An unusual presentation of brucellosis

A 26-year-old man referred to our center (Alzahra University Hospital, Isfahan, Iran) with complaints of fever, ataxia and dysarthria. His problem began 3 months prior to the recent hospitalization with low grade fever, chilly sensation, cold sweating, loss of appetite and 10 Kg weight loss. He was also complaining from arthralgia in different joints and could not walk without help. His dysarthria and vomiting started 10 and 2 days before the admission, respectively. He had undergone left nephrectomy for congenital urethropelvic junction obstruction and severe hydronephrosis 2 years ago. He was symptom free up to the recent referral.

Physical examination revealed an ill looking young man with body temperature of 39°C, pulse rate 45/Min, and blood pressure of 110/60 mm Hg. He was dysarthteric and truncal ataxia was also observed. On auscultation he had muffled heart sounds without any murmur. The abdomen was soft with tender hepatomegaly 3 cm below the costal margin.

Results of laboratory tests made on admission were as follow: White blood cell count: 17300/mm-3 with 91%Neutrophils, Patelet count: 89000/mm-3, Hemoglobin: 14.1 g/dl, C-reactive protein: 15 mg/dl, Erythrocyte sedimentation rate: 41 mm/h, Blood urea nitrogen(BUN): 40 mg/dL, Creatine(Cr): 3.9 mg/dL, SGOT: 338 mg/dL, SGPT: 151 mg/dL, Alkaline phosphatase: 593 mg/dL, Creatine phosphokinase(CPK): 468 mg/dL, CPK(MB): 38 mg/dL,. Urinalysis revealed moderate hematuria and 24-hour urinalysis (U.A) revealed proteinuria (1143 mg/dL). ANA, ANCA, anti-dsDNA, RF, Anti Cardiolopin Ab, Lupus Anticoagulant were all negative. A PPD skin test was negative.

Chest X ray was normal. Electrocardiogram showed sinus bradycardia and high T waves in the precordial leads. Echocardiographic examination revealed septal hypokinesia and ejection fraction of 30%. No vegetation was reported. Brain MRI showed white matter changes in corpus callosum, periventricular area and centrum semiovalis (Fig. 1). Liver and kidney biopsies as well as lumbar puncture were not performed due to our patient's refusal.

 

Since he was a shepherd from an endemic area for brucellosis and gave a history about the consumption of non-pasteurized dairy products, standard tube agglutination test (STAT), 2-mercaptoethanol (2-ME), and Coombs test were performed (Antigen from the Pasteur Institute of Iran, Tehran). STAT and 2-ME titers were 1:80 and 1:40 respectively. Titer of Coombs test was 1:80. Enzyme-linked immunosorbent assay (IBL, Germany) for IgG and IgM was negative. Blood culture was performed once and it was positive for brucellosis. The Brucella omp2a gene was used as target DNA for PCR amplification. Polymerase chain reaction-restriction fragment length polymorphism (PCR-RFLP) [5] demonstrated the presence of Brucella mellitensis bv 1. Doxycycline (2 × 100 mg/day p.o.) and Rifampicin (1 × 600 mg/day p.o.) were started. The patient received this treatment for six weeks. One week after the initiation of the treatment our patient's symptoms subsided and he became afebrile. After 2 weeks he had normal liver function tests, BUN, and Cr levels and transthoracic echocardiography was normal too. The patient was followed-up for 10 months. In 6th months he experienced a relapse of brucellosis with signs and symptoms similar to the previous episode of the infection. The standard treatment regimen was started again. He is now receiving this treatment and is asymptomatic but still dysarthric. Our plan is to continue the medication for at least 6 months.