Two-Year Outcome of Aflibercept for the Treatment of Choroidal Neovascularization in Punctate Inner Choroidopathy

Punctate inner choroidopathy (PIC) is a rare inflammatory chorioretinopathy that predominantly affects young myopic women. Visual prognosis is generally good, but occurrence of choroidal neovascularization (CNV) is common and may be vision threatening. Case reports and short case series support the effectiveness of intravitreal anti-vascular endothelial growth factor (VEGF) agents (ranibizumab and bevacizumab) for CNV associated with PIC given their anti-angiogenic and anti-inflammatory effects. Evidence concerning aflibercept, a more recent intravitreal anti-VEGF, is limited to a single case report. In this case report, we illustrate the case of a 43-year-old myopic woman presenting with visual acuity loss and distortion in the right eye over the last 5 days in whom CNV associated with PIC was diagnosed. Treatment with 1 injection per month of intravitreal aflibercept for 2 months and full-dose oral prednisone for 1 week, being tapered afterwards, improved visual acuity and resolved CNV, with benefits lasting up to 24 months.

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Sequential bilateral Behçet’s neuroretinitis associated with prepapillary vitreous exudate: case report

Journal of Ophthalmic Inflammation and Infection ◽

10.1186/s12348-020-00226-y ◽

2020 ◽

Vol 10 (1) ◽

Author(s):

Imen Ksiaa ◽

Safa Ben Aoun ◽

Sourour Zina ◽

Dhouha Nefzi ◽

Sana Khochtali ◽

...

Keyword(s):

Case Report ◽

Single Case ◽

Signal Loss ◽

Fiber Layer ◽

Swept Source ◽

Single Case Report ◽

Shadowing Effect ◽

Eye Involvement ◽

The Right ◽

Inflammatory Changes

Abstract Objective To describe a case of Behçet disease (BD) uveitis manifesting with sequential bilateral neuroretinitis associated with prepapillary inflammatory vitreous exudate (PIVE). Material and methods A single case report documented with multimodal imaging. Results A 37-year-old man developed neuroretinitis with associated PIVE in the left eye. He was diagnosed with ocular toxoplasmosis and treated accordingly based on positive serologic testing and negative work-up for other entities, including BD. The disease course was favorable, but 1 year later a similar neuroretinitis developed in the right eye. Extraocular features of BD became evident only at the time of the second eye involvement, and the patient received corticosteroid and immunosuppressive therapy. Swept source (SS) OCT showed at the acute phase in both eyes a typical “mushroom-shaped” prepapillary hyperreflectivity of the PIVE. SS OCT angiography (OCTA) demonstrated a corresponding prepapillary hypointense area due to shadowing effect, decreasing in size while scanning deeper layers. It also detected peripapillary retinal hypervascularity in both eyes and a sectoral area of flow signal loss in the first involved left eye. Visual acuity improved following the resolution of the PIVE and associated acute inflammatory changes in both eyes. The left eye showed residual optic disc pallor and retinal nerve fiber layer defects. Conclusion Sequential bilateral neuroretinitis associated with PIVE may occur before other clinical features of BD become evident. SS OCT and OCTA can provide useful information for the diagnosis and management of this rare, but typical, ocular manifestation of BD uveitis.

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The Neuropathology of 1p36 Deletion Syndrome: An Autopsy Case Series

Journal of Neuropathology & Experimental Neurology ◽

10.1093/jnen/nlab072 ◽

2021 ◽

Author(s):

Kyle S Conway ◽

Fozia Ghafoor ◽

Amy C Gottschalk ◽

Joseph Laakman ◽

Renee L Eigsti ◽

...

Keyword(s):

Neuronal Migration ◽

Single Case ◽

Case Reports ◽

Case Series ◽

Deletion Syndrome ◽

Cerebellar Hypoplasia ◽

Autopsy Case ◽

Single Case Report ◽

Renal Abnormalities

Abstract 1p36 deletion syndrome is the most common terminal deletion syndrome, manifesting clinically as abnormal facies and developmental delay with frequent cardiac, skeletal, urogenital, and renal abnormalities. Limited autopsy case reports describe the neuropathology of 1p36 deletion syndrome. The most extensive single case report described a spectrum of abnormalities, mostly related to abnormal neuronal migration. We report the largest published series of 1p36 autopsy cases, with an emphasis on neuropathologic findings. Our series consists of 3 patients: 2 infants (5-hours old and 23-days old) and 1 older child (11 years). Our patients showed abnormal cortical gyration together with a spectrum of neuronal migration abnormalities, including heterotopias and hippocampal abnormalities, as well as cerebellar hypoplasia. Our findings thus support the role of neuronal migration defects in the pathogenesis of cognitive defects in 1p36 deletion syndrome and broaden the reported neuropathologic spectrum of this common syndrome.

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Subungual Schwannoma, a Rare Entity: A Case Report and Literature Review

The Journal of Hand Surgery (Asian-Pacific Volume) ◽

10.1142/s2424835520720182 ◽

2020 ◽

Vol 25 (04) ◽

pp. 508-512

Author(s):

Jason D. Wink ◽

Olatomide Familusi ◽

Ines C. Lin

Keyword(s):

Case Report ◽

Literature Search ◽

Glomus Tumor ◽

Single Case ◽

Case Reports ◽

Ring Finger ◽

Dominant Male ◽

Rare Entity ◽

Single Case Report ◽

Slow Growing

We present a case of a 26-year-old right hand dominant male landscaper with a slow growing right ring finger subungual mass. MRI confirmed a 0.9 × 1.5 × 0.9 cm well circumscribed subungual mass believed to be consistent with a glomus tumor, although size and symptoms were not consistent with that diagnosis. The mass was completely excised and diagnosis of schwannoma was confirmed by H&E histology. A literature search was performed utilizing the term “subungual schwannoma.” Four case reports were found describing this diagnosis in the hand as well as a single case report describing it in the foot. In summary, this is a 26-year-old male who presents with a schwannoma in the unusual subungual location. Although rare, based on our case and the existing literature, subungual schwannomas should be included in the differential diagnosis of a slow growing subungual mass, particularly if symptoms and exam are inconsistent with more common etiologies.

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CRPS of Lower Extremity– A Case Report and Review

Indian Journal of Physical Medicine and Rehabilitation ◽

10.5005/ijopmr-27-1-22 ◽

2016 ◽

Vol 27 (1) ◽

pp. 22-28

Author(s):

Mahima Agrawal

Keyword(s):

Case Report ◽

Lower Extremity ◽

Lower Limb ◽

Relaxation Training ◽

Single Case ◽

Case Series ◽

Pain Syndrome ◽

Type I ◽

Retrospective Case ◽

Single Case Report

Abstract Complex regional pain syndrome (CRPS) of the lower limb is a relatively uncommon entity as compared to CRPS of the upper extremity. Literature search has revealed only 2 retrospective case series and a single case report of lower extremity CRPS type I from 1975 to 2014 on Pubmed, isolated cases of CRPS type I of lower extremity have also been reported following knee surgeries and arthroscopies. This report presents a case of lower limb CRPS type I following blunt trauma to right foot, treatment of which was directed towards management of allodynia, vasomotor symptoms and surgical correction of deformity which had developed because of the disease, coping mechanisms were also reinforced through counselling and relaxation training. The individual responded well to treatment with a reported 75% reduction in the disabling symptoms and improvement in ambulatory status.

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Sequential bilateral Behçet’s neuroretinitis associated with prepapillary vitreous exudate: case report

10.21203/rs.3.rs-44040/v2 ◽

2020 ◽

Author(s):

Imen Ksiaa ◽

Safa Ben Aoun ◽

Sourour Zina ◽

Dhouha Nefzi ◽

Sana Khochtali ◽

...

Keyword(s):

Visual Acuity ◽

Case Report ◽

Multimodal Imaging ◽

Single Case ◽

Signal Loss ◽

Fiber Layer ◽

Swept Source ◽

Single Case Report ◽

Shadowing Effect ◽

Inflammatory Changes

Abstract Objective: To describe a case of Behçet disease (BD) uveitis manifesting with sequential bilateral prepapillary inflammatory vitreous exudate (PIVE) documented with multimodal imaging, including swept source optical coherence tomography (SS OCT) and OCT angiography. Material and Methods: A single case report.Results: A 37-year-old man developed sequential bilateral PIVE, initially misdiagnosed as toxoplasmic neuroretinitis, before extraocular features of BD became evident and the patient treated accordingly. SS OCT showed at the acute phase in both eyes a typical “mushroom-shaped” prepapillary hyperreflectivity of the PIVE. SS OCTA demonstrated a corresponding prepapillary hypointense area due to shadowing effect, decreasing in size while scanning deeper layers. It also detected peripapillary retinal hypervascularity in both eyes and a sectoral area of flow signal loss in the first involved left eye. Visual acuity improved following the resolution of the PIVE and associated acute inflammatory changes in both eyes. The left eye showed residual optic disc pallor and retinal nerve fiber layer defects.Conclusion: Sequential bilateral PIVE may occur in the absence of other clinical features of BD. SS OCT and OCTA can provide useful information for the diagnosis and management of PIVE and underlying optic nerve inflammation.

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1271 High-Density EEG Correlates During Confusional Arousals in a Child With a Long-Lasting History Of Sleepwalking

SLEEP ◽

10.1093/sleep/zsaa056.1265 ◽

2020 ◽

Vol 43 (Supplement_1) ◽

pp. A483-A483

Author(s):

A Castelnovo ◽

N Amato ◽

S Riccardi ◽

M Pereno ◽

S Miano ◽

...

Keyword(s):

Prefrontal Cortex ◽

Case Report ◽

Sleep Stage ◽

Single Case ◽

Case Reports ◽

Nrem Sleep ◽

High Density ◽

Source Power ◽

History Of ◽

The Right

Abstract Introduction Sleepwalking belongs to a family of disorders (Disorders of Arousal - DOA) that are thought to derive from incomplete arousals out of Non Rapid Eye Movement (NREM) sleep. At yet, our knowledge about the specific neural dynamics occurring during clinical episodes is limited and relies on one SPECT case study, four stereo-EEG case reports/series and one single high-density electro-encephalography (hdEEG) case report. We herein describe a single case captured by hdEEG. Report of Case We collected two consecutive sleep recordings (using a 256-channel hdEEG coupled with standard video-polysomnography) of a non-medicated, otherwise healthy, 13-year-old male, with a history of recurrent daily sleepwalking episodes. We visually identified 17 behavioral events during sleep stage 3 and divided them into two groups: clear clinical episodes (n = 7) and simple movements associated with burst of delta waves (n = 10). Source power topography in the delta range (1-4 Hz) was computed using LORETA. Source images during selected episodes were compared to 30 second-windows of baseline stage 3 sleep. Comparisons were performed using statistical non-parametric mapping with supra-threshold cluster tests. Events were associated with an increase of delta power over the right frontopolar prefrontal cortex (rPFC) / Broadman area 10 (BA10) at their onset. This finding was clearly observable even when considering only clear-cut events, followed by the involvement of the right dorsolateral and medial prefrontal cortex / BA9 and of the left superior temporal gyrus/ BA 22. Conclusion We were able to replicate a recently published case report by our group, where we highlighted the putative role of rPFC and PFC and prefronto-temporal circuit in DOA episodes. Intriguingly, we observed a lateralization of this effect, with a prominent right frontal involvement. Novel research has shown a physiological asymmetry in the generation of large slow waves between the two hemispheres. An increased right-left unbalance might prime behavioral episodes in DOA patients.

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Sequential bilateral Behçet’s neuroretinitis associated with prepapillary vitreous exudate: case report

10.21203/rs.3.rs-44040/v3 ◽

2021 ◽

Author(s):

Imen Ksiaa ◽

Safa Ben Aoun ◽

Sourour Zina ◽

Dhouha Nefzi ◽

Sana Khochtali ◽

...

Keyword(s):

Case Report ◽

Single Case ◽

Signal Loss ◽

Fiber Layer ◽

Single Case Report ◽

Shadowing Effect ◽

Eye Involvement ◽

One Year ◽

The Right ◽

Inflammatory Changes

Abstract Objective: To describe a case of Behçet disease (BD) uveitis manifesting with sequential bilateral neuroretinitis associated with prepapillary inflammatory vitreous exudate (PIVE). Material and Methods: A single case report documented with multimodal imaging.Results: A 37-year-old man developed neuroretinitis with associated PIVE in the left eye. He was diagnosed with ocular toxoplasmosis and treated accordingly based on positive serologic testing and negative work-up for other entities, including BD. The disease course was favorable, but one year later a similar neuroretinitis developed in the right eye. Extraocular features of BD became evident only at the time of the second eye involvement, and the patient received corticosteroid and immunosuppressive therapy. SS OCT showed at the acute phase in both eyes a typical “mushroom-shaped” prepapillary hyperreflectivity of the PIVE. SS OCTA demonstrated a corresponding prepapillary hypointense area due to shadowing effect, decreasing in size while scanning deeper layers. It also detected peripapillary retinal hypervascularity in both eyes and a sectoral area of flow signal loss in the first involved left eye. Visual acuity improved following the resolution of the PIVE and associated acute inflammatory changes in both eyes. The left eye showed residual optic disc pallor and retinal nerve fiber layer defects.Conclusion: Sequential bilateral neuroretinitis associated with PIVE may occur before other clinical features of BD become evident. SS OCT and OCTA can provide useful information for the diagnosis and management of this rare, but typical, ocular manifestation of BD uveitis.

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“At-Home” Photobiomodulation: A New Approach for Bell’s Palsy Treatment

Case Reports in Neurological Medicine ◽

10.1155/2021/5043458 ◽

2021 ◽

Vol 2021 ◽

pp. 1-5

Author(s):

Carlo Fornaini ◽

Zhao Meng ◽

Elisabetta Merigo ◽

Jean-Paul Rocca

Keyword(s):

Case Report ◽

Single Case ◽

Bell’S Palsy ◽

Pharmacological Therapy ◽

Bell's Palsy ◽

Single Case Report ◽

First Case ◽

Total Fluence ◽

The Right ◽

At Home

Objective. This report is the first one to describe the possibility to use “self‐administered” photobiomodulation (PBM) for Bell’s palsy (BP) treatment. Background. BP is a peripheral disorder of the facial nerve causing sudden paralysis of unilateral facial muscles, and PBM has been successfully suggested for its treatment without any side effect. This is the first case report where a laser device was successfully used at home by the patient herself to treat BP opening new perspectives on the therapy of this disease. Methods. This report describes the “at-home PBM” treatment performed on a 15-year-old girl who presented BP consisting of acute pain on the right side of her face, difficulty in biting and dripping saliva from the right side of her lips. The treatment was performed twice a day by cutaneous applications, each of 15 minutes (total fluence 48 J/cm2) in an area corresponding to the parotid gland by a device emitting at 808 nm at 250 mW output power. Results. Two weeks after PBM treatment, performed at home twice a day by the patient herself without any kind of pharmacological therapy, the complete disappearing of the disease was noticed with no side effects. Conclusion. With the limitations due to a single case report and with the need of further clinical trials to confirm it, “at-home PBM” seems to represent a good and safe approach to the treatment of BP.

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Visibility of Transcondylar Screw Fractures in Standard and Extended Scale Computed Tomography Images

Veterinary and Comparative Orthopaedics and Traumatology ◽

10.1055/s-0041-1740620 ◽

2021 ◽

Author(s):

Robert Brash ◽

Jose Labrador ◽

Andrew Holdsworth

Keyword(s):

Computed Tomography ◽

Case Report ◽

Single Case ◽

Case Series ◽

Implant Failure ◽

Metal Implant ◽

Single Case Report ◽

Computed Tomography Images ◽

Design Case ◽

Extended Scale

Abstract Objective Application of extended computed tomography scale (ECTS) reconstruction to diagnose metal implant failure has been described in a single case report. The purpose of this study was to compare the features and visibility of humeral transcondylar screw fractures in standard CT scale (SCTS) and ECTS images. Study Design Case series: CT images of dogs with fractured transcondylar screws were retrospectively reviewed and described in both SCTS and ECTS images. Results Five dogs with a total of six transcondylar screw failures (five right and one bilateral) were reviewed. All cases had an ongoing humeral intercondylar fissure with varying degrees of stress remodelling. The fracture was seen in all screws on ECTS images, however only in three implants on SCTS images. The measured fracture gap was larger in ECTS images in all cases (range: + 0.14 mm to + 0.28mm). The three smallest fracture gaps were not seen on SCTS images. A subtle hypoattenuating streak (artefact) was visible adjacent to the screw fracture in 5/6 of cases using SCTS images. All screw fractures occurred parallel and often slightly medial to the humeral intercondylar fissure. Conclusion Implant failure is only seen with larger fracture gaps in SCTS images, with 3/6 screw fractures not visible in SCTS compared with ECTS. A hypoattenuating streak extending perpendicular to the implant in SCTS images is suggestive of screw fracture even if this is not directly visible.

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