Paroxysmal nocturnal hemoglobinuria (PNH) is a hematological disorder that affects hematopoietic stem cells. An association with other hematological diseases, such as hemolytic anemia and neutropenia, is observed with a high occurrence of aplastic anemia. The aim of the present study is to report a case of dental infection in a patient with PNH exhibiting exuberant gingival involvement. A 45‐year‐old male patient sought the Federal University of Ceara reporting severe toothache associated with tooth 24. Clinical examination revealed that the tooth was associated with an apparent fistula and a yellowish lesion with smooth surface located in the palate. The patient had interrupted the medication to control PNH. Blood transfusion was requested due to deficient hematological parameters. Tooth extraction and excisional biopsy were performed under antibiotic coverage. In the postoperative period, low‐level laser therapy (LLLT) was performed. Histopathological examination revealed connective tissue showing extensive necrotic areas, accumulation of basophilic material, numerous cyst‐like cavities, and degenerated cells. Histopathological findings were compatible with the initial clinical diagnosis of gingival necrosis. The patient evolved with febrile neutropenia, requiring hospitalization for 1 month. Improvement in the overall health was observed after the administration of antibiotics, eculizumab, and weekly LLLT at the biopsy site.

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