Imatinib, a tyrosine kinase-C protein inhibitor remains the first choice for the management of chronic myeloid leukaemia (CML) since its introduction over a decade ago. Intracranial and gastrointestinal haemorrhages have been previously reported in association with Imatinib therapy. We report the case of a 47-yr-old man who was recently diagnosed with chronic phase of CML, that was complicated by a spontaneous intramuscular haematoma in the left gluteal region after 10 days of treatment with Imatinib. To the best of our knowledge, an intramuscular haemorrhage has not been described so far in association with Imatinib therapy. Clinicians should be alert to the possibility of spontaneous haemor- rhage at unusual sites in patients being treated with Imatinib.