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Neurology. Clinical Practice Oct 2022This case report describes a patient with mesencephalic MRI signal abnormality and diplopia, possibly associated with severe acute respiratory syndrome coronavirus 2...
OBJECTIVE
This case report describes a patient with mesencephalic MRI signal abnormality and diplopia, possibly associated with severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2) infection.
METHODS
We describe a boy with binocular diplopia and nystagmus. The pattern of serology positivity and negative direct research of SARS-CoV-2 RNA in our patient allowed us to consider novel coronavirus as the trigger of possible immune-mediated phenomena against the central nervous system.
RESULTS
During hospitalization, blood tests revealed a recent SARS-CoV-2 infection. MRI revealed hyperintensity of the mesencephalic tegmentum and periaqueductal region, consistent with an inflammatory lesion of the midbrain tegmentum. Viral and bacterial molecular screening on cerebrospinal fluid and isoelectrofocusing analysis, anti-myelin oligodendrocyte glycoprotein, anti-aquaporine-4, and anti-N-methyl-d-aspartate antibodies were negative. The patient was treated with steroids and immunoglobulin therapy with complete remission of neurologic symptoms.
DISCUSSION
This report expands the spectrum of pediatric COVID-19-associated neurologic symptoms and highlights a possible isolated neurologic COVID-19-related symptom.
PubMed: 36380889
DOI: 10.1212/CPJ.0000000000200076 -
International Journal of Clinical... 2023In this study, we conducted a meta-analysis to assess the efficacy and safety of teprotumumab in treating thyroid eye disease. We searched the Cochrane Library, PubMed,... (Meta-Analysis)
Meta-Analysis Review
In this study, we conducted a meta-analysis to assess the efficacy and safety of teprotumumab in treating thyroid eye disease. We searched the Cochrane Library, PubMed, and Embase databases from inception to May 25, 2022, and included all randomized controlled trials. Odds ratios (ORs) were calculated using fixed- or random-effect models. A total of three studies involving 341 patients were identified. Overall, the analysis revealed that teprotumumab demonstrated superior integrated proptosis response compared to placebo in both the intention-to-treat (ITT) population (OR = 17.81, 95% CI = [10.32, 30.76], = 50%) and per-protocol population (OR = 24.53, 95% CI = [12.96, 46.45], = 14%). Furthermore, patients receiving teprotumumab showed significant improvement in overall response (OR = 8.35, 95% CI = [4.74, 14.71], = 79%), diplopia response (OR = 5.53, 95% CI = [3.24, 9.44], = 0%), and achieving a clinical activity score (CAS) of 0 or 1 (OR = 6.26, 95% CI = [3.87, 10.12], = 0%). Moreover, patients treated with teprotumumab experienced greater improvements in proptosis (MD = -2.49, 95% CI = [-2.54, -2.45], = 98%) and Graves' ophthalmopathy-specific quality of life (GO-QOL, MD = 11.48, 95% CI = [11.03, 11.93], = 95%). However, it is important to note that patients receiving teprotumumab had a higher risk of adverse events, including serious adverse events, gastrointestinal adverse reactions, and muscle spasms. In summary, teprotumumab demonstrated greater improvement in proptosis response, proptosis, diplopia response, overall response, GO-QOL, and CAS. Nonetheless, it should be considered that its use is associated with a higher risk of adverse events.
Topics: Humans; Graves Ophthalmopathy; Quality of Life; Diplopia; Randomized Controlled Trials as Topic; Exophthalmos
PubMed: 37588100
DOI: 10.1155/2023/6638089 -
Journal of Personalized Medicine Feb 2024Although the reported frequency of diplopia is between 10 to 40% of patients with Parkinson's disease (PD) and other movement disorders, it remains one of the most... (Review)
Review
INTRODUCTION
Although the reported frequency of diplopia is between 10 to 40% of patients with Parkinson's disease (PD) and other movement disorders, it remains one of the most undiagnosed non-motor symptoms. Furthermore, it has a major impact on the quality of life of these patients. The aim of this study is to systematically review the literature regarding the frequency, causes, and implications of diplopia in movement disorders.
METHODOLOGY
An electronic search was conducted in March and June 2023 using the PubMed database in order to identify appropriate studies. Studies that were written in English, that represented observational, analytical studies, and case reports, and that provided information regarding diplopia in movement disorders were included in the systematic review.
RESULTS
A total of 686 articles were identified out of which 43 met the inclusion criteria. The studies included in the systematic review ranged from descriptive studies (case reports and case series) to analytical-observational studies (cross-sectional studies, prospective and retrospective cohort studies, and case-control studies). In Parkinson's disease, the incidence of diplopia ranged from 10 to 38%. In these patients, diplopia was linked to the presence of visual hallucinations and cognitive decline but also to convergence insufficiency and the presence of motor fluctuations. Cases of diplopia secondary to deep brain stimulation were also reported. Diplopia was associated with longer disease duration and worse motor and non-motor scores. Diplopia was also reported in other movement disorders such as multiple system atrophy (frequency as high as 18%) and progressive supranuclear palsy (frequency as high as 39%) and was associated with increased mortality and shorter duration in life span.
CONCLUSIONS
Diplopia occurs in up to 38% of patients with movement disorders and has a negative impact on their health-related quality of life. Treating physicians should actively ask about diplopia and other ophthalmological symptoms, as many patients do not spontaneously report them. The pathophysiology of diplopia is complex, and it involves heterogeneous peripheral and central mechanisms. The management of these patients should involve a multidisciplinary team of health professionals in order to provide appropriate, tailored management.
PubMed: 38541012
DOI: 10.3390/jpm14030270 -
Annals of Maxillofacial Surgery 2020White-eyed blowout fracture is often found in pure orbital floor blowout fracture among pediatric patients. Unlike common orbital blowout fractures with apparent...
White-eyed blowout fracture is often found in pure orbital floor blowout fracture among pediatric patients. Unlike common orbital blowout fractures with apparent clinical signs, the diagnosis of white-eyed orbital blowout fractures is difficult because of minimal soft-tissue signs. This report describes an early missed-out diagnosis of a white-eyed blowout fracture in a 7-year-old child, due to negligible soft-tissue manifestation.
PubMed: 32855945
DOI: 10.4103/ams.ams_150_19 -
Therapeutics and Clinical Risk... 2019Thyroid eye disease (TED) is a complex, debilitating autoimmune disease that causes orbital inflammation and tissue remodeling, resulting in proptosis, diplopia, and in... (Review)
Review
Thyroid eye disease (TED) is a complex, debilitating autoimmune disease that causes orbital inflammation and tissue remodeling, resulting in proptosis, diplopia, and in severe cases, loss of vision. TED can lead to facial disfigurement and severely impact patients' quality of life. Although the course of TED was identified over 60 years ago, effective treatment options have proved to be challenging. Current treatments such as glucocorticoid therapy and orbital radiation focus on reducing orbital inflammation. However, these therapies fail to modify the disease outcomes, including proptosis and diplopia. Recent advances in the understanding of the molecular basis of TED have facilitated the development of targeted molecular therapies such as teprotumumab, an insulin-like growth factor-1 receptor inhibiting monoclonal antibody. In recent phase 2 and phase 3 randomized placebo-controlled trials, teprotumumab rapidly achieved improvement in clinical endpoints defining TED, including improved proptosis and diplopia. Dramatic improvement in clinical outcomes achieved after teprotumumab therapy during active TED are heretofore singular and comparable only to surgical therapies achieved during the inactive phase of TED. The advent of effective medical therapy can lead to a paradigm shift in the clinical management of TED. This review will provide an overview of TED, its epidemiology, insight into the molecular biology of the disease, clinical characteristics and diagnosis, and current and emerging treatment modalities.
PubMed: 31814726
DOI: 10.2147/TCRM.S193018 -
Clinical & Experimental Optometry Sep 2020Mucocele of the paranasal sinuses is a slowly expanding benign lesion developing when there is impeded physiological drainage of the mucous produced by the epithelial... (Review)
Review
Mucocele of the paranasal sinuses is a slowly expanding benign lesion developing when there is impeded physiological drainage of the mucous produced by the epithelial lining of the paranasal sinuses, at the sinus ostium, which is an opening that connects the sinus to the nasal cavity. Aetiologies of ostial occlusion include infection, allergy, trauma, previous surgery, benign neoplasm (osteoma or fibrous dysplasia), and malignant or metastatic tumours. Mucoceles commonly develop in the frontal sinus (70-80 per cent), followed by the ethmoid (25 per cent), frontoethmoidal (10-14 per cent), and maxillary (three per cent or less) sinuses. The most common manifestations in these cases are ocular oedema, proptosis (22-83 per cent), and diplopia (28 per cent). Due to these ocular signs and symptoms, the optometrist may be first in line managing paranasal sinus disease patients, reducing the risk of permanent damage. A case report and review of frontoethmoidal mucocele will be discussed in this report, to include the role of the optometrist in its management and treatment.
Topics: Diagnosis, Differential; Diplopia; Ethmoid Sinus; Frontal Sinus; Humans; Magnetic Resonance Imaging; Male; Middle Aged; Mucocele; Paranasal Sinus Diseases; Tomography, X-Ray Computed
PubMed: 31773805
DOI: 10.1111/cxo.13006 -
BMJ Case Reports Nov 2021A young woman presented to neurology with a 1 month history of progressive diplopia on lateral gaze and a 1 week history of headaches. On examination she was found to...
A young woman presented to neurology with a 1 month history of progressive diplopia on lateral gaze and a 1 week history of headaches. On examination she was found to have complex ophthalmoparesis with binocular horizontal diplopia, failure of abduction bilaterally and limited upgaze with convergence-retraction nystagmus. The rest of the neurological examination was normal. She was admitted for investigations: blood, CT brain, MR brain and lumbar puncture results were normal. Anti-GD1a antibodies were strongly positive; anti-GM1, anti-GM2 and anti-GD1b were also positive. On follow-up 3 weeks later, the complex ophthalmoplegia persisted. It was decided to treat with intravenous immunoglobulins (IVIgs) with good response but recurrence at 2 weeks post infusion. She was treated with 4 weekly IVIg courses and remains responsive and controlled over 1 year since presentation but becomes symptomatic in the week running up to each dose; thus, disease modifying treatment is currently being considered.
Topics: Diplopia; Female; Gangliosides; Headache; Humans; Immunoglobulins, Intravenous; Ophthalmoplegia
PubMed: 34794976
DOI: 10.1136/bcr-2021-244273 -
Journal of Binocular Vision and Ocular... 2022Divergence insufficiency-type ET is a common cause of distance diplopia in elderly adults. A recent prospective multicenter data collection study has provided additional...
Divergence insufficiency-type ET is a common cause of distance diplopia in elderly adults. A recent prospective multicenter data collection study has provided additional guidance on management. Either base-out prism glasses or strabismus surgery were found to have high success rates, based on patient report of diplopia, and health-related quality-of-life domain scores. It was concluded that either prism or surgery were reasonable initial treatment strategies. Although allocation bias precluded formal comparison of prism versus surgery (evident in baseline differences between groups), there were hints that surgery may yield superior outcomes. For surgery, the most common approach was bilateral medial rectus recession, which was highly successful when assessed 10 weeks and 12 months postoperatively. It was unclear whether adjustable sutures were helpful, but in the vast majority of adjustable cases, additional recession at the time of adjustment was performed, suggesting that larger than standard surgical doses are needed.
Topics: Adult; Humans; Aged; Esotropia; Diplopia; Ophthalmologic Surgical Procedures; Vision, Binocular; Retrospective Studies; Treatment Outcome; Strabismus
PubMed: 36279481
DOI: No ID Found -
Oxidative Medicine and Cellular... 2020Surgical management of thyroid eye disease- (TED-) associated morbidity has been plagued by the complex interplay of different operative techniques. Orbital... (Review)
Review
Surgical management of thyroid eye disease- (TED-) associated morbidity has been plagued by the complex interplay of different operative techniques. Orbital decompression is the well-recognized procedure for disfiguring exophthalmos and dysthyroid optic neuropathy (DON). There are numerous published techniques described for the removal of the orbital bone, fat, or a combination. The diverse studies are noncomparative as they include different indications, stages of disease, and methods of evaluation. Thus, it is difficult to conclude the most efficient decompression technique. To obtain effective and predictable results, it is therefore important to propose a logical and acceptable clinical guideline to customize patient treatment. Herein, we developed an algorithm based on the presence of DON, preoperative existing diplopia, and severity of proptosis which were defined by patient's disabling symptoms together with a set of ocular signs reflecting visual function or cosmesis. More specifically, we aimed to assess the minimal but effective surgical technique with acceptable potential complications to achieve therapeutic efficacy. Transcaruncular or inferomedial decompressions are indicated in restoring optic nerve function in patients with DON associated with mild or moderate to severe proptosis, respectively. Inferomedial or fatty decompressions are effective to treat patients with existing diplopia associated with mild or moderate to severe proptosis, respectively. Fatty or balanced decompressions can improve disfiguring exophthalmos in patients without existing diplopia associated with mild to moderate or severe proptosis, respectively. Inferomedial or 3-wall decompressions are preferred to address facial rehabilitation in patients associated with very severe proptosis but without preoperative diplopia.
Topics: Clinical Decision-Making; Decompression, Surgical; Exophthalmos; Graves Ophthalmopathy; Humans
PubMed: 32963693
DOI: 10.1155/2020/3537675 -
Arquivos de Neuro-psiquiatria Jan 2024(MG) is an autoimmune disease usually caused by antibodies against the acetylcholine receptor (AChR-Abs), muscle-specific tyrosine kinase (MuSK-Abs), or low-density...
BACKGROUND
(MG) is an autoimmune disease usually caused by antibodies against the acetylcholine receptor (AChR-Abs), muscle-specific tyrosine kinase (MuSK-Abs), or low-density lipoprotein receptor-related protein 4 (LRP4-Abs). However, there are MG patients who do not have these antibodies and are thus said to have triple-seronegative (triple-SN) MG.
OBJECTIVE
This study aims to describe the frequency and clinical and epidemiological characteristics of patients with triple-SN MG.
METHODS
This was a retrospective cross-sectional study carried out through the analysis of medical records. Descriptive and analytical statistical analysis was performed comparing subgroups of myasthenic patients, classified according to serological profile.
RESULTS
The sample population consisted of 93 MG patients: 85 were positive for antibodies, 80 (86%) with AChR-Abs, 5 (5.4%) with MuSK-Abs, and no MG patients with LRP4-Abs. Eight patients (8.6%) had triple-SN MG; they had a median age at disease onset of 30 years (21-45). Their most common initial symptoms were ptosis, diplopia, and generalized weakness. Most patients presented with mild symptoms at their last visit, reflecting a median MG composite scale score of 4 (0-6), and 75% of patients had an adequate response to treatment.
CONCLUSION
Our study showed a low frequency of triple-SN MG in Brazilian MG patients. Triple-SN MG was predominant in females, who presented with ptosis, diplopia, and generalized weakness, and most patients had an adequate response to immunosuppressive treatment. There was no significant difference between triple-SN MG and the other subgroups.
Topics: Female; Humans; Young Adult; Adult; Middle Aged; Retrospective Studies; Diplopia; Cross-Sectional Studies; Autoantibodies; Receptor Protein-Tyrosine Kinases; LDL-Receptor Related Proteins; Myasthenia Gravis
PubMed: 38316426
DOI: 10.1055/s-0044-1779052