable outcomes. This case underscores the importance of considering a <i>C</i>. <i>canimorsus</i> infection in patients with animal exposure and of taking diagnostic findings, precedent, and clinical response into account when determining the treatment duration.</p>","PeriodicalId":9639,"journal":{"name":"Case Reports in Neurology","volume":"16 1","pages":"269-273"},"PeriodicalIF":0.6,"publicationDate":"2024-10-04","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11521510/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142544009","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}
Pub Date : 2024-10-02eCollection Date: 2024-01-01DOI: 10.1159/000541585
Bassem Al Hariri, Muad Abdi Hassan, Muhammad Sharif, Obada Adel Alsakaji, Yussuf Abdi Hassan, Muayad Kasim Khalid
Introduction: Periodic paralysis is a condition that causes recurrent episodes of flaccid paralysis, and it can be primary or secondary. Hypokalemic periodic paralysis is the most common type of primary periodic paralysis, and it is inherited through autosomal dominant gene transmission. Males are affected three times more often than females, and the paralysis attacks usually occur at night after a period of vigorous exercise. It is crucial to exclude other diagnostic entities based on the nature of presentation, physical examination, and paraclinical studies. Thyrotoxic periodic paralysis is more prevalent in Asian or Hispanic males with thyrotoxicosis, where up to 10% of thyrotoxic patients may experience periodic paralysis.
Case presentations: Here, we present 6 cases of patients who came to our care with varying degrees of muscle weakness, each showing interesting and diverse laboratory results.
Conclusion: In patient assessment, it is crucial to consider social and family history. Even without this information, awareness of potential diagnoses is vital. The cause should be carefully considered for possible simple treatments. Failing to recognize and address this condition promptly could lead to severe outcomes. Timely identification and intervention are essential for effective disease management and patient welfare.
{"title":"Periodic Paralysis: A Case Series with a Literature Review.","authors":"Bassem Al Hariri, Muad Abdi Hassan, Muhammad Sharif, Obada Adel Alsakaji, Yussuf Abdi Hassan, Muayad Kasim Khalid","doi":"10.1159/000541585","DOIUrl":"https://doi.org/10.1159/000541585","url":null,"abstract":"<p><strong>Introduction: </strong>Periodic paralysis is a condition that causes recurrent episodes of flaccid paralysis, and it can be primary or secondary. Hypokalemic periodic paralysis is the most common type of primary periodic paralysis, and it is inherited through autosomal dominant gene transmission. Males are affected three times more often than females, and the paralysis attacks usually occur at night after a period of vigorous exercise. It is crucial to exclude other diagnostic entities based on the nature of presentation, physical examination, and paraclinical studies. Thyrotoxic periodic paralysis is more prevalent in Asian or Hispanic males with thyrotoxicosis, where up to 10% of thyrotoxic patients may experience periodic paralysis.</p><p><strong>Case presentations: </strong>Here, we present 6 cases of patients who came to our care with varying degrees of muscle weakness, each showing interesting and diverse laboratory results.</p><p><strong>Conclusion: </strong>In patient assessment, it is crucial to consider social and family history. Even without this information, awareness of potential diagnoses is vital. The cause should be carefully considered for possible simple treatments. Failing to recognize and address this condition promptly could lead to severe outcomes. Timely identification and intervention are essential for effective disease management and patient welfare.</p>","PeriodicalId":9639,"journal":{"name":"Case Reports in Neurology","volume":"16 1","pages":"274-280"},"PeriodicalIF":0.6,"publicationDate":"2024-10-02","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11501104/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142495762","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}
Introduction: This is a case of a 32-year-old woman who developed postpartum depression (PPD). She became anxious and depressive about caring for her child, and the Edinburgh Postnatal Depression Scale (EPDS) test showed a score of 9 at 2 weeks after delivery, and at 7 months postpartum, she presented with major melancholic depression followed by mild cognitive decline without any neurological symptoms except cluttering speech.
Case presentation: Cerebral magnetic resonance imaging showed confluent fluid-attenuated inversion recovery hyperintensities in the periventricular and frontal deep white matter, with multiple spotty calcifications in the frontal white matter by cerebral CT. Genetic testing revealed a mutation in the colony-stimulating factor 1 receptor (CSF1R).
Conclusion: This case report is consistent with evidence that PPD may have organic causes in some cases, including CSF1R mutations. Atypical findings such as mild cognitive decline combined with PPD in psychiatric interview may justify brain imaging to avoid misdiagnosis, since CSF1R-related leukoencephalopathy is probably an under-recognized disease in medical psychiatry. Further investigations are needed to clarify a pathophysiological correlation between CSF1R signaling abnormality and PPD as well as major depression.
{"title":"Suspected Postpartum Depression Revealed to be CSF1R-Related Leukoencephalopathy: A Case Report.","authors":"Masahiko Mikuni, Kazuhiro Horiuchi, Ayako Ishikura, Soichiro Kimura, Sho Masutani, Shinya Watanabe, Akihiro Mikami, Shuhei Ishikawa, Hisashi Narita, Ichiro Kusumi, Hidenao Sasaki","doi":"10.1159/000541551","DOIUrl":"10.1159/000541551","url":null,"abstract":"<p><strong>Introduction: </strong>This is a case of a 32-year-old woman who developed postpartum depression (PPD). She became anxious and depressive about caring for her child, and the Edinburgh Postnatal Depression Scale (EPDS) test showed a score of 9 at 2 weeks after delivery, and at 7 months postpartum, she presented with major melancholic depression followed by mild cognitive decline without any neurological symptoms except cluttering speech.</p><p><strong>Case presentation: </strong>Cerebral magnetic resonance imaging showed confluent fluid-attenuated inversion recovery hyperintensities in the periventricular and frontal deep white matter, with multiple spotty calcifications in the frontal white matter by cerebral CT. Genetic testing revealed a mutation in the colony-stimulating factor 1 receptor (CSF1R).</p><p><strong>Conclusion: </strong>This case report is consistent with evidence that PPD may have organic causes in some cases, including CSF1R mutations. Atypical findings such as mild cognitive decline combined with PPD in psychiatric interview may justify brain imaging to avoid misdiagnosis, since CSF1R-related leukoencephalopathy is probably an under-recognized disease in medical psychiatry. Further investigations are needed to clarify a pathophysiological correlation between CSF1R signaling abnormality and PPD as well as major depression.</p>","PeriodicalId":9639,"journal":{"name":"Case Reports in Neurology","volume":"16 1","pages":"281-287"},"PeriodicalIF":0.6,"publicationDate":"2024-10-02","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11527462/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142557262","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}
Pub Date : 2024-09-18eCollection Date: 2024-01-01DOI: 10.1159/000541522
Jinlong Wang, Bo Cao
Introduction: Spinal teratomas are rare, accounting for nearly 0.2-0.5% of all spinal tumors and 2% of all teratomas. Teratomas at the conus medullaris location do not inherently lead to epilepsy. However, potential epileptic seizures are caused when teratoma ruptures and the chemical stimulation of teratoma components enter the dural sac.
Case presentation: A 31-year-old Asian male patient suffering from epileptic onset and poor antiepileptic treatment was demonstrated. The spinal imaging examination was performed, and the patient suffered a space-occupying lesion within the conus medullaris related to spinal deformity, spinal embolism, etc. The autoimmune encephalitis spectrum revealed mGluR5 antibody IgG (+) 1:10 response. The patient stabilized after treatment with hormones and human immunoglobulin. Some hair and lipid droplets could be observed in the dural sac intraoperatively, and more hair and lipid-like material were present in the spinal cord. Postoperative pathology established the diagnosis as a conus medullaris teratoma in adults. Epileptic seizures stopped after surgery, and no additional seizures were reported during the 33-month follow-up period.
Conclusions: Conus medullaris teratoma rupture in adults rarely causes epileptic seizures. For spinal deformity patients with unexplained epileptic symptoms, spinal MRI can be helpful in early diagnosis, and more appropriate treatment improves disease prognosis.
{"title":"Spinal Teratoma with Recurrent Epileptic Episodes in Adults: A Case Report.","authors":"Jinlong Wang, Bo Cao","doi":"10.1159/000541522","DOIUrl":"https://doi.org/10.1159/000541522","url":null,"abstract":"<p><strong>Introduction: </strong>Spinal teratomas are rare, accounting for nearly 0.2-0.5% of all spinal tumors and 2% of all teratomas. Teratomas at the conus medullaris location do not inherently lead to epilepsy. However, potential epileptic seizures are caused when teratoma ruptures and the chemical stimulation of teratoma components enter the dural sac.</p><p><strong>Case presentation: </strong>A 31-year-old Asian male patient suffering from epileptic onset and poor antiepileptic treatment was demonstrated. The spinal imaging examination was performed, and the patient suffered a space-occupying lesion within the conus medullaris related to spinal deformity, spinal embolism, etc. The autoimmune encephalitis spectrum revealed mGluR5 antibody IgG (+) 1:10 response. The patient stabilized after treatment with hormones and human immunoglobulin. Some hair and lipid droplets could be observed in the dural sac intraoperatively, and more hair and lipid-like material were present in the spinal cord. Postoperative pathology established the diagnosis as a conus medullaris teratoma in adults. Epileptic seizures stopped after surgery, and no additional seizures were reported during the 33-month follow-up period.</p><p><strong>Conclusions: </strong>Conus medullaris teratoma rupture in adults rarely causes epileptic seizures. For spinal deformity patients with unexplained epileptic symptoms, spinal MRI can be helpful in early diagnosis, and more appropriate treatment improves disease prognosis.</p>","PeriodicalId":9639,"journal":{"name":"Case Reports in Neurology","volume":"16 1","pages":"249-255"},"PeriodicalIF":0.6,"publicationDate":"2024-09-18","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11521525/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142544015","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}
Pub Date : 2024-09-17eCollection Date: 2024-01-01DOI: 10.1159/000541424
Amber H van der Stam, Sharon Shmuely, Nienke M de Vries, Roland D Thijs, Mirjam van Kesteren-Biegstraaten, Bastiaan R Bloem
Introduction: Orthostatic hypotension is common in people with Parkinson's disease (PD) due to autonomic dysfunction and medication use and can have a significant negative impact on quality of life. Pharmacological treatment is often complicated due to complex blood pressure regulation problems. This case report presents a patient whose symptoms of orthostatic intolerance were successfully treated with the non-pharmacological method of head-up tilt sleeping (HUTS).
Case presentation: A 69-year-old man with PD and prominent autonomic failure received recommendation from the neurologist to use HUTS to battle orthostatic intolerance, of which complaints were worst in the early morning. The patient noted a marked improvement of the orthostatic intolerance after a period in which he slowly step-by-step inclined the bed to an angle just over 10°. When ceasing HUTS for a brief period, complaints of orthostatic intolerance immediately returned and the patient returned to tilted sleeping right away. After a follow-up of 3 months, the patient did not report orthostatic intolerance during a standing test.
Conclusion: This case illuminates that, despite difficulties intrinsic to this method, whole-body HUTS can ameliorate orthostatic intolerance and improve the daily life of people with advanced movement disorders.
{"title":"Head-Up Tilt Sleeping to Treat Orthostatic Intolerance in a Patient with Advanced Parkinson's Disease: A Case Report.","authors":"Amber H van der Stam, Sharon Shmuely, Nienke M de Vries, Roland D Thijs, Mirjam van Kesteren-Biegstraaten, Bastiaan R Bloem","doi":"10.1159/000541424","DOIUrl":"https://doi.org/10.1159/000541424","url":null,"abstract":"<p><strong>Introduction: </strong>Orthostatic hypotension is common in people with Parkinson's disease (PD) due to autonomic dysfunction and medication use and can have a significant negative impact on quality of life. Pharmacological treatment is often complicated due to complex blood pressure regulation problems. This case report presents a patient whose symptoms of orthostatic intolerance were successfully treated with the non-pharmacological method of head-up tilt sleeping (HUTS).</p><p><strong>Case presentation: </strong>A 69-year-old man with PD and prominent autonomic failure received recommendation from the neurologist to use HUTS to battle orthostatic intolerance, of which complaints were worst in the early morning. The patient noted a marked improvement of the orthostatic intolerance after a period in which he slowly step-by-step inclined the bed to an angle just over 10°. When ceasing HUTS for a brief period, complaints of orthostatic intolerance immediately returned and the patient returned to tilted sleeping right away. After a follow-up of 3 months, the patient did not report orthostatic intolerance during a standing test.</p><p><strong>Conclusion: </strong>This case illuminates that, despite difficulties intrinsic to this method, whole-body HUTS can ameliorate orthostatic intolerance and improve the daily life of people with advanced movement disorders.</p>","PeriodicalId":9639,"journal":{"name":"Case Reports in Neurology","volume":"16 1","pages":"256-261"},"PeriodicalIF":0.6,"publicationDate":"2024-09-17","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11521494/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142544011","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}
Introduction: Mitochondrial encephalomyopathy, lactic acidosis, and stroke-like episodes (MELAS) is reported to be a secondary cause of chronic intestinal pseudo-obstruction (CIPO). Although few case reports have described CIPO in MELAS, effective treatment for CIPO has not been established. Here, we present a case report of amelioration of CIPO symptoms using acotiamide in a patient with MELAS.
Case presentation: A 51-year-old Japanese female with a mitochondrial disorder with m.3243A>G mutation and a history of anorexia for 2 years presented to our hospital with a left temporal headache and acute paraphasia. A stroke-like episode of MELAS was suspected and combined therapy with arginine, edaravone, and levetiracetam was initiated. Although her symptoms improved, she presented with nausea and vomiting and abdominal distension 6 days following admission. Abdominal contrast-enhanced computed tomography revealed dilatation from the stomach to the intestine, particularly marked the stomach, with neither obstruction nor impaired blood flow in the intestine. CIPO exacerbation with MELAS was suspected, and the patient's symptoms gradually improved with acotiamide.
Conclusion: Patients with MELAS could possibly experience stroke-like episodes during CIPO. Since acetylcholine possibly plays an important role in the pathophysiology of CIPO and acotiamide possesses prokinetic activity by inhibiting acetylcholinesterase, acotiamide could possibly improve CIPO symptoms.
{"title":"Effective Management of Chronic Intestinal Pseudo-Obstruction in MELAS Using Acotiamide: A Case Report.","authors":"Yuri Kawano, Akira Taniguchi, Yugo Narita, Ken Kagawa, Tetsuro Harada, Akihiro Shindo","doi":"10.1159/000541012","DOIUrl":"10.1159/000541012","url":null,"abstract":"<p><strong>Introduction: </strong>Mitochondrial encephalomyopathy, lactic acidosis, and stroke-like episodes (MELAS) is reported to be a secondary cause of chronic intestinal pseudo-obstruction (CIPO). Although few case reports have described CIPO in MELAS, effective treatment for CIPO has not been established. Here, we present a case report of amelioration of CIPO symptoms using acotiamide in a patient with MELAS.</p><p><strong>Case presentation: </strong>A 51-year-old Japanese female with a mitochondrial disorder with m.3243A>G mutation and a history of anorexia for 2 years presented to our hospital with a left temporal headache and acute paraphasia. A stroke-like episode of MELAS was suspected and combined therapy with arginine, edaravone, and levetiracetam was initiated. Although her symptoms improved, she presented with nausea and vomiting and abdominal distension 6 days following admission. Abdominal contrast-enhanced computed tomography revealed dilatation from the stomach to the intestine, particularly marked the stomach, with neither obstruction nor impaired blood flow in the intestine. CIPO exacerbation with MELAS was suspected, and the patient's symptoms gradually improved with acotiamide.</p><p><strong>Conclusion: </strong>Patients with MELAS could possibly experience stroke-like episodes during CIPO. Since acetylcholine possibly plays an important role in the pathophysiology of CIPO and acotiamide possesses prokinetic activity by inhibiting acetylcholinesterase, acotiamide could possibly improve CIPO symptoms.</p>","PeriodicalId":9639,"journal":{"name":"Case Reports in Neurology","volume":"16 1","pages":"288-293"},"PeriodicalIF":0.6,"publicationDate":"2024-09-14","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11563654/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142615591","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}
Pub Date : 2024-08-19eCollection Date: 2024-01-01DOI: 10.1159/000540951
Muhammad Hamza Asif, Armeen Butt, Khadija Riaz, Faizan Shahzad, Maimoonah Asif, Muhammad Farhan, Waqas Ahmed, Besher Shami
Introduction: Acute disseminated encephalomyelitis (ADEM) is an acute autoimmune demyelinating disease of the central nervous system that typically follows an acute viral infection or post-vaccination. It is more common in children than in adults and is characterized by widespread demyelination of the white matter of the brain and spinal cord. ADEM typically presents as a monophasic illness. Multiphasic ADEM has been an emerging topic with case reports highlighting the importance of the need for further investigation and literature on this topic. Multiphasic ADEM in adults is a rare presentation with an atypical course that can cause a delay in diagnosis leading to disability in patients.
Case presentation: Multiple case reports have shown the presence of multiphasic ADEM without an established cause, emphasizing the importance of additional insight and data in this context. This is a unique case of multiphasic ADEM in a 59-year-old male with a temporal gap of 26 years with an idiopathic cause. He was suffering from many neurologic symptoms involving his upper and lower extremities and level of consciousness, and after he was diagnosed with ADEM, he was commenced on steroids and achieved a marked improvement in his clinical status.
Conclusion: Rarely, ADEM can present with recurrence or multiple phases, which requires prompt diagnosis and treatment. These phases may or may not be preceded by an identifiable trigger. Most patients respond well to steroids. However, further studies are required to achieve a good understanding and guidelines when dealing with such cases.
{"title":"Multiphasic Acute Disseminated Encephalomyelitis with a Temporal Gap of 26 Years: A Case Report with Novel Presentation.","authors":"Muhammad Hamza Asif, Armeen Butt, Khadija Riaz, Faizan Shahzad, Maimoonah Asif, Muhammad Farhan, Waqas Ahmed, Besher Shami","doi":"10.1159/000540951","DOIUrl":"https://doi.org/10.1159/000540951","url":null,"abstract":"<p><strong>Introduction: </strong>Acute disseminated encephalomyelitis (ADEM) is an acute autoimmune demyelinating disease of the central nervous system that typically follows an acute viral infection or post-vaccination. It is more common in children than in adults and is characterized by widespread demyelination of the white matter of the brain and spinal cord. ADEM typically presents as a monophasic illness. Multiphasic ADEM has been an emerging topic with case reports highlighting the importance of the need for further investigation and literature on this topic. Multiphasic ADEM in adults is a rare presentation with an atypical course that can cause a delay in diagnosis leading to disability in patients.</p><p><strong>Case presentation: </strong>Multiple case reports have shown the presence of multiphasic ADEM without an established cause, emphasizing the importance of additional insight and data in this context. This is a unique case of multiphasic ADEM in a 59-year-old male with a temporal gap of 26 years with an idiopathic cause. He was suffering from many neurologic symptoms involving his upper and lower extremities and level of consciousness, and after he was diagnosed with ADEM, he was commenced on steroids and achieved a marked improvement in his clinical status.</p><p><strong>Conclusion: </strong>Rarely, ADEM can present with recurrence or multiple phases, which requires prompt diagnosis and treatment. These phases may or may not be preceded by an identifiable trigger. Most patients respond well to steroids. However, further studies are required to achieve a good understanding and guidelines when dealing with such cases.</p>","PeriodicalId":9639,"journal":{"name":"Case Reports in Neurology","volume":"16 1","pages":"242-248"},"PeriodicalIF":0.6,"publicationDate":"2024-08-19","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11521480/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142544013","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}
Pub Date : 2024-08-14eCollection Date: 2024-01-01DOI: 10.1159/000540916
Hassan Doumiati, Ali Ezzeddine
Introduction: Anti-MuSK antibodies obstruct MuSK binding sites, leading to acetylcholine receptor (AChR) displacement within the postsynaptic membrane. MuSK MG patients often exhibit bulbar involvement and respiratory crises, setting them apart from other MG subtypes.
Case presentation: A case of a 51-year-old male with MuSK MG that presented as isolated respiratory failure was compared to similar cases in the literature. The objectives were to explore the varied clinical presentations, treatment approaches, and outcomes, and to better understand the management of this subgroup of MG. The patient responded well to treatment with pyridostigmine, steroids, and intravenous immunoglobulins and immunosuppressive therapy.
Conclusion: A review of the literature revealed varied clinical presentations and treatment approaches among reported cases. Long-term prognosis appears favorable, requiring ongoing immunosuppressive management. Although the reported outcomes of MuSK MG patients with respiratory insufficiency show substantial heterogeneity, long-term prognosis appears favorable.
{"title":"Isolated Respiratory Failure as the Presenting Symptom of Muscle-Specific Kinase Myasthenia Gravis: A Case Report and Literature Review.","authors":"Hassan Doumiati, Ali Ezzeddine","doi":"10.1159/000540916","DOIUrl":"https://doi.org/10.1159/000540916","url":null,"abstract":"<p><strong>Introduction: </strong>Anti-MuSK antibodies obstruct MuSK binding sites, leading to acetylcholine receptor (AChR) displacement within the postsynaptic membrane. MuSK MG patients often exhibit bulbar involvement and respiratory crises, setting them apart from other MG subtypes.</p><p><strong>Case presentation: </strong>A case of a 51-year-old male with MuSK MG that presented as isolated respiratory failure was compared to similar cases in the literature. The objectives were to explore the varied clinical presentations, treatment approaches, and outcomes, and to better understand the management of this subgroup of MG. The patient responded well to treatment with pyridostigmine, steroids, and intravenous immunoglobulins and immunosuppressive therapy.</p><p><strong>Conclusion: </strong>A review of the literature revealed varied clinical presentations and treatment approaches among reported cases. Long-term prognosis appears favorable, requiring ongoing immunosuppressive management. Although the reported outcomes of MuSK MG patients with respiratory insufficiency show substantial heterogeneity, long-term prognosis appears favorable.</p>","PeriodicalId":9639,"journal":{"name":"Case Reports in Neurology","volume":"16 1","pages":"233-241"},"PeriodicalIF":0.6,"publicationDate":"2024-08-14","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11521485/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142544012","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}