Buruli ulcer (BU) is a disfiguring infective skin lesion caused by Mycobacterium
ulcerans. If untreated, BU may lead to extensive soft-tissue loss and other complications. The objective of this study is to highlight the challenges encountered in the diagnosis and management of a patient with BU in our hospital. The patient is a 5-year-old boy who presented with an extensive nonhealing ulcer, knee deformity, and fever. He was initially managed as a case of nonspecific ulcer. A recurrence of the ulcer with loss of part of the skin grafts a week after discharge led to the clinical suspicion of BU which was confirmed using microscopy. In South Western Nigeria, the major constraint in the management of BU includes underdiagnosing of the disease because it is not often screened for due to absence or insufficient standard diagnostic instruments dedicated for that purpose.