Abstract

Benign Hybrid Lesions of the Jaws: A Systematic Review of the Literature

Author(s): Sarra Azzaz , Mariem Touati, Chiraz Baccouche

Background: Hybrid lesions of the jaws are rare pathological entities defined by the coexistence of two or more histopathologically distinct cystic and/or tumoral components within a single anatomical site. The World Health Organization (WHO) classification of odontogenic tumors and cysts does not currently recognize them, and their epidemiological, clinical, radiological, and therapeutic profiles remain poorly characterized. This systematic review aimed to synthesize the available literature on the epidemiology, clinical and radiological presentation, histopathology, management, and outcome of benign hybrid lesions of the jaws.

Methods: A systematic review was conducted according to PRISMA guidelines, searching PubMed/MEDLINE, Scopus, Web of Science, and Google Scholar (search finalized February 28, 2025), using a PICOS-structured question and Boolean combinations of MeSH and free-text terms (“hybrid lesion,” “odontogenic tumor,” “non-odontogenic tumor,” “jaw,” “maxilla,” “mandible,” “alveolar bone”). Case reports, case series, cohort, case-control, cross-sectional, and interventional studies in humans, in English or French, reporting hybrid lesions confined to the jaws, were eligible. Methodological quality was appraised using the Joanna Briggs Institute (JBI) critical appraisal checklists appropriate to each study design.

Results: Of 379 records identified (364 from database searching plus 15 from manual search), 56 studies were included (47 case reports, 6 case series, 3 retrospective studies), reporting 129 patients (age range 2–81 years, most frequently 15–29 years; 70 males (54.3%), 59 females (45.7%)). The mandible was affected in 96 cases (74.4%) and the maxilla in 31 (24.0%). Most lesions comprised two histopathological components (120/129, 93.0%); the most frequent combinations were Central Giant Cell Granuloma (CGCG) with central odontogenic fibroma (17 cases) and with cemento-ossifying fibroma (15 cases). Painless swelling was the most common clinical finding (67/88 evaluable cases, 76.1%). Panoramic radiography was the most frequently used imaging modality (52/70, 74.3%), most often showing well-defined (44/92, 47.8%) radiolucent (45/92, 48.9%) or mixed (43/92, 46.7%) images. Surgical treatment (enucleation, 37.9%; excision, 24.1%; resection, 9.2%, among others) was used in all definitively treated cases; intralesional corticosteroids failed in three CGCG-suspected cases. Among 53 patients with adequate follow-up (3 months–10 years), only one recurrence (1.8%) was recorded, involving a CGCG-cemento-ossifying fibroma hybrid lesion.

Conclusions: Hybrid lesions of the jaws predominantly affect the mandible of young adults, most commonly combine two components (frequently CGCG-based), and follow a generally favorable course after surgical treatment, with a low recurrence rate. Their absence from current WHO classification schemes hinders standardized diagnosis and management; formal recognition and prospective, multicenter studies are warranted.