Mesenchymal stem cell therapy (MSCT) has been shown to be a new therapeutic option for treating alopecia areata (AA). Outer root sheath cells (ORSCs) play key roles in maintaining the hair follicle structure and supporting the bulge area. In human ORSCs (hORSCs), the mechanism for this process has not been extensively studied. In this study, we aimed to examine the influence of human hematopoietic mesenchymal stem cells (hHMSCs) in the hORSCs in vitro model of AA and determine the mechanisms controlling efficacy. Interferon-gamma (IFN-γ) pretreatment was used to induce an in vitro model of AA in hORSCs. The effect of MSCT on the viability and migration of hORSCs was examined using co-cultures, the MTT assay, and migration assays. We investigated the expression of molecules related to the Wnt/β-catenin pathway, JAK/STAT pathway, and growth factors in hHMSC-treated hORSCs by reverse transcription-polymerase chain reaction (RT-PCR) and Western blot analyses. hHMSCs increased hORSC viability and migration when they were co-cultured. hHMSCs reverted IFN-γ-induced expression—including NLRP3, ASC, caspase-1, CXCL-9 through 11, IL-1β, and IL-15—and upregulated several growth factors and hair stem cell markers. hHMSCs activated several molecules in the Wnt/β-catenin signaling pathway, such as in the Wnt families, β-catenin, phosphorylated GSK-3β and cyclin D1, and suppressed the expression of DKK1 induced by IFN-γ in hORSCs. hHMSCs suppressed the phosphorylation of JAK1 to 3, STAT1, and STAT3 compared to the controls and IFN-γ-pretreated hORSCs. These results demonstrate that hHMSCs increased hORSC viability and migration in the in vitro AA model. Additionally, MSCT definitely stimulated anagen survival and hair growth in an HF organ culture model. MSCT appeared to be associated with the Wnt/β-catenin and JAK/STAT pathways in hORSCs.
PurposeTo report a case of congenital sudoriferous cyst of the orbit with esotropia.MethodsA 20-day-old male, born prematurely presented with a palpable lump on left upper lid. Orbital ultrasonography including color doppler image and orbital magnetic resonance image were performed to evaluate the lid lesion. The mass was excised and histologically examined. Complete ocular examination including visual acuity, duction, version, and the presence of strabismus were performed.ResultsA well circumscribed round cystic mass, measuring 1.4×1.3 cm was noted at medial superior aspect of the left orbit. It compressed and displaced the left globe to inferior posterior position with intact optic nerve. Histopathologic examination showed the lesion to be a solitary sudoriferous cyst lined by two layers of cuboidal epithelial cells with eosinophilic cytoplasm. After the excision of the mass, limitations of extraocular muscle movements, esotropia, and amblyopia were noted.ConclusionsIf an orbital cyst affects the globe or extraocular muscles, it should be excised as soon as possible to prevent strabismus and amblyopia especially in infant.
We report a case of regressed retinal astrocytic hamartomas (RAHs) in tuberous sclerosis complex (TSC) patients by mammalian target of rapamycin inhibitor (everolimus) treatment. Case summary: A 12-year-old girl diagnosed with TSC visited for regular checkups. The patient had undergone regular fundus examinations every year after the finding of multiple RAHs in both eyes in the initial screening at 3 months of age. There was no change in the size or thickness of the lesions until she reached 10 years of age. Two months later, the patient started systemic everolimus (5 mg, AFINITOR ® , Novartis, Basel, Switzerland) treatment for 17 months under the care of a pediatric neurologist for seizure control. Subsequent fundus examination and measurements by optical coherence tomography showed improvement in the maximal thickness of all lesions, specifically, a reduction of 25%. Conclusions: mTOR inhibitors are targeted agents that regress systemic hamartomas and control convulsions without serious side effects in TSC patients. The particular one used in this study, Afinitor everolimus, reduced the RAH size in our patient. Thus, in cases where an RAH affects vision due to its location, everolimus can considered as a therapeutic option.
Purpose: To investigate the prevalence of amblyogenic risk factors in the primary family members (parents, siblings) of patients with exotropia. Methods: The authors of the present study examined primary family members including parents and siblings of 58 exotropia patients in our clinic. Best corrected visual acuity, refractive error, and cover-uncover test were performed. A total of 49 normal children's family members (control group) were examined in a similar manner. The prevalence of significant ocular findings were determined and compared to the control group. Results: In 58 eligible families (153 subjects except proband), 65.4% had significant ocular findings: In parents, anisometopia, astigmatism, hyperopia, and strabismus were found at a rate of 18.1%, 31.1%, 2.5%, and 11.2%, respectively. In siblings, anisometropia, astigmatism, hyperopia, and strabismus were found at a rate of 24.3%, 24.3%, 5.4%, and 18.9%, respectively. Compared to the control group, in parents of exotropia patients, anisometropia, astigmastim, and strabismus were observed at a higher rate. In siblings, anisometropia, and strabismus were observed at a higher rate. Conclusions: Primary family members of exotropia patients have a high prevalence of amblyogenic risk factors. The present study offers rationale for providing comprehensive eye exams for parents and children with a family history of exotropia.
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