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.
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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)