: Introduction: In this report, we describe the case of Peripheral Hypertrophic Subepithelial Corneal Degeneration (PHSCD), managed with surgical excision and a human amniotic membrane (hAM) graft. In this article, we also propose a table for differential diagnosis, which might help physicians recognize this uncommon clinical entity in practice. Case presentation: A 45-year-old female Caucasian patient presented with decreased vision, itching and burning in both eyes, and a presence of pseudopterygium in the right eye, causing visual decline. Routine ocular exams an Anterior Segment OCT led to the diagnosis of PHSCD, after a careful differential diagnosis with other similar clinical occurrences. Subsequently, the patient was successfully managed with surgical intervention and an amniotic membrane graft placement. Conclusion: PHSCD is a rare disease, which in advanced cases can be successfully managed with hAM graft. No recurrence of the lesions was observed at one-year follow-up in our patient.
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