A case of haemolytic anemia triggered by an odontogenic infection in a patient with cold agglutinin disease

Abstract

Cold agglutinin disease (CAD) is a rare autoimmune haemolytic anaemia characterised by the presence of cold agglutinins, which are autoantibodies that agglutinate erythrocytes upon exposure to low temperatures. Although CAD patients typically develop haemolytic anaemia in response to febrile infections, no reports have been documented thus far regarding haemolytic anaemia in CAD patients resulting from odontogenic infections. A 47-year-old male patient with haemolysis because of an odontogenic infection is presented in this report. The patient had been diagnosed with CAD dating back approximately a decade, although the patient had not undergone haematological treatment. In November 2021, the patient was referred to our department from the haematology department because of haemolysis, which would have been triggered by acute right maxillary sinusitis. The sinusitis was caused by apical periodontitis of the second molar in the right maxilla. To address the condition, the patient received a red blood cell (RBC) transfusion along with antibiotics to control the sinusitis. After sinusitis had been improved, according to the suggestion from haematologists, the maxillary right second molar was extracted under local anaesthesia under body temperature control in January 2022. The patient’s body was kept warm, and infusion fluid for intravenous administration was controlled at approximately 37℃ during the peri- and postoperative period to avoid CAD. Because an odontogenic infection could be a trigger of haemolysis, maintenance of oral hygiene and early treatment of bacterial infections are important in patients with CAD.