Purpose: To evaluate the effect of periocular filler injection on the incidence of cerebral venous thrombosis (CVT). Case Report: A 41-year-old woman without a prior medical history experienced a severe headache and subsequent seizures following an eye filler injection. Diagnosis of CVT was confirmed through brain magnetic resonance venography (MRV), revealing thrombosis in the left transverse and upper sagittal sinuses. The patient was treated with intravenous heparin and oral warfarin, leading to improvement and discharge in good condition after 10 days of hospitalization. Conclusion: This case underscores the potential risk of CVT following periocular filler injections, emphasizing the need for awareness and preventive measures among medical professionals.
{"title":"Cerebral Venous Thrombosis Following Periocular Filler Injection-A Case Report and Review of Literature.","authors":"Reza Asgari, Mohammadamin Bazzazan, Fateme Jafari, Hossein Mozhdehipanah","doi":"10.1155/crnm/9363655","DOIUrl":"10.1155/crnm/9363655","url":null,"abstract":"<p><p><b>Purpose:</b> To evaluate the effect of periocular filler injection on the incidence of cerebral venous thrombosis (CVT). <b>Case Report:</b> A 41-year-old woman without a prior medical history experienced a severe headache and subsequent seizures following an eye filler injection. Diagnosis of CVT was confirmed through brain magnetic resonance venography (MRV), revealing thrombosis in the left transverse and upper sagittal sinuses. The patient was treated with intravenous heparin and oral warfarin, leading to improvement and discharge in good condition after 10 days of hospitalization. <b>Conclusion:</b> This case underscores the potential risk of CVT following periocular filler injections, emphasizing the need for awareness and preventive measures among medical professionals.</p>","PeriodicalId":9615,"journal":{"name":"Case Reports in Neurological Medicine","volume":"2025 ","pages":"9363655"},"PeriodicalIF":0.9,"publicationDate":"2025-09-03","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12422856/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145039169","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 : 2025-09-02eCollection Date: 2025-01-01DOI: 10.1155/crnm/9145358
Aimen Vanood, Ruth H McConnell, Bart M Demaerschalk
Tremor is a common symptom encountered in outpatient practice, particularly within a neurology movement disorder clinic. However, tremor can also be pertinent to inpatient medicine and can warrant emergent evaluation. We describe a case of a 72-year-old female who developed acute onset postural head tremor without appendicular tremor during a hospital admission for chest pain and leukostasis. This case explores the localization of tremor and the differential diagnosis of head tremor specifically. Additionally, this report serves as a reminder to consider ischemic stroke in the diagnostic evaluation of acute onset neurological symptoms.
{"title":"The Cerebellum in Poststroke Movement Disorders: A Case of Head Tremor Indicating a Cerebrovascular Emergency.","authors":"Aimen Vanood, Ruth H McConnell, Bart M Demaerschalk","doi":"10.1155/crnm/9145358","DOIUrl":"10.1155/crnm/9145358","url":null,"abstract":"<p><p>Tremor is a common symptom encountered in outpatient practice, particularly within a neurology movement disorder clinic. However, tremor can also be pertinent to inpatient medicine and can warrant emergent evaluation. We describe a case of a 72-year-old female who developed acute onset postural head tremor without appendicular tremor during a hospital admission for chest pain and leukostasis. This case explores the localization of tremor and the differential diagnosis of head tremor specifically. Additionally, this report serves as a reminder to consider ischemic stroke in the diagnostic evaluation of acute onset neurological symptoms.</p>","PeriodicalId":9615,"journal":{"name":"Case Reports in Neurological Medicine","volume":"2025 ","pages":"9145358"},"PeriodicalIF":0.9,"publicationDate":"2025-09-02","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12419915/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145039130","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 : 2025-08-31eCollection Date: 2025-01-01DOI: 10.1155/crnm/3106627
Andrew R Pachner
This patient had CNS demyelination in the context of stable undifferentiated connective tissue disease (sUCTD) and a relatively benign course despite minimal treatment. Her course is consistent with the course of similar patients described in the literature, which supports a relatively conservative approach to treatment in this group of patients.
{"title":"Stable Undifferentiated Connective Tissue Disease and CNS Demyelination: A Case Report and a Literature Review.","authors":"Andrew R Pachner","doi":"10.1155/crnm/3106627","DOIUrl":"10.1155/crnm/3106627","url":null,"abstract":"<p><p>This patient had CNS demyelination in the context of stable undifferentiated connective tissue disease (sUCTD) and a relatively benign course despite minimal treatment. Her course is consistent with the course of similar patients described in the literature, which supports a relatively conservative approach to treatment in this group of patients.</p>","PeriodicalId":9615,"journal":{"name":"Case Reports in Neurological Medicine","volume":"2025 ","pages":"3106627"},"PeriodicalIF":0.9,"publicationDate":"2025-08-31","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12414617/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145022871","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}
Longitudinal extensive transverse myelitis (LETM) is a rare adverse event after vaccination. We present a case of severe myelitis in a 76-year-old man with positive anti-recoverin antibodies that occurred one week after RSVPreF3 vaccination against respiratory syncytial virus (RSV). The patient presented with severe spastic paraparesis, urinary retention, postural tremor of the upper extremities, hypesthesia, severely impaired proprioception and vibration sense in the lower extremities, and tonic spasms of the lower extremities. An MRI of the spine revealed a C3-T9 LETM, with inflammatory cerebrospinal fluid (CSF). The patient was found to have positive anti-recoverin antibodies in serum and CSF. While the patient had an initial improvement on high-dose intravenous steroids, he failed to respond to plasmapheresis. Subsequently, he received intravenous immunoglobulins with mild improvement of his symptoms. The patient's symptoms could be attributed to vaccine-induced inflammatory syndrome. The relationship between anti-recoverin antibodies and central nervous system involvement is likely due to the sharing of epitopes between recoverin and endogenous antigens of the central nervous system. The association between RSV vaccination and LETM has not been previously reported.
{"title":"Longitudinal Extensive Transverse Myelitis After Respiratory Syncytial Virus Vaccination With Positive Anti-Recoverin Antibodies.","authors":"Stefania Kalampokini, Ntouigou Fountouktsi, Martha Spilioti, Stefanos Finitsis, Vasilios K Kimiskidis","doi":"10.1155/crnm/6597450","DOIUrl":"10.1155/crnm/6597450","url":null,"abstract":"<p><p>Longitudinal extensive transverse myelitis (LETM) is a rare adverse event after vaccination. We present a case of severe myelitis in a 76-year-old man with positive anti-recoverin antibodies that occurred one week after RSVPreF3 vaccination against respiratory syncytial virus (RSV). The patient presented with severe spastic paraparesis, urinary retention, postural tremor of the upper extremities, hypesthesia, severely impaired proprioception and vibration sense in the lower extremities, and tonic spasms of the lower extremities. An MRI of the spine revealed a C3-T9 LETM, with inflammatory cerebrospinal fluid (CSF). The patient was found to have positive anti-recoverin antibodies in serum and CSF. While the patient had an initial improvement on high-dose intravenous steroids, he failed to respond to plasmapheresis. Subsequently, he received intravenous immunoglobulins with mild improvement of his symptoms. The patient's symptoms could be attributed to vaccine-induced inflammatory syndrome. The relationship between anti-recoverin antibodies and central nervous system involvement is likely due to the sharing of epitopes between recoverin and endogenous antigens of the central nervous system. The association between RSV vaccination and LETM has not been previously reported.</p>","PeriodicalId":9615,"journal":{"name":"Case Reports in Neurological Medicine","volume":"2025 ","pages":"6597450"},"PeriodicalIF":0.9,"publicationDate":"2025-08-30","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12413940/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145013957","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 : 2025-08-20eCollection Date: 2025-01-01DOI: 10.1155/crnm/5382669
Karen Dos Santos Ferreira, Ana Miriam Velly
Background: Acute confusional migraine (ACM) is a migraine variant manifesting with confusion, agitation, disorientation, altered mental status, and/or memory deficits. ACM has notably been excluded from the International Classification of Headache Disorders (ICHD-3 β), despite previous literature describing 120 cases and proposing a standardized classification. Considering these findings, clinicians must be aware of this condition as it can be confounded with other serious health conditions (e.g., stroke, encephalitis, and epilepsy). Objective: Herein, we describe three cases with altered consciousness during a migraine attack, discussing diagnostic criteria, treatment, prognostic implications, and future perspectives. Results: The first case, an 18-year-old male, presented to the emergency room with migraine with visual aura, followed by confusion and torpor. After 24 h, he was completely recovered without deficits. In the second case, a 30-year-old woman in puerperium presented with a visual aura followed by headache, confusion, and disorientation. She recovered her conscience without deficits. The third patient, a 36-year-old woman, showed up in the emergency room presenting migraine, hemiplegia, and confusion. She recovered without deficits after 8 days. Finally, the genetic panel confirmed familial hemiplegic migraine (for the third patient). All the tests, including brain computed tomography (CT) scan, angioscan, brain magnetic resonance imaging (MRI), lumbar puncture (LP), and toxicological workup, had normal results for all three patients. They were treated for migraine with long-term control. Conclusion: ACM is a significant condition that can be mistaken for other serious health issues. Health professionals need to be better informed about their diagnosis and management strategies. Therefore, we proposed criteria to include ACM in the ICHD-3 β classification, and we emphasize the need for future studies to improve understanding and treatment of this condition.
{"title":"Acute Confusional Migraine: Case Reports and Discussion as a Distinct Entity.","authors":"Karen Dos Santos Ferreira, Ana Miriam Velly","doi":"10.1155/crnm/5382669","DOIUrl":"10.1155/crnm/5382669","url":null,"abstract":"<p><p><b>Background:</b> Acute confusional migraine (ACM) is a migraine variant manifesting with confusion, agitation, disorientation, altered mental status, and/or memory deficits. ACM has notably been excluded from the International Classification of Headache Disorders (ICHD-3 β), despite previous literature describing 120 cases and proposing a standardized classification. Considering these findings, clinicians must be aware of this condition as it can be confounded with other serious health conditions (e.g., stroke, encephalitis, and epilepsy). <b>Objective:</b> Herein, we describe three cases with altered consciousness during a migraine attack, discussing diagnostic criteria, treatment, prognostic implications, and future perspectives. <b>Results:</b> The first case, an 18-year-old male, presented to the emergency room with migraine with visual aura, followed by confusion and torpor. After 24 h, he was completely recovered without deficits. In the second case, a 30-year-old woman in puerperium presented with a visual aura followed by headache, confusion, and disorientation. She recovered her conscience without deficits. The third patient, a 36-year-old woman, showed up in the emergency room presenting migraine, hemiplegia, and confusion. She recovered without deficits after 8 days. Finally, the genetic panel confirmed familial hemiplegic migraine (for the third patient). All the tests, including brain computed tomography (CT) scan, angioscan, brain magnetic resonance imaging (MRI), lumbar puncture (LP), and toxicological workup, had normal results for all three patients. They were treated for migraine with long-term control. <b>Conclusion:</b> ACM is a significant condition that can be mistaken for other serious health issues. Health professionals need to be better informed about their diagnosis and management strategies. Therefore, we proposed criteria to include ACM in the ICHD-3 β classification, and we emphasize the need for future studies to improve understanding and treatment of this condition.</p>","PeriodicalId":9615,"journal":{"name":"Case Reports in Neurological Medicine","volume":"2025 ","pages":"5382669"},"PeriodicalIF":0.9,"publicationDate":"2025-08-20","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12390517/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144943216","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}
Patients with acute branch atheromatous disease often experience early neurological deterioration (END) in the first 24-h period, even after intravenous alteplase. Three cases treated with cilostazol, a phosphodiesterase 3-inhibitor, prior to intravenous alteplase with the aim of mitigating END are described. All three patients had no bleeding complications and good outcomes at 3 months, although two patients showed END within 24 h after intravenous alteplase.
{"title":"Administration of Cilostazol Prior to Intravenous Alteplase for Acute Branch Atheromatous Disease: A Report of Three Cases.","authors":"Eijirou Tanaka, Yoshinari Nagakane, Daiki Fukunaga, Daisuke Nakashima, Takehiro Yamada","doi":"10.1155/crnm/3508206","DOIUrl":"10.1155/crnm/3508206","url":null,"abstract":"<p><p>Patients with acute branch atheromatous disease often experience early neurological deterioration (END) in the first 24-h period, even after intravenous alteplase. Three cases treated with cilostazol, a phosphodiesterase 3-inhibitor, prior to intravenous alteplase with the aim of mitigating END are described. All three patients had no bleeding complications and good outcomes at 3 months, although two patients showed END within 24 h after intravenous alteplase.</p>","PeriodicalId":9615,"journal":{"name":"Case Reports in Neurological Medicine","volume":"2025 ","pages":"3508206"},"PeriodicalIF":0.9,"publicationDate":"2025-08-11","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12360887/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144882275","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}
Vitamin B12 deficiency can cause subacute combined degeneration (SACD) by disrupting myelin synthesis, leading to spinal cord degeneration. We present a unique case of SACD featuring symmetrical pseudoathetosis characterized by involuntary, slow, and writhing movements resulting from proprioceptive sensory impairment, which disrupts the brain's ability to accurately perceive limb position and movement and pulmonary embolism due to elevated homocysteine levels. A 34-year-old male presented with chest pain, generalized weakness, and numbness in his hands and feet. Two months prior, he experienced sharp chest pain, followed by progressive numbness and weakness in his upper and lower extremities. Neurological examination revealed no nuchal stiffness, normal cranial nerve function, and impaired light touch and vibration sensation in the lower extremities. Tremulousness in the hands and feet, suggestive of pseudoathetosis, had been present for 6 months. Laboratory tests confirmed severe vitamin B12 deficiency (< 159 pg/mL), elevated homocysteine, and pancytopenia. MRI of the spine showed hyperintense signals consistent with SACD, and a chest CT revealed a large saddle pulmonary embolus. Pernicious anemia was confirmed as the cause of vitamin B12 deficiency. The patient was treated with intravenous vitamin B12, leading to significant neurological improvement. This case is the first documented instance of SACD presenting with symmetrical pseudoathetosis in all four extremities. Recognizing this rare clinical sign is essential, as it can guide early diagnosis and treatment. In addition, hyperhomocysteinemia associated with vitamin B12 deficiency is a significant risk factor for thromboembolism, underscoring the need for screening in patients with unexplained thrombotic events.
{"title":"Symmetric Pseudoathetosis of Hands and Feet: A Rare Manifestation of Subacute Combined Cord Degeneration With Life-Threatening Thromboembolic Risk.","authors":"Ramsha Siddiqui, Johanna Canenguez, Nithisha Thatikonda, Awab Elaneem, Fernandez Jorge Rodriguez","doi":"10.1155/crnm/1862715","DOIUrl":"10.1155/crnm/1862715","url":null,"abstract":"<p><p>Vitamin B12 deficiency can cause subacute combined degeneration (SACD) by disrupting myelin synthesis, leading to spinal cord degeneration. We present a unique case of SACD featuring symmetrical pseudoathetosis characterized by involuntary, slow, and writhing movements resulting from proprioceptive sensory impairment, which disrupts the brain's ability to accurately perceive limb position and movement and pulmonary embolism due to elevated homocysteine levels. A 34-year-old male presented with chest pain, generalized weakness, and numbness in his hands and feet. Two months prior, he experienced sharp chest pain, followed by progressive numbness and weakness in his upper and lower extremities. Neurological examination revealed no nuchal stiffness, normal cranial nerve function, and impaired light touch and vibration sensation in the lower extremities. Tremulousness in the hands and feet, suggestive of pseudoathetosis, had been present for 6 months. Laboratory tests confirmed severe vitamin B12 deficiency (< 159 pg/mL), elevated homocysteine, and pancytopenia. MRI of the spine showed hyperintense signals consistent with SACD, and a chest CT revealed a large saddle pulmonary embolus. Pernicious anemia was confirmed as the cause of vitamin B12 deficiency. The patient was treated with intravenous vitamin B12, leading to significant neurological improvement. This case is the first documented instance of SACD presenting with symmetrical pseudoathetosis in all four extremities. Recognizing this rare clinical sign is essential, as it can guide early diagnosis and treatment. In addition, hyperhomocysteinemia associated with vitamin B12 deficiency is a significant risk factor for thromboembolism, underscoring the need for screening in patients with unexplained thrombotic events.</p>","PeriodicalId":9615,"journal":{"name":"Case Reports in Neurological Medicine","volume":"2025 ","pages":"1862715"},"PeriodicalIF":0.9,"publicationDate":"2025-08-04","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12339141/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144820673","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 : 2025-07-21eCollection Date: 2025-01-01DOI: 10.1155/crnm/6652600
Nader Pahlevan, Delora E Denney, Ezekiel Gonzalez-Fernandez, Oriana Sanchez, Mark Anderson
Introduction: Parsonage-Turner syndrome (PTS), also known as idiopathic brachial plexopathy, is an uncommon peripheral neuropathy, and the presentation of bilateral PTS is even rarer. Hemophagocytic lymphohistiocytosis (HLH) is a hyperinflammatory disorder that is normally considered hematologic but can involve the nervous system in up to 70% of cases. Case Presentation: A 56-year-old Caucasian female with a history of SLE, rheumatoid arthritis, Sjogren's disease, and Hashimoto's thyroiditis, but no prior neurologic history, was admitted for the management of isolated thrombocytopenia, diagnosed with HLH, and then developed bilateral upper extremity pain, weakness, and numbness. A diagnosis of bilateral PTS was supported by magnetic resonance imaging (MRI) findings of mild symmetric increased enhancement in the bilateral shoulder muscles, and electromyography/nerve conduction study (EMG/NCS) revealed active denervation in the shoulder muscles bilaterally. The patient was started on methylprednisolone 1000 mg for three days, followed by a long steroid taper along with physical/occupational therapy with significant improvement of her symptoms. Conclusion: It is important to maintain a high index of suspicion for PTS in patients with new-onset shoulder or upper arm pain, weakness, and sensory deficits, even if findings are bilateral. Concomitant inflammatory disorders, infection, and recent surgeries/procedures should prompt a high degree of suspicion of this disorder, and the use of relevant diagnostics, such as MRI brachial plexus and EMG/NCS, should help guide diagnosis, as this condition is very responsive to treatment.
{"title":"Bilateral Parsonage-Turner Syndrome in a Patient With Hemophagocytic Lymphohistiocytosis.","authors":"Nader Pahlevan, Delora E Denney, Ezekiel Gonzalez-Fernandez, Oriana Sanchez, Mark Anderson","doi":"10.1155/crnm/6652600","DOIUrl":"10.1155/crnm/6652600","url":null,"abstract":"<p><p><b>Introduction:</b> Parsonage-Turner syndrome (PTS), also known as idiopathic brachial plexopathy, is an uncommon peripheral neuropathy, and the presentation of bilateral PTS is even rarer. Hemophagocytic lymphohistiocytosis (HLH) is a hyperinflammatory disorder that is normally considered hematologic but can involve the nervous system in up to 70% of cases. <b>Case Presentation:</b> A 56-year-old Caucasian female with a history of SLE, rheumatoid arthritis, Sjogren's disease, and Hashimoto's thyroiditis, but no prior neurologic history, was admitted for the management of isolated thrombocytopenia, diagnosed with HLH, and then developed bilateral upper extremity pain, weakness, and numbness. A diagnosis of bilateral PTS was supported by magnetic resonance imaging (MRI) findings of mild symmetric increased enhancement in the bilateral shoulder muscles, and electromyography/nerve conduction study (EMG/NCS) revealed active denervation in the shoulder muscles bilaterally. The patient was started on methylprednisolone 1000 mg for three days, followed by a long steroid taper along with physical/occupational therapy with significant improvement of her symptoms. <b>Conclusion:</b> It is important to maintain a high index of suspicion for PTS in patients with new-onset shoulder or upper arm pain, weakness, and sensory deficits, even if findings are bilateral. Concomitant inflammatory disorders, infection, and recent surgeries/procedures should prompt a high degree of suspicion of this disorder, and the use of relevant diagnostics, such as MRI brachial plexus and EMG/NCS, should help guide diagnosis, as this condition is very responsive to treatment.</p>","PeriodicalId":9615,"journal":{"name":"Case Reports in Neurological Medicine","volume":"2025 ","pages":"6652600"},"PeriodicalIF":0.9,"publicationDate":"2025-07-21","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12303629/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144728133","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 : 2025-07-14eCollection Date: 2025-01-01DOI: 10.1155/crnm/3086387
Youjiang Tan, Tyngyu Chuah
We report an uncommon and peculiar case of a patient who developed brainstem encephalitis between three and four months after recovering from an episode of Kikuchi-Fujimoto disease (KFD). She presented acutely with oscillopsia and persistent irrepressible hiccups, for which brainstem stroke was initially suspected. Brain magnetic resonance imaging was negative for ischemic strokes but demonstrated an enhancing T2-hyperintense lesion within the area postrema of the medulla oblongata extending into the upper cervical cord. Workup for infections etiologies and demyelinating disorders of the central nervous system including neuromyelitis optica, multiple sclerosis, and antimyelin oligodendrocyte glycoprotein antibody disease, were unremarkable. Prior to the administration of immunosuppressive treatment, she spontaneously and rapidly recovered, remaining well over a 3-year period of follow-up. We reviewed prevailing scientific literature and identified similar, albeit rare, cases of encephalitis which were attributed to KFD, which we added to our discussion.
{"title":"Brainstem Encephalitis as an Atypical Neurologic Complication Following Kikuchi-Fujimoto Disease.","authors":"Youjiang Tan, Tyngyu Chuah","doi":"10.1155/crnm/3086387","DOIUrl":"10.1155/crnm/3086387","url":null,"abstract":"<p><p>We report an uncommon and peculiar case of a patient who developed brainstem encephalitis between three and four months after recovering from an episode of Kikuchi-Fujimoto disease (KFD). She presented acutely with oscillopsia and persistent irrepressible hiccups, for which brainstem stroke was initially suspected. Brain magnetic resonance imaging was negative for ischemic strokes but demonstrated an enhancing T2-hyperintense lesion within the area postrema of the medulla oblongata extending into the upper cervical cord. Workup for infections etiologies and demyelinating disorders of the central nervous system including neuromyelitis optica, multiple sclerosis, and antimyelin oligodendrocyte glycoprotein antibody disease, were unremarkable. Prior to the administration of immunosuppressive treatment, she spontaneously and rapidly recovered, remaining well over a 3-year period of follow-up. We reviewed prevailing scientific literature and identified similar, albeit rare, cases of encephalitis which were attributed to KFD, which we added to our discussion.</p>","PeriodicalId":9615,"journal":{"name":"Case Reports in Neurological Medicine","volume":"2025 ","pages":"3086387"},"PeriodicalIF":0.9,"publicationDate":"2025-07-14","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12279417/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144682103","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 : 2025-06-19eCollection Date: 2025-01-01DOI: 10.1155/crnm/9969463
Lisa B E Shields, Vasudeva G Iyer, Yi Ping Zhang, Christopher B Shields
Gorham-Stout disease (GSD), also known as vanishing bone disease or massive osteolysis, is a rare entity characterized by destruction of the osseous matrix and proliferation of vascular structures resulting in bone resorption. While neurological complications such as cerebrospinal rhinorrhea secondary to cranial involvement and paraplegia from spinal involvement have been reported, peripheral nerve complications are not known. We describe a case of radial nerve palsy that was an iatrogenic complication of shoulder replacement surgery with bone loss of the humerus resembling GSD. A 71-year-old male with a history of left total shoulder arthroplasty followed by a revision reverse total shoulder arthroplasty noted a "bone protruding" and pain in the left upper arm 12 years later. X-rays showed that the proximal portion of the humerus was not detectable. CT scan of the left upper extremity revealed loosening of the humeral component with prominent osteolysis most pronounced around the distal stem. The patient underwent a revision of the reverse total shoulder arthroplasty with replacement of the humeral head and shaft. He experienced numbness, pain, and weakness of the left shoulder and arm with wrist drop postoperatively. Physical exam revealed marked weakness of the dorsiflexors of the wrist and digits, wasting and weakness of the brachioradialis muscle, and loss of pinprick sensation of the superficial radial nerve distribution. Needle EMG showed denervation changes in the extensor digitorum communis, brachioradialis, and extensor carpi radialis longus muscles. An ultrasound (US) study showed enlargement of the left radial nerve at the spiral groove. The EDX and US findings suggested a left radial nerve palsy at the spiral groove. There were minimal EMG abnormalities in the deltoid and triceps muscles suggesting additional involvement of the posterior cord of the brachial plexus. This case illustrates the potential for iatrogenic radial nerve palsy following shoulder replacement surgery with significant bone loss of the humerus resembling GSD.
{"title":"Radial Nerve Palsy as an Iatrogenic Complication of Shoulder Replacement Surgery With Significant Bone Loss of the Humerus Resembling Gorham-Stout Disease: Case Report and Review of the Literature.","authors":"Lisa B E Shields, Vasudeva G Iyer, Yi Ping Zhang, Christopher B Shields","doi":"10.1155/crnm/9969463","DOIUrl":"10.1155/crnm/9969463","url":null,"abstract":"<p><p>Gorham-Stout disease (GSD), also known as vanishing bone disease or massive osteolysis, is a rare entity characterized by destruction of the osseous matrix and proliferation of vascular structures resulting in bone resorption. While neurological complications such as cerebrospinal rhinorrhea secondary to cranial involvement and paraplegia from spinal involvement have been reported, peripheral nerve complications are not known. We describe a case of radial nerve palsy that was an iatrogenic complication of shoulder replacement surgery with bone loss of the humerus resembling GSD. A 71-year-old male with a history of left total shoulder arthroplasty followed by a revision reverse total shoulder arthroplasty noted a \"bone protruding\" and pain in the left upper arm 12 years later. X-rays showed that the proximal portion of the humerus was not detectable. CT scan of the left upper extremity revealed loosening of the humeral component with prominent osteolysis most pronounced around the distal stem. The patient underwent a revision of the reverse total shoulder arthroplasty with replacement of the humeral head and shaft. He experienced numbness, pain, and weakness of the left shoulder and arm with wrist drop postoperatively. Physical exam revealed marked weakness of the dorsiflexors of the wrist and digits, wasting and weakness of the brachioradialis muscle, and loss of pinprick sensation of the superficial radial nerve distribution. Needle EMG showed denervation changes in the extensor digitorum communis, brachioradialis, and extensor carpi radialis longus muscles. An ultrasound (US) study showed enlargement of the left radial nerve at the spiral groove. The EDX and US findings suggested a left radial nerve palsy at the spiral groove. There were minimal EMG abnormalities in the deltoid and triceps muscles suggesting additional involvement of the posterior cord of the brachial plexus. This case illustrates the potential for iatrogenic radial nerve palsy following shoulder replacement surgery with significant bone loss of the humerus resembling GSD.</p>","PeriodicalId":9615,"journal":{"name":"Case Reports in Neurological Medicine","volume":"2025 ","pages":"9969463"},"PeriodicalIF":0.9,"publicationDate":"2025-06-19","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12202074/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144504917","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}