Introduction: Gastrointestinal stromal tumours (GISTs) are considered the most common mesenchymal lesions of the gastrointestinal tract, despite accounting for less than 1% of cancers diagnosed in this area. They frequently occur in the stomach and small intestine, rarely in the colon and esophagus. Case Report: The report concerns an unusual case of a patient referred to hospital with abdominal pain and suspected ileocecal intussusception. Ileus was confirmed during diagnostic evaluation. Surgical resection of the tumour mass was performed to release intestinal loops. Based on histology and immunohistochemical staining, GIST was diagnosed. Currently, the patient remains under constant clinical monitoring. Conclusions: The evolution of gastrointestinal stromal tumours is highly variable. Both the signs and symptoms and tumor localization in the described case are very unusual for this disease. Adequate diagnosis remains critical to implement appropriate therapeutic management. KEYWORDS: gastrointestinal stromal tumours, gastrointestinal tract, intussusception, tyrosine kinase inhibitors