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Cholecystitis as the initial manifestation of disseminated cryptococcosis

Singh, Cynthia Sudar; Rahman, Mohammad; Jamil, Syed; Sawhney, Harinder; Vernaleo, John; Thelmo, William

doi: 10.1097/QAD.0b013e32801424e3

Wyckoff Heights Medical Center, Brooklyn, New York, USA.

Received 25 September, 2006

Revised 24 October, 2006

Accepted 2 November, 2006

Cryptococcus neoformans is an important fungal pathogen causing invasive infection in recent years as a result of the frequent occurrence of the disease in patients with AIDS. Disseminated cryptococcosis presenting as cholecystitis is rare. We report a 65-year-old woman with AIDS, who presented with clinical and radiological features suggestive of cholecystitis, and was ultimately found to have cryptococcal involvement of the gall bladder as part of disseminated cryptococcosis.

A 65-year-old woman, a known case of AIDS, was admitted with complaints of poor oral intake, anorexia, nausea and vomiting on and off for 3 weeks. Laboratory results were remarkable for elevated alkaline phosphatase of 599 U/l and normocytic normochromic anaemia (haemoglobin 10.2 g/l). An abdominal sonogram demonstrated probable cholecystitis and cholelithiasis. A contrast-enhanced computerized axial tomography scan of the abdomen and pelvis demonstrated distension of the gallbladder and a stone in the neck of the gallbladder. The patient underwent a laparoscopic cholecystectomy, and biopsy showed granulomatous inflammation with cryptococcus (Fig. 1). Gallbladder washings were positive for cryptococcal fungal organisms and negative for malignant cells. Cryptococcal cholecystitis was presumptively diagnosed, and blood culture and sensitivity for cryptococcus, and serum for cryptococcal antigen were sent. The cultures came back positive for cryptococcus and the antigen titres were high, greater than 1: 512. A spinal tap was performed and the cerebrospinal fluid (CSF) was found to be positive for cryptococcal antigen with a titre greater than 1: 256. The CSF protein was 106 mg/dl, glucose 42 mg/dl, white blood cells 7, lymphocytes 93%, polymorphs 1% and eosinophils 6%.

Fig. 1

Fig. 1

C. neoformans causes most cryptococcal infections in patients with defective cell-mediated immunity [1]. The lung and meninges are the two most common sites [2]. The skin, prostate, and medullary cavity of the bones are the next most common organs involved in disseminated cryptococcosis [3]. A review of the literature revealed only nine cases of cryptococcal infection [4] presenting as acute hepatitis [5–7], acute cholangitis [8], acute cholecystitis [9], and biliary obstruction and hepatic failure [10] as initial symptoms. Four of these patients had been subjected to exploratory laparotomy for clinical suspicion of acute abdomen. Two patients died of hepatic failure with systemic cryptococcosis. One patient died of cryptococcal infection superimposed on pre-existing postnecrotic cirrhosis. One patient developed cirrhosis after hepatic involvement with disseminated cryptococcosis. A patient with AIDS developed cryptococcal hepatitis during the course of his illness. A literature review also shows one case of a 35-year-old HIV-negative man who presented with clinical features suggestive of obstructive jaundice, and radiological features suggestive of Klatskin's tumor, but who ultimately was found to have cryptococcal involvement of the liver and biliary tract as part of disseminated cryptococcosis [11].

Our patient presented as cholecystitis, and the biopsy of the gallbladder after laparoscopic cholecystectomy revealed cryptococcal infection. This had prompted further work-up to identify possible infection in the blood and CSF. The patient was found to have high titres of crptococcal antigen in the blood and CSF, confirming cryptococcal meningitis and disseminated cryptococcosis. There has only been one reported case of cryptococcal cholecystitis, but that patient was immunocompetent and also had no other organ involvement. On the contrary, our patient is the only known case of cryptococcal cholecystitis who was immunocompromised and had fungemia and meningitis in addition to cholecystitis.

Whereas cryptococcosis is greatly increased in frequency in patients with defective cell-mediated immunity, before the AIDS epidemic approximately 50% of all patients with cryptococcosis had no readily identifiable immune defect [12]. We therefore present a rare occurrence of a case, emphasizing that the gallbladder could be the initial organ involved, other than the lungs and the neural system, in disseminated cryptococcosis in immunocompromised individuals.

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