• Haixu Wang, Shuang Zhang, and Jing Hong.
• Department of Ophthalmology, Peking University Third Hospital, Key Laboratory of Vision Loss and Restoration, Ministry of Education, Beijing, China.
• Medicine (Baltimore). 2020 Oct 30; 99 (44): e22941.

IntroductionOcular benign fibrous histiocytoma can involve corneoscleral limbus and adjacent cornea and usually has a good prognosis after surgical removal. Despite the low recurrence rate, we reported a rare case of ocular benign fibrous histiocytoma with twice recurrences after excision.Patient ConcernsA 12-year-old Chinese girl presented with two painless progressively enlarging masses in the right eye for 6 years. She once had the lesions excised 1 year ago. However, the primary lesions relapsed again.DiagnosisHistopathologic and immunohistochemical examinations of the excised samples supported the diagnosis of benign fibrous histiocytomas of the corneoscleral limbus.InterventionsThe patient underwent mass resection with limbal stem cell transplantation and amniotic membrane transplantation at first. As for the tumors' second recurrence, we performed extended excision combined with lamellar keratoplasty and amniotic membrane implantation.OutcomesThe corneal graft remained clear with no sign of tumor recurrence 3 years after the second surgery.ConclusionComplete surgical resection with tumor-free margins is critical to reduce the recurrence of benign fibrous histiocytoma and appropriate ocular surface reconstruction is necessary to remedy tissue defect and maintain epithelial integrity.

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