FENO-guided asthma management during pregnancy prevented doctor-diagnosed asthma in the offspring at preschool age, in part mediated through changes in use and dosing of inhaled corticosteroids during the MAP trial.
Paediatric inflammatory multisystem syndrome temporally associated with SARS-CoV-2 (PIMS-TS) is a newly described condition. It has a spectrum of presentations proposed to occur as part of a post-infectious immune response. We report the first case of PIMS-TS in a child on established anti-Tumor Necrosis Factor-alpha (anti-TNF-α) therapy; a 10 year-old girl with ulcerative colitis treated with infliximab. The patient had 6-weeks of daily fever with mucocutaneous, gastrointestinal, renal and hematologic involvement. Biomarkers of hyperinflammation were present including: hyperferritinaemia (up to 691 µ/L; normal 15-80 µg/L), C-reactive protein (CRP) (>100mg/L for >10 days, normal 0-5 mg/L), erythrocyte sedimentation rate (ESR) consistently >100mm/hr (normal 0-15 mm/hr), raised white cell count with neutrophilia, elevated D-dimer and lactate dehydrogenase (LDH), anaemia and Mott cells on bone marrow analysis. Extensive investigations for alternative diagnoses for pyrexia of unknown origin (PUO) were negative. The condition was refractory to treatment with intravenous immunoglobulin (IVIG) but improved within 24hrs of high dose methylprednisolone. Infliximab treatment followed and the patient has remained well at follow up. Polymerase chain reaction (PCR) and serology for SARS-CoV-2 were negative. Current series report such negative findings in up to half of cases. The patient experienced a milder clinical phenotype without cardiac involvement, shock or organ failure. Accepting the wide spectrum of PIMS-TS presentations, it is possible that prior anti-TNF-α therapy may have attenuated the disease course. Given the uncertainty around therapeutic strategies for PIMS-TS this case supports the need for further investigation into continuing infliximab as a treatment option for the condition.
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.
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