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1.
Eye (Lond) ; 37(16): 3455-3460, 2023 11.
Article in English | MEDLINE | ID: mdl-37085721

ABSTRACT

OBJECTIVE: To describe the first paediatric case series of Thygesons' superficial punctate keratitis (TSPK) with management outcomes. METHODS: A retrospective chart review was done for all children either diagnosed at initial presentation or referred with TSPK from 01/2012 to 08/2021 at a tertiary children's hospital. Records were assessed for signs, symptoms, diagnosis, steroid and cyclosporine 0.05% use. The main outcome measures were visual acuity, treatment response and total steroid exposure. RESULTS: Fifteen children (7 females), mean age at presentation 8 ± 4 years were included. All had bilateral disease and a BCVA of >20/40 in the better eye. All patients received topical fluorometholone 0.1%, (FML) initially. 80% had a good response to FML. Corneal scraping was done to exclude infectious causes in four cases due to poor initial response or clinical suspicion. All 4 needed EUA for scraping and anterior segment OCT, after which 2 had molecularly confirmed TGFBI-related stromal dystrophy. For the rest, slow steroid taper was done every 4-6 weeks and recurrences were treated by increasing steroid frequency. Cyclosporine 0.05% was started in nine patients (69%), 8 ± 6 months after initial presentation. The decrease in total steroid exposure per week after starting cyclosporine was statistically significant (p < 0.05). CONCLUSION: Children with TSPK respond quickly to steroids, however, recurrences are common, necessitating a slow taper. Non-response to steroid needs careful reconsideration of the diagnosis and may necessitate the use of an EUA. Using cyclosporine 0.05% reduces the total steroid exposure in TSPK.


Subject(s)
Cornea , Keratitis , Female , Humans , Child , Child, Preschool , Fluorometholone/therapeutic use , Retrospective Studies , Keratitis/diagnosis , Keratitis/drug therapy , Keratitis/etiology , Cyclosporine/therapeutic use
2.
JCI Insight ; 3(4)2018 02 22.
Article in English | MEDLINE | ID: mdl-29467329

ABSTRACT

The inverse relationship between gestational age at birth and postviral respiratory morbidity suggests that infants born preterm (PT) may miss a critical developmental window of T cell maturation. Despite a continued increase in younger PT survivors with respiratory complications, we have limited understanding of normal human fetal T cell maturation, how ex utero development in premature infants may interrupt normal T cell development, and whether T cell development has an effect on infant outcomes. In our longitudinal cohort of 157 infants born between 23 and 42 weeks of gestation, we identified differences in T cells present at birth that were dependent on gestational age and differences in postnatal T cell development that predicted respiratory outcome at 1 year of age. We show that naive CD4+ T cells shift from a CD31-TNF-α+ bias in mid gestation to a CD31+IL-8+ predominance by term gestation. Former PT infants discharged with CD31+IL8+CD4+ T cells below a range similar to that of full-term born infants were at an over 3.5-fold higher risk for respiratory complications after NICU discharge. This study is the first to our knowledge to identify a pattern of normal functional T cell development in later gestation and to associate abnormal T cell development with health outcomes in infants.


Subject(s)
CD4-Positive T-Lymphocytes/immunology , Cell Differentiation/immunology , Gestational Age , Infant, Premature/immunology , Respiratory Tract Infections/epidemiology , CD4-Positive T-Lymphocytes/metabolism , Chronic Disease/epidemiology , Female , Humans , Infant , Infant, Newborn , Interleukin-8/immunology , Interleukin-8/metabolism , Longitudinal Studies , Male , Platelet Endothelial Cell Adhesion Molecule-1/immunology , Platelet Endothelial Cell Adhesion Molecule-1/metabolism , Pregnancy , Respiratory Tract Infections/immunology , Respiratory Tract Infections/virology
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