Cholecystitis associated with Q fever: case report and systematic review

Abstract

Background: Coxiella burnetii is an underestimated cause of acalculous cholecystitis. Large studies recently allowed to identify a dose-dependent association with IgG anticardiolipin. The management and long-term complications of Q fever-associated cholecystitis remain to be determined.

Methods: We describe a surgical case from Martigues, France, which is an endemic area and performed a systematic review of C. burnetii cholecystitis cases associated with Q Fever.

Results: Twenty-seven patients were included, including 3 (11.1%) children. All cases were reported during acute Q fever, followed by recurrent pancreatitis and chronic cholecystitis in 1 (3.7%) case. Two (2/2, 100%) were proven by a positive PCR on gallbladder but immunohistochemistry was negative in all cases. Only 3 (11.1%) cases were calculous. Coxiella burnetii cholecystitis is atypic because it is acalculous, with a flu-like syndrome, lupus anticoagulant, anticardiolipin antibodies and thrombocytopaenia.

Conclusions: Serology and PCR from blood and gallbladder biopsies are key to the aetiological diagnosis of C. burnetii cholecystitis. Inflammatory and/or autoimmune mechanism is suspected. Doxycycline remains the first-line therapy. Future prospective studies should determine whether treatment with doxycycline and hydroxychloroquine can prevent the chronic evolution of the disease in patients who initially present with antiphospholipid antibodies.

Keywords: Acalculous; Cholecystitis; Coxiella burnetii; Q fever.

Fig. 1

Study flowchart