Crohn’s disease (CD) of the vulva is a rare and under‐recognized condition. Since vulvar lesions may precede the diagnosis of digestive CD in approximately 25% of all cases, the coexistence or future onset of CD should be considered regardless of the gastrointestinal symptoms, even for pediatric patients.
Rationale:Ustekinumab is effective in the treatment of adult Crohn disease (CD) and ulcerative colitis (UC). However, data on its efficacy and safety in pediatric CD and UC are limited. To the best of our knowledge, there are no reports of Japanese children with UC treated with ustekinumab in the long-term.Patient concerns:A 14-year-old man with diarrhea and bloody stools was referred to our hospital. Colonoscopy revealed total colitis-type UC. His pediatric UC activity index score was 50, indicating moderately active UC.Diagnoses:Ulcerative colitis.Interventions:Infliximab was introduced because of steroid-resistant refractory UC; however, secondary ineffectiveness was observed 17 months later. Therefore, ustekinumab was administered along with prednisolone (16 years of age).Outcomes:The patient achieved UC remission after ustekinumab treatment, leading to maintained remission without side effects.Lessons:To the best of our knowledge, this is the first pediatric case of moderately active UC successfully treated with ustekinumab in Japan. Ustekinumab combined with steroids is an effective and safe induction therapy for UC.
Rationale: Tofacitinib is an orally administered selective Janus kinase inhibitor. Its efficacy and safety in adults with moderately to severely active ulcerative colitis (UC) have been evaluated in clinical trials; however, its efficacy in pediatric patients with UC is limited.Patient concerns: A 9-years-old boy was referred to our hospital with chief complaints of diarrhea and bloody stool for 2 months. Colonoscopy revealed total colitis-type UC. His pediatric UC activity index score was 40, indicating moderately active UC.Diagnosis: UC.Interventions: Vedolizumab, golimumab, and ustekinumab were introduced because of steroid-resistant refractory UC; however, none of these biologics were effective or the effect was short-lived. Therefore, tofacitinib was administered 5 mg twice a day.Outcomes: The patient achieved UC remission after tofacitinib treatment, leading to maintained remission without adverse events.Lessons: To the best of our knowledge, this is the first pediatric case of moderately active UC that was successfully treated with tofacitinib in Japan. Tofacitinib is a safe drug for pediatric patients with moderately active UC. Even in steroid-dependent cases refractory to other biologics, tofacitinib can result in remission induction and maintenance effects. In children and adults, high-dose tofacitinib during induction therapy may be unnecessary to reduce adverse events.Abbreviation: AEs = adverse events, JAK = janus kinase, NR = normal range, PUCAI = pediatric ulcerative colitis activity index, TCS = total colonoscopy, UC = ulcerative colitis, UCEIS = ulcerative colitis endoscopic index of severity.
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