We describe a likely case of chronic maxillary sinusitis (CMS) in a Neanderthal skeletal sample from Chagyrskaya Cave, in the Altai Mountains. Signs of CMS were recorded in the Chagyrskaya 57 specimen, which is a fragment of a left maxilla. Alveoli of the upper fi rst molar are partially preserved, and so are the second and third upper molars, with adjacent parts of the walls, and the fl oor of the maxillary sinus. The fragment was found in layer 6b, dating to 53,100–51,100 BP. We analyze the factors that had caused the development of the disease, and assess its etiology. In the 3D-model, generated by computed microtomography, and in the original specimen, porotic changes were registered, situated at the fracture line of the alveoli of M1, lost post-mortem, and near the vestibular roots of both preserved molars. Also, there were isolated bone spicules, 1.0–2.6 mm in size. These signs indicate incipient CMS, evidently caused by chronic periodontal disease combined with a deep alveolar recess of the maxillary sinus. As the periodontal gap expanded, several small nutrient foramina, piercing the bottom of the sinus, merged. As a result, several oro-antral channels formed, whereupon the infection spread into the maxillary sinus. Since the deep alveolar recess is observed in the vast majority of Neanderthal crania with published images or reconstructed maxillary cavities, it can be assumed that Neanderthals were predisposed to odontogenic CMS.