Chronic Fusarium Infection in an Adult Patient with Undiagnosed Chronic Granulomatous Diseas

Clinical Infectious Diseases, Oct 2003

Disseminated Fusarium infection is a rare disease that is usually limited to immunocompromised patients. It more commonly occurs in patients with acute leukemia and prolonged neutropenia. We report a case of chronic Fusarium infection in an adult patient with undiagnosed chronic granulomatous disease (CGD), a primary immunodeficiency disorder in which phagocytic cells are defective in generating superoxide anion and its metabolites. The case is important because the patient had no manifestations of CGD until she was almost 60 years old and because this is, to our knowledge, the first reported case of Fusarium infection in a patient with CGD.

A PDF file should load here. If you do not see its contents the file may be temporarily unavailable at the journal website or you do not have a PDF plug-in installed and enabled in your browser.

Alternatively, you can download the file locally and open with any standalone PDF reader:

https://cid.oxfordjournals.org/content/37/7/e107.full.pdf

Chronic Fusarium Infection in an Adult Patient with Undiagnosed Chronic Granulomatous Diseas

Chronic Fusarium Infection in an Adult Patient with Undiagnosed Chronic Granulomatous Disease Davoud Mansoory 1 Navid Ahmady Roozbahany () 1 Hussein Mazinany 1 Alireza Samimagam 0 0 Department of Internal Medicine, Bandarabbas Medical University , Bandarabbas , Iran 1 Research Center for Tuberculosis and Pulmonary Diseases, Masih Daneshvari Hospital , Tehran Disseminated Fusarium infection is a rare disease that is usually limited to immunocompromised patients. It more commonly occurs in patients with acute leukemia and prolonged neutropenia. We report a case of chronic Fusarium infection in an adult patient with undiagnosed chronic granulomatous disease (CGD), a primary immunodeficiency disorder in which phagocytic cells are defective in generating superoxide anion and its metabolites. The case is important because the patient had no manifestations of CGD until she was almost 60 years old and because this is, to our knowledge, the first reported case of Fusarium infection in a patient with CGD. - Fusarium infection in humans is rare. In healthy hosts, most infections occur following receipt of a traumatic soft-tissue inoculation. In immunocompromised patients, inhalation or inoculation due to a minor trauma can lead to disseminated Fusarium infection. Fusarium species, in particular, Fusarium solani, are common causes of keratitis. They are also common causes of onychomycosis, endophthalmitis, and skin and musculoskeletal infections. The disseminated form of infection most commonly occurs in patients with acute leukemia and prolonged neutropenia. Skin lesions occur in 60%–80% of patients and usually manifest as multiple papules or deep painful nodules. They are most commonly located on the trunk and face [ 1 ]. Diagnosis is made on the basis of isolation of the fungal agent from blood samples or from skin biopsy specimens of suspicious lesions. Fusarium species produce catalase. The optimal treatment for this infecB R I E F R E P O R T tion has not been confirmed, but high-dose amphotericin B can be the drug of choice [ 2 ]. Among patients with the disseminated form of Fusarium infection, the mortality rate is high (range, 50%–80%). The risk of mortality completely depends on the underlying disease(s) in and the immune function of the patient. In this article, we describe a case of chronic Fusarium infection with multiple soft-tissue abscesses in an adult patient with undiagnosed chronic granulomatous disease (CGD). Case report. A 54-year-old married Persian woman presented to the hospital in July 2002 because of skin lesions with multiple fistulas on the left upper extremity (forearm and elbow). She had a 3-year history of such lesions, and results of microbiologic studies performed in 2 separate centers demonstrated that F. solani was the causative agent. She underwent surgical drainage and received medical treatment with multiple courses of amphotericin B, fluconazole, and itraconazole. Until 1 month before presentation, she had received amphotericin B therapy (25 mg every other day). At presentation, the skin lesions were partially healed. In 1974, the patient was successfully treated for meningitis due to Mycobacterium tuberculosis, but no documents were available that mentioned that the diagnosis was based on microbiologic data. She had also a history of basal cell carcinoma on the face, which was cured by surgical excision. She had a 3-year history of respiratory symptoms that involved productive cough. Physical examination revealed skin lesions (as described above) and a coarse crackle at left lung apex; there were no other abnormal clinical findings. The hemoglobin level was 14 g/dL, the WBC count was 8100 cells/mL (57% neutrophils and 35% lymphocytes), and the platelet count was 237,000 platelets/mL. The erythrocyte sedimentation rate was 72 mm/first hour. The induration of the PPD reaction was 25 mm in diameter, and the fasting blood sugar level, the blood urea nitrogen level, and results of creatinine and liver function tests were normal. A high-resolution CT scan of the lungs showed mild bronchiectasis and air trapped in the right middle lobe and lingula. Findings of CT scan of the perinasal sinuses were normal. Results of testing for hepatitis B surface antigen (HBs), antibody to HBs, antibody to HIV, and antibody to hepatitis C virus were negative. IgG, IgM, IgE, IgA, total hemolytic activity, and complement C3 and C4 levels were within normal limits. Results of neutrophil chemotaxis testing and flow cytometric analysis of PBMCs, including CD3 (total) T cells, CD4 (helper) T cells, CD8 (suppressor) T cells, CD19 (B) cells, CD56 (natural killer) cells, and adhesion molecules (CD18, CD11a, CD11b, and CD11c on lymphocytes, neutrophils, and monocytes), were normal. The results of cutaneous Candida testing were positive. Anti-diphtheria and anti-tetanus antibodies and serum zinc level were within normal limits. CGD was confirmed by the nitroblue tetrazolium test. This test was perf (...truncated)


This is a preview of a remote PDF: https://cid.oxfordjournals.org/content/37/7/e107.full.pdf

Davoud Mansoory, Navid Ahmady Roozbahany, Hussein Mazinany, Alireza Samimagam. Chronic Fusarium Infection in an Adult Patient with Undiagnosed Chronic Granulomatous Diseas, Clinical Infectious Diseases, 2003, pp. e107-e108, 37/7, DOI: 10.1086/377608