We present a case, with a 2-year follow-up, of the use of topical insulin in managing neurotrophic keratopathy in a 3-year-old boy diagnosed with congenital insensitivity to pain with anhidrosis (CIPA). Initially, the patient had a corneal perforation in the right eye, for which he underwent tectonic amniotic membrane transplantation. On examination, a central corneal ulcer with stromal infiltration was identified in the left eye, accompanied by a complete absence of corneal sensation in both eyes. Systemic features consistent with CIPA, such as the absence of a normal response to pain stimuli, anhidrosis, and self-injurious behavior, were also observed. Topical insulin, along with preservative-free lubricants (PF-L) and prophylactic antibiotics, was prescribed for both eyes. Within 2 weeks, the left corneal ulcer had healed, and treatment with topical insulin and PF-L was continued. After a 2-year follow-up, the left cornea exhibited a stable small paracentral nebula, whereas the right cornea showed a central non-adherent leucoma. No recurrence of corneal ulceration occurred.