Olfactory reference syndrome is characterised by the erroneous belief that one emits an unpleasant body odour. This results in significant distress and is often accompanied by repetitive behaviour such as frequent showering in an attempt to camouflage the perceived odour. The body odour concerns may have a delusional quality and do not respond to simple reassurance or counterexample. Herein, we report the case of an olfactory reference disorder (ORD) patient who had received multiple medical interventions and undergone polysurgery prior to an accurate diagnosis being established. ORD may lead to significant disability, yet often goes unrecognised for many years. For many patients, poor insight will contribute to their reluctance to consider psychiatric treatment. This case demonstrated that a multimodal treatment approach comprising judicious medication use, combined with cognitive behavioural therapy, in the context of a therapeutic alliance yielded therapeutic success.