Background: Hypospadias is a congenital malformation, rarely reported in dogs, caused by a failure of the urogenital folds to fuse during fetal development resulting in incomplete formation of the penile urethra. The inadequate production of fetal androgens is believed to be the etiology of this condition. It is characterized by urethra and corpus spongiosum defciency, often accompanied by penile underdevelopment, and cryptorchidism. Classifcation is based on anatomical location of the urethral meatus opening as glandular, penile, perineal, anal, and scrotal. The aim of this paper is to report a case of perineal hypospadias in a male, six-month old French bulldog, describing the therapeutic approach. Case: A six month old male French bulldog, weighing 14.3 kg, was presented for a veterinarian appointment at the Department of Veterinary Medicine (Federal University of Sergipe, Brazil) due to a prolapsed nictitans gland and abnormalities of its genitalia. The medical records included recurrent urinary infection, presence of ectoparasites (thick) and ehrlichiosis, previously treated with doxycycline for 28 days (5 mg/kg/BID). During physical examination, the patient was bright and alert showing a good overall body condition. Temperature, pulse, respiration, thoracic auscultation and lymph nodes were within normal limits. Abdominal palpation was unremarkable. Inspection of the external genitalia was conclusive for bifd scrotum containing both testicles, incomplete fusion of the distal penile urethra, a vestigial penis, and an incomplete preputial cover. The urethral meatus was located ventral to the anus, in the perineal region between the testicles. It was also observed the presence of fecal material in the region where the urethra was opened, predisposing the animal to bacterial infections. However, no macroscopic sign of bacterial infection was noticed at the time of the appointment. Complementary exams such as complete blood count (CBC), urinalysis, two projections of contrast radiography, and abdominal ultrasound were requested. Orchiectomy and penile amputation were undertaken. Postoperative Enrofloxacin and Ketoprofen were prescribed. The dog returned for reevaluation and showed excellent recovery. Discussion: French bulldog seems to be more affected than other breeds by several congenital diseases, including hypospadias. The reported hypospadias was classifed as perineal, that is considered the most common type of hypospadias among dogs. Urinary infection was related by the tutor and that most likely occurred due to the urethral meatus opening location, which facilitates contamination by fecal coliforms. The CBC showed an absolute lymphopenia probably caused by the infection previously diagnosed. Urinalysis showed no alterations. The contrast radiography showed that there was not penile urethra, which one was ending ventral to the anus. The detailed examination was important to establish a defnitive diagnosis, to choose the best surgical technique and the most effective therapy. Both testicles and penis were nonfunctional, leading to the choice of orchiectomy and penile amputation, avoiding dryness and/or injuries due to penile and preputial mucosa exposure. Urethrostomy was not performed given the location of the urethra and the fact that he did not expressed signs of dysuria, not justifying the use of this technique. Although it is rare, hypospadias is a pathological condition that must be properly diagnosed and treated because it interferes not only in the animal esthetics, but also in its health and quality of life, as it predisposes the occurrence of infections and other possible complications. Keywords: malformation, penis, prepuce, canine