Hemophilic Pseudotumor of the Maxilla Secondary to Endodontic Treatment: Case Report and Systematic Review.

Abstract

Hemophilic pseudotumor (HP) is a rare but severe complication of hemophilia, characterized by progressive bleeding in the muscles, joints, and bone tissue, which can lead to lytic lesions. Its prevalence is approximately 1-2% among patients with hemophilia. This report presents a male patient with mild hemophilia A who developed an intraosseous lesion in the posterior region of the right maxilla, with a prior history of endodontic treatment in the area. Surgical excision was performed and, following clinicopathological correlation, the lesion was diagnosed as HP. <b>Background/Objectives:</b> This review aims to identify previously reported cases of HP located in the maxilla. <b>Methods:</b> The study protocol followed the Preferred Reporting Items for Systematic Review and Meta-Analyses (PRISMA) guidelines. The databases PubMed, Scopus, and ScienceDirect were searched, and Google Scholar was used to identify gray literature. The Joanna Briggs Institute (JBI) tool was employed to assess the risk of bias and the quality of the included reports and case series. <b>Results:</b> A total of 1487 publications were identified using specific keywords. After removing duplicates and non-relevant titles/abstracts, 42 full-text articles were reviewed. Of these, 10 met the inclusion criteria: 7 case reports and 3 case series, comprising 13 cases of HP in the maxilla, including the case presented here. Although rare in the maxillofacial region, when it does occur, it is more commonly seen in the mandible and is often linked to prior trauma. In this case, endodontic treatment may have triggered lesion development. <b>Conclusions:</b> This report highlights that, although uncommon, HP can manifest with involvement of the maxilla, and that specific dental interventions may represent potential triggering events.