Bisphosphonate-induced orbital inflammation in a patient on chronic immunosuppressive therapy.

BACKGROUND: To report a case of orbital inflammation after bisphosphonate infusion in a patient who was already receiving immunosuppressive therapy. CASE PRESENTATION: A 56-year-old woman presented to the ophthalmology clinic with acute onset of right eye pain 24 h after receiving her first Zolendronic acid infusion. She has a past medical history of chronic inflammatory demyelinating polyneuropathy, Sjogren's syndrome, and systemic lupus erythematosus that have been controlled with immunosuppressive therapy for three years. Clinical ophthalmic exam and MRI studies were significant for right orbital inflammation. The patient was started on oral prednisone with rapid resolution of symptoms. CONCLUSIONS: This is the first case report of a patient receiving chronic immunosuppressive therapy to develop orbital inflammation after Zoledronic acid infusion. In addition, it demonstrates that corticosteroids can be an effective first line therapy in treating orbital inflammation in similar patients. Physicians should be aware of this rare but serious potential side effect of bisphosphonates, and have bisphosphonate-related orbital inflammation on their differential for proper initiation of treatment.

What the tip-of-the-tongue (TOT) says about homophone frequency inheritance.

The present study uses tip-of-the-tongue (TOT) states as a unique source of evidence to test the hypothesis of lexical access benefits for homophones--that is, whether low-frequency homophones, such as tee, inherit the lexical access benefits of their high-frequency homophonic counterparts, such as tea. We compared retrieval success rates for low-frequency homophones, for matched low-frequency controls, and for high-frequency controls with the combined frequency of the homophone set. In correct retrievals, low-frequency homophones behaved according to their specific frequency, not differing from the low-frequency controls. However, retrieval failures revealed a different kind of homophone effect. When retrieval failed for targets with a homophone partner, access difficulties tended to be less profound than for low-frequency controls, ending closer to target retrieval more often than low-frequency controls (at Step 2; in a self-resolved TOT or in a TOT with a strong feeling of knowing), and ending far away from target retrieval less often than low-frequency controls (at Step 1; in a notGOT). These results provide evidence against the notion of shared word-form representations for homophonic targets but leave open a door for a weaker form of homophone effects, possibly arising from feedback activation that influences retrieval only when access is sufficiently slowed (as when retrieval fails).

Frosted branch angiitis presenting after a SARS-CoV-2 infection.

PURPOSE: To report a case of frosted branch angiitis presenting in a pediatric patient with unremarkable laboratory work-up apart from SARS-CoV-2 IgG antibodies. OBSERVATIONS: Less than four weeks after a SARS-CoV-2 infection, a 10 year-old female presented to the emergency department with severe headache and intermittent fevers. During her hospital admission, the ophthalmology service was consulted for blurry vision. Subsequent eye examination revealed frosted branch angiitis. The patient initially received intravenous corticosteroids but was escalated to plasmapheresis to achieve resolution of her symptoms. Outpatient maintenance therapy consisted of an oral Prednisone taper and Infliximab infusion. CONCLUSION AND IMPORTANCE: This case represents a unique ocular manifestation of COVID-19, as recent SARS-CoV-2 was the sole identifiable cause of the patient's frosted branch angiitis. Additionally, this patient required plasmapheresis to control disease progression.

Uveo-meningeal syndrome secondary to Herpes Simplex Virus related acute retinal necrosis.

PURPOSE: To present a rare case of uveo-meningeal syndrome secondary to herpes simplex virus (HSV-1) in a patient with acute retinal necrosis. OBSERVATIONS: A 49-year-old female with a past medical history of herpes simplex encephalitis 18 years prior presented with a 3-day history of right sided headache and decreased vision of the right eye. Her visual acuity was 20/30 in the right eye and 20/20 in the left eye. Clinical examination revealed right relative afferent pupillary defect, panuveitis, and retinal necrosis. Examination of the left eye was unremarkable. Cerebral spinal fluid (CSF) analysis by polymerase chain reaction (PCR) was negative for herpes simplex virus 1 (HSV-1) but did reveal pleocytosis consistent with meningitis. The patient was admitted and empirically treated with intravenous acyclovir (10 mg/kg every 8 hours) and systemic steroids. Topical steroids and cycloplegia were also started. Magnetic resonance imaging revealed no leptomeningeal, pachymeningeal, or parenchymal enhancement. Systemic autoimmune and infectious workup were unremarkable. Based on clinical exam findings and negative PCR results, an anterior chamber tap was performed with aqueous fluid PCR testing which revealed 71,000 copies of HSV-1. A repeat lumbar puncture was performed on day three of admission and revealed a decrease in pleocytosis after initiation of acyclovir therapy and remained negative for HSV on PCR testing. She was discharged home on intravenous acyclovir, topical steroids, and topical cycloplegics. Her retinal necrotic lesions continued to regress and her headaches continued to improve. CONCLUSIONS AND IMPORTANCE: Uveo-meningeal syndromes are a rare clinical entity that involve the uvea, retina, and meninges. This case highlights the importance of aqueous fluid PCR testing despite negative CSF PCR, as it may hasten treatment with antiviral therapies to preserve vision and limit neurologic sequelae.

Unilateral retinopathy post perilesional interferon α2b injections for ocular surface squamous cell carcinoma.

PURPOSE: To describe the clinical course of a patient presenting with unilateral retinopathy after perilesional interferon alpha injections for treatment of ocular surface squamous cell carcinoma. OBSERVATIONS: A patient, who was being treated with interferon alpha for ocular squamous cell carcinoma, presented with new onset decreased vision in her left eye. Upon examination, she was found to have cotton wool spots and retinal hemorrhages in the affected eye. CONCLUSIONS AND IMPORTANCE: Retinopathy is a well-documented side effect of systemic usage of interferon alpha. However, retinopathy has not been well discussed in the scenario of perilesional injections of interferon. It is important for clinicians to monitor for such pathology when using interferon alpha not only systemically, but also locally.

INTRAVITREAL METHOTREXATE FOR MANTLE CELL LYMPHOMA INFILTRATION OF THE OPTIC NERVES: A CASE REPORT.

PURPOSE: To report the successful treatment of a 78-year-old woman with bilateral mantle cell lymphoma involving the optic nerves. Chemotherapy initially was administered in the form of intravitreal methotrexate (MTX) monotherapy and was subsequently combined with systemic ibrutinib. METHODS: Retrospective case report. The diagnosis of CD5-negative mantle cell lymphoma was confirmed via immunohistopathological analysis of an axillary lymph node. Serial ophthalmologic examinations in conjunction with fluorescein angiography, fundus photography, and spectral domain optical coherence tomography were used to assess the treatment response. RESULTS: Prompt improvement in optic nerve infiltration, no significant side effects, and excellent tolerability were noted after two weekly injections of unilateral intravitreal MTX monotherapy. Combined systemic treatment with ibrutinib and bilateral weekly MTX intravitreal injections then resulted in continued regression of optic nerve infiltration bilaterally as confirmed by serial fundus photography and optical coherence tomography. After eight additional bilateral weekly injections, a mild MTX-associated keratopathy developed, which resolved promptly with cessation of injections and administration of topical lubrication. Six weeks after MTX cessation, but with continued ibrutinib treatment, the optic nerves revealed near-complete resolution of the lymphomatous infiltration and the visual acuity improved. CONCLUSION: Intravitreal MTX injections and systemic ibrutinib may represent effective treatment options for patients diagnosed with intraocular mantle cell lymphoma.