Objective: To report a case of lichen planopilaris in a patient treated with infliximab for longstanding refractory psoriasis. Case Summary: A 37-year-old man with recalcitrant plaque psoriasis was being treated with infliximab at a dosage of 5 mg/kg every 8 weeks, with good response. However, 11 months later the patient developed follicular keratotic papulo-pustules, perifollicular erythema, and scaling, with progressive hair toss of the frontal and parietal regions of the scalp and eyebrows. A skin biopsy from a representative lesion was consistent with the diagnosis of lichen planopilaris. Discussion: Anti-tumor necrosis factor (TNF) agents have been associated with numerous cutaneous adverse events. Lichenoid reactions are uncommon but are an emerging cutaneous adverse effect. At least 13 cases of these eruptions have been recently described. Although lichenoid reactions in patients treated with TNF-α inhibitors may be clinically very diverse, we have found no previously reported cases of lichen planopilaris induced by these agents. An objective causality assessment revealed that the adverse event was probable. Conclusions: Since anti-TNF agents are being used for a rapidly expanding number of rheumatic, digestive, and dermatologie diseases, it is expected that lichenoid eruptions and other skin toxicities are likely to be seen with increasing frequency in clinical practice.