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Cureus Jul 2020Pembrolizumab, one of many novel immune checkpoint inhibitors (ICPi), is a monoclonal antibody that enhances immunity against cancer cells. Extensive escalation in...
Pembrolizumab, one of many novel immune checkpoint inhibitors (ICPi), is a monoclonal antibody that enhances immunity against cancer cells. Extensive escalation in immune activity predisposes to unsought immune-related adverse events. Due to progressive mesothelioma, a 67-year-old man was referred to the research unit and enrolled in a clinical trial with a cluster of differentiation (CD) 27 chemotherapeutic agent. He began crossover treatment and received just two doses of pembrolizumab, 33 and 16 days prior to admission. He subsequently presented to the emergency department with three days of acute onset severe diplopia and a drooping left eye. Acetylcholine receptor (AChR) antibodies returned positive at 13.9 nmol/L, and a diagnosis of ocular myasthenia gravis (OMG) was made. During his hospitalization, the patient was managed with methylprednisolone 80 mg intravenously daily, with conversion to prednisone 60 mg by mouth daily at time of discharge. Neuro-ophthalmology consultation was sought in the outpatient setting, and the patient was started on pyridostigmine. He was readmitted two weeks later with symptoms of progressive diffuse weakness, unsteady gait, and dysphagia, all in the setting of persistent diplopia. Intravenous immunoglobulin (IVIG) was promptly initiated, in addition to the pyridostigmine previously initiated in the outpatient setting. Unfortunately, after three IVIG treatments, the patient had experienced little improvement in his symptoms, and therefore elected hospice care. Although ICPis have revolutionized the management of a multitude of malignancies, recognition of immune-related adverse events is of critical importance.
PubMed: 32685327
DOI: 10.7759/cureus.9192 -
Indian Journal of Ophthalmology Feb 2022We aimed to study the success of prism in regard to diplopia resolution score and associated factors in patients presenting with symptomatic diplopia arising from...
PURPOSE
We aimed to study the success of prism in regard to diplopia resolution score and associated factors in patients presenting with symptomatic diplopia arising from various etiologies.
METHODS
In this descriptive, retrospective study diplopia resolution among 31 patients who were prescribed prism were analyzed.
RESULTS
Fifty-four patients were evaluated for diplopia and 31 were included for the study done over 3 years. The mean follow-up was 15 months. Esotropia, exotropia, and hypertropia were seen in 39%, 51%, and 19.4% of patients, respectively. Furthermore, 71% received Fresnel prism and 29% were given ground glass prism. The mean prism power prescribed was 13.3 PD. 87% had complete resolution of diplopia; 96.8% continued usage of prism. High success rates were seen among patients with decompensated strabismus, sixth and fourth nerve palsy. Horizontal prism and oblique prism in the form of Fresnel prism yielded complete resolution of diplopia (P = 0.028). There was no association between the success of prism and etiology (P 0.058), history of trauma (P = 0.212), and type of deviation (P = 0.387). The study showed that oblique Fresnel prism can be considered for combined deviation.
CONCLUSION
Our study showed prism to be effective in alleviating diplopia over a varied range of etiologies.
Topics: Diplopia; Esotropia; Humans; Oculomotor Muscles; Retrospective Studies; Strabismus; Trochlear Nerve Diseases; Vision, Binocular
PubMed: 35086246
DOI: 10.4103/ijo.IJO_939_21 -
Neurology India 2021Rise in intracranial tension (ICT) has varied clinical presentation which can range from subtle disturbances like headache to frank neurologic impairment. An important...
Rise in intracranial tension (ICT) has varied clinical presentation which can range from subtle disturbances like headache to frank neurologic impairment. An important aspect is rapidity of rise of ICT. Pseudotumor cerebri is associated with many syndromes, toxication, and drugs. Our case is a unique one given the rarity of eltroxin, which is otherwise relatively safe drug and commonly used in this part of the world, induced Pseudotumor cerebri. Our patient had dramatic response to discontinuation of levothyroxine.
Topics: Headache; Humans; Pseudotumor Cerebri; Syndrome; Thyroxine
PubMed: 34747819
DOI: 10.4103/0028-3886.329602 -
Romanian Journal of Ophthalmology 2022A 36-year-old female patient presented to our clinic with a two months history of diplopia and dizziness. The symptoms appeared gradually and increased in frequency and...
A 36-year-old female patient presented to our clinic with a two months history of diplopia and dizziness. The symptoms appeared gradually and increased in frequency and intensity. She had no significant medical history and she did not take any medication. A full ophthalmological consult was performed, which revealed restricted ocular motility in the left eye (LE), in left gaze. Otherwise, the examination showed no pathological findings: best corrected visual acuity (BCVA) both eyes (OU) 1 (Snellen chart), normal slit lamp examination and pupillary reflexes, normal intraocular pressure (IOP) and fundus aspect. Diplopia tests revealed a horizontal diplopia, exacerbated in left gaze. Sixth nerve palsy suspicion was raised and the patient was directed to the neurology department. Following magnetic resonance imaging, with angiographic sequence, a complex intracerebral vascular malformation that interacted with the cranial nerves and determined horizontal diplopia, was found. For a correct diagnosis, we needed a good collaboration between various medical specialties, especially ophthalmology and neurology, because patients with diplopia often present for the first time at the ophthalmologist. BCVA = best corrected visual acuity, IOP = intraocular pressure, LE = left eye, RE = right eye.
Topics: Abducens Nerve Diseases; Adult; Diplopia; Female; Humans; Intraocular Pressure; Tonometry, Ocular; Visual Acuity
PubMed: 35531446
DOI: 10.22336/rjo.2022.15 -
Neurology. Genetics Aug 2023The objective of this study was to describe the first patient with recurrent ataxia and diplopia in association with a pathogenic variant in .
OBJECTIVES
The objective of this study was to describe the first patient with recurrent ataxia and diplopia in association with a pathogenic variant in .
METHODS
We identified a girl with a heterozygous pathogenic variant and performed thorough phenotyping.
RESULTS
A 10-year-old girl was previously well with normal intelligence. She had recurrent diplopia, dysmetria, and unsteady gait, which occurred only in the context of febrile illnesses. EEG during her initial acute episode showed multifocal epileptiform discharges, with similar findings seen on a follow-up study 3 months later when she was well. Brain MRI finding was normal. A gene panel identified a de novo variant, p.Arg847Gln, classified as likely pathogenic. One year after her initial presentation, the girl is well and developmentally normal and has never had an event concerning for seizure.
DISCUSSION
This case presentation demonstrates that pathogenic variants should be considered in children with transient ataxia, dysmetria, and diplopia in the context of viral febrile illnesses, even if there is no history of seizures. While there are clinical and molecular data suggesting that SCN8A dysfunction can cause temperature-sensitive phenotypes, further research is necessary to determine how the functional changes caused by our patient's variant result in her unique phenotype.
PubMed: 37440794
DOI: 10.1212/NXG.0000000000200085 -
Revue Medicale de Liege Jul 2020We report the story of an 11-year-old girl admitted to the emergency room for diplopia and divergent squint. Promptly apparead a fluctuating ptosis, a nasal voice and...
We report the story of an 11-year-old girl admitted to the emergency room for diplopia and divergent squint. Promptly apparead a fluctuating ptosis, a nasal voice and swallowing disorders, evoking the diagnosis of autoimmune myasthenia. The latter has been confirmed with electromyogram. Treatment with corticoids, plasmapheresis and pyridostigmine allowed symptoms control. Thymectomy by left thoracoscopy, non-robot assisted, was performed 6 months after the appearance of the first clinical signs for the purpose of remission.
Topics: Adrenal Cortex Hormones; Child; Diplopia; Female; Humans; Myasthenia Gravis; Thoracoscopy; Thymectomy
PubMed: 32779904
DOI: No ID Found -
Cureus Sep 2022Sixth nerve palsies present with horizontal diplopia and typically have a neurological or neurovascular aetiology. They can be confirmed by clinically evaluating the...
Sixth nerve palsies present with horizontal diplopia and typically have a neurological or neurovascular aetiology. They can be confirmed by clinically evaluating the velocity of the abducting saccade, which is slowed. Three cases are presented in which the patients had apparent defective abduction of one eye, resulting from not only neurological causes but also orbital causes. Clinicians should have a high index of suspicion in patients with defective abduction without diplopia and should include apparent defective abduction without diplopia (ADAD) in the list of potential differential diagnoses, considering not only neurological involvement but also orbital involvement.
PubMed: 36258930
DOI: 10.7759/cureus.29155 -
Neuro-ophthalmology (Aeolus Press) 2022Giant cell arteritis (GCA) is the most common vasculitis in older adults with permanent vision loss as a feared complication. Diplopia has been reported in a small...
Giant cell arteritis (GCA) is the most common vasculitis in older adults with permanent vision loss as a feared complication. Diplopia has been reported in a small percentage of patients with visual manifestations. The goal of this study was to determine the population-based rates and patterns of binocular diplopia from GCA. The Rochester Epidemiology Project (REP), a medical records linkage system was used to identify all residents of Olmsted County, Minnesota, USA, diagnosed with GCA between January 1, 1950 and December 31, 2019. Medical records were then reviewed to identify patients with binocular diplopia from GCA. There were 301 incident cases of GCA from 1950 to 2019. Fourteen (5%) patients presented with binocular diplopia. Of these 14 patients, nine (3%) had constant diplopia and five (2%) had transient diplopia. Among patients with constant diplopia, cranial nerve VI involvement was suspected in four (44%) cases. Systemic symptoms and inflammatory markers were similar in patients with and without diplopia. There was no difference in the rate of anterior ischaemic optic neuropathy between the two groups (7% vs. 7%, = 1.00). In conclusion, this population-based study showed that binocular diplopia was present in 5% of patients with GCA, which could either be transient or constant. GCA patients with diplopia had similar systemic manifestations and risk of vision loss as GCA patients without diplopia.
PubMed: 35273408
DOI: 10.1080/01658107.2021.1965627 -
Thyroid : Official Journal of the... Apr 2022Graves' eye disease, also called Graves' orbitopathy (GO), is a potentially debilitating autoimmune disease associated with retro-orbital inflammation and tissue...
Graves' eye disease, also called Graves' orbitopathy (GO), is a potentially debilitating autoimmune disease associated with retro-orbital inflammation and tissue expansion, involving both fibroblasts and adipocytes, resulting in periorbital edema, worsening proptosis, and muscle dysfunction with diplopia and may ultimately threaten sight. Accumulating evidence has indicated that autoantibodies to the thyrotropin receptor (TSHR), which induce the hyperthyroidism of Graves' disease, also help mediate the pathogenesis of the eye disease in susceptible individuals through TSHR expression on retro-orbital cells. Since it has long been known that the effects of insulin-like growth factor 1 (IGF-1) and thyrotropin are additive, recent clinical trials with a human monoclonal IGF-1 receptor blocking antibody (teprotumumab; IGF-1R-B-monoclonal antibody [mAb]) have demonstrated its ability to induce significant reductions in proptosis, diplopia, and clinical activity scores in patients with GO. However, the molecular mechanisms by which such an antibody achieves this result is unclear. We have used Li-Cor In-Cell Western, Western blot, and immunohistochemistry to define levels of different proteins in mouse and human fibroblast cells. Proteomic array was also used to define pathway signaling molecules. Using CCK-8 and BrdU cell proliferation ELISA, we have analyzed proliferative response of these cells to different antibodies. We now show that a stimulating TSHR antibody was able to induce phosphorylation of the IGF-1R and initiate both TSHR and IGF-1R signaling in mouse and human fibroblasts. IGF-1R-B-mAb (1H7) inhibited all major IGF-1R signaling cascades and also reduced TSHR signaling. This resulted in the antibody-induced suppression of autophagy as shown by inhibition of multiple autophagy-related proteins (Beclin1, LC3a, LC3b, p62, and ULK1) and the induction of cell death by apoptosis as evidenced by activation of cleaved caspase 3, FADD, and caspase 8. Furthermore, this IGF-1R-blocking mAb suppressed serum-induced perkin and pink mitophagic proteins. Our observations clearly indicated that stimulating TSHR antibodies were able to enhance IGF-1R activity and contribute to retro-orbital cellular proliferation and inflammation. In contrast, an IGF-1R-B-mAb was capable of suppressing IGF-1R signaling leading to retro-orbital fibroblast/adipocyte death through the cell-extrinsic pathway of apoptosis. This is likely the major mechanism involved in proptosis reduction in patients with Graves' eye disease treated by IGF-1R inhibition.
Topics: Animals; Antibodies, Monoclonal; Apoptosis; Diplopia; Fibroblasts; Graves Disease; Graves Ophthalmopathy; Humans; Immunoglobulins, Thyroid-Stimulating; Inflammation; Insulin-Like Growth Factor I; Mice; Proteomics; Receptor, IGF Type 1; Receptors, Thyrotropin; Thyrotropin
PubMed: 34927457
DOI: 10.1089/thy.2021.0176 -
Archives of Craniofacial Surgery Apr 2020Due to the different handling properties of unsintered hydroxyapatite particles/poly-L-lactic acid (uHA/PLLA) and polycaprolactone (PCL), we compared the surgical...
BACKGROUND
Due to the different handling properties of unsintered hydroxyapatite particles/poly-L-lactic acid (uHA/PLLA) and polycaprolactone (PCL), we compared the surgical outcomes and the postoperative implantation accuracy between uHA/PLLA and PCL meshes in orbital fracture repair.
.METHODS
Patients undergoing orbital wall reconstruction with PCL and uHA/PLLA mesh, between 2017 and 2019, were investigated retrospectively. The anatomical accuracy of the implant in bony defect replacement and the functional outcomes such as diplopia, ocular motility, and enophthalmos were evaluated.
.RESULTS
No restriction of eye movement was reported in any patient (n=30 for each group), 6 months postoperatively. In the PCL group, no patient showed diplopia or enophthalmos, while the uHA/PLLA group showed two patients with diplopia and one with enophthalmos. Excellent anatomical accuracy of implants was observed in 27 and 22 patients of the PCL and uHA/PLLA groups, respectively. However, this study showed that there were neither any significant differences in the surgical outcomes like diplopia and enophthalmos nor any complications with the two well-known implants.
.CONCLUSION
PCL implants and uHA/PLLA implants are safe and have similar levels of complications and surgical outcomes in orbital wall reconstruction.
PubMed: 32380809
DOI: 10.7181/acfs.2020.00010