Twelve-year follow up of a case of autosomal recessive bestrophinopathy with transient resolution of retinal edema in one eye

2022 ◽  
pp. 112067212110732
Author(s):  
Lidia Remolí Sargues ◽  
Clara Monferrer Adsuara ◽  
Raquel Rodríguez López ◽  
Roberto Gallego Pinazo ◽  
Javier Montero Hernández ◽  
...  
Keyword(s):  
Case Report ◽  
Fundus Autofluorescence ◽  
Anterior Segment ◽  
Subretinal Fluid ◽  
Posterior Pole ◽  
Dietary Therapy ◽  
Retinal Edema ◽  
Blurred Vision ◽  
First Case

Purpose To report 12-year follow-up of a patient with ARB. Case report A 25-year-old man presented with blurred vision in his both eyes (OU). Best-corrected visual acuity (BCVA) was 20/63 Snellen equivalent in the right eye (OD) and 20/32 Snellen equivalent in the left eye. The intraocular pressures and anterior segment examination were unrevealing in OU. Posterior segment examination revealed multiple yellowish flecks and dots in the posterior pole in OU. Optical coherence tomography (OCT) showed subretinal fluid (SRF), intraretinal hyporeflective spaces, elongated and shaggy photoreceptors and outer retinal defects. Fundus autofluorescence demonstrated mottling hyperautofluorescence and hypoautofluorescence in the posterior pole in OU. Fluorescein angiography illustrated hyperfluorescence in the posterior pole and surrounding the arcades in OU. Multifocal electroretinography objectified mild to markedly abnormal responses in all ring areas in OU. Molecular genetic testing confirmed two heterozygous sequence variations in the BEST1 gene. At 4 years of follow-up, OCT revealed a complete resolution of SRF and a partial resolution of intraretinal hyporeflective spaces in the OD with corresponding improvement in the BCVA to 20/23 Snellen equivalent in the OD, even though outer retinal defects persisted. Our patient denied recent changes in his alimentary habits and medical history at that time. Posteriorly, SRF and intraretinal hyporeflective spaces reappeared in the OD. Conclusion To the best of our knowledge, this is the first case report of ARB with a transient resolution of retinal edema in one eye without medical treatment and dietary therapy.

Chronic Central Serous Chorioretinopathy; Signs, Diagnosis and Treatment

2020 ◽  
pp. 104-111
Keyword(s):  
Central Serous Chorioretinopathy ◽  
Pigment Epithelium ◽  
Fundus Autofluorescence ◽  
Retinal Pigment ◽  
Subretinal Fluid ◽  
Fluid Accumulation ◽  
Fundus Fluorescein Angiography ◽  
Blurred Vision ◽  
Chronic Central Serous Chorioretinopathy

Central serous chorioretinopathy (CSCR) is a maculopathy characterized by the separation of the neurosensory layer as a result of fluid accumulation between the retinal pigment epithelium (RPE) and the photoreceptor layer. Classically it is classified as acute and chronic forms. When the disease lasts longer than 4-6 months, it is called a chronic form and comprises 15% of all CSCR cases. Although the exact etiology is unknown; studies emphasize changes in choroidal circulation causing choroidal ischemia and vascular hyperpermeability as well as subretinal fluid accumulation due to deterioration pump function of RPEs. Subretinal fluid accumulation can lead to photoreceptor dysfunction and loss of vision. Classical findings in patients are a decrease in visual acuity, blurred vision, metamorphopsia, micropsia, disturbance in color vision and dark adaptation, and scotomas. Diagnosis and follow-up depend on fundoscopy as well as imaging. Optical coherent tomography is the primary method. Fundus autofluorescence (FAF) is useful in defining RPE changes noninvasively. Fundus fluorescein angiography (FFA) shows the source of leakage. In recurrent, unresolved and chronic cases, OCT, FAF, FFA, and indocyanine green angiography can be used all together to manage the disease, to follow-up its extension, and to diagnose possible neovascular as well as polypoidal component. For the treatment of chronic CSCR patients, besides medical treatments such as carbonic anhydrase inhibitors, mineralocorticoid receptor, and glucocorticoid antagonists and intravitreal vascular endothelial growth factor antagonist (Anti-VEGF) injections, half-dose photodynamic therapy and subthreshold micropulse laser treatment are used. Prospective, controlled trials with large series for the treatment of chronic CSCR warranted.

scholarly journals Increase in Central Retinal Edema after Subthreshold Diode Micropulse Laser Treatment of Chronic Central Serous Chorioretinopathy

2015 ◽  
Vol 2015 ◽  
pp. 1-4 ◽  
Author(s):  
Maciej Gawęcki
Keyword(s):  
Central Serous Chorioretinopathy ◽  
Fundus Autofluorescence ◽  
Subretinal Fluid ◽  
Intravitreal Bevacizumab ◽  
Thermal Reaction ◽  
Retinal Edema ◽  
Chronic Central Serous Chorioretinopathy ◽  
Intravitreal Bevacizumab Injection ◽  
Micropulse Laser

Purpose. Subthreshold diode micropulse laser (SDM) treatment is believed to be safe method of treating clinical entities involving retinal edema. We present a case of serous edematous reaction of the retina to SDM treatment.Methods. Case report.Results. A patient with chronic central serous chorioretinopathy (CSCR) was treated with SDM Yellow multispot laser. Procedure had been preceded by careful titration of the laser power, which after achieving of the threshold parameter was decreased by 50%. The follow-up visit two days after treatment revealed significant central retinal edema and subretinal fluid. Fundus autofluorescence image showed thermal reaction from the RPE in the form of small spots of hyperfluorescence corresponding to the laser multispot pattern used for treatment. Retinal edema resolved after topical bromfenac and single intravitreal bevacizumab injection. Slight pigmentary reaction from the RPE persisted.Conclusion. In the treatment of CSCR, there is a need to significantly reduce threshold SDM power parameters or simply use very low power without titration.

scholarly journals Acute Hydrops with Micro-Cornea – an Extremely Rare Condition: A Case Report

2017 ◽  
Vol 1 (1) ◽  
pp. oapoc.0000003
Author(s):  
Tuhin Chowdhury ◽  
Aditya Pradhan
Keyword(s):  
Optical Coherence Tomography ◽  
Case Report ◽  
General Anaesthesia ◽  
Systemic Disease ◽  
Anterior Segment ◽  
Rare Condition ◽  
First Case ◽  
Intracameral Injection ◽  
Lost To Follow Up

Purpose To report the first case of a one-eyed child with micro-cornea, coloboma of iris and acute hydrops without any other disorder of the cornea or systemic disease. Methods Intracameral injection of perfluoro propane (C3F8) gas was performed under general anaesthesia. Results Resolution of the hydrops was observed at 1st week of follow-up, which was confirmed by anterior segment optical coherence tomography. Patient was subsequently lost to follow-up. Conclusions Intracameral injection of perfluoro propane gas is an effective way to quickly resolve acute hydrops. In this specific case, the patient presented with micro-cornea and no other corneal abnormalities.

Optic disc and choroidal metastases secondary to breast cancer: A case report

2021 ◽  
pp. 112067212110362
Author(s):  
Rim Bouraoui ◽  
Khaled El Matri ◽  
Yousra Falfoul ◽  
Zied Chelly ◽  
Ahmed Chebil ◽  
...  
Keyword(s):  
Breast Cancer ◽  
Case Report ◽  
Optic Disc ◽  
Infrared Imaging ◽  
Subretinal Fluid ◽  
Posterior Pole ◽  
Wide Field ◽  
Fundus Examination ◽  
Blurred Vision ◽  
Macular Lesion

Aim: To report an uncommon case of optic disc and multiple choroidal metastases secondary to breast cancer, assessed with swept source optical coherence tomography (SS-OCT), fluorescein (FA), and infracyanine (ICGA) angiographies. Methods: Observational case report. Case presentation: A 40-year-old woman with history of left breast carcinoma presented with blurred vision in her right eye (RE). Her visual acuity was 1/20 in the RE and 10/10 in the left eye. Fundus examination of the RE showed a large yellowish elevation of the posterior pole and a particular whitish nodular papillary cluster protruding from the optic disc into the vitreous. Infrared imaging enhanced the papillary nodular infiltrates. Characteristic findings of choroidal metastasis were noted within the macular lesion on SS-OCT and ICGA. SS-OCT showed specific “lumpy bumpy” irregularity of the anterior surface of the choroid and elevated hyperreflective nodular lesions of the optic disc associated to peripapillary subretinal fluid. The papillary lesions appeared as a bunch of hypofluorescent dots on both FA and ICGA, and ultra-wide field FA was helpful clearly delimiting the large macular lesion. Besides, comprehensive imaging and especially ICGA could detect two asymptomatic choroidal metastases in a systematic assessment of the fellow eye. Conclusion: Optic disc metastases are extremely rare. Their diagnosis can be easily done on fundus examination when presenting with characteristic whitish cluster nodular infiltrates of the optic disc. However multimodal imaging remains very useful for the assessment of the local extension of the lesion and for diagnosing associated asymptomatic choroidal lesions gone unnoticed at the fundus examination.

scholarly journals First reported case of penile prosthesis infection from brucellosis: case report

2020 ◽  
Vol 26 (1) ◽  
Author(s):  
Nabil Nabil Moohialdin ◽  
Ahmad Shamsodini ◽  
Steven K. Wilson ◽  
Osama Abdeljaleel ◽  
Ibrahim Alnadhari ◽  
...  
Keyword(s):  
Case Report ◽  
Broad Spectrum ◽  
Surgical Intervention ◽  
Penile Prosthesis ◽  
Raw Milk ◽  
Prosthesis Infection ◽  
Case Presentation ◽  
First Case ◽  
Milk Ingestion

Abstract Background Infection after the penile prosthesis can be devastating to both the patient and surgeon with various complications and consequences. After introduction of antibiotic-coated implants, the rate of infection has dramatically decreased, but still we see uncommon organisms causing infection. We present a first case report of penile prosthesis infection by brucellosis due to raw milk ingestion. To our knowledge, this is the first reported case of brucellosis penile prosthesis infection. Case presentation We present a first case report of penile prosthesis infection by brucellosis due to raw milk ingestion. A 75-year-old, diabetic male patient presented with penile prosthesis infection 5 months post-penile exchange surgery due to mechanical malfunctioning of 2-piece penile prosthesis which was inserted 11 years ago. The initial treatment with broad spectrum antibiotics did not subside the infection. After diagnosis of brucellosis, the antibiotic was changed to anti-brucellosis (Rifampicin + Tetracycline). The patient improved dramatically and was discharged home with smooth follow-up course. Conclusion Brucellosis can cause infection of penile prosthesis and can be treated with anti-brucellosis antibiotics without necessitating surgical intervention and removal of prosthesis.

scholarly journals Posterior Uveal Effusion after Cataract Surgery

2021 ◽  
pp. 62-67
Author(s):  
Annegret Abaza ◽  
Özlem Dikmetas ◽  
Irmingard Neuhann ◽  
Faik Gelisken
Keyword(s):  
Cataract Surgery ◽  
Corticosteroid Therapy ◽  
Anterior Segment ◽  
Subretinal Fluid ◽  
Posterior Pole ◽  
Functional Improvement ◽  
Long Term Observation ◽  
The Right ◽  
Uveal Effusion

We report a case of posterior uveal effusion (UE) with a long-term follow-up that has occurred following cataract surgery. A 64-year-old woman presented with diminished vision of the right eye (RE) 3 weeks after an uneventful phacoemulsification and intraocular lens implantation. Complete ophthalmic examination including fluorescein angiography (FA), indocyanine green angiography (ICGA), echography and optical coherence tomography (OCT) were performed. Best corrected visual acuity (BCVA) of the RE was 20/50. Anterior segment and intraocular pressure were unremarkable. OCT revealed prominent folds of the choroid and retina, subretinal fluid and darkening of the choroid with reduced visibility of the choroidal vessels and the scleral border. The left eye (LE) was unremarkable. BCVA of the LE was: 20/20. After topical anti-inflammatory and systemic corticosteroid therapy for 5 months, no morphological change of the macula was seen. The patient was observed without any treatment. Forty-three months after the cataract surgery and 38 months after cessation of the corticosteroid therapy, OCT revealed a normal macular morphology and the BCVA improved to 20/25. Even though rare, UE at the posterior pole may occur after modern cataract surgery. OCT examination is a reliable tool in monitoring the macular morphology. Since morphological and functional improvement can be seen in long-term, observation may be considered for some cases of posterior UE with resistance to the therapy.

CONGENITAL MICROGASTRIA, A CASE REPORT WITH A 26-YEAR FOLLOW-UP

PEDIATRICS ◽  
1973 ◽  
Vol 51 (6) ◽  
pp. 1037-1041
Author(s):  
Eugene Blank ◽  
Alvin Chisolm
Keyword(s):  
Case Report ◽  
English Language ◽  
Normal Adult ◽  
Healthy Children ◽  
Adult Size ◽  
Persistent Vomiting ◽  
First Case ◽  
English Language Journal ◽  
Roentgenographic Examination

A 27-year-old woman had congenital microgastria, which was apparent in roentgenographic examination when she was 1 year of age. Despite inability to eat anything but pureed foods for the first 13 years and despite persistent vomiting during that time, she has reached normal adult size and has three healthy children. This report represents two firsts: the first case of congenital microgastria in an English language journal of pediatrics and the first with a long follow-up.

scholarly journals Teratoma of larynx: case report

2021 ◽  
Vol 7 (10) ◽  
pp. 1700
Author(s):  
Rakesh Srivastava ◽  
Vini Tandon
Keyword(s):  
Carbon Dioxide ◽  
Case Report ◽  
Germ Cells ◽  
Carbon Dioxide Laser ◽  
Rare Case ◽  
Histopathological Examination ◽  
Flexible Laryngoscopy ◽  
First Case ◽  
Congenital Teratoma

<p class="abstract">Teratomas are embryonal neoplasm arises from totipotent germ cells. They are having tissues from all the three blastodermic layers. There are various locations of congenital teratoma. Naso-oropharyngeal site teratoma are either sessile or pedunculated. We describe a rare case of laryngeal teratoma in a five years old patient presented with change in voice and breathing difficulty. On flexible laryngoscopy, it appeared like supraglottic cyst but on CT scan it was confirmed as teratoma. Pre-operative tracheostomy and transoral carbon dioxide laser assisted excision done. Histopathological examination showed osteoid trabeculae, chondroid tissue with loose myxoid islands and adipose tissue. No recurrence of tumor on 18 months follow-up. This is the first case report of pediatric larynx teratoma reported in present century.</p>

scholarly journals Inflammatory Myofibroblastic Tumor of the Epiglottis Excised with a Carbon Dioxide Laser: Case Report and Literature Review

2018 ◽  
Vol 97 (8) ◽  
pp. E31-E33 ◽  
Author(s):  
Blake Raggio ◽  
Neil Chheda
Keyword(s):  
Carbon Dioxide ◽  
Case Report ◽  
Adjuvant Therapy ◽  
Literature Review ◽  
Carbon Dioxide Laser ◽  
Inflammatory Myofibroblastic Tumor ◽  
Benign Neoplasm ◽  
First Case ◽  
Laser Excision

Inflammatory myofibroblastic tumor (IMT) is a benign neoplasm of intermediate biologic potential. It rarely occurs in the larynx, and it has not been previously reported in the epiglottis. We treated a 66-year-old woman who presented with progressive dysphonia and a mass on her suprahyoid epiglottis. The tumor was completely excised with a CO2 laser; no adjuvant therapy was administered. Histopathology revealed that the mass was an IMT. No evidence of recurrence was noted after 6 months of follow-up. We present what we believe is the first case of an epiglottic IMT to be reported in the literature, and we propose CO2 laser excision without adjuvant therapy as an acceptable treatment.

Is choriocapillaris the early target in acute maculopathy secondary to hand, foot, and mouth disease? A case report

2020 ◽  
pp. 112067212098252
Author(s):  
Pietro Maria Talli ◽  
Edlira Bendo ◽  
Emilio Pedrotti ◽  
Alberto Pazzaglia
Keyword(s):  
Case Report ◽  
Foot And Mouth Disease ◽  
Mouth Disease ◽  
Ocular Involvement ◽  
Primary Target ◽  
Retrospective Case ◽  
First Case ◽  
Foot And Mouth ◽  
Acute Maculopathy

Purpose: To report the case of a 33-year old man who disclosed the first case of bilateral ocular involvement of hand, foot, and mouth disease (HFMD) with a different stage of the disease in each eye. Methods: Retrospective case report. Results: The study included a patient who incurred in a bilateral HFMD maculopathy. During 6 months follow-up period persistent abnormalities in fundus examination and in OCT scans were reported. Conclusion: We consider that our case shows that choriocapillaris is the primary target of HFMD maculopathy, for this reason we consider that ICGA is mandatory for early diagnosis and an effective treatment.

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