ESPE Abstracts

Background: Craniopharyngioma is a benign tumour that develops in the sellar and surrounding parasellar regions, including the hypothalamus and the pituitary gland responsible for the production and regulation of neuropeptides. Oxytocin is a hypothalamic neuropeptide that has been identified as a key modulator of appetite drive and social cognition. Here, we present a case of parent-observed improvements in neurobehavioural dysfunction following administration of intranasal of oxytocin in a male, post-craniopharyngioma resection.

Case presentation: A 16-year-old male with panhypopituitarism following surgical resection and proton beam therapy for adamantinomatous craniopharyngioma developed severe hyperphagia, food-related obsessive-compulsive features, and obesity. Specific neurobehavioural features included an overall preoccupation with food (e.g., writing food lists, watching food-related shows, constant food seeking), emotional lability, and anxious-depressive mood. Intranasal oxytocin was administered on a daily dosage of 4 IU/day (increasing to 8 IU/day) and the patient demonstrated improvements in food-related obsessive-compulsive features (e.g., decreased anxiety about obtaining food), reduced desire to eat (e.g., fewer snacks, no longer waking up during night to eat), and improved overall anxious-depressive mood and behavioural functioning. Naltrexone (100 mg/ day) was added after 6 months. The patient was treated with intranasal oxytocin for more than 3 years with no side-effects. Despite neurobehavioural and emotional improvements, his weight gain continued and he maintained a body mass index (BMI) within the obese range (BMI z score from 2.62 to 3.92). Due to shortages in supply, the patient is no longer on oxytocin and his food-related obsessive-compulsive features and symptoms of anxiety and depression have returned, and continue to severely impact his quality of life.

Conclusion: In this patient with post-operative craniopharyngioma, treatment with low dose intranasal oxytocin resulted in reduced food-related obsessive-compulsive features and improved mood symptomatology. The treatment trial was well tolerated, but had no effect on BMI. Future investigations into the potential therapeutic benefits of intranasal oxytocin are required to develop optimal treatment plans for patients with hypothalamic dysfunction and associated neurobehavioural difficulties.