A Case of Delayed Oculo-Palato-Brachial Tremor after Pontine Infarction.
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ABSTRACT:
SUBMITTER: Ryu HS
PROVIDER: S-EPMC8175821 | biostudies-literature |
REPOSITORIES: biostudies-literature
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| S-EPMC6739662 | biostudies-literature
Project description:Although pontine strokes account for a small percentage of all ischaemic events, they can be associated with significant initial disability. These lesions may be missed on computed tomography and therefore magnetic resonance imaging is generally preferred for the assessment of brainstem strokes. The aetiopathogenesis of isolated pontine infarcts, not due to a significant compromise (occlusion or dissection) in the vertebrobasilar territory, still remains to be fully characterised. These strokes present with different symptoms, depending on the lesion location and size, partly reflecting the anatomical variability of the vertebrobasilar vessels. Progressive neurological deterioration is relatively common and has been associated with the extension of such lesions. However, many patients with significant infarcts in the pons will do well in the future and initial diffusion-weighted imaging may not add useful prognostication to the clinical assessment. We discuss here a case where an initially progressive presentation was associated with a marked improvement in both clinical and radiological assessments at 42 days.A 49-year-old white British man presented with left-sided weakness, incoordination, unsteadiness, cerebellar ataxic dysarthria and dysphonia. A baseline magnetic resonance imaging scan with diffusion-weighted imaging, T1-weighted and T2-weighted sequences showed an acute bilateral pontine infarct. On a repeat scan at 42 days, there was a 57.5% decrease in the size of the lesion on the high-resolution three-dimensional T1-weighted image and a corresponding improvement in the symptoms and the clinical assessments of this patient. The reduction in infarct size was also comparable to the decrease calculated between the baseline diffusion-weighted and the follow-up fluid attenuated inversion recovery sequences.This case report discusses the significant clinical improvement and corresponding lesion reduction in a patient that presented with worsening neurological symptoms and was diagnosed with acute bilateral ischaemic pontine infarction. Further studies, utilising structural and functional magnetic resonance imaging with follow-up scans, are needed to provide better insights into the underlying aetiopathology and recovery mechanisms of pontine stroke. These will help define the relationship between imaging parameters and outcome allowing for better prognosis along with the development of relevant rehabilitation programs for this group of patients.
| S-EPMC4203895 | biostudies-other
Project description:Little is known about dysphagia after pontine infarction. In this study, we evaluated the incidence of dysphagia after isolated pontine infarction and identified the predictive factors for the occurrence of dysphagia. A total of 146 patients were included in this study. All patients underwent clinical testing for dysphagia within 1 day after admission and at the time of discharge. We compared the incidence of dysphagia between patients with unilateral pontine infarction and those with bilateral pontine infarction. To evaluate the functional status of patients, we investigated their initial modified Rankin Scale (mRS) score and initial National Institutes of Health Stroke Scale (NIHSS) score within 1 day of admission. Of 146 patients, 50 (34.2%) had dysphagia initially within 1 day after admission. At the second evaluation at the time of discharge, dysphagia was diagnosed in 24 patients (16.4%). Patients with bilateral pontine infarction were more likely to present with dysphagia. In addition, clinical severity (in terms of mRS and NIHSS scores) was identified as a predictor of dysphagia in patients with cerebral infarction (multiple binary logistic regression analysis, mRS: P = 0.011, NIHSS: P = 0.004). Dysphagia frequently occurs in patients with isolated pontine infarction. Clinicians should pay particular attention to the occurrence of dysphagia, especially in patients with bilateral pontine infarction or high functional disability.
| S-EPMC6199935 | biostudies-literature
Project description:BackgroundOculo-facio-cardio-dental (OFCD) syndrome is a rare condition that affects the eyes, face, heart, and teeth of patients. One notable dental characteristic of OFCD is radiculomegaly, or root gigantism, which highlights the role of dentists in detecting this syndrome. OFCD is an X-linked dominant syndrome that results from a variant in the BCOR gene. Our study presents the first documented case of OFCD in Vietnam and reports a novel BCOR gene variant observed in this case.Case presentationA 19-year-old Vietnamese female patient with an extremely long root with an abscess was clinically examined for the expression of OFCDs. The radiograph and the variant in BCOR gene were also evaluated. We identified abnormalities in the teeth, as well as ocular, facial, and cardiac features, with radiculomegaly of the canines being a specific symptom for OFCDs. The patient's genetic analysis revealed a pathogenic heterozygous deletion at intron 11 of the BCOR gene, representing a novel variant.ConclusionOculo-facio-cardio-dental syndrome (OFCD) is an extremely rare condition characterized by abnormalities in the eyes, face, heart, and teeth, often caused by variants in the BCOR gene. Radiculomegaly, or enlarged dental roots, is a key diagnostic feature of OFCD, and early detection is crucial for preventing future dental complications.
| S-EPMC10765806 | biostudies-literature
Project description:A 2-month-old boy presented to us with bilateral microtia, left lower motor neuron facial palsy, micrognathia, hemivertebra, bifid rib, bifid thumb and absent/hypoplastic right-sided depressor anguli oris. He had bilateral external auditory canal atresia, although response to loud sound was present. Brain stem evoked response audiometry (BERA) was advised at 3 months of age. Karyotype was normal. We diagnosed him as a case of oculo-auriculo-vertebral spectrum. Child was discharged on request by the family with the plan for bone-anchored hearing aid after BERA and plan for pinna and ear canal reconstruction at a later age but child did not come for any follow-up visit. On telephonic enquiry, it was found that he is thriving well but has developmental delay including speech delay. We conclude that children presenting with external ear abnormalities should be screened for multiple congenital anomalies so that a multidisciplinary approach to management can be planned.
| S-EPMC7931756 | biostudies-literature
Project description:BACKGROUND AND PURPOSE:Subclinical atherosclerotic plaques are common in patients with pontine infarctions (PIs) but without basilar artery (BA) stenosis. We hypothesized that BA plaque locations may differ by PI type and vertical location as well as vertebrobasilar artery geometry. METHODS:Ninety-six patients with PI but without BA stenosis on magnetic resonance imaging (MRI) and magnetic resonance angiography were enrolled. PIs were classified by type (paramedian, deep, or lateral) and vertical location (rostral, middle, or caudal). Patients underwent high-resolution MRI to evaluate BA plaque location (anterior, posterior, or lateral). The mid-BA angle on anteroposterior view and angle between the BA and dominant vertebral artery (BA-VA angle) on lateral view were measured. RESULTS:The PIs were paramedian (72.9%), deep (17.7%), and lateral (9.4%) type with a rostral (32.3%), middle (42.7%), and caudal (25.0%) vertical location. The BA plaque locations differed by PI type (P=0.03) and vertical location (P<0.001); BA plaques were most frequent at the posterior wall in paramedian (37.1%) and caudal (58.3%) PIs and at the lateral wall in lateral (55.5%) and middle (34.1%) PIs. The BA-VA and mid-BA angles differed by BA plaque and PI vertical location; the greatest BA-VA angle was observed in patients with posterior plaques (P<0.001) and caudal PIs (P<0.001). Greatest mid-BA angles were observed with lateral BA plaques (P=0.03) and middlelocated PIs (P=0.03). CONCLUSIONS:Greater mid-BA angulation may enhance lateral plaque formation, causing lateral and middle PIs, whereas greater BA-VA angulation may enhance posterior plaque formation, causing paramedian or caudal PIs.
| S-EPMC5836573 | biostudies-literature
Project description:Progressive motor deficits are relatively common in acute pontine infarction and frequently associated with increased functional disability. However, the factors that affect the progression of clinical motor weakness are largely unknown. Previous studies have suggested that pontine infarctions are caused mainly by basilar artery stenosis and penetrating artery disease. Recently, lower pons lesions in patients with acute pontine infarctions have been reported to be related to progressive motor deficits, and ensuing that damage to the corticospinal tracts may be responsible for the worsening of neurological symptoms. Here, we review studies on motor weakness progression in pontine infarction and discuss the mechanisms that may underlie the neurologic worsening.
| S-EPMC4396117 | biostudies-other
Project description:IntroductionCentral pontine myelinolysis (CPM) is an acquired demyelinating lesion of the basis pontis that typically occurs after rapid correction of hyponatremia. There are only a few reported cases of patients without symptoms that have demonstrated CPM on imaging.Case presentationWe report the case of a 26-year-old Hispanic male with history of alcohol abuse who was transferred to our medical center for acute onset diffuse abdominal pain. During his work up, a computed tomography scan demonstrated a large pancreatic mass. He underwent an endoscopic guided biopsy which demonstrated a rare and aggressive natural killer T cell lymphoma. His laboratory values were consistent with hyponatremia, which the medical team gently corrected. An MRI was performed for staging purposes which revealed findings consistent with CPM. A full neurological exam demonstrated no deficits.Materials and methodsWE CONDUCTED A PUBMED SEARCH USING THE FOLLOWING KEYWORDS: asymptomatic, central, pontine, and myelinolysis in order to find other case reports of asymptomatic CPM.ResultsOf the 29 results, only 6 previous case reports with English language abstracts of asymptomatic CPM were present since 1995.ConclusionDespite slow correction of hyponatremia, CPM can be an important consequence, especially in patients with chronic alcoholism. Although this patient did not demonstrate any neurological deficits, the fact that there were changes seen on MRI should caution physicians in aggressively treating hyponatremia. Furthermore, if there is a decision to treat, then fluid restriction and reversal of precipitating factors (i.e. diuretics) should be used initially, unless there is concern for hypovolemia.
| S-EPMC3506049 | biostudies-literature
Project description:Myopericytoma (MPC) is a benign soft tissue tumor that develops from perivascular myoid cells and is part of the perivascular tumor group. MPC most commonly occurs in the subcutaneous soft tissues of the extremities, while intracranial MPC is remarkably rare. Herein, we report the case of a 45-year-old woman with myopericytoma who had a 2-week history of recurrent dizziness. Magnetic resonance imaging (MRI) revealed an irregular mass in the pons, with nodular enhancement of the mass on contrast-enhanced scans. The mass was considered a vascular lesion and was highly suspected to be a hemangioblastoma, prompting surgical intervention for the patient. The postoperative pathological report corrected the initial diagnosis, hemangioblastoma, to MPC. Intracranial MPC is extremely rare and there are no detailed imaging sources for this condition; furthermore, MPC occurrence in the pons has not been reported previously. This report presents the etiological characteristics intracranial MPC as visualized through MRI data alongside a comparative discussion on other reported diagnoses that resemble MPC. The case findings will provide a more widespread understanding for radiologists regarding the differential diagnosis of intracranial blood-rich supply lesions.
| S-EPMC9148993 | biostudies-literature
Project description:•We report 2 patients with pontine infarcts showing transient conjugate eye deviation.•Conjugate eye deviation resolved within a few days in both patients.•Small restricted dorsomedial pontine lesions can produce conjugate eye deviation.
| S-EPMC6006905 | biostudies-literature