Unusual Neurological Manifestations Research Articles

Incessant Ectopic Atrial Tachycardia Accompanied by Unusual Neurological Manifestations – Case Report and Literature Review

Incessant ectopic atrial tachycardia (EAT) is well-known in the electrophysiology world. However, once accompanied by neurological manifestations, hence is a dilemma. We are reporting a 26-year-old male presenting with incessant symptomatic EAT. Once cardioverted, he began to manifest cardiovascular stroke clinical symptoms. Further objective investigation revealed no evidence of cardioembolic stroke. Our aim of this case report is to explore if there is a true correlation between EAT and possible neurological association and open further guidelines related to research.

Pediatric Neurotuberculosis: A cases series and review of the literature.

Neurotuberculosis or central nervous system tuberculosis is a form of tuberculous infection that affects any part of the nervous system. Although it is more frequent in adults, pediatric cases have been reported in endemic countries and it is potentially a deadly affection. Therefore, any unusual neurological manifestation in a formerly healthy child, independently of their vaccination status, must bring suspicion of CNS tuberculosis among other diagnoses. We report four cases of pediatric neurotuberculosis with various clinical presentations and outcome and a brief review of the litterature. We conclude that clinical manifestations of pediatric neurotuberculosis are extremely variable and could be misleading. Extra-neurological sites are a key element for diagnosis especially in the pediatric population. A diagnosis and clinical outcome score, especially designed for children might help personalize the therapeutic approach and outcome measures.

Posterior reversible encephalopathy syndrome in juvenile lupus- a case series and literature review.

PRES, as a complication of juvenile lupus, is rarely reported in the literature. In this study, six juvenile lupus patients admitted with diagnosis of PRES were assessed on the basis of clinical characteristics, imaging findings, disease activity status, treatment response and prognosis. Six juvenile (≤ 16 years) lupus patients with a diagnosis of PRES were included. Demographic, clinical, and laboratory features and outcomes of all six patients were noted. Literature review was performed on PubMed search forum. Search terms in English included Juvenile SLE, Lupus and PRES. The youngest patient was seven years old while the oldest was sixteen years. All patients had history of lupus nephritis , presented with seizure and hypertension. In imaging, four out of six patients had hyperintensities in atypical distribution suggesting atypical PRES. All the patients had significant clinical recovery with resolution of hyperintensities in five out of six patients on repeat imaging. Juvenile lupus with PRES is considered an unusual neurologic manifestation triggered by multiple factors. It can be stipulated that PRES in juvenile lupus cases often remain undiagnosed. Early suspicion and treatment institution with reversal of triggers can result in a favorable outcome in these patients.

Unusual neurologic manifestations of Vogt-Koyanagi-Harada disease: a systematic literature review

Background and PurposeThe usual neurologic manifestations of Vogt-Koyanagi-Harada (VKH) disease include aseptic meningitis and headaches. We performed the present study to review all unusual neurologic manifestations reported in VKH disease to summarize them.MethodsA literature search was performed in the English language on Scopus and Medline via PubMed from 1946 to July 31, 2021, by using the following terms: “Vogt Koyanagi Harada disease” OR “VKH disease” AND “Brain” OR “Spinal cord” OR “CNS” OR “Central nervous system” OR “Neurologic” OR “Peripheral nervous system” OR “Polyneuropathies. Our inclusion criteria were unusual neurologic manifestations of VKH disease.ResultsOur literature search yielded 417 total articles (PubMed = 334, Scopus = 83) from which 32 studies comprising 43 patients (22 men and 21 women, of which 62.8% were younger than 50 years) were included in this systematic literature review. Regarding the study design, all studies were case reports and published between 1981 and 2021. CNS involvement was the most reported (93%) in VKH disease. Peripheral nervous system involvement represents 7% of cases. The cerebral lesions were parenchymal inflammatory lesions in the white matter or posterior fossa with or no contrast enhancement (16.3%), leptomeningitis (9.3%), pachymeningitis (7%), meningoencephalitis (2.3%), ischemic stroke (4.6%), hemorrhagic stroke (2.3%), transient ischemic attack (2.3%), and hydrocephalus (2.3%). The optic nerve lesions were anterior ischemic optic neuropathy (20.9%) and optic neuritis (9.3%). Concerning spinal cord lesion, it was mainly myelitis (14%).ConclusionThis systematic literature review provides a summary of the different unusual neurologic manifestations reported in VKH disease.

The Spectrum of Neuro-COVID: A Study of a Comprehensively Investigated Large Cohort from India.

Background:Though reports of neurological manifestations of COVID-19 have emerged from various parts of the world, the cohorts reported are from the West and mostly derived from electronic databases. Much remains unknown regarding neuro-COVID in developing countries. India is the second-worst affected country, and this study reports the neurological manifestations of COVID-19 in a comprehensively evaluated cohort.Objective:The aim of this study was to describe the range of neurological manifestations of COVID-19 in India with an emphasis on the risk factors, laboratory and imaging findings and short-term outcome.Methods:Retrospective review of hospital records of all confirmed COVID-19 patients with neurological manifestations, receiving inpatient care in two neurology referral hospitals were done. All demographic, clinical details, investigations, and treatment were analysed.Results:A total of 120 confirmed COVID-19 cases presenting with neurological symptoms were included. The mean age of illness and duration of illness was 48.03 ± 17.3 years and 10.9 ± 17.3 days respectively. New onset of neurological symptoms occurred in 100 cases while 20 patients had worsening of pre-existing neurological illness. Stroke was the commonest neurological disorder (43%), followed by encephalopathy (23%) and Guillain-Barre syndrome (10%). Other unusual neurological manifestations included new-onset headache (7%), seizures including denovo status epilepticus (5%) and meningo-encephalitis (5%). Nearly half of the patients had preceding COVID-19 symptoms. Poor outcome at discharge was seen in 40% and mortality occurred in 15%.Conclusion:Stroke and encephalopathy constitute the most common neurological manifestations. The absence of preceding COVID-19 symptoms in nearly half the cases is striking. Poor outcome was seen in nearly 50% despite early recognition and management.

Unusual neurological manifestations of scrub typhus.

Unusual neurological manifestations of scrub typhus.

Double Trouble of Severe Hyperkalemia: Syncope and No Significant ECG Changes

Potassium is an important ion capable to maintain intra-extracellular electric gradient. Hyperkalemia is a common and potential life-threatening electrolyte disorder in patients presenting to the emergency setting. Variations in the intra-extracellular ionic flow may alter cells functions, skeletal and smooth muscle contractility and electric activity of myocardial cells. Hyperkalemia can be difficult to diagnose clinically because symptoms may be vague. Patients may be asymptomatic or report non-specific symptoms such as generalized fatigue, weakness, paralysis or palpitations. Syncope is unusual neurological manifestation. An increase in serum potassium levels is followed by progressively severe electrophysiological derangements in cardiac impulse generation and conduction, which are reflected in the electrocardiogram (ECG).

Cranial polyneuropathy and triparesis due to mononeuritis multiplex in a case of scrub typhus

Scrub typhus with neurological manifestations are often described. We present here unusual neurological manifestations in a patient with Scrub typhus. A 49 years old male presented with continued fever for the last 12 days along with headache and myalgia. He had left sided oculomotor nerve palsy along with palatal palsy and triparesis (except left upper limb) of lower motor neurone type. Electro-diagnostic studies showed asymmetric axonal sensorimotor neuropathies of multiple nerves in paretic limbs, suggestive of mononeuritis multiplex. Scrub typhus was diagnosed by the presence of IgM antibody in serum. He responded well with doxycycline therapy. This type of neurological presentation is extremely rare in scrub typhus and possibly has not been described earlier to the best of our knowledge.

Molecular Epidemiology of Enterovirus in Children with Central Nervous System Infections.

Limited recent molecular epidemiology data are available for pediatric Central Nervous System (CNS) infections in Europe. The aim of this study was to investigate the molecular epidemiology of enterovirus (EV) involved in CNS infections in children. Cerebrospinal fluid (CSF) from children (0–16 years) with suspected meningitis–encephalitis (ME) who were hospitalized in the largest pediatric hospital of Greece from October 2017 to September 2020 was initially tested for 14 common pathogens using the multiplex PCR FilmArray® ME Panel (FA-ME). CSF samples positive for EV, as well as pharyngeal swabs and stools of the same children, were further genotyped employing Sanger sequencing. Of the 330 children tested with FA-ME, 75 (22.7%) were positive for EV and 50 different CSF samples were available for genotyping. The median age of children with EV CNS infection was 2 months (IQR: 1–60) and 44/75 (58.7%) of them were male. There was a seasonal distribution of EV CNS infections, with most cases detected between June and September (38/75, 50.7%). EV genotyping was successfully processed in 84/104 samples: CSF (n = 45/50), pharyngeal swabs (n = 15/29) and stools (n = 24/25). Predominant EV genotypes were CV-B5 (16/45, 35.6%), E30 (10/45, 22.2%), E16 (6/45, 13.3%) and E11 (5/45, 11.1%). However, significant phylogenetic differences from previous described isolates were detected. No unusual neurologic manifestations were observed, and all children recovered without obvious acute sequelae. Specific EV circulating genotypes are causing a significant number of pediatric CNS infections. Phylogenetic analysis of these predominant genotypes found genetic differences from already described EV isolates.

A case of vitamin B12 deficiency with some unusual features

Background: Vitamin B12 (VB12) deficiency is a pleomorphic disease with a wide range of manifestations. Its major cause is pernicious anemia, an autoantibody-mediated disease that results in low levels of intrinsic factor [1,2]. Case Presentation: We present a case report of a middle-aged man with severe megaloblastic anemia due to VB12 deficiency in relation to an unsuspected Crohn’s disease with exclusive ileum involvement. He also had some unusual neurological manifestations of this vitamin’s deficiency, such as hypersudoresis. Unlike most cases, the anemia was not hypoproliferative and hemolysis existed. Conclusion: This case reports some unusual aspects of VB12 deficiency and reminds us that ileum disease should be considered in patients with a negative autoimmune profile.

An unusual concurrent dermatological and neurological manifestation in an elderly woman with eosinophilic granulomatosis with polyangiitis: a case report and literature review

: Eosinophilic granulomatosis with polyangiitis (EGPA) is a rare disease, occurs commonly in middle-aged females. EGPA involves multiple systems and hallmarked by small and medium vessel entanglement. Concurrent basal ganglia lacunar infarctions (BLI) and peripheral nervous (PN) manifestation rarely reported in dermatological co-morbidity. We encountered an 81-year-old female with controlled hypertension, presented at the Neurology department with an antecedent ten years history of tuberculosis and asthma, and five days of complaints of left limb weakness accompanied by bilateral feet numbness associated with abdominal ache. Two weeks before, she had attended the dermatology clinic for lower limb purpuric rashes, which not resolved at the presentation. Examination and series of investigations revealed eosinophilia, negative anti-neutrophil cytoplasmic antibodies (ANCA), mild anemia secondary to gastrointestinal involvement, and purpuric rash. Further, peripheral polyradiculoneuropathy noted. Radiographically, paraventricular, and bilateral basal ganglia lacunar infarction and cerebral atrophy documented. Nasosinusitis, multiple lung fibrous, and sclerotic foci, and aortic regurgitation observed. On a clinical evaluation basis, the patient managed with corticosteroids and supportive symptomatic treatment, the 7th day’s improvement observed. To the authors’ knowledge, this is the oldest EGPA patient reported with co-occurrence of nervous system and cutaneous manifestation. Further, the patient had a unique presentation of the occurrence of both BLI and PN findings. Yet, the cardiac manifestation of aortic valve regurgitation rather than mitral valve regurgitation. Through case-reports, when EGPA nervous and dermatological manifestation co-occurs, the likelihood of encountering negative ANCA result >70% yet this necessitate vast future researches.

Hemorrhagic PRES: an unusual neurologic manifestation in two COVID-19 patients.

Hemorrhagic PRES: an unusual neurologic manifestation in two COVID-19 patients.

Thrombotic thrombocytopenic purpura with reversible splenial lesion syndrome: a case report

BackgroundReversible splenial lesion syndrome (RESLES) is known to cause severe psychiatric symptoms but is also a very rare clinical disease in which the specific aetiology is unknown. According to current reports, there are major causes of the disease, including viral or bacterial infection, epilepsy, anti-epileptic drug withdrawal, high-altitude cerebral oedema, and metabolic disorders such as hypoglycaemia and hypernatraemia. In this article, we report a patient with thrombotic thrombocytopenic purpura (TTP) who presented with RESLES.Case presentationA 34-year-old female patient who presented with fever and progression of disorder of consciousness was eventually diagnosed with RESLES based on brain imaging. Moreover, clinical features and peripheral smears demonstrating schistocytes and thrombocytopenia confirmed a diagnosis of TTP. RESLES can be improved by plasma exchange therapy.ConclusionThis rare case highlights the occurrence of RESLES as a presenting feature of the expanding list of unusual neurological manifestations of TTP.

An unusual case of inflammatory meningitis in a young man with systemic lupus erythematosus

We describe a man presenting with unusual neurological manifestations of systemic lupus erythematosus (SLE) including pachymeningitis, aseptic meningitis and encephalitis with grossly elevated cerebrospinal fluid protein, responding to immunosuppression. Initially he had intermittent dysarthria, dysphasia and unilateral upper limb weakness. One month later he experienced dysphasia, right-sided hemiparesis and confusion. Cerebrospinal fluid (CSF) analysis showed a white cell count of 70 x 106/litre and an unusually elevated protein level of 5.39 g/litre. An MRI brain showed dural and leptomeningeal enhancement compatible with a meningitic process. He improved with cefotaxime and aciclovir. On day seven of antimicrobials he developed left-sided weakness, sensory inattention and a left homonymous hemianopia. He responded well to intravenous methylprednisolone. On switching to oral prednisolone he developed expressive dysphasia, a right inferior quadrantanopia and seizures. His bloods were suggestive of macrophage activation syndrome. The patient improved with methylprednisolone and intravenous immunoglobulins, and the improvement was sustained on switching back to oral prednisolone. The prevalence of neuropsychiatric manifestations of SLE varies between 14 and 80% and according to the American College of Rheumatology includes 19 conditions. This case is unique because although some features were in keeping with aseptic meningitis the MRI appearances were also suggestive of pachymeningitis.

Reversible splenial lesion syndrome associated with dengue fever: a case report

BackgroundDengue virus infection in humans can lead to a wide range of clinical manifestations, from mild fever to potentially fatal dengue shock syndrome. The incidence of dengue fever is on the rise in tropical countries. Due to the increasing incidence of dengue fever worldwide, atypical manifestations of the disease are increasingly reported. In this article we report a patient with dengue haemorrhagic fever who presented with reversible splenial lesion syndrome.Case presentationA 24-year-old Sri Lankan man who presented with fever and confusion was eventually diagnosed to have reversible splenial lesion syndrome based on brain imaging. Clinical, serological and haematological parameters confirmed a diagnosis of dengue haemorrhagic fever. His presentation, assessment, and management are described in this case report.ConclusionReversible splenial lesion syndrome is a condition which is radiologically characterized by reversible lesion in the splenium of the corpus callosum. It is associated with infectious and non-infectious aetiologies. This case report highlights the occurrence of reversible splenial lesion syndrome as a presenting feature of the expanding list of unusual neurological manifestations of dengue infection.

F56. Description of neurophysiological findings in mononeuritis multiple as a neurologic manifestation of borrelia burgdorferi infection - Case report

Introduction The infection for Borrelia burgdorferi is a zoonosis, in which the spirochaetes can be transmitted to human beings only by the bite of infected ixodid ticks, the spirochaete is maintained at high levels in populations of field mice or birds, the likelihood of having it is dependent on geography, recreational habits of the patient, and season. It is a multisistemic disease it can affect central nervous system. Neurological manifestations often referred to as Lyme neuroborreliosis are reported in up to 12% of patients with Lyme disease. In Europe and the United States seroprevalencia is 1–27%. This has not been determined in Mexico yet. In Mexico are few reports and series of cases published about Lyme neuroborreliosis, being a subdiagnosed disease in patients with neurological clinical manifestations. Methods Male 23 yo, carpenter. He started eight months earlier with symmetric paresthesias of both hands with ascending distribution from distal to proximal, presented inability to hold work tools with both hands, paresthesias in the plantar region and paresis of the lower limbs, ascending from distal to proximal, preventing him from walking and requiring help, finally prostration. PE: Generalized atrophy of distal predominance, interosseous atrophy of thenar and hypothenar, proximal upper limb 4/5, distal 3/5, proximal and distal lower limbs 4/5, absent bilateral patellar reflex. Bilateral plantar flexor response. Hypopallesthesia, altered arthrokinesia in both lower limbs and right hand. Unsteady gait with a broad base, short and slow steps with help. Romberg positive. Results Neurophysiological studies. Somatosensory evoked potentials (SSEP) of upper limbs with peripheral response integration at the level of the brachial plexus (N9) asymmetric by left prolongation and absence of responses from posterior cords (N13). SSEP of lower limbs with complete and bilateral absence of responses from popliteal fossa. N. median left, N. ulnar right, delay in nerve conduction velocity. Bilateral tibial with prolonged latency. N. median left with prolongation of peak latency. N. ulnar voltage asymmetry by right depression. F response of bilateral ulnar nerve did not elicit response. Bilateral tibial nerve with prolonged latencies and frequency of reduced evocation. The neurophysiological study showed evidence of multiple mononeuropathy that involves all four limbs asymmetrically. Ig G by Western blot versus Borrelia burgdorferi was positive in blood. It was prescribed ceftriaxone 1 g IV BID for 14 days, after this treatment showed neurological improvement, he continued with doxycycline 100 mg orally BID for a month. Conclusion Neuropathies in patients with chronic Lyme disease commonly present as non-vasculitic multiple mononeuritis, the motor deficit follows the multiple pattern. This case is based on an unusual neurological manifestation together with the exclusive predilection for affection of the central nervous system, having as main geographical and occupational risk factors.

Dengue encephalitis with cerebellar involvement: A rare case report

Dengue is the most important arthropod-borne viral infection of humans. Clinical presentation of dengue fever ranged from mild clinical febrile illness to severe life-threatening conditions. Many unusual neurological manifestations have been reported with dengue fever. These include aseptic meningitis, encephalitis, myelitis, intracranial hemorrhage, and mono/polyneuropathies. Here, we report a case who presented with cerebellar syndrome with sinus bradycardia as the initial manifestation of dengue fever.

Internuclear Ophthalmoplegia in Pontomesencephalic Stroke as the First Manifestation of Takayasu Arteritis

Takayasu arteritis (TA) is a chronic inflammatory disease affecting the aorta and its main branches. Patients generally present with symptoms such as fever, myalgia, limb claudication, carotid vessel tenderness, hypertension, and asymmetric pulses. Neurological manifestations are present in a small percentage of patients, with stroke occurring in about 10% of cases. Stroke, occurring as the very first presentation of the disease, is however unusual. We present an atypical case of unilateral internuclear ophthalmoplegia (INO) due to acute pontomesencephalic stroke in the absence of cerebral angiographic abnormality, in a 36-year-old woman as the initial presenting feature of TA. The patient had angiographic evidence of bilateral subclavian artery stenosis but no abnormality in the cerebral vessels, thus pointing toward a possible embolic origin from inflamed vessels. Though other unusual neurological manifestations have been reported, INO as the first manifestation has not been found in medical literature so far.

Unusual Neurologic Manifestations of a Patient with Cyanotic Congenital Heart Disease after Phlebotomy

Secondary erythrocytosis in cyanotic congenital heart disease (CCHD) is a compensatory response to chronic hypoxia which should be managed with caution. CCHD patients, who have compensated erythrocytosis but do not manifest significant neurologic symptoms, may experience secondary life-threatening complications such as stroke in case of inappropriate phlebotomy. This study reports a young man with CCHD who developed frequently repeated transient neurologic deficits with various presentations after one session of phlebotomy. The symptoms resolved a few days after the hematocrit (Hct) level returned to the prephlebotomy level.

Case Report: Thyroid Storm Presenting with Unusual Neurologic Manifestations

Clinical ThyroidologyVol. 26, No. 7 SummariesCase Report: Thyroid Storm Presenting with Unusual Neurologic ManifestationsStephen Ku and Dorothy S. MartinezStephen KuSearch for more papers by this author and Dorothy S. MartinezSearch for more papers by this authorPublished Online:14 Jul 2014https://doi.org/10.1089/ct.2014;26.194-199AboutSectionsView articleView Full TextPDF/EPUB ToolsPermissionsDownload CitationsTrack CitationsAdd to favorites Back To Publication ShareShare onFacebookTwitterLinked InRedditEmail View articleFiguresReferencesRelatedDetails Volume 26Issue 7Jul 2014 InformationCopyright 2014 American Thyroid Association, Inc.To cite this article:Stephen Ku and Dorothy S. Martinez.Case Report: Thyroid Storm Presenting with Unusual Neurologic Manifestations.Clinical Thyroidology.Jul 2014.194-199.http://doi.org/10.1089/ct.2014;26.194-199Published in Volume: 26 Issue 7: July 14, 2014PDF download