Isotretinoin is a retinoid derivative drug used in acne vulgaris treatment. Sacroiliitis has been reported as an uncommon adverse effect following isotretinoin treatment. We report a 30-year-old male patient who developed sacroiliitis after isotretinoin use. Stopping treatment with isotretinoin resulted in a complete disappearance of inflammatory back pain. A literature search was performed to assess the relevance of this association in everyday clinical practice, and to discuss whether the link between isotretinoin and sacroiliitis is causal or coincidental.