Aim of the study: To present a case of lateral rectus myositis in a 19-month-old male patient. Case report: The 19-month-old male patient was admitted to the clinic because of exophthalmos of the right eye for diagnosis and further treatment. Based on the clinical manifestation, and after other potential causes had been ruled out, the patient was diagnosed with ocular myositis. Treatment with steroids and antibiotics resulted in resolution of symptoms. Conclusions: Ocular myositis is an isolated inflammation of the extraocular muscles, manifested by swollen eyelids, exophthalmos, pain upon eye movement and diplopia. Ocular myositis is an extremely rare disease which occurs in pediatric populations. To the best of our knowledge, this is the first documented case of myositis in a very young child.