Rationale:Pediatric Graves’ disease at preschool age is a rare condition. Previous reports have indicated that adolescents with this disease are girls. Pediatric Graves’ ophthalmopathy in young children is rare, and long-term follow-up reports are lacking.Patient concerns:The patient had hyperthyroidism and bilateral proptosis for 2 years, but she was only 4 years old.Diagnoses:The blood test revealed hyperthyroidism and the ophthalmic examination revealed proptosis. The patient had Graves’ disease and Graves’ ophthalmopathy.Interventions:Initially, she was followed up in the pediatric department. Bilateral proptosis developed, and she was brought to the ophthalmology department for assistance. Orbital computed tomography revealed borderline enlargement of the extraocular muscles bilaterally. Other initial clinical findings included bilateral upper and lower eyelid trichiasis and mild punctate epithelial erosions of the cornea. She received conservative medical treatment in the ophthalmology department.Outcomes:Remission of hyperthyroidism was achieved 2 years after medical control. No elevated intraocular pressure, strabismus, or optic neuropathy developed during follow-up. Significant cosmetic improvement and gradual resolution of punctate epithelial erosions were found over 10 years. Finally, the patient had only mild bilateral lower trichiasis.Lessons:Longitudinal follow-up revealed that the ocular manifestations of proptosis and eyelid trichiasis may have good outcomes. Proptosis gradually improved as the patient grew up.