Visual Outcomes after Proton Therapy for Recurrent Uveal Melanoma.

OBJECTIVE: Proton beam reirradiation remains an effective and globe-preserving alternative to enucleation in the treatment of local recurrence in uveal melanoma. The study aimed to assess visual outcomes and prognostic factors in visual acuity following proton beam salvage therapy. DESIGN: Retrospective study SUBJECTS: A retrospective study evaluated patients with recurrent uveal melanoma treated with proton beam irradiation (PBI) from 1984 through 2019 at a single academic tertiary center. METHODS: Patient and tumor characteristics were collected from the medical record, as well as best visual acuity (BVA) and ocular outcomes following treatment of recurrent uveal melanoma with PBI. MAIN OUTCOME MEASURES: The primary outcome of the study was the visual acuity of patients following proton beam irradiation for recurrent uveal melanoma. Additional outcome measures included enucleation rate of patients following salvage PBI and analysis of tumor and patient characteristics in the prognostication of visual acuity. RESULTS: The study comprised 67 patients who received PBI for recurrent uveal melanoma. The median age at recurrence was 67.6 years (range 31.6-91.0 years) and median follow-up from the time of recurrence to last examination was 4.4 years (range 0.23-17.1 years). The median final BVA was hand motion (range 20/20-no light perception) and 6 (9.1%) patients maintained a Snellen VA 20/40 or better. The 5-year probability of visual acuity retention of 20/200 or better was 19%. In a multivariable Cox model, VA at tumor recurrence of less than 20/40 was found to be significantly associated with a VA of 20/200 or worse following retreatment with PBI. Twelve (18%) patients underwent enucleation following retreatment with PBI. CONCLUSIONS: PBI allows for ocular preservation and functional vision in the treatment of recurrent uveal melanoma in select patients.

Service innovations for people with multiple long-term conditions: reflections of a rapid evaluation team.

Background: People living with multiple long-term conditions represent a significant concern for National Health Service policy and practice, and their care is a major theme in the 2019 National Health Service Long Term Plan. The Birmingham RAND and Cambridge Rapid Evaluation Centre team has undertaken a thematic synthesis of the 10 evaluations it has conducted from 2018 to 2023, exploring the needs, priorities and implications for people with multiple long-term conditions. Objectives: The aims for this overarching study were to: (1) build a body of learning about service innovations in primary and community settings for people of all ages with multiple long-term conditions, focused on questions that matter most to people with multimorbidity; and (2) develop methodological insights about how rapid evaluation can be used to inform the scoping, testing and implementation of service innovations for people with multiple long-term conditions. Design: The focus on multiple long-term conditions came from a Birmingham RAND and Cambridge Rapid Evaluation Centre prioritisation process undertaken in 2018 using James Lind Alliance methods. Cross-analysis of the findings from the 10 individual rapid evaluations was supplemented by (1) building aspects of multimorbidity into the design of later evaluations; (2) interviewing national and regional stakeholders (n=19) working in or alongside integrated care systems; (3) undertaking a rapid review of evidence on remote monitoring for people with multiple long-term conditions (19 papers included); and (4) testing overall insights with organisations representing patients and carers through a patient, public and professional engagement workshop with 10 participants plus members of the research team. Results: While living with multiple long-term conditions is common and is the norm for people over the age of 50 using health and care services, it is not often a focus of health service provision or innovation, nor of research and evaluation activity. We discuss six themes emerging from the totality of the study: (1) our health system is mainly organised around single conditions and not multiple long-term conditions; (2) research calls and studies usually focus on single conditions and associated services; (3) building opportunities for engaged, informed individuals and carers and improved self-management; (4) the importance of measures that matter for patients and carers; (5) barriers to developing and implementing service innovations for people with multiple long-term conditions; and (6) what is needed to make patients with multiple long-term conditions a priority in healthcare planning and delivery. Limitations: Care of people with multiple long-term conditions was not the principal focus of several of the rapid evaluations. While this was a finding in itself, it limited our learning about designing and implementing, as well as methodological approaches to evaluating, service innovations for people with multiple long-term conditions. Conclusions: Through a thematic analysis of the portfolio of evaluations, we have deduced a set of suggested implications for how the needs of people with multiple long-term conditions can be better embedded in policy, research and practice. Future work: Areas of uncertainty related to the care of people with multiple long-term conditions should be further explored, including developing and testing measures of patient experience of (un)co-ordinated care across settings, and interrogating the experience of health and care staff when working with people with multiple long-term conditions, to understand what works. Funding: This award was funded by the National Institute for Health and Care Research (NIHR) Health and Social Care Delivery Research programme (NIHR award ref: NIHR134284) and is published in full in Health and Social Care Delivery Research; Vol. 12, No. 15. See the NIHR Funding and Awards website for further award information.

Many people in England live with two or more physical and/or mental health conditions that are expected to last for years. Estimates vary, but it is likely that a majority of National Health Service services are serving people living with two or more long-term conditions. We wanted to find out how well the needs of this group are taken into account when new types of health care are introduced, or existing services are reorganised. To do this, we went back to all 10 of the health service innovations that had been studied by our Birmingham RAND and Cambridge Evaluation Centre from 2018 to 2023. We did some extra research, including discussions with patient representatives and interviews with National Health Service policy-makers and managers at national and regional levels. We also looked at what new research had been published about one example of a new healthcare technology that is intended to help people who have several long-term health conditions: monitors that can be used by health service staff to measure patients' symptoms when they are in their own home. Our main finding was that most National Health Service attention is given to organising care for single conditions, often treating them in isolation. Patients' many treatments and needs are not routinely considered all at the same time by healthcare staff, nor by researchers. Care for one condition is too often not co-ordinated with care for other health problems that a patient may have. Although the situation of people living with several long-term health conditions is in principle understood by healthcare staff, managers and researchers, relatively little is done in practice to meet their needs. We conclude by suggesting ways that policy-makers, healthcare staff and researchers could improve how they help people living with multiple long-term conditions.

Epiretinal Membrane Formation following Rhegmatogenous Retinal Detachment Repair: A Retrospective Cohort Study.

INTRODUCTION: This study aimed to investigate the incidence of and risk factors for epiretinal membrane (ERM) formation following primary rhegmatogenous retinal detachment (RRD) repair. METHODS: This comparative, retrospective, single-center cohort study included eyes with primary RRD treated between 2011 and 2023 at Massachusetts Eye and Ear, Boston, Massachusetts, with pars plana vitrectomy (PPV), scleral buckle (SB), PPV+SB, or pneumatic retinopexy (PnR). Demographic, clinical, and surgical parameters were collected from medical records. The primary outcome was the risk of ERM formation, while the secondary outcome was the risk of ERM requiring surgery. Univariable and multivariable Cox regression were performed, and a hazard ratio (HR) and 95% confidence interval (95% CI) were reported. RESULTS: Overall, 394 eyes were included. The mean age was 58.49 ± 12.8 years, and most patients were male. There was a significantly lower risk of ERM formation following SB compared to PPV in the univariable analysis (HR = 0.2, 95% CI = 0.08-0.60, p = 0.003); however, there was no significant association between treatment modality and ERM formation on multivariable Cox regression controlling for confounding factors (p = 0.24). ERM formation was found more commonly in patients who were older (HR = 1.0 per 1 year increase in age, 95% CI = 1.01-1.04, p = 0.001), those with worse baseline visual acuity (HR = 1.3, 95% CI = 1.09-1.71, p = 0.008), and those with macula-off RRDs (HR = 2.1, 95% CI = 1.41-3.32, p < 0.001). CONCLUSION: Surgical modality does not have a significant impact on the risk of ERM following retinal detachment repair. However, age, baseline visual acuity, and macular status are important predictors of ERM formation after RRD repair.

Outcomes after Proton Beam Irradiation in Patients with Choroidal Melanoma Eligible for Investigational AU-011 Treatment.

Introduction: Vision loss is common in patients treated with radiotherapy for uveal melanoma. With proton beam irradiation (PBI), the prescribed dose is delivered to the tumor with a sharp dose reduction outside the target volume. However, radiation complications are likely to develop when tumors are located near the optic nerve or fovea. Treatment with light-activated AU-011 (belzupacap sarotalocan), an investigational drug which specifically targets tumor cells, may avoid these complications. We evaluated outcomes in a historical group of patients who fit eligibility criteria for AU-011 therapy and were treated with PBI. Methods: A consecutive series of patients who received PBI for small choroidal melanoma at a single center between 1986 and 2016 were identified. Consistent with eligibility criteria in clinical trials of AU-011, patients were included when tumor dimensions did not exceed 2.5 mm in maximum thickness and 10.0 mm in largest basal diameter (LBD). Snellen visual acuities were converted to logMAR for analysis. Visual acuity outcomes were analyzed in patients with an initial acuity of logMAR 0.7 or better (equivalent to Snellen 20/100). Rates of visual acuity loss and mortality were calculated using the Kaplan-Meier method. Acuity loss by tumor location was compared using log-rank testing. Rates of tumor recurrence, neovascular glaucoma (NVG), and eye loss were also described. Results: Two hundred and 22 patients were included in the study. The median age was 60.7 years (range 21.3-94.8 years). Median tumor thickness was 2.0 mm (range 1.2-2.5 mm), and median LBD was 8.0 mm (range 4.0-10.0 mm). Median follow-up was 6.9 years (range 1.0-30.2 years). In 204 patients with a baseline logMAR visual acuity of 0.7 or better, the mean baseline acuity was 0.15 (equivalent to Snellen 20/25), which decreased to 0.52 (approximately Snellen 20/70) by 5 years after PBI. Visual outcomes were significantly worse for patients with tumors located within 3 mm of the optic disc and/or fovea. Tumor recurrence (1.4%), NVG (4.5%), and eye loss (2.7%) were uncommon. Discussion: Despite the advantageous dose distribution of protons, over half of patients with small choroidal melanomas located near the optic disc or fovea had a visual acuity equivalent to 20/80 or worse at 5 years after PBI. Treatment with AU-011 may allow better vision preservation in small tumors that carry a high risk of vision loss with radiotherapy.

Vertical integration of general practices with acute hospitals in England: rapid impact evaluation.

Background: Vertical integration means merging organisations that operate at different stages along the patient pathway. We focus on acute hospitals running primary care medical practices. Evidence is scarce concerning the impact on use of health-care services and patient experience. Objectives: To assess the impact of vertical integration on use of hospital services, service delivery and patient experience and whether patients with multiple long-term conditions are affected differently from others. Design: Rapid, mixed methods evaluation with four work packages: (1) review of NHS trust annual reports and other sources to understand the scale of vertical integration across England; (2) development of the statistical analysis; (3) analysis of national survey data on patient experience, and national data on use of hospital services over the 2 years preceding and following vertical integration, comparing vertically integrated practices with a variety of control practices; and (4) focus groups and interviews with staff and patients across three case study sites to explore the impact of vertical integration on patient experience of care. Results: At 31 March 2021, 26 NHS trusts were in vertically integrated organisations, running 85 general practices across 116 practice sites. The earliest vertical integration between trusts and general practices was in 2015; a mean of 3.3 practices run by each trust (range 1-12). On average, integrated practices have fewer patients, are slightly more likely to be in the most deprived decile of areas, are more likely to hold an alternative provider medical services contract and have worse Quality and Outcomes Framework scores compared with non-integrated practices. Vertical integration is associated with statistically significant, modest reductions in rates of accident and emergency department attendances: 2% reduction (incidence rate ratio 0.98, 95% confidence interval 0.96 to 0.99; p < 0.0001); outpatient attendances: 1% reduction (incidence rate ratio 0.99, 95% confidence interval 0.99 to 1.00; p = 0.0061), emergency inpatient admissions: 3% reduction (incidence rate ratio 0.97, 95% confidence interval 0.95 to 0.99; p = 0.0062) and emergency readmissions: 5% reduction (incidence rate ratio 0.95, 95% confidence interval 0.91 to 1.00; p = 0.039), with no impact on length of stay, overall inpatient admissions or inpatient admissions for ambulatory care sensitive conditions. The falls in accident and emergency department and outpatient attendance rates are temporary. Focus groups and interviews with staff (N = 22) and interviews with patients (N = 14) showed that with vertical integration, health service improvements are introduced following a period of cultural interchange. Patients with multiple long-term conditions continue to encounter 'navigation work' choosing and accessing health-care provision, with diminishing continuity of care. Limitations: In the quantitative analysis, we could not replicate the counterfactual of what would have happened in those specific locations had practices not merged with trusts. There was imbalance across three case study sites with regard to staff and patients recruited for interview, and the latter were drawn from patient participation groups who may not be representative of local populations. Conclusions: Vertical integration can lead to modest reductions in use of hospital services and has minor or no impact on patient experience of care. Our analysis does not reveal a case for widespread roll-out of the approach. Future research: Further quantitative follow-up of the longer-term impact of vertical integration on hospital usage and more extensive interviewing of patients and their carers about patient experiences of navigating care. Funding: This project was funded by the National Institute for Health and Care Research (NIHR) Health and Social Care Delivery Research programme (BRACE Project no. 16/138/31) and will be published in full in Health and Social Care Delivery Research; Vol. 11, No. 17. See the NIHR Journals Library website for further project information.

Induction of Posterior Vitreous Detachment Using Perfluorooctane Dissection in a Pediatric Patient.

BACKGROUND AND OBJECTIVE: Inducing a posterior vitreous detachment (PVD) may be challenging, especially in pediatric patients with firm vitreo-retinal adherence. This case report will present an alternative method of PVD induction using adjunctive perfluorooctane (PFO). MATERIALS AND METHODS: An 11-year-old boy underwent scleral buckle placement and 23-gauge pars plana vitrectomy for macula-off retinal detachment. Triamcinolone acetonide was used for vitreous staining. A flexible loop was used to peel the hyaloid membrane from attached retina nasal to the optic nerve. A small hole was created in the posterior hyaloid, and a small PFO bubble was injected under the detached vitreous to further dissect it from the retina and propagate the vitreous detachment anteriorly. The vitrector was used to lift and complete the PVD peripherally. RESULTS: PVD induction was obtained with no iatrogenic retinal breaks or postoperative complications. CONCLUSION: PVD induction using a stepwise PFO approach is a safe and effective alternative to conventional techniques. [Ophthalmic Surg Lasers Imaging Retina 2023;54:543-546.].

Survival of patients with recurrent uveal melanoma after treatment with radiation therapy.

BACKGROUND/AIMS: We evaluated a large cohort of patients treated for local recurrence of choroidal or ciliary body melanomas at the Massachusetts Eye and Ear (MEE) to quantify the risk of melanoma-related mortality associated with recurrence, independent of other risk factors. METHODS: Patients treated with radiation therapy from 1982 to 2017 were identified through the Uveal Melanoma Registry at MEE. Competing risks regression was performed to investigate the risk of melanoma-related mortality associated with recurrence, treating recurrence as a time-varying covariate. RESULTS: Of 4196 patients treated, 4043 patients remained recurrence-free and 153 patients experienced a recurrence (median follow-up: 9.9 years). Median time from initial treatment to recurrence was 30.5 months (range: 2.0-238.7). Seventy-nine (69.9%) patients with recurrences and 826 (37.9%) patients in the recurrence-free group died of metastatic uveal melanoma (p<0.001). Median time from initial treatment to melanoma-related death was 4.9 years (1.0-31.8) for patients who developed recurrences and 4.3 years (0.59-33.8) for patients who did not (p=0.17). Five-year and 10-year probabilities of melanoma-related mortality were 9.5% and 15.0%, respectively, in patients without local recurrences compared with 32.0% and 46.6% in patients with recurrences (p<0.001). CONCLUSION: These data confirm previous reports that local recurrence is associated with an increased risk of dying of melanoma and quantify the risk that can be attributed to local recurrence independent of other risk factors. This group of patients should be strongly considered for adjuvant therapies when available.

Growth rate of indeterminate choroidal lesions prior to melanoma diagnosis.

PURPOSE: Small choroidal melanocytic lesions have a low rate of metastasis and can be reasonably managed with surveillance until they demonstrate evidence of growth or clinical risk factors for melanoma. However, even choroidal nevi are not stationary, with many exhibiting slow growth over time. We sought to quantify the growth rates of indeterminate choroidal lesions that were initially observed prior to a clinical diagnosis of melanoma. METHODS: A single-center retrospective study was performed of patients diagnosed with choroidal melanoma based upon clinical characteristics who were initially followed for indeterminate lesions over at least 6 months. Subjects were included if they had a minimum of two B-scan ultrasound measurements prior to the visit at which melanoma was diagnosed. Demographic and tumor characteristics were collected from the medical record. Growth rates were calculated as the change in lesion thickness in mm per month and were recorded at 6-month intervals; ultrasound measurements less than 1 month apart were excluded. The characteristics of indeterminate lesions with faster versus slower growth rates prior to melanoma diagnosis were compared. RESULTS: Fifty-four patients met inclusion criteria. The mean age at melanoma diagnosis was 67.4 years, and 53.7% were female. Subjects had a median of four B-scan ultrasound measurements prior to melanoma diagnosis (range 2-19) and were followed for a median of 40.6 months (range 9.9-138.0 months). The mean lesion thickness was 1.4 mm (range 0.5-2.2 mm) at presentation, and increased to 2.3 mm (range 1.5-5.7 mm) at melanoma diagnosis. The mean growth rate did not exceed 0.021 mm/month (95% CI: 0.004-0.039; equivalent to 0.25 mm/year) for indeterminate lesions, but increased to 0.057 mm/month (95% CI: 0.043-0.071 mm/month; equivalent to 0.68 mm/year) at the time of melanoma diagnosis. Rapidly growing lesions had a greater tumor thickness and shorter duration of observation at the time of melanoma diagnosis. CONCLUSION: For most indeterminate choroidal lesions eventually diagnosed as melanoma, the lesion thickness was relatively stable for a period of time, then rose significantly between the penultimate visit and the final visit. These findings confirm the recommendation for continued monitoring of suspicious choroidal lesions, as the growth rate may accelerate just prior to melanoma diagnosis. Lesions with a mean growth rate of up to 0.25 mm/year were observed, whereas lesions clinically determined to have transformed into melanoma demonstrated a mean growth rate of 0.68 mm/year. These values provide a baseline for future studies and potential therapies directed at stabilizing or reducing the growth of indeterminate choroidal lesions or small choroidal melanomas. Limitations of this study include its retrospective nature and reliance on clinical diagnostic criteria.

Refractory Choroidal Neovascularization in a Patient With Pseudoxanthoma Elasticum and Cowden Syndrome.

Purpose: To describe a patient with concurrent pseudoxanthoma elasticum (PXE) and Cowden syndrome who developed choroidal neovascularization (CNV) secondary to angioid streaks. The CNV presented at a young age and was relatively refractory to intravitreal antivascular endothelial growth factor (anti-VEGF) therapy. Methods: A retrospective chart review was performed. Results: A 32-year-old man was treated for bilateral sequential CNV over 11 years. Good visual acuity was maintained with 53 anti-VEGF injections in the right eye and 82 injections in the left eye. On average, 1 injection was administered every 1.7 months in each eye to control the exudation. A skin biopsy and genetic testing confirmed a diagnosis of PXE. He was also found to harbor a PTEN mutation consistent with Cowden syndrome. Conclusions: The concurrent PTEN mutation lends a possible explanation for the relative resistance of CNV to anti-VEGF therapy in this patient with PXE. Phosphatase and tensin homolog is a tumor suppressor that negatively regulates the VEGF pathway.

OUTCOMES OF RHEGMATOGENOUS RETINAL DETACHMENT REPAIR FOLLOWING INFECTIOUS ENDOPHTHALMITIS.

PURPOSE: To determine the incidence of and identify risk factors for redetachment in eyes that underwent rhegmatogenous retinal detachment (RRD) repair after infectious endophthalmitis. METHODS: Retrospective chart review of patients who had RRD surgery following a diagnosis of endophthalmitis from 2008 to 2021 at a tertiary referral center. Demographic and clinical characteristics as well as operative details were collected. Subjects with prior RRD, trauma, keratoprosthesis, or less than 3 months of postoperative follow-up were excluded. Univariate logistic regression analyses were performed. RESULTS: Thirty-four eyes of 34 patients were included. Most subjects were male with a mean age of 60.1 years. Exogenous (58.8%) was more common than endogenous (41.2%) sources of endophthalmitis. Seventeen of 34 eyes (50.0%) experienced redetachment. Significant risk factors were infection with virulent organisms, hypotony, and postoperative proliferative vitreoretinopathy. Initial management with vitreous tap or vitrectomy, exogenous versus endogenous source, and surgical procedure were not associated with recurrent RRD. At the final follow-up, 32 of 34 eyes (91.2%) were attached, although more than one third of these had silicone oil. CONCLUSION: Redetachment occurred in half of eyes that underwent surgical repair of RRD after endophthalmitis. Early vitrectomy, with or without prior vitreous tap, was not associated with a higher risk of redetachment and improved the likelihood of a positive culture result. Culture data can aid in risk stratification because organism virulence was a risk factor for recurrent RRD. Although visual outcomes were generally poor, most retinas remained attached at the final visit.

Acute Idiopathic Maculopathy Following SARS-CoV-2 Vaccination.

A 49-year-old man presented with acute unilateral blurred vision one week after SARS-CoV-2 vaccination. A unilateral serous detachment of the macula, intraretinal hemorrhages, vitritis, and anterior chamber cell was found. Diagnostic testing was negative for infectious and inflammatory causes, and a diagnosis of acute idiopathic maculopathy (AIM) was made. Symptoms and serous detachment resolved over 12 weeks, with residual retinal pigment epithelial changes consistent with the disease course. AIM is a rare diagnosis that presented in close proximity to SARS-CoV-2 vaccination without evidence of coxsackievirus infection. Further research is necessary to clarify an association between this vaccine and uveitis.

Towards cataloguing and characterising advance care planning and end-of-life care resources.

BACKGROUND: Resources for healthcare professionals, patients and those important to them relating to planning and coordinating treatment and care at the end of life are abundant, and can be difficult to navigate. However, they have not been systematically collated or catalogued in terms of their purpose, scope or intended audience. AIM: To collate, categorise and characterise advance care planning and end-of-life treatment and care (EoLT + C) resources directed towards healthcare professionals, patients and their families. METHODS: Rapid review and thematic synthesis of resources available in the United Kingdom. Google searches and reviews of websites belonging to selected organisations that develop and publish materials relating to EoLT + C, and advance care planning were used. Materials were included if they were intended for those over 18 living in the UK and pertained to five domains of EoLT + C: identifying those approaching end of life; accessing EoLT + C services; conducting important conversations about EoLT + C and preferences; advance care planning, including recording of preferences and plans; and ensuring that plans and preferences are accessed and used by health and social care services. RESULTS: 246 resources directed at healthcare professionals, patients and their families were identified, collated, catalogued and made internationally available for clinicians, researchers, patients and the public. 61 were classified as interactive, providing decision support in EoLT + C that went beyond simply providing information. Of these, there was notable content overlap among tools for identifying patients in their last year of life. There was variation in the development of tools across all domains of end-of-life care by geography and patient group. Few interactive resources integrated seamlessly with a digital interface or healthcare provider workflows. Incentives for the adoption of best-practice appeared rare. CONCLUSIONS: We present a repeatable and scalable approach to the cataloguing and characterisation of palliative care resources. The identified resources will be of benefit not only to those in the UK but to those in other countries, developing or evaluating their own resources for aiding professionals and patients to plan and deliver excellent treatment and care at the end of life.

Treatment of Aggressive Retinal Astrocytic Hamartoma with Oral Mechanistic Target of Rapamycin Inhibition.

PURPOSE: To describe the clinical course and outcomes of aggressive retinal astrocytic hamartoma (RAH) treated with oral mechanistic target of rapamycin inhibitors (mTORis). DESIGN: A retrospective clinical case series. PARTICIPANTS: Five patients with genetically confirmed tuberous sclerosis complex and visually significant RAH due to tumor growth or exudation. METHODS: In this retrospective clinical case series, a review of electronic medical records was performed to determine baseline and follow-up ophthalmic examination characteristics, along with ancillary imaging findings, in patients receiving off-label treatment with either oral sirolimus or everolimus for symptomatic RAH. MAIN OUTCOME MEASURES: Visual acuity, change in tumor size, degree of exudation, and adverse effects of the mTORis were evaluated. RESULTS: The 5 patients in this series ranged in age from 8 months to 54 years. Four were treated with sirolimus, and 1 received everolimus. In all the cases, the tumor height was stable or decreased after the treatment (median follow-up duration, 39 months; range, 11-73 months). Exudation improved after the treatment in all the cases. In an 8-month-old infant, frequent upper respiratory tract infections prompted the cessation of treatment. In 1 patient, the mTORi was temporarily withheld because of elevated liver enzyme levels. No other significant adverse effects were noted. CONCLUSIONS: Sirolimus and everolimus should be considered in the management of vision-threatening RAH, particularly in the setting of exudative and rapidly growing tumors. Four of the 5 patients in this series tolerated the oral mTORi and continued with the therapy. There were no serious complications.

Bacillary Layer Detachment Associated With Endogenous Fungal Endophthalmitis.

Purpose: To recognize the novel finding of a bacillary layer detachment (BALAD) secondary to endogenous fungal endophthalmitis. Methods: Chart review, literature review. Results: BALAD is a recently described condition in which the photoreceptor layer splits at the level of the inner segment myoid. We describe a case of BALAD associated with endogenous fungal endophthalmitis and subsequent development of choroidal neovascularization, although it is unclear if BALAD contributed to neovessel formation. Conclusions: BALAD is generally seen in the setting of inflammatory or infectious retinal diseases. This is the first report of BALAD secondary to endogenous fungal endophthalmitis.

COCCIDIOIDAL ENDOPHTHALMITIS: EXCELLENT RECOVERY OF VISION WITH AGGRESSIVE USE OF INTRAVITREAL ANTIFUNGALS AND VITRECTOMY.

PURPOSE: To report a case of Coccidioides immitis endophthalmitis with severe vision loss and a return to excellent vision after aggressive intervention. METHODS: Case report. RESULTS: A 41-year-old man with a history of solid organ transplantation who complained of floaters and decreased vision in the setting of disseminated Coccidioides infection was found to have presumed coccidioidal endophthalmitis with visual acuities of 20/20 in the right eye and 20/200 in the left eye. The patient was managed with intravenous amphotericin B, oral voriconazole, and intravitreal injections of amphotericin B and voriconazole in the left eye every three days. Five weeks after presentation, his visual acuity remained 20/20 in the right eye and improved to 20/40 in the left eye. The patient was transitioned to twice weekly intravitreal injections and oral voriconazole on hospital discharge. One week later, vision in the left eye decreased to 20/500 with worsening vitritis, prompting vitrectomy. Vision in the left eye subsequently improved to 20/30. Five weeks later, the patient developed a macula-on inferior rhegmatogenous retinal detachment in the left eye and underwent a second vitrectomy, with scleral buckle, laser, and gas injection. Vision in the left eye returned to 20/25. In total, the patient received 22 amphotericin B and 17 voriconazole intravitreal injections in the left eye with two vitrectomies. Vision in the right eye remained 20/20 throughout his treatment course. At four months after presentation, the patient remained on oral voriconazole with no evidence of active intraocular infection on examination. CONCLUSION: Aggressive medical and surgical management can be successful in ocular conservation and restoration of vision in coccidioidal endophthalmitis. Very mild disease may be conservatively monitored and managed with systemic antifungal therapy alone. In severe disease, early diagnosis and prompt and aggressive use of systemic and intravitreal antifungals may spare panophthalmitis and preserve vision.

The role of the informal and formal organisation in voice about concerns in healthcare: A qualitative interview study.

The importance of employee voice-speaking up and out about concerns-is widely recognised as fundamental to patient safety and quality of care. However, failures of voice continue to occur, often with disastrous consequences. In this article, we argue that the enduring sociological concepts of the informal organisation and formal organisation offer analytical purchase in understanding the causes of such problems and how they can be addressed. We report a qualitative study involving 165 interviews across three healthcare organisations in two high-income countries. Our analysis emphasises the interdependence of the formal and informal organisation. The formal organisation describes codified and formalised elements of structures, procedures and processes for the exercise of voice, but participants often found it frustrating, ambiguous, and poorly designed. The informal organisation-the informal practices, social connections, and methods for making decisions that are key to coordinating organisational activity-could facilitate voice through its capacity to help people to understand complex processes, make sense of their concerns, and frame them in ways likely to prompt an appropriate organisational response. Sometimes the informal organisation compensated for gaps, ambiguities and inconsistencies in formal policies and systems. At the same time, the informal organisation had a dark side, potentially subduing voice by creating informal hierarchies, prioritising social cohesion, and providing opportunities for retaliation. The formal and the informal organisation are not exclusive or independent: they interact with and mutually reinforce each other. Our findings have implications for efforts to improve culture and processes in relation to voice in healthcare organisations, pointing to the need to address deficits in the formal organisation, and to the potential of building on strengths in the informal organisation that are crucial in supporting voice.

Bilateral anterior segment dysgenesis and peripheral avascular retina with tractional retinal detachment in an infant with multiple congenital anomalies-hypotony-seizures syndrome 3.

Background: Multiple congenital anomalies-hypotony-seizures syndrome 3 (MCAHS3) is a rare autosomal recessive disorder caused by mutations in the PIGT gene. PIGT encodes phosphatidylinositol-glycan biosynthesis class T, which plays a crucial role in protein anchoring to cell membranes. The clinical presentation of MCAHS3 is variable in expression and severity, but can be characterized by developmental delay, seizures, hypotonia, facial dysmorphism, and other abnormalities.Materials and Methods: Case report.Results: We report unusual ocular findings including bilateral anterior segment dysgenesis, avascular retinal periphery, and tractional retinal detachment in a 1-month-old male infant with compound heterozygous PIGT mutations consistent with MCAHS3. Whole-exome sequencing did not detect any other genetic abnormalities.Conclusions: This case expands the clinical spectrum of MCAHS3 to include anomalies in ocular anterior segment and retinal vascular development. Given the rarity and the genetic heterogeneity of MCAHS3, giving rise to varied non-ocular phenotypes, it is possible that milder intraocular phenotypes could have gone unrecognized in the past.

Molecular Targets for Proliferative Vitreoretinopathy.

While innovations in the surgical management of retinal detachment (RD) with proliferative vitreoretinopathy (PVR) have significantly improved anatomic and visual outcomes over the years, recurrent RD due to PVR remains the major limitation to success. There are currently no medical therapies proven to be effective against PVR in humans. Increased understanding of the pathophysiology and risk factors for PVR have helped guide investigations for molecular targets. Drugs that counteract inflammation, proliferation, and growth factors are the leading candidates for treatment of PVR. This review discusses the ongoing search for pharmacologic therapies, with an emphasis on the results of recent clinical investigations.

Specialty COPD care during COVID-19: patient and clinician perspectives on remote delivery.

INTRODUCTION: The COVID-19 pandemic has impacted specialty chronic obstructive pulmonary disease (COPD) care. We examined the degree to which care has moved to remote approaches, eliciting clinician and patient perspectives on what is appropriate for ongoing remote delivery. METHODS: Using an online research platform, we conducted a survey and consensus-building process involving clinicians and patients with COPD. RESULTS: Fifty-five clinicians and 19 patients responded. The majority of clinicians felt able to assess symptom severity (n=52, 95%), reinforce smoking cessation (n=46, 84%) and signpost to other healthcare resources (n=44, 80%). Patients reported that assessing COPD severity and starting new medications were being addressed through remote care. Forty-three and 31 respondents participated in the first and second consensus-building rounds, respectively. When asked to rate the appropriateness of using remote delivery for specific care activities, respondents reached consensus on 5 of 14 items: collecting information about COPD and overall health status (77%), providing COPD education and developing a self-management plan (74%), reinforcing smoking cessation (81%), deciding whether patients should seek in-person care (72%) and initiating a rescue pack (76%). CONCLUSION: Adoption of remote care delivery appears high, with many care activities partially or completely delivered remotely. Our work identifies strengths and limitations of remote care delivery.