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2.
J Complement Integr Med ; 20(3): 577-589, 2023 Sep 01.
Article in English | MEDLINE | ID: mdl-37311120

ABSTRACT

OBJECTIVES: Dopamine-related movement disorders are associated with a loss of visual acuity. Studies have shown that chemical stimulation of the vitamin D3 receptor (VDR) ameliorates movement disorders; however, the chemical stimulation is not effective when there is a deficiency of vitamin A in the cells. In the study, we examine the role of VDR and its interplay with vitamin A in impaired visual function in the dopamine deficit model. METHODS: Thirty (30) male mice with an average weight of 26 g ± (2) were divided into six group (NS,-D2,-D2 + VD D2 + VD, -D2 + VA, -D2 + (VD + VA) and -D2 + D2 groups). Dopamine deficit models of movement disorders were created using 15 mg/kg of haloperidol (-D2) injected intraperitoneally daily for 21 days. In the -D2 + (VD + VA) group, 800 IU/day of vitamin D3 (VD) and 1000 IU/day of vitamin A were concurrently used, while in the -D2 + D2 group, bromocriptine (+D2) was used as the standard treatment of the model. At the end of the treatment phase, the animals were subjected to visual water box test for visual acuity. The level of oxidative stress was measured using Superoxide dismutase (SOD) and malondialdehyde (MDA) in the retina and visual cortex. The level of cytotoxicity in these tissues was measured using Lactate dehydrogenase (LDH) assay, while the structural integrity of these tissues was assessed using a light microscope by assessing slide mounted sections that were stained with haematoxylin and eosin. RESULTS: A significant decline in time taken to reach the escape platform in the visual water box test was observed in the -D2 (p<0.005) and -D2 + D2 (p<0.05) group. In the retina and the visual cortex, a significant increase in LDH, MDA and the density of degenerating neurons was observed in the -D2 and -D2 + D2 groups. LDH level in the retina was also found to be significantly increased in (-D2 + VD, -D2 + VA, -D2 + (VD + VA). A Significant decrease in SOD was found in the retina and visual cortex of -D2 and -D2 + D2 group. In the histology of the retina, thinning of the retina, retinal fold, distortion and retinal detachment were all seen in the -D2 group. These structural alterations were not seen in other groups. Histological hallmarks of degeneration were observed in the visual cortex of the mice from the -D2 (p<0.001), -D2 + D2 (p<0.005) and -D2 + VD (p<0.05) groups only. CONCLUSIONS: Dopamine-deficient models of movement disorders are associated with loss of visual functions, especially due to thinning of the retina, retinal fold, retinal detachment, and neurodegeneration in the visual cortex. Supplementation during the development of the model with vitamin D3 and vitamin A prevented the deterioration of the retina and visual cortex by reducing the degree of oxidative stress and cytotoxicity.


Subject(s)
Movement Disorders , Parkinsonian Disorders , Retinal Detachment , Male , Animals , Mice , Cholecalciferol/pharmacology , Cholecalciferol/therapeutic use , Receptors, Dopamine , Dopamine , Vitamin A/pharmacology , Parkinsonian Disorders/drug therapy , Mice, Knockout
3.
Transl Vis Sci Technol ; 12(5): 19, 2023 05 01.
Article in English | MEDLINE | ID: mdl-37191619

ABSTRACT

Purpose: Proliferative vitreoretinopathy (PVR) is the dreaded cause of failure following retinal detachment repair; however, no cures or preventative therapies exist to date. The purpose of this study was to use bioinformatics tools to identify drugs or compounds that interact with biomarkers and pathways involved in PVR pathogenesis that could be eligible for further testing for the prevention and treatment of PVR. Methods: We queried PubMed to compile a comprehensive list of genes described in PVR to date from human studies, animal models, and genomic studies found in the National Center for Biotechnology Information database. Gene enrichment analysis was performed using ToppGene on PVR-related genes against drug-gene interaction databases to construct a pharmacome and estimate the statistical significance of overrepresented compounds. Compounds with no clinical indications were filtered out from the resulting drug lists. Results: Our query identified 34 unique genes associated with PVR. Out of 77,146 candidate drugs or compounds in the drug databases, our analysis revealed multiple drugs and compounds that have significant interactions with genes involved in PVR, including antiproliferatives, corticosteroids, cardiovascular agents, antioxidants, statins, and micronutrients. Top compounds, including curcumin, statins, and cardiovascular agents such as carvedilol and enalapril, have well-established safety profiles and potentially could be readily repurposed for PVR. Other significant compounds such as prednisone and methotrexate have shown promising results in ongoing clinical trials for PVR. Conclusions: This bioinformatics approach of studying drug-gene interactions can identify drugs that may affect genes and pathways implicated in PVR. Predicted bioinformatics studies require further validation by preclinical or clinical studies; however, this unbiased approach could identify potential candidates among existing drugs and compounds that could be repurposed for PVR and guide future investigations. Translational Relevance: Novel repurposable drug therapies for PVR can be found using advanced bioinformatics models.


Subject(s)
Cardiovascular Agents , Hydroxymethylglutaryl-CoA Reductase Inhibitors , Retinal Detachment , Vitreoretinopathy, Proliferative , Animals , Humans , Vitreoretinopathy, Proliferative/drug therapy , Vitreoretinopathy, Proliferative/genetics , Retinal Detachment/complications , Retinal Detachment/prevention & control , Computational Biology
4.
Retina ; 43(8): 1246-1254, 2023 08 01.
Article in English | MEDLINE | ID: mdl-37027819

ABSTRACT

PURPOSE: To evaluate visual acuity and morphologic changes after photobiomodulation (PBM) for patients affected with large soft drusen and/or drusenoid pigment epithelial detachment associated with dry age-related macular degeneration. METHOD: Twenty eyes with large soft drusen and/or drusenoid pigment epithelial detachment age-related macular degeneration were included and treated using the LumiThera Valeda Light Delivery System. All patients underwent two treatments per week for 5 weeks. Outcome measures included best-corrected visual acuity, microperimetry-scotopic testing, drusen volume, central drusen thickness, and quality of life score at baseline and month 6 (M6) follow-up. Data of best-corrected visual acuity, drusen volume, and central drusen thickness were also recorded at week 5 (W5). RESULTS: Best-corrected visual acuity significantly improved at M6 with a mean score gain of 5.5 letters ( P = 0.007). Retinal sensitivity decreased by 0.1 dB ( P = 0.17). The mean fixation stability increased by 0.45% ( P = 0.72). Drusen volume decreased by 0.11 mm 3 ( P = 0.03). Central drusen thickness was reduced by a mean of 17.05 µ m ( P = 0.01). Geographic atrophy area increased by 0.06 mm 2 ( P = 0.01) over a 6-month follow-up, and quality of life score increased by 3,07 points on average ( P = 0.05). One patient presented a drusenoid pigment epithelial detachment rupture at M6 after PBM treatment. CONCLUSION: The visual and anatomical improvements in our patients support previous reports on PBM. PBM may provide a valid therapeutic option for large soft drusen and drusenoid pigment epithelial detachment age-related macular degeneration and may potentially slow the natural course of the disease.


Subject(s)
Geographic Atrophy , Low-Level Light Therapy , Macular Degeneration , Retinal Detachment , Retinal Drusen , Humans , Pilot Projects , Prospective Studies , Quality of Life , Macular Degeneration/complications , Retinal Drusen/complications , Retinal Detachment/complications , Geographic Atrophy/complications , Tomography, Optical Coherence , Follow-Up Studies
5.
Graefes Arch Clin Exp Ophthalmol ; 261(6): 1723-1729, 2023 Jun.
Article in English | MEDLINE | ID: mdl-36595051

ABSTRACT

BACKGROUND: Stickler syndrome (STL) is an inherited progressive connective tissue collagen disorder. STL is the most common hereditary cause of retinal complications, retinal tears, and the development of retinal detachment (RD) in childhood. The aim of the study was to evaluate the long-term anatomical and functional results of surgical treatment of retinal complications in children and adolescents affected by STL. METHODS: A retrospective, single-center study was performed a cohort of children with STL who underwent retinal surgery between 2004 and 2021. RESULTS: The study group consisted of nine children; the mean age at the time of the retinal tear with/without retinal detachment was 7.2 (2-10) years, and the mean follow-up period was 9.6 (5-16) years. Pathogenic variants COL2A1 (5 children) and COL11A1 (3 children) were confirmed in our cohort. In total, we operated on 13 eyes, 11 eyes with complicated RD and two eyes with multiple retinal defects, but without RD. At the end of the follow-up period, an attached retina was achieved 77% (10 eyes) with or without silicone oil tamponade: cryopexy alone was successful in one eye (10%), scleral buckling (EB) in five eyes (50%), and vitrectomy with silicone oil tamponade combined with EB in four eyes (40%). The mean number of surgeries was 2.3 per eye. The resulting best corrected visual acuity ranged from 0.03 to 0.1 in one eye, from 0.16 to 0.4 in two eyes, and from 0.5 to 1.0 in 7 eyes. CONCLUSION: Repair of retinal tears with/without retinal detachment in patients with Stickler syndrome often requires multiple surgeries with combinations of cryopexy, scleral buckling, and/or vitrectomy with silicone oil tamponade. Treatment of the ocular complications arising from STL requires long-term comprehensive care.


Subject(s)
Eye Diseases, Hereditary , Retinal Detachment , Retinal Perforations , Humans , Child , Adolescent , Retinal Detachment/diagnosis , Retinal Detachment/etiology , Retinal Detachment/surgery , Retinal Perforations/surgery , Retrospective Studies , Silicone Oils , Retina/pathology , Scleral Buckling , Vitrectomy/methods , Eye Diseases, Hereditary/surgery , Treatment Outcome
6.
Retin Cases Brief Rep ; 17(1): 41-43, 2023 Jan 01.
Article in English | MEDLINE | ID: mdl-33323898

ABSTRACT

PURPOSE: To report a case of early postoperative scleral buckle slippage because of the dehiscence of scleral belt loop tunnels. METHOD: Case report. RESULTS: A 54-year-old woman presented with painful diplopia after a combination pars plana vitrectomy and scleral buckling procedure. Ocular movements were limited. Forced duction testing was restricted in all directions. Anterior slippage of the silicone band was suggested on computed tomography (CT) scans and was confirmed with surgical exploration. During surgery, it was found that thin-roofed scleral belt loop tunnels were dehisced in three quadrants leading to anterior slippage of the buckle. The displaced buckle was removed. Diplopia and pain resolved, and ocular motility improved immediately afterward. The retina remained attached at six months follow-up. A supplemental video summarizes the surgical findings and postoperative results. CONCLUSION: Spontaneous dehiscence of scleral belt loops may occur in thinly dissected scleral tunnels. Painful eye movement, diplopia, and a positive forced duction test should raise suspicion about a displaced scleral buckle. A CT scan may help with the diagnosis. Early diagnosis and immediate surgical intervention are needed to minimize patient discomfort and to improve long-term ocular motility.


Subject(s)
Pain, Intractable , Retinal Detachment , Female , Humans , Middle Aged , Scleral Buckling/adverse effects , Scleral Buckling/methods , Diplopia/diagnosis , Diplopia/etiology , Diplopia/surgery , Pain, Intractable/complications , Pain, Intractable/surgery , Retinal Detachment/surgery , Retrospective Studies , Treatment Outcome , Vitrectomy/methods
7.
Retin Cases Brief Rep ; 17(6): 775-778, 2023 Nov 01.
Article in English | MEDLINE | ID: mdl-35972814

ABSTRACT

PURPOSE: To describe the use of the amniotic membrane for the repair of the exit wound of a perforating injury involving the retina and the choroid. METHODS: Case report. RESULTS: A 46-year-old man presented one day after a perforating ocular injury with an exit wound close to the inferior temporal retinal vascular arcade. The next day, the patient underwent a combined vitrectomy and phacoemulsification with intraocular lens implantation. During vitrectomy, a retinochoroidectomy was performed at the exit wound and the internal limiting membrane was peeled over the macula and up to the exit wound. Laser retinopexy was followed by plugging of a piece of the amniotic membrane to the exit wound. A second piece of the amniotic membrane was used to cover the bare retinochoroidectomy area. The surgery was concluded with a silicone exchange. Postoperatively, no sign of proliferative vitreoretinopathy was observed, and at 3 months, the silicone oil was removed. The follow-up was uneventful, and the eye achieved a final visual acuity of 20/30. CONCLUSION: The amniotic membrane may offer a simple and safe solution for the repair of the exit wounds of perforating injuries involving the retina and the choroid. The use of the amniotic membrane for this purpose may afford the opportunity for early vitrectomy in the management of perforating ocular injuries.


Subject(s)
Eye Injuries, Penetrating , Retinal Detachment , Vitreoretinopathy, Proliferative , Humans , Male , Middle Aged , Amnion , Eye Injuries, Penetrating/complications , Eye Injuries, Penetrating/surgery , Eye Injuries, Penetrating/diagnosis , Retina/surgery , Retinal Detachment/surgery , Retrospective Studies , Silicone Oils , Vitrectomy , Vitreoretinopathy, Proliferative/complications
8.
Altern Ther Health Med ; 29(1): 80-84, 2023 Jan.
Article in English | MEDLINE | ID: mdl-36112791

ABSTRACT

Context: Epidemiological data has shown that retinal detachment (RD) can occur at any age but has a poor prognosis in the adolescent population, which can cause huge obstacles to their life and learning. Although medications can achieve some curative effect, they have potential side effects and differences in individual efficacy. Objective: • The study intended to explore the clinical significance of visual training in improving recovery of postoperative visual function after external reduction of retinal detachment in adolescent patients. Design: The research team designed a prospective randomized controlled study. Setting: The study took place at Guiyang First People's Hospital in Guiyang, Guizhou, China. Participants: Participants were 110 adolescents with retinal detachments who underwent external reduction surgery, each on one eye for 110 eyes in total, and who were patients at the hospital between June 2015 and June 2019. Intervention: The research team assigned 52 participants to the visual-function training group, the intervention group, and 58 participants to the control group, according to the random-number-table method. Each group participated in training method for six months. Outcome Measures: To compare the groups, the research team measured visual function using the Visual Function scale (VF), binocular fusion function using the Worth Four Light Test (W4LT), and stereoscopic vision using the Titmus Stereo Test. The team obtained preoperative and postoperative data-at baseline, one day before surgery and postoperatively at one month and 3 months, and postintervention at 6 months. Results: Both groups had visual impairment after surgery. For visual function, the intervention group's scores after surgery increased gradually and were significantly higher than those of the control group at each follow-up time (P < .05). No significant difference in binocular fusion function existed between the groups at baseline or at one month after surgery (P > .05). At 3 months after surgery and postintervention, the proportions of participants in the intervention group with normal binocular fusion function were 86.54% and 88.46%, respectively, compared to that of the control group, at 68.97% and 70.69%, respectively. The intervention group's recovery was significantly better than that of the control group at 3 months after surgery and postintervention, at P < .028 and P < .022, respectively. No significant difference existed between the groups in stereoscopic vision at baseline, at 9.62% and 12.07%, respectively (P > .05). The proportion of participants in the intervention group with normal, binocular, stereoscopic vision increased gradually, and at one and 3 months after surgery and postintervention was 67.31%, 82.69%, and 88.46%, respectively. The intervention group's recovery was significantly better than that of the control group at one and 3 months after surgery and postintervention, at P < .028, P < .010, and P < .013, respectively. Conclusions: Visual training can effectively promote the recovery of visual function after external reduction of RD in adolescents and improve patients' prognoses. Moreover, long-term persistence can achieve significant effects, making the training worthy of clinical promotion.


Subject(s)
Retinal Detachment , Humans , Adolescent , Retinal Detachment/surgery , Retinal Detachment/etiology , Prospective Studies , Clinical Relevance , Visual Acuity , China
9.
Indian J Ophthalmol ; 70(8): 3167, 2022 08.
Article in English | MEDLINE | ID: mdl-35919014

ABSTRACT

Background: A 40-year-old male presented with a complaint of sudden onset diminution of vision in the left eye for 2 weeks. He was a follow-up case with retinal hemangioblastoma in both eyes. He underwent two sittings of fundus fluorescein angiography-guided trans-pupillary thermotherapy 2 years back. Since then, he was regularly followed up for 2 years with stable vision and stable retinal findings. At present, the best-corrected visual acuity (BCVA) in the right eye is 6/6, and in the left eye, it is counting fingers 2 meters. On fundus examination, he had one active hemangioblastoma in the right eye and total retinal detachment in the left eye with multiple active lesions. The right eye was treated with a single sitting of thermotherapy, and the left eye underwent pars plana vitrectomy and angioma excision, followed by silicone oil tamponade. The immediate and late post-operative periods were uneventful, with successful anatomical and functional outcomes. The left eye BCVA on late follow-up was 6/36, no further treatment was advised, and the patient was kept under follow-up and observed closely. Purpose: : To educate regarding the systemic workup, diagnosis, and surgical management of complicated retinal detachment in retinal hemangioblastoma. Synopsis: : Systemic workup, diagnosis, and surgical steps in the management of complicated retinal detachment in retinal hemangioblastoma were performed. Highlights: : Close follow-up, keen observation, and prompt treatment in the early stages of the disease are indispensable to prevent untoward sequelae of retinal hemangioblastoma. A thorough systemic workup is necessary to diagnose the systemic involvements early. Surgery, if indicated for the retinal hemangioblastoma or its associated sequelae, should be performed diligently and with careful handling of blood vessels and anomalous tissues. Online Video Link: https://youtu.be/CkoqWEnaPB8.


Subject(s)
Hemangioblastoma , Retinal Detachment , Retinal Neoplasms , Adult , Hemangioblastoma/complications , Hemangioblastoma/diagnosis , Hemangioblastoma/surgery , Humans , Male , Retina/surgery , Retinal Detachment/diagnosis , Retinal Detachment/etiology , Retinal Detachment/surgery , Retinal Neoplasms/complications , Retinal Neoplasms/diagnosis , Retinal Neoplasms/surgery , Retrospective Studies , Silicone Oils , Visual Acuity , Vitrectomy/adverse effects
10.
JAMA Netw Open ; 5(8): e2224892, 2022 08 01.
Article in English | MEDLINE | ID: mdl-35917124

ABSTRACT

Importance: Population-based data are needed to inform the safe prescribing of fluoroquinolone antibiotics to patients with advanced chronic kidney disease (CKD). Objective: To quantify the 14-day risk of a hospital visit with nervous system and/or psychiatric disorders, hypoglycemia, or a collagen-associated event in patients with advanced CKD newly prescribed a fluoroquinolone at a higher vs a lower dose. Design, Setting, and Participants: This population-based cohort study in Ontario, Canada (January 1, 2008, to March 17, 2020) used linked health care data to identify new users of fluoroquinolone antibiotics. Participants included adults 66 years or older with advanced CKD (an estimated glomerular filtration rate [eGFR] <30 mL/min/1.73 m2 but not receiving dialysis). Data analysis was performed from January 1 to April 30, 2021. Exposures: A new prescription for a higher-dose fluoroquinolone (ciprofloxacin, 501-1000 mg/d; levofloxacin, 501-750 mg/d; or norfloxacin, 401-800 mg/d) vs a lower-dose fluoroquinolone (ciprofloxacin, 500 mg/d; levofloxacin, 250-500 mg/d; or norfloxacin, 400 mg/d). Main Outcomes and Measure: The primary outcome was the 14-day risk of a hospital visit with nervous system and/or psychiatric disorders, hypoglycemia, or a collagen-associated event. Secondary outcomes included a hospital visit with sepsis, retinal detachment or other tendinopathies, all-cause hospitalization, all-cause mortality, and sudden cardiac death. Inverse probability of treatment weighting on the propensity score was used to balance comparison groups on baseline health. Weighted risk ratios and risk differences were obtained using modified Poisson regression and binomial regression, respectively. Results: Of 11 917 patients (median age, 83 years [IQR, 77-89 years]; 7438 women [62.4%]; median eGFR, 25 [IQR, 21-28] mL/min/1.73 m2) included in the analysis, 5482 (46.0%) received a higher-dose and 6435 (54.0%) received a lower-dose fluoroquinolone. After weighting, the primary composite outcome-a hospital visit with nervous system and/or psychiatric disorders, hypoglycemia, or a collagen-associated event-occurred in 68 of 5482 patients (1.2%) treated with a higher-dose fluoroquinolone and in 47 of 5516 (0.9%) treated with a lower-dose fluoroquinolone (weighted risk ratio, 1.45 [95% CI, 1.01-2.08]; weighted risk difference, 0.39% [95% CI, 0.01%-0.76%]). The risk of sepsis, retinal detachment, all-cause hospitalization, all-cause mortality, and sudden cardiac death did not differ significantly between groups. Conclusions and Relevance: These findings suggest that older patients with advanced CKD who were prescribed a fluoroquinolone at a higher-than-recommended dose were significantly more likely to experience the composite outcome of a hospital visit with nervous system and/or psychiatric disorders, hypoglycemia, or a collagen-associated event, although the absolute risk of these events was less than 2%.


Subject(s)
Hypoglycemia , Renal Insufficiency, Chronic , Retinal Detachment , Sepsis , Aged , Aged, 80 and over , Anti-Bacterial Agents/adverse effects , Ciprofloxacin , Cohort Studies , Death, Sudden, Cardiac , Female , Fluoroquinolones/adverse effects , Humans , Hypoglycemia/chemically induced , Hypoglycemia/epidemiology , Levofloxacin , Norfloxacin , Ontario/epidemiology , Renal Dialysis , Renal Insufficiency, Chronic/chemically induced , Renal Insufficiency, Chronic/complications , Renal Insufficiency, Chronic/drug therapy , Sepsis/complications
11.
Ophthalmology ; 129(10): 1129-1141, 2022 10.
Article in English | MEDLINE | ID: mdl-35680097

ABSTRACT

PURPOSE: Proliferative vitreoretinopathy (PVR) is the major cause for surgical failure after primary rhegmatogenous retinal detachment (RRD). So far, no therapy has been proven to prevent PVR. Promising results for 5-fluorouracil (5-FU) and low-molecular weight heparin (LMWH) in high-risk eyes have been reported previously. The objective of this trial was to examine the effect of adjuvant intravitreal therapy with 5-FU and LMWH compared with placebo on incidence of PVR in high-risk patients with primary RRD. DESIGN: Randomized, double-blind, controlled, multicenter, interventional trial with 1 interim analysis. PARTICIPANTS: Patients with RRD who were considered to be at high risk for PVR were included. Risk of PVR was assessed by noninvasive aqueous flare measurement using laser flare photometry. METHODS: Patients were randomized 1:1 to verum (200 mg/ml 5-FU and 5 IU/ml dalteparin) and placebo (balanced salt solution) intravitreally applied during routine pars plana vitrectomy. MAIN OUTCOME MEASURES: Primary end point was the development of PVR grade CP (full-thickness retinal folds or subretinal strands in clock hours located posterior to equator) 1 or higher within 12 weeks after surgery. For grading, an end point committee assessed fundus photographs. Secondary end points included best-corrected visual acuity and redetachment rate. A group sequential design with 1 interim analysis was applied using the O'Brien and Fleming boundaries. Proliferative vitreoretinopathy grade CP incidence was compared using a Mantel-Haenszel test stratified by surgeon. RESULTS: A total of 325 patients in 13 German trial sites had been randomized (verum, n = 163; placebo, n = 162). In study eyes, mean laser flare was 31 ± 26 pc/ms. No significant difference was found in PVR rate. Primary analysis in the modified intention-to-treat population results were: verum 28% vs. placebo 23% (including not assessable cases as failures); odds ratio [OR], 1.25; 95% confidence interval [CI], 0.76-2.08; P = 0.77. Those in the per-protocol population were: 12% vs. 12%; OR, 1.05; 95% CI, 0.47-2.34; P = 0.47. None of the secondary end points showed any significant difference between treatment groups. During the study period, no relevant safety risks were identified. CONCLUSIONS: Rate of PVR did not differ between adjuvant therapy with 5-FU and LMWH and placebo treatment in eyes with RRD.


Subject(s)
Retinal Detachment , Vitreoretinopathy, Proliferative , Dalteparin/therapeutic use , Double-Blind Method , Fluorouracil , Heparin/therapeutic use , Heparin, Low-Molecular-Weight/therapeutic use , Humans , Retinal Detachment/surgery , Vitrectomy/adverse effects , Vitreoretinopathy, Proliferative/drug therapy , Vitreoretinopathy, Proliferative/etiology , Vitreoretinopathy, Proliferative/prevention & control
12.
Ophthalmic Surg Lasers Imaging Retina ; 52(7): 400-402, 2021 07.
Article in English | MEDLINE | ID: mdl-34309433

ABSTRACT

A 9-year-old female with a history of Bohring-Opitz syndrome (BOS), Down syndrome, and autism initially presented with bilateral cataracts and a total retinal detachment in her left eye secondary to chronic self-injurious behavior. The authors report the first case of self-induced retinal detachment and traumatic cataracts in a patient with BOS. For patients who present with self-injurious behavior, the authors advocate for behavioral modifications at home, including the use of "no-no's," supplemental medication if necessary, and behavioral therapy to reduce the risk of self-induced visual injury. The authors also suggest the use of 25-gauge vitrectomy with silicone oil for retinal detachment repair. Finally, given the high risk of irreversible vision loss from amblyopia and recurrent retinal detachments in children with BOS and self-injurious behavior, the authors recommend regular 2-month interval ophthalmic follow-up. [Ophthalmic Surg Lasers Imaging Retina. 2021;52:400-402.].


Subject(s)
Cataract , Craniosynostoses , Retinal Detachment , Cataract/complications , Cataract/diagnosis , Child , Female , Humans , Intellectual Disability , Retinal Detachment/diagnosis , Retinal Detachment/etiology , Retinal Detachment/surgery , Silicone Oils , Treatment Outcome , Vitrectomy
13.
Am J Med Genet A ; 185(12): 3694-3700, 2021 12.
Article in English | MEDLINE | ID: mdl-34291880

ABSTRACT

Robin sequence (RS) has many genetic and nongenetic causes, including isolated Robin sequence (iRS), Stickler syndrome (SS), and other syndromes (SyndRS). The purpose of this study was to determine if the presence and type of cleft palate varies between etiologic groups. A secondary endpoint was to determine the relationship of etiologic group, cleft type, and mortality. Retrospective chart review of patients with RS at two high-volume craniofacial centers. 295 patients with RS identified. CP was identified in 97% with iRS, 95% with SS, and 70% of those with SyndRS (p < .0001). U-shaped CP was seen in 86% of iRS, 82% with SS, but only 27% with SyndRS (p < .0001). At one institution, 12 children (6%) with RS died, all from the SyndRS group (p < .0001). All died due to medical comorbidities related to their syndrome. Only 25% of children who died had a U-shaped CP. The most common palatal morphology among those who died was an intact palate. U-shaped CP was most strongly associated with iRS and SS, and with a lower risk of mortality. RS with submucous CP, cleft lip and palate or intact palate was strongly suggestive of an underlying genetic syndrome and higher risk of mortality.


Subject(s)
Arthritis/genetics , Cleft Lip/genetics , Cleft Palate/genetics , Connective Tissue Diseases/genetics , Hearing Loss, Sensorineural/genetics , Pierre Robin Syndrome/genetics , Retinal Detachment/genetics , Arthritis/diagnostic imaging , Arthritis/mortality , Arthritis/pathology , Child , Child, Preschool , Cleft Lip/diagnostic imaging , Cleft Lip/mortality , Cleft Lip/pathology , Cleft Palate/diagnostic imaging , Cleft Palate/mortality , Cleft Palate/pathology , Connective Tissue Diseases/diagnostic imaging , Connective Tissue Diseases/mortality , Connective Tissue Diseases/pathology , Female , Hearing Loss, Sensorineural/diagnostic imaging , Hearing Loss, Sensorineural/mortality , Hearing Loss, Sensorineural/pathology , Humans , Infant , Male , Pierre Robin Syndrome/diagnostic imaging , Pierre Robin Syndrome/mortality , Pierre Robin Syndrome/pathology , Retinal Detachment/diagnostic imaging , Retinal Detachment/mortality , Retinal Detachment/pathology , Retrospective Studies
14.
Medicine (Baltimore) ; 100(11): e25189, 2021 Mar 19.
Article in English | MEDLINE | ID: mdl-33726010

ABSTRACT

RATIONALE: In this report, we present an extremely rare case of recurrent monocular exudative retinal detachment without concomitant ocular metastases. This turned out to be the first symptom of squamous cell lung cancer. PATIENT CONCERNS: A 63-year-old woman was referred to our ophthalmology clinic by her primary care physician with a complaint of deteriorating vision in her right eye that had started four months prior, without concomitant pain. DIAGNOSES: We observed a detachment in the lower part of the retina during her ophthalmoscopy. We did not find any tears, holes, or degenerative changes in the periphery of the retina of the right eye during the surgery. In addition, plaques, tumor masses, and metastases were absent. Therefore, we diagnosed her with unilateral paraneoplastic exudative retinal detachment. Imaging tests performed before surgery revealed perihilar density with a visible air bronchogram in the middle field of the left lung. This turned out to be squamous cell carcinoma. INTERVENTIONS: Patient underwent pars plana vitrectomy and routine laboratory and imaging tests before the procedure that utilized 20-gauge instrumentation. The subretinal fluid and was drained and a tamponade using Densiron (Fluoron Co, Neu-Ulm, Germany) was applied. After ophthalmic treatment, patient underwent complex oncological treatment based on chemotherapy and radiotherapy. OUTCOMES: Despite the application of heavy silicone oil (Densiron) into the vitreous chamber, we observed a recurrence of retinal detachment in the right eye during the follow-up visit, 13 months after the first ophthalmic surgery. Following subsequent pars plana vitrectomy, the Densiron and subretinal membranes were removed. Despite oncological treatment, the patient died, twenty months after the appearance of the first ocular symptoms. LESSONS: Exudative retinal detachment without tumor metastasis to the eyeball can be one of the first signs of lung cancer in rare cases. Multidisciplinary care and imaging methods with greater accuracy will provide comprehensive care to the patients. It will not only facilitate timely detection and treatment of lung tumors but also for a plethora of oncological diseases.


Subject(s)
Carcinoma, Squamous Cell/complications , Lung Neoplasms/complications , Paraneoplastic Syndromes, Ocular/pathology , Retinal Detachment/pathology , Fatal Outcome , Female , Humans , Middle Aged , Paraneoplastic Syndromes, Ocular/etiology , Recurrence , Retinal Detachment/etiology
15.
J Med Case Rep ; 15(1): 16, 2021 Jan 20.
Article in English | MEDLINE | ID: mdl-33468218

ABSTRACT

BACKGROUND: Use of perfluorocarbon liquid (PFCL) has been increasingly growing as an adjuvant in vitreo-retina surgeries. Some commonly encountered complications with its use include subretinal migration, formation of sticky silicone oil or retained PFCL in vitreous cavity and anterior chamber. Scleral rupture during PFCL injection has a rare occurrence. We report an unexpected event of scleral rupture during PFCL injection and discuss the management challenges faced by the surgeon. CASE PRESENTATION: A 66 year indo-aryan male was undergoing pars-plana vitrectomy (PPV) with diagnosis of subtotal rhegmatogenous retinal detachment (RD) with Proliferative Vitreo-retonipathy (PVR)-B. After near total vitrectomy PFCL was being injected and then there was sudden poor visualization of fundus with development of bullous RD and globe hypotony. The surgeon was not able to figure out the cause of hypotony and air was switched on in the infusion cannula. This further complicated the situation resulting in migration of air in the anterior chamber, posterior dislocation of intraocular lens complex, 180° inferior retinal dialysis and ballooning of the conjunctiva which gave a clue of probable scleral rupture. Conjunctival peritomy was performed superiorly and scleral defect was noted. Intraocular tissue incarceration and air leak was visible from the wound. This confirmed scleral rupture during PFCL injection. Repositioning of incarcerated retina was not possible and retinectomy was performed followed by repair of scleral rupture with lots of difficulty in a vitrectomised eye. CONCLUSION: PFCL injection, a crucial step of vitreoretina surgery, should be performed slowly with extreme caution maintaining an optimal intraocular pressure to prevent devastating complications like scleral rupture.


Subject(s)
Fluorocarbons/administration & dosage , Intraoperative Complications/etiology , Intravitreal Injections/adverse effects , Retinal Detachment/surgery , Rupture/etiology , Sclera/injuries , Vitrectomy/methods , Vitreoretinopathy, Proliferative/surgery , Aged , Eye Injuries/etiology , Humans , Male , Vitreoretinal Surgery/methods
16.
Sci Rep ; 10(1): 14370, 2020 09 01.
Article in English | MEDLINE | ID: mdl-32873842

ABSTRACT

This study aims to report the 12 months results of efficacy and safety of laser photocoagulation and anti-vascular endothelial growth factor (VEGF) injections for drusenoid pigment epithelial detachment (dPED). In this prospective study, patients with treatment naïve bilateral intermediate age-related macular degeneration, featuring dPED, with visual acuity ≤ 83 letters were enrolled. The study group received PASCAL laser (532 nm) along the periphery of the dPED, and the fellow eye served as a control group. To prevent complications of choroidal neovascularization, intravitreal anti-VEGF injections to laser treated eye were performed on a 3-month interval up to 1 year. Primary outcomes-drusen area, PED height-and secondary outcomes-best-corrected visual acuity (BCVA), contrast sensitivity, degree of metamorphopsia, NEI-VFQ 25, and fundus autofluorescence-were analyzed. Among 21 patients, a total of 20 patients satisfied the 12 months follow-up. Drusen area and PED height decreased significantly in the laser group, while no significant change appeared in the control group (74.1% vs. - 3.5%, P < 0.001; 76.6% vs. 0.1%, P < 0.001). Mean BCVA improved 4.6 letters in the laser group (vs. 1.1 letters in the control group, P = 0.019). As for safety, one study eye developed retinal pigment epithelial tear, and one control eye developed retinal angiomatous proliferation. Low energy laser photocoagulation and anti-VEGF injection in eyes with dPED showed some improvement in visual acuity. dPED regressed without developing center involving GA in the study eye, but a longer term follow-up is necessary to reveal the efficacy and safety of these treatments. The 2-year results of this study will be followed to reveal long term efficacy and safety of the treatment for dPED.


Subject(s)
Angiogenesis Inhibitors/administration & dosage , Bevacizumab/administration & dosage , Geographic Atrophy/complications , Low-Level Light Therapy/adverse effects , Macular Degeneration/drug therapy , Macular Degeneration/radiotherapy , Retinal Detachment/drug therapy , Retinal Detachment/radiotherapy , Vascular Endothelial Growth Factor A/antagonists & inhibitors , Aged , Aged, 80 and over , Choroidal Neovascularization/prevention & control , Female , Follow-Up Studies , Humans , Male , Middle Aged , Pilot Projects , Prospective Studies , Retinal Perforations/etiology , Treatment Outcome , Visual Acuity
17.
Optom Vis Sci ; 97(7): 482-484, 2020 Jul.
Article in English | MEDLINE | ID: mdl-32697553

ABSTRACT

SIGNIFICANCE: The case report highlights the possible complications of undergoing neck manipulation within a critical time period after intravitreal injection. PURPOSE: This study aimed to describe a case of traumatic hemorrhagic choroidal detachment after cervical manipulation during a chiropractic treatment session. CASE REPORT: A 43-year-old male patient with a history of complex rhegmatogenous retinal detachment repair and recurrent cystoid macular edema presented with decreased vision and sudden pain in the right eye after chiropractic manipulation of the neck, status post-intravitreal injection of triamcinolone, which was performed earlier that day. Vision in the right eye was hand motion and 20/20 in the left eye. IOPs were 8 and 11 mmHg, respectively. Slit lamp examination of the right eye revealed blood-tinged steroid residues in the anterior chamber. There was no view to the posterior pole. Ultrasonography showed a lobulated mass with heterogeneous echogenicity consistent with a large hemorrhagic choroidal detachment. No central kissing was observed. Left eye examination was unremarkable. CONCLUSIONS: With the increasing use of complementary and alternative medicine, a better understanding of potential complications to raise awareness is becoming essential.


Subject(s)
Choroid Hemorrhage/etiology , Manipulation, Chiropractic/adverse effects , Neck Pain/therapy , Adult , Choroid Hemorrhage/diagnostic imaging , Glucocorticoids/therapeutic use , Humans , Intravitreal Injections , Macular Edema/drug therapy , Male , Retinal Detachment/surgery , Slit Lamp Microscopy , Triamcinolone Acetonide/therapeutic use , Ultrasonography
18.
Rev. cuba. oftalmol ; 33(2): e855, graf
Article in Spanish | CUMED, LILACS | ID: biblio-1139079

ABSTRACT

RESUMEN Los drusen de nervio óptico fueron descritos por primera vez por Liebreich en el año 1868. Otros términos para designar esta entidad incluyen cuerpos hialinos y cuerpos coloides del disco óptico. Tienen una prevalencia de 1 por 500 y el 60 por ciento de los casos se encuentran profundos en la cabeza del nervio óptico. La patogenia primaria de los drusen puede ser una displasia hereditaria del canal óptico del disco óptico y su vasculatura, lo que predispone a la formación de estos. La evolución natural de los drusen es un proceso dinámico que transcurre durante toda la vida. Entre las complicaciones asociadas se presentan defectos de campo visual, pérdida de visión central (rara pero bien documentada), neuropatía óptica isquémica, oclusiones vasculares retinales, pérdidas transitorias de la visión, neovascularización subretinal peripapilar, corioretinopatia serosa central peripapilar y hemorragias pre y peripapilares. Se presenta una paciente de 64 años de edad con antecedente de haber sido operada de desprendimiento de retina del ojo izquierdo, y en el ojo derecho presentaba una hemorragia peripapilar subretinal profunda asociada a drusen(AU)


ABSTRACT Optic nerve drusens were first described by Liebreich in the year 1868. Other terms to designate this condition are optic disc hyaline bodies and colloid bodies. They have a prevalence of 1 per 500 and 60 percent of the cases occur deep in the optic nerve head. The primary pathogenesis of drusens may be an inherited dysplasia of the optic canal of the disc and its vasculature, which leads to their formation. The natural evolution of drusens is a lifelong dynamic process. Associated complications include visual field defects, central vision loss (rare but well documented), ischemic optic neuropathy, retinal vascular occlusion, transient sight loss, peripapillary subretinal neovascularization, central serous peripapillary chorioretinopathy, and pre- and peripapillary bleeding. A case is reported of a 64-year-old female patient with a history of surgery for retinal detachment of the left eye. In the right eye the patient presented deep peripapillary subretinal bleeding associated to drusen(AU)


Subject(s)
Humans , Female , Middle Aged , Retinal Detachment/etiology , Homeopathic Pathogenesy/epidemiology , Optic Neuropathy, Ischemic/diagnostic imaging , Optic Nerve Neoplasms/epidemiology
19.
J Ocul Pharmacol Ther ; 36(5): 304-310, 2020 06.
Article in English | MEDLINE | ID: mdl-32186940

ABSTRACT

Purpose: To assess the clinical effects of preoperative, intraoperative, or preoperative combined with intraoperative intravitreal conbercept (IVC) injection in vitrectomy with silicone oil tamponade for severe proliferative diabetic retinopathy (PDR). Methods: Ninety-eight eyes of 98 severe PDR patients undergoing vitrectomy with silicone oil tamponade were randomly assigned to 3 groups: Group 1 (34 eyes) received IVC injections 3 to 5 days before surgery; Group 2 (35 eyes) received IVC injections at the end of surgery; and Group 3 (29 eyes) received IVC injections 3 to 5 days before and at the end of operation. Follow-up examinations were performed for 6 months. Results: The incidence and severity of intraoperative bleeding were not significantly different (P = 0.233). However, the duration of surgery was significantly shorter in Group 1 and Group 3 compared with Group 2 (P < 0.001). The incidences of early and late recurrent vitreous hemorrhage (VH) were 32.35%, 28.57%, and 13.80%, respectively. At 6-month follow-up, mean best-corrected visual acuity had significantly increased to 1.25 ± 0.45 logMAR in Group 1, 1.29 ± 0.46 logMAR in Group 2, 1.16 ± 0.44 logMAR in Group 3 (all P < 0.001). The incidence of postoperative VH, neovascular glaucoma, and retinal detachment in Group 3 was slightly lower, however, no significant differences were observed (all P > 0.05). In young patients, similar results were observed and Group 3 had better visual improvements (P = 0.037). Conclusions: Preoperative IVC injection could be a safe and effective adjunct in pars plana vitrectomy with silicone oil tamponade for severe PDR. Preoperative combined with intraoperative IVC are promising, especially in young patients.


Subject(s)
Angiogenesis Inhibitors/therapeutic use , Chemotherapy, Adjuvant/methods , Diabetic Retinopathy/drug therapy , Recombinant Fusion Proteins/therapeutic use , Vascular Endothelial Growth Factor A/antagonists & inhibitors , Vitrectomy/methods , Adult , Aged , Angiogenesis Inhibitors/administration & dosage , Chemotherapy, Adjuvant/statistics & numerical data , Combined Modality Therapy , Diabetic Retinopathy/diagnosis , Diabetic Retinopathy/surgery , Female , Follow-Up Studies , Glaucoma, Neovascular/epidemiology , Hemorrhage/epidemiology , Humans , Incidence , Intraoperative Care/statistics & numerical data , Intravitreal Injections , Male , Middle Aged , Operative Time , Postoperative Complications/epidemiology , Preoperative Care/statistics & numerical data , Prospective Studies , Recombinant Fusion Proteins/administration & dosage , Retinal Detachment/epidemiology , Severity of Illness Index , Silicone Oils/administration & dosage , Silicone Oils/therapeutic use , Visual Acuity/physiology , Vitreous Hemorrhage/epidemiology
20.
Ophthalmic Physiol Opt ; 40(3): 281-288, 2020 05.
Article in English | MEDLINE | ID: mdl-32196734

ABSTRACT

PURPOSE: To identify the genetic defect causing early-onset high myopia (eoHM)/ocular-only Stickler syndrome (ocular-STL) in a large Chinese family. METHODS: Genomic DNA and clinical data from a four-generation family with eoHM/ocular-STL were collected. Whole-exome sequencing was performed on one affected member in initial screening. Linkage scan based on microsatellite markers was carried out initially from candidate loci associated with autosomal dominant eoHM and Stickler syndrome. Sanger sequencing was used to detect potential variants. The pathogenicity of candidate variants was evaluated using mini genes ex vivo. RESULTS: Eight patients and five unaffected members in the family participated in the study, in which the patients had high myopia with other variable ocular phenotypes but without extraocular abnormalities. Whole exome sequencing did not detect any potential pathogenic variant in all genes known to associate with the disease. The eoHM/ocular-STL in the family was mapped to markers around COL2A1 by candidate loci linkage scan, with a maximum lod score of 3.31 for D12S1590 at θ = 0. A novel deep intronic variant, c.86-50C > G in intron 1 of COL2A1, was detected by Sanger sequencing and co-segregated with eoHM/ocular-STL in the family. Ex vivo splicing test using mini genes confirmed that the variant created a new splicing acceptor 49 bp before the canonical splicing site of exon 2, resulted in addition of 49 bp fragment in the transcript (from c.86-49 to c.86-1) and premature termination. CONCLUSIONS: Linkage study, bioinformatics prediction, and ex vivo transcript analysis suggest a novel deep intronic variant adjacent to 5-prime of exon 2 of COL2A1, affecting exon 2 splicing, as a potential cause of ocular-STL in a large family. To our knowledge, this is the first report of an intronic variant around exon 2 as a cause of ocular-STL while a series of variants in the coding region of exon 2, a dispensable alternative-splicing exon for extraocular tissues, in COL2A1 have been reported to cause Stickler syndrome-related ocular phenotype alone.


Subject(s)
Arthritis/genetics , Collagen Type II/genetics , Connective Tissue Diseases/genetics , DNA/genetics , Hearing Loss, Sensorineural/genetics , Myopia/genetics , Retinal Detachment/genetics , Adolescent , Adult , Aged , Arthritis/metabolism , Child , Collagen Type II/metabolism , Connective Tissue Diseases/metabolism , DNA Mutational Analysis , Female , Hearing Loss, Sensorineural/metabolism , Humans , Male , Middle Aged , Mutation , Myopia/metabolism , Pedigree , Retinal Detachment/metabolism , Time Factors , Young Adult
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