Aims Children with severe neurodisability presenting with pain behaviour, retching, bloating, abdominal distension, and constipation/pseudo-obstruction can be referred to as having Gastro-Intestinal Dystonia. Management can be extremely challenging, with evidence base for therapy extremely limited, and a spectrum of symptomology that can be severely debilitating.¹

Nabilone is a synthetic cannabinoid designed to mimic the activity of delta-9-Tetrahydrocannabinol (THC). Nabilone is a partial agonist of both CB1 and CB2 receptors, and is licensed, and indicated by NICE guidance, for use in adults as adjunctive therapy alongside standard anti-emetic for chemotherapy induced nausea and vomiting. It has also been used widely in paediatrics for the same indication, despite not being licensed for use in children.²

There has been significant interest in the use of medical cannabis to treat the spectrum of symptomology that manifest in gut dystonia yet limited published work in reporting the effectiveness of nabilone in treating gut dystonia. However, there is emerging anecdotal and case reports from clinical practice suggesting a role for nabilone.¹

Method From October 2022 to June 2023, patients referred and accepted to the Great Ormond Street Hospital palliative care service, for end-of-life care, were considered for use of nabilone to manage gut dystonia that had proven resistant to normal dystonia management strategies (e.g., optimisation of gabapentin, clonidine, baclofen etc.). Nabilone was initiated following approval for use by trust DTC and in discussion with the neuro-disability team. Families of patients commenced on nabilone were requested to complete a dystonia diary and reviewed weekly assessing for effectiveness and toxicity. Notes and charts were reviewed in triplicate by consultant in paediatric palliative, specialty registrar and specialist pharmacist, to ensure consensus agreement on the effectiveness of nabilone as well as any potential incidence of toxicity.

Results One patient was initiated on nabilone for gut dystonia during the study period. Weight at initiation of nabilone was 16.45 kg. Weight after 9 months (June 2023) was 19.45 kg. Observed dystonia and feed tolerance improved significantly, enabling the child to return to school, and there was a significant reduction in number of dystonic breakthrough medications required, from daily use of chloral hydrate to infrequent use at night. Following optimisation of nabilone, gabapentin has been weaned with no observed increase in dystonic episodes. Additionally, despite increased weight there has been no requirement to increase doses of clonidine. There were no parenteral or professional observations of any adverse effects associated with nabilone initiation or titration.

Conclusion Nabilone can be safely initiated for children with gut dystonia that has proven resistant to other conventional medications used for dystonia management. Use of nabilone has demonstrated a subjective and objective improvement in tone and an increase in ability to tolerate feeds with weight gain, that has persisted despite weaning of other concomitant medications.

References

1. McConnel N, Beattie LM, Richards CE, Protheroe S, Barclay AR. Nabilone for gastro-intestinal dystonia. a single centre case series. Journal of Paediatric Gastroenterology and Nutrition 2018;66(suppl 2):1002.

2. Nabilone capsules 250 micrograms and 1 mg capsules Summary of Product Characteristics, UK. Last Updated 07/2021. www.medicines.org.uk (Accessed 09 March 2023).