{
  "abstract": "Gastrointestinal dystonia (GID) is a rare but a challenging motility disorder in children with neurodisabilities, frequently resulting in profound feeding intolerance and, in some cases, presumed intestinal failure (IF). Conventional prokinetic therapies don’t always provide full resolution of symptoms. Emerging evidence suggests that Neurokinin-1 (NK1) receptor antagonists, such as aprepitant, may exert prokinetic effects, but clinical data, particularly in paediatric IF, remain limited. This late-breaking abstract presents three paediatric cases in which aprepitant facilitated successful restoration of enteral feeding after failure of standard therapies.We retrospectively reviewed three children (aged 8, 12, and 16 years) with GID and severe feed intolerance or presumed IF. Clinical notes were examined to assess gastric output, feed tolerance, prokinetic regimens, and parenteral nutrition (PN) dependence before and after aprepitant introduction.Case A An 8-year-old with neurodysability due to Cerebral palsy presented with severe Clostridium difficile enterocolitis. The patient developed high gastric output and inability to tolerate enteric feed despite infection resolution. Multiple prokinetics including azithromycin, domperidone, and alimemazine were ineffective, with ongoing gastric losses of 10–30 mL/kg/day that would increase if feed was increased, hence inability to progress enteral feeds and PN dependence.Following aprepitant initiation, gastric output decreased by 25–30%,enabling gradual advancement to full enteral feeds and eventual discontinuation of PN.Case B A 12-year-old, neurodysability due to Wang syndrome, dependent on gastrojejunal (G-J) tube feeding, was admitted with suspected autonomic dysfunction. They required cessation of all prokinetics with arrhythmogenic risk. Furthermore, the patient’s G-J tube kept recoiling in the stomach, with subsequent PN dependence. After aprepitant initiation, gastric output fell from 200–350 ml/day to near zero. Notably, the patient tolerated full gastric feeding for the first time in years.Case C A 16-year-old with Myotonic dystrophy, admitted with recurrent episodes of chronic intestinal pseudo-obstruction failed trials of conventional prokinetics and required repeated PN-dependent admissions. Introduction of aprepitant restored bowel motility, allowing tolerance of full gastrostomy tube feeds at 100 mL/hr.An inadvertent interruption of aprepitant therapy resulted in rapid clinical deterioration, reversed promptly upon re-initiation; further supporting its direct therapeutic effect.Across all three cases, aprepitant demonstrated a consistent and clinically meaningful impact on gut motility, gastric output reduction, and re-establishment of enteral nutrition, despite failure of multiple standard promotility therapies. None of the three patient developed any concerning side effects. This emerging pattern, particularly given the diverse underlying pathologies and ages, suggests aprepitant may address an unmet therapeutic need in children with GID and presumed IF. The rapid recurrence of symptoms after unintentional withdrawal in one case strengthens the causal relevance of NK1 receptor blockade.These late-breaking findings highlight aprepitant as a promising, under-recognised prokinetic therapy in paediatric GID and presumed IF. While limited by the small case series, the consistent clinical improvements support the need for prospective, controlled studies to evaluate efficacy, dosing, safety, and long-term outcomes. Aprepitant may represent a significant therapeutic advancement for children with complex motility disorders unresponsive to standard treatments. The Graph shows the pattern of gastric output in case2(red arrow-Aprepitant)Reference Barclay AR, Meade S, Richards C, Warlow T, Lumsden DE, Fairhurst C, Paxton C, Forrest K, Mordekar SR, Campbell D, Thomas J. Definition, investigation and management of gastrointestinal dystonia in children and young people with neurodisability. Archives of Disease in Childhood 2025.Abstract LBA4 Figure 1",
  "authors": [
    {
      "affiliations": [
        "Noah’s Ark Children Hospital for Wales - University Hospital of Wales - Cardiff"
      ],
      "name": "Huda Atta"
    },
    {
      "affiliations": [
        "Noah’s Ark Children Hospital for Wales - University Hospital of Wales - Cardiff"
      ],
      "name": "Jessica Girvin"
    },
    {
      "affiliations": [
        "Noah’s Ark Children Hospital for Wales - University Hospital of Wales - Cardiff"
      ],
      "name": "Amar Wahid"
    }
  ],
  "title": "LBA4 Aprepitant as a novel prokinetic in paediatric gastrointestinal dystonia and presumed intestinal failure: three case reports",
  "uid": "2f69164c-ecef-5258-a29f-a72a39579c87"
}