Pub Date : 2024-12-01DOI: 10.1016/j.nerep.2024.100233
Mario B Prado , Karen Joy Adiao
For NMOSD and MOGAD, onset and relapses are often preceded by non-specific infections, but up until now no known microorganism has been identified to be strongly associated with these conditions. Syphilis as inciting event for NMOSD or MOGAD has only been reported once (Wilcox et al., 2008). This case report examines whether syphilis triggers autoimmune CNS condition or autoimmune CNS disease may cause false positive confirmatory treponemal test. The patient is a 30-year-old who came in with 8-month history of sudden onset spasticity and ataxia of bilateral lower extremities and blindness, confirmed to be transverse myelitis via imaging and optic neuritis by optic coherence tomography respectively. Anti-AQ4 and anti-MOG were negative, however, the patient repeatedly tested positive in FTA-ABS. We suspect that through molecular mimicry and bystander activation, there is a possibility that syphilis may have incited the onset and relapse of his autoimmune CNS demyelinating condition.
{"title":"Autoimmune demyelinating central nervous system disease in young male with persistently positive fluorescent treponemal antibody absorption test: A case report","authors":"Mario B Prado , Karen Joy Adiao","doi":"10.1016/j.nerep.2024.100233","DOIUrl":"10.1016/j.nerep.2024.100233","url":null,"abstract":"<div><div>For NMOSD and MOGAD, onset and relapses are often preceded by non-specific infections, but up until now no known microorganism has been identified to be strongly associated with these conditions. Syphilis as inciting event for NMOSD or MOGAD has only been reported once (Wilcox et al., 2008). This case report examines whether syphilis triggers autoimmune CNS condition or autoimmune CNS disease may cause false positive confirmatory treponemal test. The patient is a 30-year-old who came in with 8-month history of sudden onset spasticity and ataxia of bilateral lower extremities and blindness, confirmed to be transverse myelitis via imaging and optic neuritis by optic coherence tomography respectively. Anti-AQ4 and anti-MOG were negative, however, the patient repeatedly tested positive in FTA-ABS. We suspect that through molecular mimicry and bystander activation, there is a possibility that syphilis may have incited the onset and relapse of his autoimmune CNS demyelinating condition.</div></div>","PeriodicalId":100950,"journal":{"name":"Neuroimmunology Reports","volume":"6 ","pages":"Article 100233"},"PeriodicalIF":0.0,"publicationDate":"2024-12-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142744787","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 : 2024-11-13DOI: 10.1016/j.nerep.2024.100231
João Hugo Abdalla Santos , Ligia Fernandes Abdalla , Luana Catarina Marinho Serruya , Wolfgang Lucas Silva de Paula , Felipe Gomes Naveca
This study reports three cases of Guillain-Barré Syndrome (GBS) associated with SARS-CoV-2 infection at a tertiary hospital in Manaus, Brazil. The patients presented with atypical epidemiological profiles and manifestations, deviating from the classic presentation of the syndrome. In the first case, a 20-year-old patient exhibited lower limb paresthesias and respiratory symptoms. The second case involved a 29-year-old patient with a range of symptoms, including asthenia, diarrhea, and vertigo. The third case, a 51-year-old patient with a history of COVID-19, initially manifested dysautonomia and later developed weakness in the lower limbs. The administration of human immunoglobulin led to significant improvements in all cases within a period of one month to six weeks. Despite these outcomes, the underlying mechanisms varied between cases, highlighting the complexity of this association and emphasizing the need for further research to better understand the factors involved in the link between GBS and COVID-19.
{"title":"Guillain-Barré syndrome associated with COVID-19 infection: A case series","authors":"João Hugo Abdalla Santos , Ligia Fernandes Abdalla , Luana Catarina Marinho Serruya , Wolfgang Lucas Silva de Paula , Felipe Gomes Naveca","doi":"10.1016/j.nerep.2024.100231","DOIUrl":"10.1016/j.nerep.2024.100231","url":null,"abstract":"<div><div>This study reports three cases of Guillain-Barré Syndrome (GBS) associated with SARS-CoV-2 infection at a tertiary hospital in Manaus, Brazil. The patients presented with atypical epidemiological profiles and manifestations, deviating from the classic presentation of the syndrome. In the first case, a 20-year-old patient exhibited lower limb paresthesias and respiratory symptoms. The second case involved a 29-year-old patient with a range of symptoms, including asthenia, diarrhea, and vertigo. The third case, a 51-year-old patient with a history of COVID-19, initially manifested dysautonomia and later developed weakness in the lower limbs. The administration of human immunoglobulin led to significant improvements in all cases within a period of one month to six weeks. Despite these outcomes, the underlying mechanisms varied between cases, highlighting the complexity of this association and emphasizing the need for further research to better understand the factors involved in the link between GBS and COVID-19.</div></div>","PeriodicalId":100950,"journal":{"name":"Neuroimmunology Reports","volume":"6 ","pages":"Article 100231"},"PeriodicalIF":0.0,"publicationDate":"2024-11-13","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142698245","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 : 2024-11-01DOI: 10.1016/j.nerep.2024.100232
Nikoloz Karazanashvili , Monica M. Diaz , Jorge L. Almodovar , Bushra Javed , Stephanie Iyer , Irena Dujmovic Basuroski
We report a case of leptomeningeal and trigeminal nerve enhancement in an African American male patient with pediatric onset (age 17) relapsing-remitting multiple sclerosis (MS) during fingolimod rebound. The patient was clinically and radiologically stable while on fingolimod for 3.5 years, but developed disease rebound following fingolimod self-discontinuation. During this episode, several new T2/fluid-attenuated inversion recovery (FLAIR) lesions, enlarging T2/FLAIR lesions and contrast-enhancing lesions in the brain (supratentorial, cerebellum, brainstem) and spinal cord, enhancement of both trigeminal nerves and mild leptomeningeal enhancement (LME) around the brainstem were seen on brain magnetic resonance imaging (MRI). LME or cranial nerve enhancement (CNE) were not present on prior MRI studies, or on MRI studies performed after the patient recovered from fingolimod rebound. LME and CNE in MS may occur during rebound MS activity following fingolimod discontinuation as a transient phenomenon.
{"title":"Brain leptomeningeal enhancement and cranial nerve enhancement in a pediatric-onset multiple sclerosis patient during fingolimod rebound: A case report","authors":"Nikoloz Karazanashvili , Monica M. Diaz , Jorge L. Almodovar , Bushra Javed , Stephanie Iyer , Irena Dujmovic Basuroski","doi":"10.1016/j.nerep.2024.100232","DOIUrl":"10.1016/j.nerep.2024.100232","url":null,"abstract":"<div><div>We report a case of leptomeningeal and trigeminal nerve enhancement in an African American male patient with pediatric onset (age 17) relapsing-remitting multiple sclerosis (MS) during fingolimod rebound. The patient was clinically and radiologically stable while on fingolimod for 3.5 years, but developed disease rebound following fingolimod self-discontinuation. During this episode, several new T2/fluid-attenuated inversion recovery (FLAIR) lesions, enlarging T2/FLAIR lesions and contrast-enhancing lesions in the brain (supratentorial, cerebellum, brainstem) and spinal cord, enhancement of both trigeminal nerves and mild leptomeningeal enhancement (LME) around the brainstem were seen on brain magnetic resonance imaging (MRI). LME or cranial nerve enhancement (CNE) were not present on prior MRI studies, or on MRI studies performed after the patient recovered from fingolimod rebound. LME and CNE in MS may occur during rebound MS activity following fingolimod discontinuation as a transient phenomenon.</div></div>","PeriodicalId":100950,"journal":{"name":"Neuroimmunology Reports","volume":"6 ","pages":"Article 100232"},"PeriodicalIF":0.0,"publicationDate":"2024-11-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142593072","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}
Collapsin response-mediator protein 5 (CRMP-5) is a cytoplasmic regulator of neurite outgrowth. Antibodies against CRMP-5 are associated with various neurologic diseases, including myelitis. Underlying malignancy is present in 70 – 90 % of patients with CRMP-5 autoimmunity. We present two patients with myelitis and transiently elevated anti-CRMP-5 without evidence of malignancy and discuss the relevance of the antibody in these cases.
Case Report
1. A 44-year-old male presented with symptoms of subacute thoracic myelitis and was found to have a persistently enhancing cord lesion on MRI. Serum anti-CRMP-5 antibody levels were initially elevated but absent on subsequent testing. Three whole-body PET scans during a three-year follow-up failed to uncover a malignancy. Neurologic condition improved on steroids. 2. A 65-year-old female presented with symptoms of a cervical myelitis followed by left facial weakness. MRI demonstrated multiple brain and spinal cord lesions as well as evidence of cranial neuritis, which persisted despite pulse steroid courses. Elevated serum anti-CRMP-5 was noted nine months after symptom onset. Malignancy workup failed to identify neoplasm and anti-CRMP-5 level subsequently seroreverted. Clinical and radiographic improvement occurred over several years of follow-up.
Conclusion
CRMP-5 autoantibody is a marker for paraneoplastic autoimmune neurologic syndromes. However, these two cases illustrate the uncertainty regarding its significance, as anti-CRMP-5 was only transiently elevated and not associated with an underlying malignancy. The possibilities that anti-CRMP-5 autoantibodies were an incidental or false-positive finding are discussed.
{"title":"Transiently elevated anti-CRMP-5 autoantibodies in two patients with myelitis without underlying malignancy","authors":"Kennan Negrete , Zeinab Awada , Asaff Harel , Ilya Kister","doi":"10.1016/j.nerep.2024.100229","DOIUrl":"10.1016/j.nerep.2024.100229","url":null,"abstract":"<div><h3>Introduction</h3><div>Collapsin response-mediator protein 5 (CRMP-5) is a cytoplasmic regulator of neurite outgrowth. Antibodies against CRMP-5 are associated with various neurologic diseases, including myelitis. Underlying malignancy is present in 70 – 90 % of patients with CRMP-5 autoimmunity. We present two patients with myelitis and transiently elevated anti-CRMP-5 without evidence of malignancy and discuss the relevance of the antibody in these cases.</div></div><div><h3>Case Report</h3><div>1. A 44-year-old male presented with symptoms of subacute thoracic myelitis and was found to have a persistently enhancing cord lesion on MRI. Serum anti-CRMP-5 antibody levels were initially elevated but absent on subsequent testing. Three whole-body PET scans during a three-year follow-up failed to uncover a malignancy. Neurologic condition improved on steroids. 2. A 65-year-old female presented with symptoms of a cervical myelitis followed by left facial weakness. MRI demonstrated multiple brain and spinal cord lesions as well as evidence of cranial neuritis, which persisted despite pulse steroid courses. Elevated serum anti-CRMP-5 was noted nine months after symptom onset. Malignancy workup failed to identify neoplasm and anti-CRMP-5 level subsequently seroreverted. Clinical and radiographic improvement occurred over several years of follow-up.</div></div><div><h3>Conclusion</h3><div>CRMP-5 autoantibody is a marker for paraneoplastic autoimmune neurologic syndromes. However, these two cases illustrate the uncertainty regarding its significance, as anti-CRMP-5 was only transiently elevated and not associated with an underlying malignancy. The possibilities that anti-CRMP-5 autoantibodies were an incidental or false-positive finding are discussed.</div></div>","PeriodicalId":100950,"journal":{"name":"Neuroimmunology Reports","volume":"6 ","pages":"Article 100229"},"PeriodicalIF":0.0,"publicationDate":"2024-10-30","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142539516","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 : 2024-10-14DOI: 10.1016/j.nerep.2024.100228
Hamna Khan , Michelle Maynard , Ahmed Z. Obeidat
Background
Despite its rarity, concerns about cardiac complications could be linked to ofatumumab in line with its cardiac risks in non-MS treatments. Here, we provide a first-time report of two instances of acute myocardial events in women under 50 temporally associated with ofatumumab for the treatment of multiple sclerosis (MS). Both patients were also on stimulant medications, suggesting that clinicians should take caution when prescribing anti-CD20 agents to patients with cardiovascular risk factors or concomitant use of stimulants.
Discussion
Further research is needed to confirm and quantify the association between anti-CD20 agents in use in patients with cardiovascular risk factors on concomitant use of stimulants.
{"title":"Cardiac events in the setting of ofatumumab treatment: An association or A Co-incidence?","authors":"Hamna Khan , Michelle Maynard , Ahmed Z. Obeidat","doi":"10.1016/j.nerep.2024.100228","DOIUrl":"10.1016/j.nerep.2024.100228","url":null,"abstract":"<div><h3>Background</h3><div>Despite its rarity, concerns about cardiac complications could be linked to ofatumumab in line with its cardiac risks in non-MS treatments. Here, we provide a first-time report of two instances of acute myocardial events in women under 50 temporally associated with ofatumumab for the treatment of multiple sclerosis (MS). Both patients were also on stimulant medications, suggesting that clinicians should take caution when prescribing anti-CD20 agents to patients with cardiovascular risk factors or concomitant use of stimulants.</div></div><div><h3>Discussion</h3><div>Further research is needed to confirm and quantify the association between anti-CD20 agents in use in patients with cardiovascular risk factors on concomitant use of stimulants.</div></div>","PeriodicalId":100950,"journal":{"name":"Neuroimmunology Reports","volume":"6 ","pages":"Article 100228"},"PeriodicalIF":0.0,"publicationDate":"2024-10-14","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142527059","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 : 2024-09-14DOI: 10.1016/j.nerep.2024.100227
S Gandelman , S Parauda , C Dohle
Background
New-onset Neuromyelitis Optica Spectrum Disorder (NMOSD) has rarely been reported in adults over age 70. We describe a severe presentation in a 77-year-old female.
Case
The patient presented with left arm numbness, followed by left leg numbness and bilateral optic neuritis. Neuroimaging showed a C2-T1 lesion with C4-C7 holocord enhancement, and bilateral optic nerve enhancement extending to the optic chiasm. Serum testing detected anti-aquaporin-4 antibodies. She was left with dyschromatopsia, myelopathy, and ambulatory dysfunction. She had a history notable for rheumatoid arthritis, treated with rituximab, which had been discontinued six years prior to presentation.
Discussion
Very-late-onset NMOSD encompasses a unique cohort of patients with significant immunogenicity despite immunosenescence. Limited data suggest diagnostic delays, severe morbidity, and decreased utilization of disease-modifying therapies.
{"title":"Very late-onset neuromyelitis optica presenting with simultaneous bilateral optic neuritis and myelitis: A case report","authors":"S Gandelman , S Parauda , C Dohle","doi":"10.1016/j.nerep.2024.100227","DOIUrl":"10.1016/j.nerep.2024.100227","url":null,"abstract":"<div><h3>Background</h3><p>New-onset Neuromyelitis Optica Spectrum Disorder (NMOSD) has rarely been reported in adults over age 70. We describe a severe presentation in a 77-year-old female.</p></div><div><h3>Case</h3><p>The patient presented with left arm numbness, followed by left leg numbness and bilateral optic neuritis. Neuroimaging showed a C2-T1 lesion with C4-C7 holocord enhancement, and bilateral optic nerve enhancement extending to the optic chiasm. Serum testing detected anti-aquaporin-4 antibodies. She was left with dyschromatopsia, myelopathy, and ambulatory dysfunction. She had a history notable for rheumatoid arthritis, treated with rituximab, which had been discontinued six years prior to presentation.</p></div><div><h3>Discussion</h3><p>Very-late-onset NMOSD encompasses a unique cohort of patients with significant immunogenicity despite immunosenescence. Limited data suggest diagnostic delays, severe morbidity, and decreased utilization of disease-modifying therapies.</p></div>","PeriodicalId":100950,"journal":{"name":"Neuroimmunology Reports","volume":"6 ","pages":"Article 100227"},"PeriodicalIF":0.0,"publicationDate":"2024-09-14","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.sciencedirect.com/science/article/pii/S2667257X24000287/pdfft?md5=911d5b73fdd8fbd05a4f3ee1c3b475a6&pid=1-s2.0-S2667257X24000287-main.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142241537","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 : 2024-09-08DOI: 10.1016/j.nerep.2024.100226
Krithika Arrabothu , Srinivas Govindan , Wilson Rodriguez , Christopher Tapia , Robert L. White III
Autoimmune glial fibrillary acidic protein (GFAP) astrocytopathy is a novel central nervous system disorder characterized by subacute neuropsychiatric symptom development and rapid remission with corticosteroid therapy. In this paper, we describe a case of GFAP encephalitis found to have renal cell carcinoma. The patient was a 71-year-old man with a history of transient ischemic attacks, severe diabetic retinopathy and monoclonal gammopathy of undetermined significance, who presented with subacute cognitive decline. Anti-GFAP antibodies were detected in the cerebrospinal fluid. Computer Tomography (CT) of chest, abdomen and pelvis identified a renal mass that was resected. Subsequent pathology confirmed clear cell renal cell carcinoma. The patient was treated with corticosteroids with cognitive recovery near to baseline. In conclusion, we encourage the early consideration of autoimmune GFAP astrocytopathy for patients with clinical manifestation of subacute neuropsychiatric symptoms after other common conditions have been ruled out, as it usually has a great response to immunosuppressive therapy. In addition, this case highlights the importance of full body scan to identify possible underlying malignancy as a possible precipitating factor.
{"title":"A case of autoimmune glial fibrillary acidic protein astrocytopathy with concurrent renal cell carcinoma","authors":"Krithika Arrabothu , Srinivas Govindan , Wilson Rodriguez , Christopher Tapia , Robert L. White III","doi":"10.1016/j.nerep.2024.100226","DOIUrl":"10.1016/j.nerep.2024.100226","url":null,"abstract":"<div><p>Autoimmune glial fibrillary acidic protein (GFAP) astrocytopathy is a novel central nervous system disorder characterized by subacute neuropsychiatric symptom development and rapid remission with corticosteroid therapy. In this paper, we describe a case of GFAP encephalitis found to have renal cell carcinoma. The patient was a 71-year-old man with a history of transient ischemic attacks, severe diabetic retinopathy and monoclonal gammopathy of undetermined significance, who presented with subacute cognitive decline. Anti-GFAP antibodies were detected in the cerebrospinal fluid. Computer Tomography (CT) of chest, abdomen and pelvis identified a renal mass that was resected. Subsequent pathology confirmed clear cell renal cell carcinoma. The patient was treated with corticosteroids with cognitive recovery near to baseline. In conclusion, we encourage the early consideration of autoimmune GFAP astrocytopathy for patients with clinical manifestation of subacute neuropsychiatric symptoms after other common conditions have been ruled out, as it usually has a great response to immunosuppressive therapy. In addition, this case highlights the importance of full body scan to identify possible underlying malignancy as a possible precipitating factor.</p></div>","PeriodicalId":100950,"journal":{"name":"Neuroimmunology Reports","volume":"6 ","pages":"Article 100226"},"PeriodicalIF":0.0,"publicationDate":"2024-09-08","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.sciencedirect.com/science/article/pii/S2667257X24000275/pdfft?md5=2065be24e880ef5864893cd0798d0453&pid=1-s2.0-S2667257X24000275-main.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142241536","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 : 2024-08-24DOI: 10.1016/j.nerep.2024.100225
Bhanu B Gowda , Snehal Shah , Rahul Lakshmanan , Jonathan Silberstein
{"title":"Hypertrophic polyneuropathy in CMV related GBS in a paediatric patient","authors":"Bhanu B Gowda , Snehal Shah , Rahul Lakshmanan , Jonathan Silberstein","doi":"10.1016/j.nerep.2024.100225","DOIUrl":"10.1016/j.nerep.2024.100225","url":null,"abstract":"","PeriodicalId":100950,"journal":{"name":"Neuroimmunology Reports","volume":"6 ","pages":"Article 100225"},"PeriodicalIF":0.0,"publicationDate":"2024-08-24","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.sciencedirect.com/science/article/pii/S2667257X24000263/pdfft?md5=40986e4a76b366f1461794031233cb24&pid=1-s2.0-S2667257X24000263-main.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142097485","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 : 2024-07-29DOI: 10.1016/j.nerep.2024.100224
Nipith Charoenngam , Michael F. Holick
A 36-year-old male presented with 2 months of left-eye visual disturbance and was diagnosed with optic neuritis due to probable multiple sclerosis (MS). He was advised to undergo periodic ophthalmology follow-up without immunosuppressive treatment. Due to persistent symptoms, he expressed interest in very high-dose vitamin D3 therapy of 54,000 IUs/day (1,000 IUs/kg/day) along with a zero-calcium diet. After starting the therapy, he experienced sustained symptomatic improvement of visual symptoms over 4 years, along with radiological stability of the optic neuritis lesion without developing hypercalcemia. This case supports the potential therapeutic efficacy of very high-dose vitamin D for MS.
{"title":"Resolution of optic neuritis and probable multiple sclerosis after long-term ingestion of very high doses of vitamin D3: A case report","authors":"Nipith Charoenngam , Michael F. Holick","doi":"10.1016/j.nerep.2024.100224","DOIUrl":"10.1016/j.nerep.2024.100224","url":null,"abstract":"<div><p>A 36-year-old male presented with 2 months of left-eye visual disturbance and was diagnosed with optic neuritis due to probable multiple sclerosis (MS). He was advised to undergo periodic ophthalmology follow-up without immunosuppressive treatment. Due to persistent symptoms, he expressed interest in very high-dose vitamin D<sub>3</sub> therapy of 54,000 IUs/day (1,000 IUs/kg/day) along with a zero-calcium diet. After starting the therapy, he experienced sustained symptomatic improvement of visual symptoms over 4 years, along with radiological stability of the optic neuritis lesion without developing hypercalcemia. This case supports the potential therapeutic efficacy of very high-dose vitamin D for MS.</p></div>","PeriodicalId":100950,"journal":{"name":"Neuroimmunology Reports","volume":"6 ","pages":"Article 100224"},"PeriodicalIF":0.0,"publicationDate":"2024-07-29","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.sciencedirect.com/science/article/pii/S2667257X24000251/pdfft?md5=7b1df6ff327984b259e7f1d7ec65313f&pid=1-s2.0-S2667257X24000251-main.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141952470","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 : 2024-07-29DOI: 10.1016/j.nerep.2024.100223
Antonella Frattari , Maria Vittoria De Angelis , Mariangela Battilana , Ennio Polilli , Alessandro Ferrieri , Daniela Onofrillo , Nicole Santoro , Antonella Sau , Anna D'Andreagiovanni , Pierluigi Tocco , Donatella Bosco , Giustino Parruti
Varicella is a benign, self-limiting disease, but both primary VZV infection and VZV reactivation can be life-threatening in immunocompromised children, due to CNS and systemic dissemination of the virus. Reactivation of the vaccine-type VZV (vOka) has been reported sporadically, although SARS COV2 infection may have recently played a role in facilitating VZV reactivation. Here we report on the case of a young boy with Acute Lymphocytic Leukemia, in remission after induction chemotherapy, and recent SARS COV 2 infection. He developed VZV encephalitis due to massive reactivation of VZV virus, complicated by PRES, diagnosed with brain MRI, and Super Refractory Status Epilepticus, lasting until substantial suppression of VZV replication in the CNS. We also report and discuss the role of the remarkably augmented renal clearance persistently observed in our patient, complicating CNS involvement and making both antiviral and antiepileptic treatments more difficult to manage. Effective neuroprotection was completed by physical hypothermia and infusion of IVIG and steroids. The patient obtained complete functional recovery, with reversion of MRI signs of occipital involvement at presentation, at 6-month follow-up. Intense and daily interplay of intensivists, neurologists, hematologists and infectious disease experts likely made his uneventful recovery possible, combining all skills necessary to manage his unusual and complex clinical picture.
{"title":"High viral load VZV encephalitis complicated by super refractory status epilepticus in a vaccinated child with in Acute Lymphocytic Leukemia: Case report and review of the literature","authors":"Antonella Frattari , Maria Vittoria De Angelis , Mariangela Battilana , Ennio Polilli , Alessandro Ferrieri , Daniela Onofrillo , Nicole Santoro , Antonella Sau , Anna D'Andreagiovanni , Pierluigi Tocco , Donatella Bosco , Giustino Parruti","doi":"10.1016/j.nerep.2024.100223","DOIUrl":"10.1016/j.nerep.2024.100223","url":null,"abstract":"<div><p>Varicella is a benign, self-limiting disease, but both primary VZV infection and VZV reactivation can be life-threatening in immunocompromised children, due to CNS and systemic dissemination of the virus. Reactivation of the vaccine-type VZV (vOka) has been reported sporadically, although SARS COV2 infection may have recently played a role in facilitating VZV reactivation. Here we report on the case of a young boy with Acute Lymphocytic Leukemia, in remission after induction chemotherapy, and recent SARS COV 2 infection. He developed VZV encephalitis due to massive reactivation of VZV virus, complicated by PRES, diagnosed with brain MRI, and Super Refractory Status Epilepticus, lasting until substantial suppression of VZV replication in the CNS. We also report and discuss the role of the remarkably augmented renal clearance persistently observed in our patient, complicating CNS involvement and making both antiviral and antiepileptic treatments more difficult to manage. Effective neuroprotection was completed by physical hypothermia and infusion of IVIG and steroids. The patient obtained complete functional recovery, with reversion of MRI signs of occipital involvement at presentation, at 6-month follow-up. Intense and daily interplay of intensivists, neurologists, hematologists and infectious disease experts likely made his uneventful recovery possible, combining all skills necessary to manage his unusual and complex clinical picture.</p></div>","PeriodicalId":100950,"journal":{"name":"Neuroimmunology Reports","volume":"6 ","pages":"Article 100223"},"PeriodicalIF":0.0,"publicationDate":"2024-07-29","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.sciencedirect.com/science/article/pii/S2667257X2400024X/pdfft?md5=78f3514e7bb97f38fd7091c4f3ca5aaf&pid=1-s2.0-S2667257X2400024X-main.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141952469","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}
扫码关注我们
求助内容：
应助结果提醒方式：
京公网安备 11010802042870号