Oral Verruciform Xanthoma as a Manifestation of Chronic Graft-Versus-Host Disease in Fanconi Anemia: A Rare Case Report.

BACKGROUND

Oral verruciform xanthoma (OVX) is an uncommon benign epithelial lesion, typically observed in otherwise healthy individuals. It has occasionally been reported in association with chronic graft-versus-host disease (cGVHD) following hematopoietic stem cell transplantation (HSCT). To date, only 12 cases of OVX associated with cGVHD have been reported in the literature.

CASE REPORT: We present the first documented case of OVX in a 29-year-old male with Fanconi anemia (FA), occurring 19 years post-HSCT. The patient initially exhibited oral leukoplakia attributed to cGVHD, followed by the development of an OVX lesion on the posterolateral tongue. Clinically, the lesion appeared as a whitish plaque with a verrucous, irregular surface and well-demarcated borders. Histopathological examination revealed stratified squamous epithelium with papillary projections and keratin-filled clefts, along with numerous xanthoma cells confined to the connective tissue papillae.

CONCLUSION

This case contributes to the understanding of OVX in the context of FA and cGVHD, highlighting the critical importance of accurate diagnosis and individualized management of oral lesions in this high-risk population. Early recognition and intervention are essential to prevent progression and ensure optimal outcomes.