Limbal stem cell deficiency arising from systemic chemotherapy with hydroxycarbamide

Abstract

Purpose: The purpose of this study was to report a case of limbal stem cell deficiency (LSCD) after systemic chemotherapy with hydroxycarbamide.

Methods: Clinical manifestations and pathology are detailed.

Results: We describe the case of a woman with sickle cell disease, who developed bilateral LSCD after treatment with hydroxycarbamide. Histologic examination confirmed the diagnosis of LSCD, revealing goblet cells, inflammatory cells, deposits of new collagen components, and neovascularization in the peripheral cornea. Matrix metalloproteinase-3, fibronectin, and collagen III were also detected in the lesions.

Conclusions: The systemic use of the antineoplastic drug, hydroxycarbamide, may cause severe LSCD. We recommend that a medication history, including that of cytotoxic drugs, be considered in evaluating LSCD.

Figure 1

Ocular photography in May 2003 (upper panels) and February 2007 (lower panels). Upper panel: Pterygium-like lesions cover 270 degree of the conjunctiva and peripheral cornea involving the nasal, superior, and temporal areas of both eyes. Lower panel: Progressive pterygium-like growths extend to the central and inferior cornea with neovascularization, more severe in the left eye than in the right eye.

Figure 2

Photomicrographs of the left eye. A, An aggregation of inflammatory cells (long arrow) and neovascular vessels (short arrow) is observed in the limbal cornea. B, The inflammatory cells are mainly T lymphocytes (arrow). C, MMP-3 (arrows) is detected in the limbus and central cornea. D, Fibronectin (arrow) is detected in the central cornea with abrasion. E, Collagen III (arrow) is detected in the limbus with neovascularization. (A, hematoxylin and eosin; B–E, avidin–biotin complex immunoperoxidase; ×100.)