We describe an 8-week-old infant with severe gastrointestinal symptoms, significant hypoalbuminemia, and mild carditis following asymptomatic infection with SARS-CoV-2. The infant's symptoms, including their temporal appearance, were consistent with multisystem inflammatory syndrome in children (MIS-C). A unique finding on colonic histology which may shed light on the pathogenesis of MIS-C was identified. The patient improved significantly following several anti-inflammatory treatments. The lag between the presentation of MIS-C and initial SARS-CoV-2 exposure, which may often be asymptomatic, together with the young age of our patient, make this a challenging diagnosis. Clinicians should be aware of this entity, even in the neonatal and infantile age groups, to facilitate timely identification and treatment.
Background and aims: Ulcerative proctitis (UP) is an uncommon presentation in pediatric patients with ulcerative colitis. We aimed to characterize the clinical features and natural history of UP in children, and to identify predictors of poor outcomes. Methods:The retrospective cohort study involved 37 sites affiliated with the IBD Interest group of ESPGHAN. Data were collected at different time points from patients aged<18 years diagnosed with UP between 01/01/2016-31/12/2020. Results:We identified 250 patients with UP with a median follow-up of 2.7 (IQR 1.7-3.9) years. were included. Median age at diagnosis was 14.5 (IQR 12.3-15.9) years. Median follow-up was 2.7 (IQR 1.7-3.9) years. The most common presenting symptoms were bloody stools (93.6%), abdominal pain (60.4%) and diarrhea (52.8%). At diagnosis, the median pediatric ulcerative colitis activity index (PUCAI) score was 25 (IQR 20-35), the median fecal calprotectin level was 720 mcg/g (IQR 310-1800), notably 16 patients (11.7%) had a calprotectin level <100mcg/g. Most patients exhibited moderate-severe endoscopic inflammation. Oral, topical or By the end of induction, administration of orally, topically or combination of both resulted in clinical remission rates of 51.8%, 50.0% 73.3%, respectively at weeks 8-12?. The rates of treatment escalation to biologics at 1, 3 and 5 years were 10.6%, 22.7% and 44.6%. in multivariate analysis, Tthe PUCAI score at diagnosis was highly associated with escalation of therapy and subsequent events with acute severe colitis eventsand or IBD-associated admissions (multivariate analysis). By the end of follow-up, 3.4% of patients underwent colectomy. Cecal patch (P=0.009), higher PUCAI score (P=0.009) and lack of steroid-free clinical remission (P=0.005) by the end of induction were associated with proximal disease extension, identified in 48.3%.. Conclusion:Pediatric patients with UP exhibit high rates of proximal disease extension and treatment escalation.
Background Several groups have proposed models to predict disease outcomes in pediatric Crohn’s disease (CD), notably the RISK, GROWTH, and the Porto group, but none were externally validated. We aimed to explore these predictive models and individual predictors summarized by the PIBD-ahead project in a prospective inception cohort of pediatric CD. Methods We included children who were diagnosed with CD at two medical centers and followed them at 3 and 12 months thereafter as well as at the last follow-up . Outcomes included steroid-free remission (SFR), surgery and stricturing/fistulizing disease Results 155 children were included (median follow-up of 31 [16–48] months, 107 [71%] had moderate-to-severe disease). Stricturing and penetrating disease at diagnosis were noted in 34 (22%) and two (1.3%) children, respectively, and these were excluded from the relevant analyses. At 1 year, 10 (8.3%) developed new stricturing disease, two (1.7%) developed penetrating disease, seven (5%) required intestinal surgery, and 15 (10%) required perianal surgery. The sensitivity/specificity/PPV/NPV of the GROWTH criteria for predicting SFR at 12 months (occurring in 70% of children) were 20%/85%/76%/31% and for surgery at two years 96%/20%/16%/96%. Strictures were predicted by the RISK model with sensitivity/specificity/PPV/NPV of 33%/73%/18%/86%. The sensitivity/specificity/PPV/NPV of the Porto criteria to predict surgery were 86%/10%/4%/94%. None of the PIBD-ahead predictors were associated with surgery or stricturing disease. Conclusion None of the three main predictive models in pediatric CD achieved sufficient accuracy, far from that reported in the original cohorts. This highlights the necessity of external validation in any prediction model prior to its implementation in clinical practice.
In healthy individuals, HR and renin responses to dobutamine were more than three-fold greater among ADRB1 Arg389 compared with Gly389 homozygotes. Future studies on the effect of the ADRB1 389 polymorphism on dobutamine stress echocardiography should compare Arg389 and Gly389 homozygotes.
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