Adult vasovagal syncope with abdominal pain diagnosed by head-up tilt combined with transcranial doppler: a preliminary study.

BACKGROUND: Syncope is a common condition that increases the risk of injury and reduces the quality of life. Abdominal pain as a precursor to vasovagal syncope (VVS) in adults is rarely reported and is often misdiagnosed.​. METHODS: We present three adult patients with VVS and presyncopal abdominal pain diagnosed by synchronous multimodal detection (transcranial Doppler [TCD] with head-up tilt [HUT]) and discuss the relevant literature. RESULTS: Case 1: A 52-year-old man presented with recurrent decreased consciousness preceded by six months of abdominal pain. Physical examinations were unremarkable. Dynamic electrocardiography, echocardiography, head and neck computed tomography angiography, magnetic resonance imaging (MRI), and video electroencephalogram showed no abnormalities. Case 2: A 57-year-old woman presented with recurrent syncope for 30 + years, accompanied by abdominal pain. Physical examination, electroencephalography, and MRI showed no abnormalities. Echocardiography showed large right-to-left shunts. Case 3: A 30-year-old woman presented with recurrent syncope for 10 + years, with abdominal pain as a precursor. Physical examination, laboratory analysis, head computed tomography, electrocardiography, and echocardiography showed no abnormalities. Syncope secondary to abdominal pain was reproduced during HUT. Further, HUT revealed vasovagal syncope, and synchronous TCD showed decreased cerebral blood flow; the final diagnosis was VVS in all cases. CONCLUSIONS: Abdominal pain may be a precursor of VVS in adults, and our findings enrich the clinical phenotypic spectrum of VVS. Prompt recognition of syncopal precursors is important to prevent incidents and assist in treatment decision-making. Abdominal pain in VVS may be a sign of sympathetic overdrive. Synchronous multimodal detection can help in diagnosing VVS and understanding hemodynamic mechanisms.

Successful robot-assisted laparoscopic resection of pheochromocytoma in a patient with dilated cardiomyopathy: A case report on extremely high-risk anesthesia management.

RATIONALE: Anesthetic management during resection of pheochromocytoma is a huge challenge, especially when accompanied by dilated cardiomyopathy (DCM). However, there is a lack of research evidence in this area. PATIENT CONCERNS: A 36-year-old man was admitted with a left retroperitoneal space-occupying lesion, present for 2 years. The patient also had DCM for 2 years. Blood analysis on admission showed elevated levels of norepinephrine and the N-terminus of the brain natriuretic peptide precursor. Abdominal computed tomography revealed a circular shadow in the left adrenal area. Echocardiography showed a cardiac ejection fraction of 31% to 37%, markedly enlarged left atrium and left ventricle, extensive cardiac hypokinesia, and reduced left ventricular diastolic and systolic functions. DIAGNOSES: The preoperative diagnosis was left paraganglioma/pheochromocytoma with DCM. INTERVENTIONS: Multidisciplinary consultation, blood pressure measurements, and volume expansion measurements were performed preoperatively. Invasive arterial blood pressure, central venous pressure, depth of anesthesia, cardiac function, left heart volume, and body temperature were monitored intraoperatively. OUTCOMES: The adrenal pheochromocytoma was successfully removed, and the patient recovered well. LESSONS: The anesthetic management for adrenal pheochromocytoma resection in adult patients with DCM is extremely high-risk but is evidently not impossible. Adequate preoperative evaluation and preparation, optimization of the anesthesia induction plan, close intraoperative monitoring of cardiac function and hemodynamic changes, and robot-assisted laparoscopic technology are the key success factors. The challenges to anesthetic management may be partly prevented with invasive monitoring techniques and minimally invasive surgery. This case confirms the importance of individual management and multidisciplinary cooperation for a successful outcome.

Metabotropic glutamate receptor 5-related autoimmune encephalitis with reversible splenial lesion syndrome following SARS-CoV-2 vaccination.

RATIONALE: Metabotropic glutamate receptor 5 (mGluR5)-related autoimmune encephalitis (AE) has been rarely reported; however, there are no reports on mGluR5-related AE with reversible splenial lesion syndrome following vaccination against severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2). PATIENT CONCERNS: A 29-year-old man was admitted with a history of headache and fever for 9 days and 6 days, respectively. DIAGNOSIS: He was initially diagnosed with an intracranial infection, however the final diagnosis was corrected as anti-mGluR5-related AE with reversible splenial lesion syndrome. INTERVENTIONS: He had received an inactivated SARS-CoV-2 vaccine 3 weeks prior to the examination and was initially diagnosed with an intracranial infection. Physical examination revealed bilateral horizontal nystagmus, ataxia, and neck rigidity. Antiinfective therapy was minimally helpful. An analysis of the cerebrospinal fluid did not reveal pathogens for sequencing. Magnetic resonance imaging displayed abnormal signals in the splenium of the corpus callosum. OUTCOMES: We identified mGluR5 antibodies in the cerebrospinal fluid and serum. Subsequently, intravenous methylprednisolone pulse and gamma-globulin pulse therapies were administered, which substantially improved the symptoms. Follow-up did not reveal abnormal neurological symptoms, and the lesion in the corpus callosum had resolved. LESSONS: AE with mGluR5 antibodies could arise from SARS-CoV-2 vaccination, which warrants the awareness of healthcare workers. Reversible splenial lesion syndrome may accompany mGluR5-related AE and mimic intracranial infection. Thus, early treatment can prevent serious residual signs and symptoms.

X-linked Charcot-Marie-Tooth disease after SARS-CoV-2 vaccination mimicked stroke-like episodes: A case report.

BACKGROUND: Severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2) vaccinations have been administered worldwide, with occasional reports of associated neurological complications. Specifically, the impact of vaccinations on individuals with X-linked Charcot-Marie-Tooth disease type 1 (CMTX1) is unclear. Patients with CMTX1 can have stroke-like episodes with posterior reversible encephalopathy syndrome on magnetic resonance imaging (MRI), although this is rare. CASE SUMMARY: A 39-year-old man was admitted with episodic aphasia and dysphagia for 2 d. He received SARS-CoV-2 vaccination 39 d before admission. Physical examination showed pes cavus and reduced tendon reflexes. Brain MRI showed bilateral, symmetrical, restricted diffusion with T2 hyperintensities in the cerebral hemispheres. Nerve conduction studies revealed peripheral nerve damage. He was diagnosed with Charcot-Marie-Tooth disease, and a hemizygous mutation in the GJB1 gene on the X chromosome, known to be pathogenic for CMTX1, was identified. Initially, we suspected transient ischemic attack or demyelinating leukoencephalopathy. We initiated treatment with antithrombotic therapy and immunotherapy. At 1.5 mo after discharge, brain MRI showed complete resolution of lesions, with no recurrence. CONCLUSION: SARS-CoV-2 vaccination could be a predisposing factor for CMTX1 and trigger a sudden presentation.

Clinical characteristics and long-term follow-up outcomes of myelin oligodendrocyte glycoprotein antibody-associated disease in Han Chinese participants.

Myelin oligodendrocyte glycoprotein (MOG) antibody-associated disease (MOGAD) is an immune-mediated inflammatory demyelinating disease of the central nervous system. This study aimed to delineate the clinical manifestations, imaging features, and long-term outcomes in Chinese patients with MOGAD and analyze the recurrence-associated factors. The phenotypic and neuroimaging characteristics of 15 Han Chinese patients with MOGAD were retrospectively analyzed. Demyelinating attacks, MOG antibodies in the cerebrospinal fluid/serum, response to immunotherapy, follow-up outcomes, and recurrence-associated factors were recorded. The median age at disease onset was 34 years (range, 4-65 years). The most common initial presentations included vision loss (10/15, 66.7%) and seizures (5/15, 33.3%). Serum MOG-Ab titers in 14/15 cases were higher than those in the cerebrospinal fluid and were detected in 3/6 relapsed patients. Brain magnetic resonance imaging during acute attacks showed lesions in 10/15 patients (66.7%), mostly in the cortex/subcortical white matter (5/15, 33.3%). Recurrence occurred in 6/15 patients (40.0%); in 4 patients, recurrence occurred shortly after immunotherapy discontinuation. Residual neurological deficits were present in 5/15 patients (33.3%), including visual impairment, incapacitation, cognitive impairment, and speech reduction. Optic neuritis was the most common clinical manifestation of MOGAD. magnetic resonance imaging findings were heterogeneous and the cerebral cortex/subcortical white matter was the most susceptible brain region. Although patients in the acute phase responded well to methylprednisolone pulse therapy, the long-term recurrence rate was high. Consistently detected serum MOG antibodies and inappropriate maintenance immunotherapy may be associated with recurrence, and residual neurological deficits should not be ignored.

Corrigendum: Case report: Cryptogenic giant brain abscess caused by Providencia rettgeri mimicking stroke and tumor in a patient with impaired immunity.

[This corrects the article DOI: 10.3389/fneur.2022.1007435.].

Case report: Cryptogenic giant brain abscess caused by Providencia rettgeri mimicking stroke and tumor in a patient with impaired immunity.

The highly lethal cryptogenic brain abscess can be easily misdiagnosed. However, cryptogenic brain abscess caused by Providencia rettgeri is rarely reported. We present the case of a cryptogenic Providencia rettgeri brain abscess and analyze the clinical manifestations, imaging findings, treatment, and outcome to improve the level of awareness, aid in accurate diagnosis, and highlight effective clinical management. A 39-year-old man was admitted to the hospital after experiencing acute speech and consciousness disorder for 1 day. The patient had a medical history of nephrotic syndrome and membranous nephropathy requiring immunosuppressant therapy. Magnetic resonance imaging revealed giant, space-occupying lesions involving the brain stem, basal ganglia, and temporal-parietal lobes without typical ring enhancement, mimicking a tumor. Initial antibiotic treatment was ineffective. Afterward, pathogen detection in cerebrospinal fluid using metagenomic next-generation sequencing revealed Providencia rettgeri. Intravenous maximum-dose ampicillin was administered for 5 weeks, and the patient's symptoms resolved. Cryptogenic Providencia rettgeri brain abscess typically occurs in patients with impaired immunity. Our patient exhibited a sudden onset with non-typical neuroimaging findings, requiring differentiation of the lesion from stroke and brain tumor. Metagenomic next-generation sequencing was important in identifying the pathogen. Rapid diagnosis and appropriate use of antibiotics were key to obtaining a favorable outcome.

Healthy adults with Streptococcus pneumoniae meningitis and Streptococcus pneumoniae subdural abscess: two case reports and a literature review.

We present the cases of two otherwise healthy adults, one with meningitis and another with a subdural abscess, with both conditions attributable to Streptococcus pneumoniae. A 31-year-old man was admitted with a 3-day history of fever, headache, and vomiting. Physical examination revealed intermittent confusion, irritability, and neck stiffness. Cerebrospinal fluid (CSF) culture was positive for S. pneumoniae. Contrast-enhanced magnetic resonance imaging (C-MRI) revealed multiple small lesions on the bilateral frontal lobes. Intravenous ceftriaxone and vancomycin were administered, followed by intravenous moxifloxacin. His symptoms resolved within 3 months. Additionally, a 66-year-old man was admitted for acute fever with confusion, abnormal behavior, and a recent history of acute respiratory infection. Physical examination revealed confusion, neck stiffness, and a positive right Babinski sign. CSF metagenomic analysis detected S. pneumoniae. C-MRI disclosed left occipitotemporal meningoencephalitis with subdural abscesses. Intravenous ceftriaxone was administered for 3 weeks. His condition gradually improved, with resorbed lesions detected on repeat MRI. This study expanded the clinical and imaging spectra of S. pneumoniae meningitis. In healthy adults, S. pneumoniae can invade the brain, but subdural abscess is a rare neuroimaging manifestation. Early diagnosis of S. pneumoniae meningitis by high-throughput sequencing and flexible treatment strategies are necessary for satisfactory outcomes.

Case Report: Early-Onset Charcot-Marie-Tooth 2N With Reversible White Matter Lesions Repeatedly Mimicked Stroke or Encephalitis.

Introduction: Charcot-Marie-Tooth (CMT) disease is a rare group of peripheral neuropathies with high clinical and genetic heterogeneity. CMT type 2N (CMT 2N) is a rare subtype of CMT with few clinical reports. The clinical presentation mimics that of other diseases, frequently leading to misdiagnoses. We present a case of CMT 2N with reversible white matter lesions (WMLs), which repeatedly mimicked stroke or encephalitis. We include a literature review to the improve management of this disease. Case Description: An 8-year-old boy was admitted to the hospital with slurred speech and limb weakness that had persisted for 1 day. Physical examination revealed lethargy, dysarthria, and a positive bilateral Babinski sign. Cerebrospinal fluid (CSF) analysis showed no abnormalities. Brain magnetic resonance imaging (MRI) revealed symmetrical abnormal signal areas in the paraventricular white matter and corpus callosum. The patient was suspected of having viral encephalitis and recovered rapidly after treatment.He was hospitalized 3 years later for limb weakness, barylalia, and facial paralysis persisting for 1 day. MRI showed an abnormal signal in the bilateral corona radiata. He was suspected of having a stroke or encephalitis. He was completely recovered after treatment.After a second 3-year span, he was admitted for another stroke-like episode. Physical examination revealed facial-lingual hemiparesis, mild atrophy of the left thenar muscle, decreased muscle strength in the extremities, and disappearance of the tendon reflex. MRI revealed more pronounced abnormal signals in the bilateral centrum semiovale and corpus callosum. Antibodies against autoimmune encephalitis were negative. A nerve conduction velocity (NCV) study showed motor and sensory four-limb nerve demyelination with axonal damage, most notably at the distal end. His symptoms were resolved after active treatment. A follow-up MRI showed the complete disappearance of the abnormal white matter signal. Whole exon sequencing showed a heterozygous mutation [c.2093C > T(p.Ser698Phe)] in the alanyl-tRNA synthetase 1 gene (AARS1). His mutation, clinical features, and electrophysiological testing led to a diagnosis of CMT 2N. Discussion: Early-Onset CMT 2N with reversible WMLs can often mimic stroke or encephalopathy. Affected individuals may show an atypical posterior reversible encephalopathy syndrome (PRES) on MRI. Careful family history assessment, physical examination, nerve conduction studies, MRIs, and genetic testing are essential for early diagnosis. Further studies are required to confirm these findings.