Severe Leptospirosis Presenting as Acalculous Cholecystitis

Leptospirosis is a worldwide zoonosis caused by spirochetes, more common in humid climates and rural areas, although in recent years an increase in cases in urban areas in developing countries has been described.¹ Severe presentations (Weil disease) with acute renal failure, respiratory failure due to adult respiratory distress or pulmonary hemorrhage are variable in frequency, but could appear in approximately 10%–50% of cases.^2–4 We present a case of severe leptospirosis and an uncommon clinical presentation, acute acalculous cholecystitis (AC).

A 55-year-old man, who worked in a farm, with a prior history of a lacunar stroke, came to the Emergency department of our Hospital with a 3-week history of myalgia and articular pains. In the last few days he had also presented abdominal pain radiated to the right shoulder, diarrhea and jaundice. On arrival, the patient was in mild distress, diaphoretic, jaundiced, with right conjunctival chemosis and intense pain in the right upper quadrant of the abdomen. He was in shock and in atrial fibrillation at 120 ppm. Blood tests revealed urea: 190 mg/dl (vn: 10–50), creatinine: 3 mg/dl (vn: 0.5–1.4), total bilirubin: 12.4 mg/dl (vn: 0.2–1.2), direct bilirubin: 12.1 mg/dl (vn: 0.1–0.3), ALT: 134 U/l (vn: 5–37), AST: 61 U/l (vn: 5–45), amylase: 157 U/l (vn: 20–115), CRP: 16.5 mg/dl (vn<0.5), neutrophilic leucocytosis: 27 100/mm³ and 84.20% respectively, thrombocytopenia: 57 000 platelets/mm³ and hyperfibrinogenemia: 1.291 mg/dl (nv: 150–400). An arterial gasometry reveals severe hypoxemia and hypocapnia. A chest X-ray showed a bilateral interstitial infiltrate and an abdominal CT scan revealed a gallbladder with thickened and oedematous walls without cholelithiasis or common bile duct dilatation. With a diagnosis of cholecystitis and possible cholangitis emergency surgery was performed. A cholecystectomy and live biopsy were performed. The patient was admitted in the Intensive Care Unit due to multiorgan failure, and mechanical ventilation, ionotropic support and antibiotherapy with ceftriaxone and ciprofloxacin.

During the next few hours the patient deteriorated, presenting a leukemoid reaction in blood tests and increase in bilirubin levels. Surgical re-exploration was performed. A thrombosis of the portal vein was observed, and the patient died in the operating room.

Due to prior contact with animals, serology for Mycoplasma, Chlamydia, Brucella and Coxiella were performed, which were negative; however, using indirect immunofluorescence, IgG and IgM antibodies for Leptospira interrogans were positive at 1/512 (nv<1/256) and 1/2.560 (nv<1/40), respectively. Microscopic agglutination (MAT) showed a title of 1/100 to the hardjo serovariety, and negative titles to bratislava, canicola, copenhageni, castellonis, grippotyphosa, pomona and pyrogenes serovarieties; polymerase chain reaction in a urine sample was positive for Leptospira spp. Histological examination of the gallbladder revealed an unspecific cholecystitis without detecting microbes and the liver biopsy revealed an infiltration of inflammatory cells, mostly neutrophils in the portal spaces.

An AC due to Leptospira can appear in isolation,^5–7 or associated with pancreatitis, with high levels of amylase and lipase associated with radiological findings due to pancreatitis.^8,9 The pathogenesis of AC is unknown, although as leptospirosis is considered a generalized vasculitis, a microvascular involvement could play an important role.

Occasionally the bacteria can be detected in the gallbladder,⁷ bile or intraabdominal fluid¹⁰ or by inmunohistochemistry finding bacterial antigens in the gallbladder wall.⁷ Treatment is controversial; in some cases the AC can resolve with medical treatment, without surgery^6,8; however, there have been cases described of clinical deterioration after an initial stabilization and several days of hospitalization^5,6,10 and a period of several days of intensive monitoring is recommended. In our case, due to the severity of the clinical presentation, emergency surgery was indicated, and in the re-operation an extensive thrombosis of the portal vein was found, that could have contributed to the fatal outcome.

It is therefore important, in young patients with AC, to perform a directed clinical anamnesis on possible epidemiological risk of leptospirosis, in order to start an adequate antibiotic treatment and in occasions avoid surgery using a conservative approach.