Background: The relationship between anti-estrogen drugs and macular telangiectasia type 2 (MacTel-2) remains unknown. Here we report a case with anti-estrogen maculopathy resembling MacTel-2 with improved visual function and macular morphology following cessation of anti-estrogen drugs. Case presentation: A 53-year-old woman presented with a 5-month history of central vision loss and anorthopia in both eyes. She had received oral tamoxifen followed by toremifene for 69 months. Funduscopy, fluorescein angiography, and optical coherence tomography (OCT) revealed MacTel-2-like findings OU. Fundus autofluorescence (FAF) showed hyper-autofluorescence at the fovea OU. Visual acuity, macular morphology on OCT, and FAF findings gradually improved after cessation of anti-estrogen drugs. Conclusions: In the present case, visual acuity, macular morphology, and impairment of the retinal pigment epithelium (RPE) improved following cessation of anti-estrogen drugs, suggesting the relationship between retinal toxicity of anti-estrogen drugs and the development of MacTel-2-like findings. From these results and the previous observations, toxicity of both photoreceptor and RPE cells caused by anti-estrogen drugs may contribute to the development of anti-estrogen maculopathy similar to MacTel-2.
To describe a rare case of cerebrospinal fluid hypovolemia syndrome after a traffic accident with abnormal eye movements. A 19-year-old man was referred to our clinic after being hit by a car five months ago while riding a bicycle. After the accident, he sometimes noticed oscillopsia, and had postural headaches and reading difficulties. His eye movement recording revealed square wave jerks during fixation and decreased pursuit gain during horizontal smooth pursuit. MR myelography detected cerebrospinal fluid leakage and the patient was diagnosed with cerebrospinal fluid hypovolemia. After undergoing epidural blood patch therapy, the leakage disappeared, and his postural headaches improved immediately. Square wave jerks and decreased pursuit gain improved, and his oscillopsia and reading difficulty also improved after therapy. A patient with cerebrospinal fluid hypovolemia presented with square wave jerks and decreased pursuit gain. Epidural blood patch therapy was effective for the symptoms. When treating patients with oscillopsia and postural headaches, we should consider the possibility of cerebrospinal fluid hypovolemia syndrome in the differential diagnosis.
This open-label, uncontrolled, single-arm, multicenter, phase 3 study evaluated the efficacy and safety of incobotulinumtoxinA in Japanese patients with blepharospasm. Eligible patients received incobotulinumtoxinA injections at fixed doses (50, 75, or 100 units [U] for those who had previously received botulinum toxin treatment; 50 U for treatment-naïve patients), followed by flexible doses up to 100 U for 48 weeks, with at least 6-week intervals. In total, 29 Japanese patients were enrolled (26 [89.7%] women, mean age 64.6 years, mean baseline Jankovic Rating Scale [JRS] severity score 3.24). The primary endpoint, the least squares mean of change in JRS severity scores from baseline to 6 weeks after the first injection, was −2.08 (95% confidence interval: −2.49, −1.66), meeting the prespecified efficacy criteria. The secondary endpoint results (JRS severity, frequency, and total scores for 48 weeks; Blepharospasm Disability Index; Patient Evaluation of Global Response; and fast blinking test) supported the efficacy of repeated incobotulinumtoxinA injections. Adverse events (AEs) occurred in 19 (65.5%) patients, with eyelid ptosis being the most common treatment-related AE (4 [13.8%] patients). No severe or serious AEs were reported. IncobotulinumtoxinA demonstrated sustained efficacy in Japanese patients with blepharospasm, without new safety concerns. (Japan Registry of Clinical Trials identifier, jRCT2031230711)
Interleukin-6 (IL-6) and insulin-like growth factor-1 (IGF-1) are involved in immune regulation and tissue repair, but their combined effects on gastrointestinal mucosa remain unclear. We report a case of new-onset ulcerative colitis with pyoderma gangrenosum after sequential IL-6 and IGF-1 receptor inhibition. The patient developed severe ulcerative colitis and pyoderma gangrenosum refractory to corticosteroids but showed rapid improvement after infliximab therapy, with endoscopic evidence of mucosal improvement. This case highlights a potential role for tumor necrosis factor-α (TNF-α) blockade in reversing severe immune dysregulation induced by IL-6 and IGF-1 inhibition.
15 March 2022 in American Journal of Ophthalmology Case Reports
Cerebrospinal fluid hypovolemia syndrome (CHS) is a rare clinical entity that can be caused by spontaneous cerebrospinal fluid (CSF) leakage. The aim of this study is to report a rare case of CHS after a traffic accident in a patient who presented with diplopia and ptosis with fluctuation and was initially diagnosed with ocular myasthenia gravis. A 29-year-old man exhibited fluctuating left ptosis and diplopia after a traffic accident. Although he was suspected of having myasthenia gravis and was treated using oral pyridostigmine bromide, his symptoms did not improve. He also had orthostatic headaches and malaise after the accident. His symptoms were suspected to be associated with traumatic cerebrospinal fluid hypovolemia. After 1000-mL fluid replacement, his diplopia and ptosis improved, and orbital T2-weghted MRI detected a high-signal zone around the optic nerve. We diagnosed him with oculomotor nerve paresis associated with cerebrospinal fluid hypovolemia. The symptoms, including ptosis, diplopia, orthostatic headaches, and malaise, disappeared after epidural blood patch therapy. When treating patients with fluctuating ocular symptoms, such as diplopia and ptosis, who have a history of trauma and orthostatic headaches, the possibility of CHS should be considered in the differential diagnosis.