Ocular Dirofilariasis: A Clinicopathologic Case Series and Literature Review.

Purpose: Ocular dirofilariasis is an uncommon zoonotic infection that is usually associated with a carnivore host. In this case series and literature review, we investigate the clinical presentation, management, and histopathology of ocular dirofilariasis. Methods: The database at the Florida Lions Ocular Pathology Laboratory was searched for surgical specimens at the Bascom Palmer Eye Institute under approval of the Institutional Review Board. Patients with a histopathologic diagnosis of dirofilariasis between the years 1962 and 2022 from the Florida Lions Ocular Pathology Laboratory database were included (n = 3). A systematic PubMed search was conducted by two independent authors to identify published cases of ophthalmic dirofilariasis worldwide. Keywords were used to identify articles, and exclusion criteria were applied. Results: Three patients, two males and one female, were identified from the Florida Lions Ocular Pathology Laboratory database with a diagnosis of ocular dirofilariasis. The mean age was 46.7 years (with a range 33-57 years). There were two eyelid lesions (Cases 1 and 3) and one involving the subconjunctival space (Case 2). All three organisms were excised and presumptively identified as Dirofilaria tenuis. All 3 patients were managed with curative surgical removal and recovered completely. Our review of the literature identified 540 published reports and 142 published reports with 186 cases that met the exclusion criteria. Conclusion: We present a case series and literature review of ocular dirofilariasis. Knowledge of the incidence, risk factors, prevention, and diagnosis of this unique parasitic infection will help in proper management and prevent further ocular complications.

Burn Induced Cicatricial Eyelid Retraction: A Challenging Case and Review of Management Principles.

Thermal and chemical burns can result in cicatricial eyelid retraction, characterized by an abnormal resting position of the eyelid margin and increased palpebral fissure height. Eyelid retraction often leads to exposure keratopathy, which can cause complications ranging from mild dry eye to globe-threatening ulceration and perforation. Prompt intervention includes aggressive lubrication, moisture chambers, eyelid tarsorrhaphy and retraction repair surgery. Discussed here is a burn patient with severe cicatricial retraction and ectropion leading to severe exposure keratopathy and infectious corneal ulceration with perforation. The patient required aggressive medical intervention, as well as two surgeries to restore the normal eyelid anatomy to protect the globe.

Teprotumumab-Related Adverse Events in Thyroid Eye Disease: A Multicenter Study.

PURPOSE: To assess the duration, incidence, reversibility, and severity of adverse events (AEs) in patients with thyroid eye disease (TED) treated with teprotumumab. DESIGN: Multicenter, retrospective, observational cohort study. PARTICIPANTS: Patients with TED of all stages and activity levels treated with at least 4 infusions of teprotumumab. METHODS: Patients were treated with teprotumumab between February 2020 and October 2022 at 6 tertiary centers. Adverse event metrics were recorded at each visit. MAIN OUTCOME MEASURES: The primary outcomes measure was AE incidence and onset. Secondary outcome measures included AE severity, AE reversibility, AE duration, proptosis response, clinical activity score (CAS) reduction, and Gorman diplopia score improvement. RESULTS: The study evaluated 131 patients. Proptosis improved by 2 mm or more in 77% of patients (101/131), with average proptosis improvement of 3.0 ± 2.1 mm and average CAS reduction of 3.2 points. Gorman diplopia score improved by at least 1 point for 50% of patients (36/72) with baseline diplopia. Adverse events occurred in 81.7% of patients (107/131). Patients experienced a median of 4 AEs. Most AEs were mild (74.0% [97/131]), 28.2% (37/131) were moderate, and 8.4% (11/131) were severe. Mean interval AE onset was 7.9 weeks after the first infusion. Mean resolved AE duration was 17.6 weeks. Forty-six percent of patients (60/131) demonstrated at least 1 persistent AE at last follow-up. Mean follow-up was 70.2 ± 38.5 weeks after the first infusion. The most common type of AEs was musculoskeletal (58.0% [76/131]), followed by gastrointestinal (38.2% [50/131]), skin (38.2% [50/131]), ear and labyrinth (30.5% [40/131]), nervous system (20.6% [27/131]), metabolic (15.3% [20/131]), and reproductive system (12.2% [16/131]). Sixteen patients (12.2%) discontinued therapy because of AEs, including hearing loss (n = 4), inflammatory bowel disease flare (n = 2), hyperglycemia (n = 1), muscle spasms (n = 1), and multiple AEs (n = 8). CONCLUSIONS: Adverse events are commonly reported while receiving teprotumumab treatment. Most are mild and reversible; however, serious AEs can occur and may warrant treatment cessation. Treating physicians should inform patients about AE risk, properly screen patients before treatment, monitor patients closely throughout therapy, and understand how to manage AEs should they develop. FINANCIAL DISCLOSURE(S): Proprietary or commercial disclosure may be found in the Footnotes and Disclosures at the end of this article.

Teprotumumab for the Treatment of Recalcitrant Thyroid Eye Disease.

PURPOSE: Teprotumumab, an insulin-like growth factor 1 receptor monoclonal antibody, is FDA-approved to treat thyroid eye disease (TED). The initial clinical trials excluded patients with previous orbital irradiation, surgery, glucocorticoid use (cumulative dose >1 gm), or prior biologic treatment. Information on the use of teprotumumab for patients who failed prior therapy is limited. Our purpose is to characterize the efficacy of teprotumumab for the treatment of recalcitrant TED. METHODS: This is a multicenter retrospective study of all patients treated with teprotumumab for moderate-to-severe TED after failing conventional therapy with corticosteroids, orbital radiation, surgical decompression, biologics, or other steroid-sparing medications. Treatment failure was defined as an incomplete response to or reactivation after previous treatment. Only patients who received at least 4 infusions of teprotumumab were included in the analysis. Primary outcome measures comprised proptosis response (≥2 mm reduction in the study eye without a similar increase in the other eye), clinical activity score (CAS) response (≥2-point reduction in CAS), and diplopia response (≥1 point improvement in Gorman diplopia score in patients with baseline diplopia) following treatment. Adverse events and risk factors for recalcitrant disease were also evaluated. RESULTS: Sixty-six patients were included in this study, 46 females and 20 males. Average age was 59.3 years (range 29-93). The mean duration of disease from TED diagnosis to first infusion was 57.8 months. The proptosis, CAS, and diplopia responses in this recalcitrant patient population were 85.9%, 93.8%, and 69.1%, respectively. Patients experienced a mean reduction in proptosis of 3.1 ± 2.4 mm and a mean improvement in CAS of 3.8 ± 1.6. Patients who underwent prior decompression surgery experienced a statistically significant decrease in diplopia response (46.7% vs. 77.5%, p = 0.014) and proptosis response (75.0% vs. 90.9%, p = 0.045) when compared with nondecompression patients. Additionally, there were no significant differences in proptosis, CAS, and diplopia responses between patients with acute (defined as disease duration <1 year) versus chronic (disease duration ≥1 year) TED. While most adverse events were mild to moderate, 4 patients reported serious adverse events related to persistent hearing loss. CONCLUSIONS: Patients with recalcitrant TED demonstrated a significant improvement after teprotumumab in each of the primary study outcomes. The degree of proptosis reduction, diplopia response, and CAS improvement in the recalcitrant group were similar to those of treatment-naïve patients from the pivotal clinical trials. Patients with a prior history of orbital decompression, however, demonstrated poor improvement in diplopia and less reduction in proptosis than surgery naïve patients. These results indicate that teprotumumab is a treatment option for the treatment of patients with TED recalcitrant to prior medical therapies.

The effects of topical oxymetazoline on eyelid position, eye redness, and patient-reported eye appearance: A randomized controlled trial.

PURPOSE: This study assesses the effects of topical oxymetazoline 0.1% on eyelid position, eye redness, and patient-perceived eye appearance in patients without severe ptosis. METHODS: This is a randomized double-blinded controlled trial conducted at a single institute. Patients aged 18-100 years were randomized to receive one drop of oxymetazoline hydrochloride 0.1% or placebo bilaterally. Marginal reflex distance (MRD) 1 and 2, palpebral fissure height, eye redness, and patient-perceived eye appearance were assessed at baseline and two hours after drop instillation. Primary outcome measures included the change in MRD1, MRD2, and palpebral fissure height. Secondary outcome measures included changes in eye redness and patient-perceived eye appearance after drop instillation. RESULTS: In total, 114 patients were included, 57 treatment patients (mean age 36.4 ± 12.7 years, 31.6% male) and 57 controls (mean age 31.3 ± 10.1 years, 33.3% male). Baseline mean MRD1, MRD2, and palpebral fissure were similar between groups (p = 0.24, 0.45, and 0.23, respectively). Changes in MRD1 and eye redness in the treatment group were significantly greater than those in the control group (0.9 ± 0.9 mm vs. - 0.3 ± 0.4 mm, p < 0.001; - 2.6 ± 4.4 vs. - 0.5 ± 2.3, p = 0.002, respectively). Patient-perceived eye appearance was significantly improved in the treatment group compared to the controls (p = 0.002), with more treatment group patients also reporting increased eye size and decreased eye redness (p = 0.008, p = 0.003, respectively). There were 9 treatment-emergent adverse events (TEAEs) in 7 treatment group patients and 5 TEAEs in 5 control patients (p = 0.25), all of which were mild in severity. CONCLUSIONS: Topical oxymetazoline 0.1% increases MRD1 and palpebral fissure height, decreases eye redness, and improves patient-perceived eye appearance.

Spontaneous Subperiosteal Orbital Hematoma Associated With Cocaine-Induced Midline Destructive Lesion.

The authors present a case of a non-traumatic, spontaneous subperiosteal orbital hematoma in a woman with a history of chronic pansinusitis and absence of midline nasal cavity structures due to chronic inhalational cocaine use. The patient underwent left orbitotomy and drainage of the lesion, showing mostly blood with a small amount of purulence that grew methicillin-resistant Staphylococcus aureus when cultured. The patient received 4 weeks of intravenous antibiotics in addition to functional endoscopic sinus surgery. At 1 month after surgery, her vision had returned to baseline, and proptosis was resolved. Fewer than 20 cases of subperiosteal orbital hematomas associated with chronic sinusitis have been reported. To the authors' knowledge, this is the first reported case of a subperiosteal orbital hematoma associated with cocaine-induced midline destructive lesions. Patient consent to obtain photographs was obtained and archived. All collection and evaluation of patient health information were compliant with the Health Insurance Portability and Accountability Act, and this report adheres to the Declaration of Helsinki.

Anatomic and physiologic approach for trans-conjunctival needle decompression of orbital emphysema.

A 19-year-old with blunt trauma and repeated nose blowing presented with orbital emphysema and orbital compartment syndrome. Orbital emphysema is the abnormal presence of air within the orbit, typically secondary to trauma. Most cases will resolve with observation alone, however orbital compartment syndrome is a feared complication that necessitates urgent decompression. A superior fornix, trans-conjunctival approach was safely utilized to decompress the orbit while avoiding unwanted complications. Various decompression techniques have previously been described, most of which describe trans-palpebral approaches. The key safety benefit to the trans-conjunctival approach is direct visualization of the needle tip adjacent to the superior fornix, therefore posterior to the equator of the globe. With the needle positioned parallel to the curvature of the globe at the equator, the posterior sclera surface curves away from the tip, rendering it difficult to pierce the globe. In addition, the needle needs to be advanced only 3-4 millimeters and traverses only the conjunctiva and Tenon's to enter the central surgical space - the most direct route to the air pocket. The trans-conjunctival approach lowers the risk of damage to eyelid neurovascular structures and is less painful.

Antimicrobial-eluting cement orbital implant: a case report of staged enucleation for infectious panophthalmitis with extraocular extension.

There are various orbital implant options following enucleation. In cases of severe infection, such as panophthalmitis with extraocular extension, it is reasonable to consider a two-staged approach to decrease the risk of infectious complications. One option, illustrated by this case, is enucleation with insertion of an antimicrobial-eluting cement implant, followed by a secondary procedure to exchange the cement with a permanent orbital implant. We report on a patient with clinical, ultrasound, and radiographical findings consistent with infectious panophthalmitis with extra-scleral extension. Intolerable pain and progressive orbital involvement in a blind eye were the indications for enucleation. To reduce the risk of persistent infection, a gentamycin-eluting cement implant (Palacos® R + G as an intraorbital implant) was utilized in the initial procedure. Two months later, the cement implant was removed, and a scleral-wrapped porous implant was placed into a quiet socket without signs of inflammation or infection. In the setting of severe infection, a two-staged procedure utilizing an antimicrobial-eluting implant can be considered.

Outcomes of Patients With Thyroid Eye Disease Partially Treated With Teprotumumab.

PURPOSE: In response to the coronavirus (COVID-19) pandemic, teprotumumab production was temporarily halted with resources diverted toward vaccine production. Many patients who initiated treatment with teprotumumab for thyroid eye disease were forced to deviate from the standard protocol. This study investigates the response of teprotumumab when patients receive fewer than the standard 8-dose regimen. METHODS: This observational cross-sectional cohort study included patients from 15 institutions with active or minimal to no clinical activity thyroid eye disease treated with the standard teprotumumab infusion protocol. Patients were included if they had completed at least 1 teprotumumab infusion and had not yet completed all 8 planned infusions. Data were collected before teprotumumab initiation, within 3 weeks of last dose before interruption, and at the visit before teprotumumab reinitiation. The primary outcome measure was reduction in proptosis more than 2 mm. Secondary outcome measures included change in clinical activity score (CAS), extraocular motility restriction, margin reflex distance-1 (MRD1), and reported adverse events. RESULTS: The study included 74 patients. Mean age was 57.8 years, and 77% were female. There were 62 active and 12 minimal to no clinical activity patients. Patients completed an average of 4.2 teprotumumab infusions before interruption. A significant mean reduction in proptosis (-2.9 mm in active and -2.8 mm in minimal to no clinical activity patients, P < 0.01) was noted and maintained during interruption. For active patients, a 3.4-point reduction in CAS ( P < 0.01) and reduction in ocular motility restriction ( P < 0.01) were maintained during interruption. CONCLUSIONS: Patients partially treated with teprotumumab achieve significant reduction in proptosis, CAS, and extraocular muscle restriction and maintain these improvements through the period of interruption.

Lateral Tarsal Strip Complications With and Without Conjunctiva Stripping.

PURPOSE: The lateral tarsal strip (LTS) procedure is commonly used to correct eyelid malposition. When performing LTS, some surgeons elect to remove conjunctiva from the tarsal strip, while others do not. It has been hypothesized that without conjunctival stripping, the buried conjunctival tissue can cause complications such as inclusion cysts and granulomas. However, there is limited data comparing LTS cases with and without conjunctiva removal. The authors sought to evaluate whether conjunctival stripping had any impact on complication rates with LTS. METHODS: LTS operations for ectropion correction were retrospectively reviewed and were separated into 2 cohorts, Con (conjunctiva not removed) or Coff (conjunctival removed). Charts were reviewed for outcomes and complications including inclusion cyst formation, granuloma formation, wound dehiscence, infection, and focal rim tenderness. RESULTS: The complication rate was 10% versus 8% for Con versus Coff respectively ( p = 0.54). The common complications of LTS surgery were granuloma (4%), wound dehiscence (3%), focal rim tenderness (3%), and infection requiring antibiotics (<1%). There was no significant difference in these complications between the Con and Coff cohorts. CONCLUSIONS: Complications in both groups were minimal, similar to prior studies, and there was no difference between the 2 cohorts. While it has been suggested that buried conjunctiva may result in increased complication rates, the author's findings suggest that removing the tarsal conjunctiva is a superfluous step in the LTS surgery and does not affect complication rates.

Bilateral iPhone Gorilla Glass Intraocular Foreign Bodies in a Pediatric Patient.

A 13-year old male presented with bilateral intraocular foreign bodies (IOFBs) after a bullet struck his iPhone. The right eye vision was 20/50 and examination disclosed a limbal entry wound at 6:00, dense traumatic cataract, and vitreous hemorrhage. The left eye was 20/40 and an entry wound located roughly 4mm posterior to the limbus was noted. Fundoscopy revealed multiple refractile particles with pre-retinal and vitreous hemorrhage. CT scan demonstrated hyperdensities in the vitreous cavity bilaterally. Globe closure, cataract removal, vitrectomy, and endolaser were undertaken in the right eye, while only globe closure was performed in the left eye. On latest follow-up, the patient had 20/20 corrected vision without inflammation bilaterally. This case highlights challenges associated with the management of IOFBs of unknown material in a pediatric patient. A conservative approach led to full visual recovery, despite retained intraocular smartphone Gorilla glass.

Primary evisceration for neonatal endogenous endophthalmitis: A report of two cases.

PURPOSE: To present two cases of neonatal endophthalmitis with poor prognosis that were managed with primary evisceration. OBSERVATIONS: Case 1 is a 27-weeks' gestation neonate who developed Pseudomonas aeruginosa endophthalmitis complicated by globe rupture. Case 2 describes a 34-weeks' gestation neonate with Serratia marcescens endophthalmitis. Both patients had poor prognosis and thus underwent primary evisceration with good long-term cosmetic outcomes at 15 years and 17 months, respectively. CONCLUSIONS AND IMPORTANCE: Primary evisceration should be considered in neonates with endophthalmitis with a poor prognosis and can result in good long-term cosmesis.