Purpose: Internuclear ophthalmoplegia (INO) is an eye movement disorder caused by a lesion of the medial longitudinal fasciculus. It typically manifests bilaterally and is commonly caused by multiple sclerosis. We report a case of pediatric unilateral INO, which is rare in the pediatric population.Case summary: A 14-year-old female, with a history of traumatic head injury 9 days previously, presented with diplopia, nausea and vomiting for the past 5 days. Her ophthalmic examination revealed severely impaired adduction of the right eye and abduction nystagmus in the left eye. Orbital computed tomography showed no evidence of orbital wall fracture or muscle impingement. However, an magnetic resonance imaging of her brain demonstrated an infarct in the right paramedian midbrain, leading to a diagnosis of unilateral INO. Diplopia and impaired adduction of the right eye resolved completely 7 weeks after the trauma. Conclusions Demyelinating diseases, such as multiple sclerosis, account for most of the pediatric INO cases. Although most of these cases are bilateral, this reports highlights a pediatric case of unilateral INO following head injury, which is an uncommon occurrence in this age group.

BACKGROUND: Interstitial cystitis (IC) is a chronic and intractable disease that can severely deteriorate patients’ quality of life. Recently, stem cell therapy has been introduced as a promising alternative treatment for IC in animal models. We aimed to verify the efficacy and safety of the human perirenal adipose tissue-derived stromal vascular fraction (SVF) in an IC rat model. METHODS: From eight-week-old female rats, an IC rat model was established by subcutaneous injection of 200 lg of uroplakin3A. The SVF was injected into the bladder submucosal layer of IC rats, and pain scale analysis, awakening cytometry, and histological and gene analyses of the bladder were performed. For the in vivo safety analysis, genomic DNA purification and histological analysis were also performed to check tumorigenicity and thrombus formation. RESULTS: The mean pain scores in the SVF 20 ll group were significantly lower on days 7 and 14 than those in the control group, and bladder intercontraction intervals were significantly improved in the SVF groups in a dose-dependent manner. Regeneration of the bladder epithelium, basement membrane, and lamina propria was observed in the SVF group.In the SVF groups, however, bladder fibrosis and the expression of inflammatory markers were not significantly improved compared to those in the control group. CONCLUSION This study demonstrated that a perirenal adipose tissue-derived SVF is a promising alternative for the management of IC in terms of improving bladder pain and overactivity.

Background: The primary goal of scabies treatment is to eliminate mites; however, but post-scabetic itch persists in many patients. Since long-standing itching can lead to the development of other comorbidities, appropriate interventions are necessary. Objective: This study aimed to investigate the effects of inappropriate (continuous or prolonged) scabicide use and oral steroids on the duration of pruritus in patients with scabies. Methods: The duration of pruritus, use of oral corticosteroids, total immunoglobulin E (IgE) levels, specific IgE levels for house dust mites, and eosinophil counts were evaluated. Post-scabetic itch was defined as itching that persisted for more than 3 weeks, although scabies was no longer detected; 208 participants with post-scabetic itch were included. Results: We recruited 265 participants with confirmatory diagnosis of scabies and 116 with clinical diagnosis. We treated 124 participants with scabicides based on the guidelines, whereas 257 participants were not. The type of scabicide did not affect the duration of pruritus. Scabicide overuse did not shorten the duration of pruritus; however, its prolonged use lengthened the treatment period. Although oral corticosteroids did not shorten the duration of pruritus in the patients, they were effective when the patient had post-scabetic itch. In addition, a positive correlation between the total IgE level and duration of pruritus was observed. Conclusion This study investigated the effect of treatment modality on the clinical course of scabies. Our findings suggest that overuse of scabicides might be associated with post-scabetic itch, and short-term use of oral corticosteroids may be effective in reducing its duration.

Erythema multiforme (EM) is an acute, self-limited mucocutaneous disease with diverse triggering factors, and the recurrences are quite common. A 24-year old male presented with multiple erythematous, itchy papules and plaques on multiple sites. He has worked in a lithium battery factory and experienced the chemical burn 2 weeks ago. A histopathologic examination on right wrist showed a scattered lymphocytic infiltration, vacuolar degeneration, and necrotic keratinocyte. The final diagnosis was EM after occupational lithium exposure. He was treated by oral methylprednisolone and experienced recurrences after returning to the same workplace after remission. Although the precise pathogenesis is unknown, the pathogenesis of EM by lithium is related to the effect of lithium on immune system, different from other etiologies. To our knowledge, our case is the first report of EM following the chemical burn and occupational lithium exposure. We report this as an interesting case of EM.

Epithelioid hemangioma is a rare benign vascular neoplasm. Recently, the term “multiple eruptive epithelioid hemangioma” has been proposed for epithelioid hemangioma with distinct features. It is different from usual epithelioid hemangioma because of the multifocal distributions in various body regions with an eruptive onset. In addition, the histopathology of multiple eruptive epithelioid hemangioma shows increased cellular proliferation, mitosis, and nuclear pleomorphism and positive findings for FOS-B compared to classic epithelioid hemangioma. Herein, we report the case of a 59-year-old man with unusual manifestations suitable for multiple eruptive epithelioid hemangioma. He had multiple erythematous to purple-red dome-shaped nodules on the right hand, arm, and shoulder. The initial lesion was a solitary erythematous nodule on the right hand that abruptly extended to the right arm and shoulder. Microscopically, the tumor was a well-demarcated dermal nodule and showed capillary sized vascular structures. Vascular structures had epithelioid endothelial cells with abundant eosinophilic cytoplasm and vesicular nuclei. The tumor cells showed mild nuclear pleomorphism and a few mitosis and feature of resembling cobble stone was observed. In immunohistochemistry, CD31 and CD34 were positive in the endothelial cells.The endothelial cells showed nuclear positivity in FOS-B. Based on the clinical and histopathological findings, the final diagnosis was multiple eruptive epithelioid hemangiomas.This is the first report of multiple eruptive epithelioid hemangiomas in an Asian man after the term had been introduced.

Purpose: To report a rare case of severe ocular hypertension following intravenous methylprednisolone treatment in a child with bilateral optic neuritis.Case summary: A 6-year-old girl presented with bilateral severe visual loss and disc swelling. Magnetic resonance imaging revealed optic nerve enhancement, and she received high-dose intravenous methylprednisolone for 3 days and oral prednisolone 30 mg/day for 2 weeks. Two weeks after initiating treatment, ophthalmologic examination revealed an intraocular pressure (IOP) of 46 mmHg in the right eye and 42 mmHg in the left eye. The best-corrected visual acuity was 1.0 in the right eye and 0.6 in the left eye. Relative afferent pupillary defect was noted in the left eye. Fundus examination showed normal cup-to-disc ratio (0.4) in both eyes. The iridocorneal angle was open in all directions. The treatment plan included the cessation of oral prednisolone and initiation of latanoprost and dorzolamide-timolol fixed combination agent. After 12 hours, the IOP decreased to 18 mmHg in the right eye and 14 mmHg in the left eye. Six months later, her visual acuity was 1.0 in both eyes, and IOP was maintained at 11 and 13 mmHg in the right and left eye, respectively. Optic disc examination showed no sign of glaucomatous change with normal cup-to disc ratio in both eyes. Conclusions Since steroid medication may cause asymptomatic IOP elevation, regular intraocular pressure measurements are required during steroid treatment for pediatric optic neuritis. The administration of IOP-lowering eyedrops returned IOP to the normal range without damaging the optic nerve.

Purpose: To evaluate the long-term results of 2-muscle surgery in children with large-angle intermittent exotropia (IXT), comparing bilateral lateral rectus recession (BLR) with unilateral lateral rectus recession & medial rectus resection (RR). Methods: Ninety-two children with IXT of 40 prism diopter or more, who underwent BLR or RR were included in this retrospective study. Final successful alignment rates, and cumulative probabilities of surgical success at 3 years postoperatively were analyzed and compared using Kaplan-Meier survival analysis between the two groups. Results: At 3 years after surgery, 37 (67.3%) of 55 patients in the BLR group achieved successful alignment; 18 (32.7%) had undercorrection, and none of them had overcorrection. In the RR group, 34 (92%) of 37 patients were successfully aligned, two (5%) had undercorrection, and one (3%) had an overcorrection at 3 years after surgery. There was a statistically significant difference in postoperative results between the two groups (p = 0.0012). The cumulative probabilities of surgical success at 3 years postoperatively analyzed using the Kaplan-Meier survival curve were 63% in the BLR group and 92% in the RR group. Postoperative mean recurrence time was 45 months in the BLR group and 50 months in the RR group, showing a significant difference between the two groups (p = 0.0006). Conclusions The unilateral RR procedure demonstrated more favorable long-term postoperative outcomes than BLR procedure in patients with large-angle IXT.

Purpose: Dominant optic atrophy is one of the most common hereditary optic neuropathies, causing progressive bilateral vision loss that begins early in life. Optic atrophy 1 (OPA1) gene mutation brings about mitochondrial dysfunction, which results in clinical manifestations of dominant optic atrophy. Here, we report a case of dominant optic atrophy caused by the c.1334G>A mutation of the OPA1 gene, the first known case in Korea to our knowledge.Case summary: A 12-year-old female patient with no specific medical history or systemic symptoms visited our clinic complaining of a progressive decrease in vision in either eye. Slit-lamp microscopy, intraocular pressure, ocular motility, and pupil reflex were normal. However, her best-corrected visual acuity in both eyes was 20/100, and her color vision was reduced to 8/12 in Ishihara’s test. Fundus examination showed temporal pallor of the optic nerve head in both eyes, and a corresponding cecocentral scotoma was observed on Goldmann visual field examination. Optical coherence tomography revealed significant thinning of the peripapillary retinal fiber layer and macular ganglion cell layer in both eyes. Genetic examination confirmed the c.1334G>A mutation of the OPA1 gene. Conclusions We report a case of dominant optic nerve atrophy caused by c.1334G>A mutation of the OPA1 gene and its clinical manifestations.

Background: Thiazolidinediones (TZDs) have been associated with various safety concerns including weight gain, bladder cancer, and congestive heart failure (CHF). This study evaluated the efficacy and safety of lobeglitazone, a novel TZD in patients with type 2 diabetes mellitus (T2DM) in real practice. Methods: In this non-interventional, multi-center, retrospective, and observational study conducted at 15 tertiary or secondary referral hospitals in Korea, a total of 2,228 patients with T2DM who received lobeglitazone 0.5 mg for more than 1 year were enrolled. Results: Overall adverse events (AEs) occurred in 381 patients (17.10%) including edema in 1.97% (n=44). Cerebrovascular and cardiovascular diseases were identified in 0.81% (n=18) and 0.81% (n=18), respectively. One case of CHF was reported as an AE. Edema occurred in 1.97% (n=44) of patients. Hypoglycemia occurred in 2.47% (n=55) of patients. Fracture occurred in 1.17% (n=26) of all patients. Lobeglitazone significantly decreased HbA1c level, resulting in a mean treatment difference of -1.05%± 1.35% (P<0.001), and decreased total cholesterol, triglyceride, and low-density lipoprotein cholesterol. However, it increased high-density lipoprotein cholesterol, regardless of statin administration. The patients who received lobeglitazone 0.5 mg showed an apparent reduction in glycosylated hemoglobin (HbA1c) from baseline during the first 6 months of treatment. The HbA1c levels remained stable between months 6 and 42. Conclusion Lobeglitazone has long-term safety profile, good glycemic-lowering effect and long-term durability of glycemic control in real-world clinical settings.

The complexity in the molecular mechanism of the internal anal sphincter (IAS) limits preclinical or clinical outcomes of fecal incontinence (FI) treatment. So far, there are no systematic reviews of IAS translation and experimental studies that have been reported. This systematic review aims to provide a comprehensive understanding of IAS critical role in FI. Previous studies revealed the key pathway for basal tone and relaxation of IAS in different properties as follows; calcium, Rho-associated, coiled-coil containing serine/threonine kinase, aging-associated IAS dysfunction, oxidative stress, renin-angiotensin-aldosterone, cyclooxygenase, and inhibitory neurotransmitters. Previous studies have reported improved functional outcomes of cellular treatment for regeneration of dysfunctional IAS, using various stem cells, but did not demonstrate the interrelationship between those results and basal tone or relaxation-related molecular pathway of IAS. Furthermore, these results have lower specificity for IAS-incontinence due to the included external anal sphincter or nerve injury regardless of the cell type. An acellular approach using bioengineered IAS showed a physiologic response of basal tone and relaxation response similar to human IAS. However, in both cellular and acellular approaches, the lack of human IAS data still hampers clinical application. Therefore, the IAS regeneration presents more challenges and warrants more advances.