{
  "abstract": "Inflammatory bowel diseases (IBD), including ulcerative colitis and Crohn’s disease , are chronic immune-mediated inflammatory disorders of the gastrointestinal tract that often emerge in adolescence or early adulthood. Approximately one quarter of IBD cases present before 20 years of age, and 4% occur before age five. Very early-onset IBD (VEO-IBD) is defined as diagnosis before six years, and infantile-onset IBD (IO-IBD) refers to diagnosis before two years. IO-IBD constitutes roughly 1–2% of all paediatric IBD cases. Known causative monogenic mutations account for only about 7.8% of VEO-IBD, representing 30% of IO-IBD. Consequently, non-monogenic IO-IBD remains underreported.A three-month-old male presented with frequent bloody diarrhoea to local hospital, he was suspected to have infection therefore was started on ceftriaxone, gentamicin and metronidazole. Also, breastfeeding was switched to amino-acid formula. Initial workup at the referring hospital revealed positive cytomegalovirus (CMV) serology and DNA; ganciclovir was commenced. Despite all treatments and feeding change his symptoms persisted, prompting referral to our hospital. On admission, he continued to have bloody, loose stools, requiring multiple blood and albumin transfusions. He was maintained nil by mouth and on parenteral nutrition (PN). Esophagogastroduodenoscopy (OGD) revealed erythematous, exudative mucosa in the stomach and duodenum, while colonoscopy (limited by a descending colon stricture) demonstrated friable, ulcerated mucosa. Histopathology confirmed active inflammation consistent with IO-IBD and CMV was ruled out. Comprehensive immunological and genetic evaluation for monogenic IBD, revealed no abnormalities. Intravenous corticosteroid induction was initiated but discontinued after 11 days due to non-response and worsening gastrointestinal bleeding. Emergency endoscopy identified large ulcerations in the descending colon requiring haemostatic spray. As steroid-refractory disease was evident, infliximab (5 mg/kg) was commenced. After two loading doses, clinical and biochemical remission was achieved: normalised CRP, improved albumin and haemoglobin, and reduction in stool frequency to 3–4 formed motions daily without blood or mucus (table 1). Follow-up endoscopy showed marked mucosal healing though residual oedema and skip lesions persisted. Histopathology confirmed quiescent proctocolitis, and the patient was discharged on oral sulfasalazine.From a nutritional perspective, the infant received tailored PN for 68 days, providing ~100 kcal/kg/day and 3.3 g/kg/day of protein per ASPEN and BSPGHAN recommendations. Adjustments for catabolic stress and protein loss were made, and metabolic stability was maintained through PN cycling and lipid modulation. A gradual oral transition using an amino-acid-based formula achieved full enteral feeds over 18 days, resulting in significant catch-up growth from below the 1st percentile to above the 3rd percentile.This case represents one of the youngest reported examples of non-monogenic IO-IBD, an under-reported cohort. It highlights the importance of considering broader differential diagnosis in infants presenting with bloody diarrhoea. Also, our experience with this case demonstrated safe use of infliximab in very young infants.Early multidisciplinary management—including full endoscopic evaluation with OGD, exclusion of immunodeficiency and monogenic causes, early biologic therapy with proactive therapeutic drug monitoring, and comprehensive nutritional support—is essential for optimising outcomes in steroid-refractory IO-IBD.References Kelsen JR, Sullivan KE, Rabizadeh S, Singh N, Snapper S, Elkadri A, et al. North American society for pediatric gastroenterology, hepatology, and nutrition position paper on the evaluation and management for patients with very early-onset inflammatory bowel disease. J Pediatr Gastroenterol Nutr 2020;70(3):389–403.Guz-Mark A, Aloi M, Scarallo L, Bramuzzo M, Escher JC, Alvisi P, et al. Infantile and very early onset inflammatory bowel disease: a multicenter study. Pediatrics 2024;154(2).Chapuy L, Leduc B, Godin D, Damphousse A, Patey N, Dal Soglio D, et al. Phenotype and outcomes of very early onset and early onset inflammatory bowel diseases in a Montreal pediatric cohort. Front Pediatr 2023;11:1157025.Kammermeier J, Dziubak R, Pescarin M, Drury S, Godwin H, Reeve K, et al. Phenotypic and genotypic characterisation of inflammatory bowel disease presenting before the age of 2 years. J Crohns Colitis 2017;11(1):60–9.Mesotten D, Joosten K, van Kempen A, Verbruggen S, nutrition EEECwgopp. ESPGHAN/ESPEN/ESPR/CSPEN guidelines on pediatric parenteral nutrition: Carbohydrates. Clin Nutr 2018;37(6 Pt B):2337–43.Uhlig HH, Charbit-Henrion F, Kotlarz D, Shouval DS, Schwerd T, Strisciuglio C, et al. Clinical genomics for the diagnosis of monogenic forms of inflammatory bowel disease: a position paper from the paediatric ibd porto group of european society of paediatric gastroenterology, hepatology and nutrition. J Pediatr Gastroenterol Nutr 2021;72(3):456–73.Abstract OC43 Table 1Comparison of basic blood test and faecal calprotectin pre and post treatment",
  "authors": [
    {
      "affiliations": [
        "King’s College Hospital, Paediatric Gastroenterology"
      ],
      "name": "Enes Coskun"
    },
    {
      "affiliations": [
        "King’s College Hospital, Paediatric Gastroenterology"
      ],
      "name": "Sorcha Mullen"
    },
    {
      "affiliations": [
        "King’s College Hospital, Paediatric Pharmacy Department"
      ],
      "name": "Joanne Crook"
    },
    {
      "affiliations": [
        "King’s College Hospital, Paediatric Gastroenterology Dietetic Department"
      ],
      "name": "Chelsea Edgcumbe"
    },
    {
      "affiliations": [
        "King’s College Hospital, Paediatric Gastroenterology"
      ],
      "name": "Babu Vadamalayan"
    }
  ],
  "title": "OC43 The successful use of infliximab in a 3-month-old infant with non-monogenic inflammatory bowel disease",
  "uid": "dfa4fe86-5d43-5f8b-84c1-69be30e3c684"
}