Background <p><i>Coxiella burnetii</i> is an underestimated cause of <i>acalculous</i> 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.</p> Methods <p>We describe a surgical case from Martigues, France, which is an endemic area and performed a systematic review of <i>C. burnetii</i> cholecystitis cases associated with Q Fever.</p> Results <p>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. <i>Coxiella burnetii</i> cholecystitis is <i>atypic</i> because it is <i>acalculous</i>, with a flu-like syndrome, lupus anticoagulant, anticardiolipin antibodies and thrombocytopaenia.</p> Conclusions <p>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.</p>

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Cholecystitis associated with Q fever: case report and systematic review

  • Aymery Stheme de Jubécourt,
  • Marie Hocquart,
  • Olivier Picaud,
  • Georges Farvacque,
  • Sophie Edouard,
  • Louis Beaudoin,
  • Romain Bitoun,
  • Hubert Lepidi,
  • Florence Fenollar,
  • Pierre-Edouard Fournier,
  • Matthieu Million

摘要

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.