<b><i>Background:</i></b> Multiple cutaneous piloleiomyomas are rare, frequently painful and difficult-to-treat benign tumours originating in the arrectores pilorum muscles of the hair follicles. <b><i>Objective:</i></b> The aim of this study was to determine the efficacy of a local injection of triamcinolone acetonide in relieving cutaneous piloleiomyoma-related pain. <b><i>Methods:</i></b> A patient with multiple painful piloleiomyomas was treated with weekly local injections of triamcinolone acetonide into the nodules and papules for 3 weeks. <b><i>Results:</i></b> The patient's nodules and papules became smaller, and the pain disappeared. At 1-year follow-up, there were no side effects, pain recurrence or significant enlargement of any of the injected lesions. <b><i>Conclusion:</i></b> Our study demonstrates that local injection of triamcinolone acetonide may be a promising new treatment option for multiple painful cutaneous piloleiomyomas.