Inherited chromosomally-integrated human herpes virus 6 confounding test results in a patient with myelitis and optic neuritis - A case report.

A case of the 'perfect storm' of myelin oligodendrocyte glycoprotein (MOG) antibody-mediated myelitis, human herpesvirus 6 (HHV-6) reactivation, and COVID-19 infection was reported in 2021. This article reports a case of a similarly observed clinical triad, but with a different conclusion and explanation supported by laboratory test results and evidence from our literature review.

Multifocal strokes and vision loss from PDLLA filler injections.

BACKGROUND: Poly-D, L-lactic acid (PDLLA) is increasingly used as a commercial dermal filler due to its lasting cosmetic properties. Consequently, PDLLA-related vascular complications are increasingly recognized and described. Herein, we describe the first known occurrence of multifocal strokes from the use of PDLLA as a cosmetic dermal filler, and discuss the mechanisms facilitating PDLLA's entry into the intracranial arterial system. CASE PRESENTATION: A middle-aged female presented with acute vision loss of both eyes immediately after dermal injections of PDLLA to her nasolabial folds and infraorbital regions. There were no additional neurological deficits. Dilated fundal examination revealed retinal edema bilaterally, with deposition of filler material in the retinal arteries. Magnetic resonance imaging of her brain and orbits demonstrated multifocal strokes (left caudate head, right medial frontal lobe) and ischemia of the left optic nerve. The temporal proximity of the dermal injections to her symptoms, guided by fundal examination and neuroimaging findings, allowed us to attribute her strokes and ischemic optic neuropathy to PDLLA's entry into, and embolism within, the intracranial arterial system. She was treated with hyperbaric oxygen therapy and experience improvement to her right eye's vision, although poor vision persisted in her left eye. CONCLUSION: While PDLLA is generally considered safe, its increasing use as a cosmetic filler renders it crucial for physicians to be cognizant of its vascular complications, especially when early recognition and treatment are essential in mitigating their devastating ramifications.

Painless nondiabetic lumbosacral radiculoplexus neuropathy with complete recovery: a case report.

BACKGROUND: Lumbosacral radiculoplexus neuropathy, also known as amyotrophy, is an uncommon monophasic disorder characterized by inflammation of the lumbosacral nerve roots and plexuses. Lumbosacral radiculoplexus neuropathy is usually associated with diabetes mellitus, is typically painful at presentation, and often associated with long-term residual neurologic deficits. We report a case of painless, nondiabetic lumbosacral radiculoplexus neuropathy in a young Chinese woman, who made a full recovery after treatment with intravenous immunoglobulin, adding an atypical case to the scarce literature on lumbosacral radiculoplexus neuropathy. CASE PRESENTATION: A 35-year-old Chinese woman presented to our emergency department with 1-week history of painless left lower limb weakness and numbness. Examination revealed weakness confined to the left lower limb but spanning various nerves and myotomes, with abnormal sensation. Clinical localization to the lumbosacral plexus was supported by neurodiagnostic tests, and magnetic resonance imaging of the lumbosacral plexus showed that the nerve roots were also involved. After treatment with intravenous immunoglobulin for nondiabetic lumbosacral radiculoplexus neuropathy, the patient had a full recovery. CONCLUSION: Our patient's case highlights that lumbosacral radiculoplexus neuropathy, an already rare disorder, can occur in the absence of diabetes mellitus and pain, making it even harder to recognize. A systematic and meticulous clinical approach, supported by intelligent selection of adjunctive tests, is required for localization and diagnosis. With an accurate diagnosis, our case also demonstrates that appropriate and prompt treatment can lead to complete recovery, despite previous reports suggesting a high prevalence of long-term residual deficits after lumbosacral radiculoplexus neuropathy.

Anti-NMDAR encephalitis in Southeast Asia - A single-centre, longitudinal study.

AIMS: To describe the clinical features and outcomes of anti-NMDA receptor encephalitis (ANMDARE) in Southeast Asian (SEA) patients. METHOD: SEA patients diagnosed and treated for ANMDARE at Singapore General Hospital between January 2010 and June 2020 were included in this observational study, in which their clinical features and outcomes were retrospectively analysed. RESULTS: We studied 20 patients: 11 Chinese, 3 Tagalogs, 2 Malays, 2 Indians, 1 Eurasian and 1 Javanese. Their median age was 28 years. 15 were females, amongst whom teratomas were demonstrated in 13 (12 ovarian, 1 mediastinal). Delirium and seizures were the two commonest events leading to their presentation at our facility. 1 male had biliary neuroendocrine tumour. Comparison between genders revealed a strong male predilection for early seizures and insomnia; females were four times likelier than males to develop movement disorders or have underlying neoplasms. Patients with dysautonomia required longer ICU stay beyond 14 days, but their outcomes at 1 year did not differ. When reviewed at 1 year, none had clinical relapses, and outcomes were favourable (mRS 0-2) in nearly two-thirds. CONCLUSIONS: SEA patients with ANMDARE frequently present with delirium and seizures. Underlying neoplasms are very common in females. Differences in clinical characteristics may exist between the two genders. Recognition of these can facilitate diagnosis, and permit earlier initiation of appropriate treatment strategies, and thus improve outcomes of SEA patients.

COVID-19 and isolated oculomotor nerve palsy: Clinical features and outcomes.

AIM: This study aims to describe the clinical characteristics of patients with isolated oculomotor nerve palsy from COVID-19 infection, and provide guidance on their treatment and management. METHODS: We performed a systematic review and retrospective analysis on the clinical features and outcomes of patients with isolated oculomotor nerve palsy from COVID-19 reported in literature over the past three years. RESULTS: We analyzed a total of 11 cases; 9 identified in literature from January 2020 to September 2022, together with our two patients. Their median age was 46 years (range 2-65), and three were children. More than half (6/11, 55 %) were without medical history. Oculomotor nerve palsies tended to occur early (longest interval of 16 days), but they can also occur concurrently (2/11, 18 %) or before the appearance of COVID-19 symptoms (1/11, 9 %). COVID-19 symptoms tended to be mild (8/11, 73 %). Oculomotor nerve palsies, however, displayed neither a clear gender predilection, nor consistent clinical features in terms of the severity of extraocular weakness and the involvement of pupillary light responses. Nearly two-thirds (7/11, 64 %) received no pharmacological treatment. Regardless, recovery was complete in nearly all (9/10, 90 %), with most occurring within a month (8/9, 89 %) CONCLUSION: Isolated oculomotor nerve palsies are early but uncommon complications of COVID-19. They affect patients with mild infections, and can be the first symptom. Prognosis is excellent, with recovery being often complete and early. Early discharge and outpatient clinical review, with or without short courses of oral steroids, are reasonable treatment measures.

Capecitabine related neurotoxicity: Clinical and radiologic features.

AIM: To study the clinical and radiologic features of patients with capecitabine neurotoxicity. METHODS: We performed a retrospective analysis and systematic review on the clinical and radiologic characteristics of all patients with capecitabine neurotoxicity reported in literature between 2003 and 2020. RESULTS: 24 cases including our patient were retrospectively analysed, with their clinical and radiologic features summarized. Their median age was 59 years old (ranges from 31 to 82 years old). Encephalopathy was the predominant clinical symptom affecting more than half (15/24, 63%) of the patients. This was followed by cerebellar ataxia (10/24, 42%). Amongst the patients who had magnetic resonance imaging(MRI) brain imaging performed, majority of them (18/23, 78%) had acute radiologic abnormalities. Leukoencephalopathy was the commonest radiologic abnormality seen in more than half of the patients (15/23,65%). Despite the preponderance of female patients in our study, there were no significant statistical differences in the clinical and radiologic features. Short term prognosis was excellent with complete resolution of neurologic symptoms observed in nearly all of the patients (22/23, 96%). CONCLUSION: Capecitabine-related neurotoxicity is an uncommon cause of toxic encephalopathy, with a predilection for females. Clinical features are non-specific, with encephalopathy being the commonest. Prognosis remains good with timely recognition, and cessation of capecitabine. Future research looking into other pathogenic pharmacogenetic processes should be conducted for further elucidation of these associations.

Acute metaldehyde poisoning from ingestion: clinical features and implications for early treatment.

Aims: To describe and compare the clinical features of patients with acute metaldehyde toxicity from suicidal and accidental ingestion of metaldehyde, and to elucidate factors influencing early treatment and disposition. Methods: We undertook a systematic review and retrospective analysis of the clinical characteristics and outcomes of patients with acute toxicity from ingesting metaldehyde. Results: Twenty-one cases identified between 1965 and 2021 were analyzed. The median age was 32 years (range, 3-68 years), and two-thirds of patients experienced symptoms (14/21, 67%). In symptomatic patients, gastrointestinal symptoms were present in two-thirds (9/14, 64%), and half experienced neurologic complications (8/14, 57%); of those with neurologic complications, half experienced seizures (8/14, 57%). There were near-equal cases of accidental and suicidal poisoning. Those who attempted suicide were likelier to develop symptoms (90% versus 45%, P = 0.031), experience seizures (60% versus 18%, P = 0.049), require intensive care (50% versus 9%, P = 0.038), and suffer longer hospitalizations (13.3 days versus 2.9 days, P = 0.005), despite no statistically significant differences in the doses of metaldehyde consumed when compared against patients with accidental ingestion (9.04 g versus 2.03 g, P = 0.09). Conclusion: The circumstances in which metaldehyde is consumed heavily influence clinical symptoms and outcomes. Early and close observation for seizures and adopting a lowered threshold for escalation to the intensive care unit are recommended in patients attempting suicide even when the dose ingested cannot be determined at that time, which is common during the early phases of treatment.

Orbital apex syndrome secondary to herpes zoster ophthalmicus: Clinical features and outcomes case report and systematic review.

BACKGROUND: This study aimed to describe the clinical features of patients with orbital apex syndrome (OAS) as a complication of herpes zoster ophthalmicus (HZO) and to identify factors associated with poor visual acuity outcomes. METHODS: We performed a systematic review and retrospective analysis of the clinical characteristics and outcomes of patients with OAS secondary to HZO reported in the literature over 42 years (1978-2020). RESULTS: We analysed 21 cases, 20 of which were identified in the literature, together with our patient. Their median age was 65 years, with equal involvement in both sexes. The median onset of OAS due to HZO was 10 days (range 1-28 days). The median time of treatment initiation was five days (range 1-21 days). All patients presented with reduced visual acuity, complete ophthalmoplegia, and ptosis. Most patients (17/21, 80.95%) were treated with systemic antiviral and corticosteroid therapy. Three (3/21, 14.29%) patients were immunocompromised. Recovery for ophthalmoplegia (19/21, 90.48%) and ptosis (16/21, 76.19%) was good. Half of the patients (9/18, 50%) showed poor vision recovery. Starting treatment more than 72 h after HZO onset (p = 0.045) was more likely to cause poor vision recovery. CONCLUSION: OAS is a rare, serious, and potentially late complication of HZO and continued observation up to and perhaps beyond four weeks is justifiable, if not encouraged. Early initiation of treatment with systemic antiviral and/or corticosteroids within 72 h of onset of HZO appears beneficial for the recovery of visual acuity.

Cerebellar Stroke in a COVID-19 Infected Patient. a Case Report.

BACKGROUND: Recent studies have reported that COVID-19 infected patients with stroke, who were often in the older age group, had a higher incidence of vascular risk factors, and more severe infection related respiratory symptoms. These observations provided little evidence to suggest that COVID-19 infection is a potential causative factor for stroke. This report describes a young patient with a cerebellar stroke secondary to COVID-19 infection. CASE PRESENTATION: A 45-year old male presented at a hospital, reporting a two-day history of headache, vertigo, persistent vomiting, and unsteady gait. Physical examination revealed gaze-evoked nystagmus on extraocular movement testing, left-sided dysmetria and dysdiadochokinesia. He was diagnosed with a left cerebellar stroke. An external ventricular drain was inserted, and sub-occipital craniectomy was performed to manage the effects of elevated intracranial pressure due to the extent of oedema secondary to the infarct. He also underwent screening for the COVID-19 infection, which was positive on SARS-COV-2 polymerase chain reaction testing of his endotracheal aspirate. Blood and cerebrospinal fluid samples were negative. After the surgery, the patient developed atrial fibrillation and had prolonged vomiting symptoms, but these resolved eventually with symptomatic treatment. He was started on aspirin and statin therapy, but anticoagulation was withheld due to bleeding concerns. The external ventricular drain was removed nine days after the surgery. He continued with active rehabilitation. CONCLUSIONS: Young patients with COVID-19 infection may be more susceptible to stroke, even in the absence of risk factors. Standard treatment with aspirin and statins remains essential in the management of COVID-19 related stroke. Anticoagulation for secondary prevention in those with atrial fibrillation should not be routine and has to be carefully evaluated for its benefits compared to the potential harms of increased bleeding associated with COVID-19 infection.

Isolated Infarctions of the Conus Medullaris: Clinical Features and Outcomes.

OBJECTIVE: This study aims to describe the clinical features and outcomes of patients with isolated infarctions of the conus medullaris, and to identify factors associated with poor functional outcomes. MATERIALS AND METHODS: We performed a systematic review and retrospective analysis on the clinical characteristics and outcomes of patients with isolated conus medullaris infarctions reported in literature over the past 30 years. RESULTS: We analyzed a total of 19 cases; 18 identified in literature from January 1991 to June 2021, together with our patient. Their median age was 56 years (range 28-79), with twice as many females as males. Pain was prominent at onset (15/19, 79%), only a third had vascular risk factors (7/19, 37%), and half had no significant preceding activities or events (9/19, 47%). Almost all experienced paraplegia or paraparesis (16/19, 84%), in which upper motor neuron features were rare (3/19, 16%). The underlying cause was unknown in half (10/19, 53%). Functional outcomes appeared fair, with nearly half being capable of unassisted ambulation (9/11, 82%). Patients with vascular risk factors (67% vs 13%, p = 0.024) or with identified underlying causes (78% vs 13%, p = 0.007) were less likely to walk unassisted. CONCLUSION: Isolated conus medullaris but should be considered in patients with acute cauda equina syndrome, especially in females. Patients with vascular risk factors, or with known causes of infarction, are less likely to walk unassisted. DWI sequences should be included in conventional MRI sequences when evaluating patients with acute cauda equina syndrome.

Sulcal artery syndrome: A Three-patient series and review of literature.

OBJECTIVE: This study aims to describe the clinical characteristics of patients with sulcal artery syndrome, and between those with vertebral artery dissection against those without. METHODS: We report three cases of sulcal artery syndrome without vertebral artery dissection, performed a systematic review and retrospective analysis of the characteristics of patients with sulcal artery syndrome in available literature, and compared the clinical features of those with vertebral artery dissection against those without. RESULTS: We report 3 patients with sulcal artery syndrome, and analysed them with 17 other cases identified in literature between January 1990 till April 2020. The mean age was 47 years (range 10-80), with twice as many males as females. Pain at onset was a prominent feature (17/18, 94.4%). Preceding trauma occurred in less than half (7/18, 38.9%). Most had cervical cord infarctions (18/20, 90%), often over the high cervical cord (16/18, 88.9%). Good functional recovery (mRS 0-2) was observed in 86.7% (13/15). While vertebral artery dissection was the leading aetiology (11/20, 55.5%), about half of the cases were due to other causes. Cervical cord involvement was significantly associated with vertebral artery dissection (p = 0.026). CONCLUSION: Sulcal artery syndrome should be suspected in patients with acute hemicord syndrome, especially in males with cervical cord involvement or pain at onset. High cervical cord involvement was strongly suggestive of underlying vertebral artery dissection. Additionally, DWI sequences are useful when evaluating acute myelopathies, and its inclusion in conventional MRI sequences is supported in prevailing literature.

Recurrent ipsilateral hemiparesis in a patient with both uncrossed corticospinal tracts and reorganization of cortical motor areas - An opportune visitation of the motor tracts.

We report the case of a patient who experienced recurrent ipsilateral hemiparesis in the setting of predominantly-uncrossed corticospinal tracts, with concomitant neuronal reorganization of the cortical motor maps, and the presence of aberrant interhemispheric connections. Their presence was supported by our results from diffusion tensor imaging tractography, functional magnetic resonance imaging, and transcranial magnetic stimulation. To our knowledge, this has never been reported before, and provides valuable insights into the mechanisms behind post-stroke motor recovery.

Stroke Patients Without COVID-19 Symptoms: Is There a Need to Screen?

INTRODUCTION: COVID-19 patients who present with strokes but without typical COVID-19 symptoms have been described in small numbers. Despite the paucity of fever and respiratory symptoms, they remain capable of infecting others. The patient we discuss herein highlights the important issues of strokes as presenting events of COVID-19 infections, and how testing for COVID-19 in stroke patients, even when asymptomatic for COVID-19, can play an important role in infection control, clinical management and outcomes amidst this global pandemic. CASE REPORT: A 45-year-old male resident of a dormitory presented to our unit with acute vertigo and left-sided dysmetria. NIHSS was 2. The initial magnetic resonance imaging demonstrated infarction of the left cerebellar hemisphere, middle cerebellar peduncle and hemipons. An extensive work-up for stroke etiologies was unremarkable. Despite having no fever, respiratory symptoms, anosmia or ageusia, he was isolated and screened for COVID-19 due to his epidemiologic risks, with multiple residents from his dormitory being recently diagnosed with COVID-19. Confirming our suspicion, his respiratory samples returned positive for COVID-19. His D-dimer levels returned normal. Thereafter, the patient underwent posterior decompression surgery due to worsening edema caused by the cerebellar infarct. He was started on antiplatelet therapy and recovered significantly a month from presentation with an modified Rankin Sore of 2. He remained without typical COVID-19 symptoms. CONCLUSION: Our patient's case clearly supports the screening for COVID-19 in stroke patients who are without COVID-19 symptoms, appreciating the significant value it adds to infection control, clinical management, and outcomes amidst this global pandemic.

Idiopathic Intracranial Hypertension - Characteristic MRI Features.

We present the case of 24-year-old woman who presented with chronic headaches with features of raised intracranial pressure and bilateral papilledema. Brain magnetic resonance imaging revealed the characteristic features of idiopathic intracranial hypertension. A diagnosis supported by clinical features and other ancillary tests. These features are important for physicians to recognize early, so that timely treatment may prevent permanent complications from this rare but potentially sight-threatening headache.

Genetic diversity of Chinese rabies viruses: evidence for the presence of two distinct clades in China.

There have been three major rabies epidemics in China since the 1950s. To gain more insights into the molecular epidemiology of rabies viruses (RVs) for the third (the current) epidemic, we isolated RV from dogs and humans in major endemic areas, and characterized these isolates genetically by sequencing the entire glycoprotein (G) gene and the G-L non-coding region. These sequences were also compared phylogenetically with RVs isolated in China during previous epidemics and those around the world. Comparison of the entire G genes among the Chinese isolates revealed up to 21.8% divergence at the nucleotide level and 17.8% at the amino acid level. The available Chinese isolates could be divided into two distinct clades, each of which could be further divided into six lineages. Viruses in clade I include most of the Chinese viruses as well as viruses from southeast Asian countries including Indonesia, Malaysia, the Philippines, Thailand, and Vietnam. The viruses in the other clade were found infrequently in China, but are closely related to viruses distributed worldwide among terrestrial animals. Interestingly, most of the viruses isolated during the past 10 years belong to lineage A viruses within clade I whereas most of the viruses isolated before 1996 belong to other lineages within clades I and II. Our results indicated that lineages A viruses have been predominant during the past 10 years and thus are largely responsible for the third and the current epidemic in China. Our results also suggested that the Chinese RV isolates in clade I share a common recent ancestor with those circulating in southeast Asia.