OBJECTIVE: A patient with rhabdomyosarcoma of the left eye with a history of recurrent sinusitis. Rhabdomyosarcoma is the most common malignant tumor of the soft tissues in children and mainly occurs in the head and neck. Nasal obstruction, rhinorrhea, and recurrent otitis media are the most common presenting symptoms. Treatment must be individualized for each patient, including chemotherapy, surgical resection, and radiotherapy. In this case, a 5-year-old girl (LG) was referred to pediatrics because of left-sided purulent rhinorrhea, fever, and leukocytosis, as well as a previous history of uncountable exacerbations of acute sinusitis. The patient's left-eye rhabdomyosarcoma had been treated years ago with otorhinolaryngological surgery, chemotherapy, and 12 radiotherapy sessions. The following were revealed during a physical examination: no alterations were apparent with oroscopy and otoscopy; with rhinoscopy, an abundant yellowish secretion was seen in the left nostril, as well as a purulent secretion in the left eye and edema of the ipsilateral hemiface. The child was admitted for ceftriaxone treatment and computed tomography of the sinuses. After a 14-day treatment with the medication without recovery, maxillary antrostomy and ethmoidal sinusotomy were performed. CONCLUSION: Rhabdomyosarcoma presents with various symptomatology and complications according to the involved location. If located in the head or neck, one of the first presenting symptoms is recurrent sinusitis despite having prior treatment, as in the described case.