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Background: Wry Nose (Campylorrhinus lateralis) is a congenital shortening and deviation of the maxillae, premaxillae, nasal bones, vomer, and nasal septum that affects mainly foals. Intrauterine positioning as an etiology of this deformity is highly unlikely. Clinically, varying degree of premaxillar deviation is noticeable upon inspection of the foal's head. The deviation usually results in malocclusion, with no contact between the incisors of the mandible and maxilla. Some foals experience breathing difficulties or have increased respiratory sounds. This condition will not self-correct, and a decision should be made to treat the foal using reconstructive techniques or to euthanize it to prevent it from suffering. The objective of this work was to describe a case of wry nose in an adult male equine. Case: A 4-year-old, 300 kg, male SRD equine was submitted for necropsy. There was no history of respiratory problems and trouble with mastication or prehension of food. Three days before death the horse showed clinical sings characterized by no ingestion of food and water, changing behavior and locomotion, weakness, and not respond to calls, seemingly oblivious to the environment evolving into permanent recumbency and death after about 24 h of the decline. The horse was treated by a veterinarian who suspected of leucoencephalomalacea. The animal died 24 h after her condition worsened. Macroscopically, the main lesion observed was in the nose. The nasal septum was deviated to the left with the sigmoid appearance and a dorsal hump on the right side of the nasal bone. The radiographs of dorsoventral view of the jaws showing premaxillae deviated approximately 90 degrees to the left. The left premaxillae was shorter than the right premaxilla, thus creating a lesser and greater curvature of the premaxilla. Macroscopic and Microscopic lesions in the brain were not observed discarding the possibility of leucoencephalomalacea and other neurological diseases of infectious origin. Discussion: Wry nose is a relatively rare disease and have being reported in foals. In our study, it was not possible to determine the cause of this abnormality, but a computed tomography study has been associated this deformity with an underdeveloped pregnant uterus during the second half of pregnancy witch the narrow tip of the uterine horns provides only limited space for the developing cranial half of the fetus. Although the disease have been reported in young animals in which the surgical procedure in moderate to severe cases it is recommended, in the present case, the equine was four years old indicating that there is a possibility of the animal to reach adulthood, but with their commitment to quality of life. Several diseases, such as cystic degeneration, malformation of normal tissue, abscesses, fractures and neoplasms may cause thickening, malformation or deviation of the nasal septum in horse producing similar clinical sings. In the present case, the macroscopic lesions and radiography exam were useful to determinate the exact site, extent and nature of the involvement of the lesion. Despite the owner to report a clinical picture indicative of neurological disease, histological analysis of the brain ruled out this possibility. These findings indicate that wry nose disease should be kept in mind when investigating causes of deviation of the nasal septum in equines. [ABSTRACT FROM AUTHOR]