Here we report the case of a 42-yr-old patient who presented himself to us with a serpiginous erythematous lesion from the wrist of the right forearm up the arm to the right shoulder A similar lesion of a smaller size was also present in the left forearm. On the basis of clinical manifestations and progression of the lesion, combined with previous treatments and different diagnostic investigations, hookworm-related cutaneous larva migrans (HrCLM) disease was hypothesized. Albendazole was employed as treatment and the resolution of the symptoms confirmed the diagnosis. The relevance of the reported case relies on 3 main aspects: the acquisition of the disease in Italy, the initial treatment with topical corticosteroids that sped up the progression of the cutaneous trail, and the uncommon location of the lesions. Furthermore, the anamnestic data and the laboratory/clinical investigations strongly suggested an occupational exposure to the etiological agent. As illustrated here, HrCLM might represent a challenge for Western physicians in terms of diagnosis, treatment, and ways of acquisition. Describing the clinical presentation and the treatment of cases of cutaneous larva migrans might contribute to early and correct diagnosis, to an increase of our knowledge on this disease, and to an update on its epidemiology.