A case has been reported of a 49-year-old man with a history of brucellosis, cryoglobulinemia, and antineutrophil cytoplasmic autoantibodies (ANCA) associated vasculitis (AAV).
The case, reported in BMC Infectious Diseases, also presented hypertension, renal failure, nephrotic syndrome, livedo reticularis on the left foot, and congestive heart failure.
“Here we presented a rare case of chronic brucellosis with acute nephritic syndrome accompanied by cryoglobulinemia, AAV, superimposed on iliac aortic stent implantation,” the authors wrote. “The diagnosis and treatment of this case were intricate and experiences in such a case are valuable.”
The patient underwent pulmonary and abdominal computed tomography, which revealed bilateral exudative lesions, bilateral pleural effusion, and pulmonary atelectasis with pericardial effusion, along with peritoneal and pelvic effusion, and diffused abdominal wall edema.
Read more about AAV comorbidities
Kidney ultrasound showed normal-sized kidneys with increased parenchymal echogenicity, and an electrocardiogram revealed sinus rhythm with left ventricular high voltage. Finally, a transthoracic echocardiogram observed enlargement in all chambers of the heart and myocardial wall hypokinesia.
The authors note that recurrent attacks of brucellosis likely led to an immunological disturbance reflected by the coexistence of ANCA and cryoglobulinemia, which is a rare occurrence. Livedo reticularis is often seen in cryoglobulinemia-related vasculitis but not in AAV.
The patient responded well to treatment with oral doxycycline and rifampin for 6 weeks, with complete symptom disappearance and renal function improvement. He relapsed 3 times, and subsequent treatments were also effective in resolving symptoms, but his Brucella antibody levels continued to persist, possibly reflecting inadequate treatment.
The authors highlighted the importance of suspecting infection-related glomerulonephritis in the presence of both ANCA and cryoglobulinemia, rather than assuming it is AAV. They recommend a careful review of past medical records, renal biopsy, and actively treating coexisting brucellosis even if there are no symptoms of active infection, with the aim of avoiding brucellosis-associated renal complications.
Reference
Yang X, Jiao, C, Liu X, Zhang Y, Zhou H, Wang Y. Coexistence of cryoglobulinemia and ANCA-associated vasculitis in a chronic brucellosis patient – a case report and literature review. BMC Infect Dis. Published online May 2, 2023. doi.org/10.1186/s12879-023-08232-w
