Brucellosis, a zoonotic disease caused by the Gram-negative bacterium Brucella infection, has a high incidence and morbidity rates in both humans and animals, which has imposed a heavy burden on society [1]. There are many kinds of Brucella, the most pervasive type is Brucella melitensis. The main source of infection of Brucella is animal food, and it can also be spread through contact with sick animals, its secretions or excrements. Once infected, clinical manifestations of brucellosis are protean. The common clinical manifestations include fever, sweating, abdominal pain, etc. But none of them was specific. Patients with brucellosis frequently experience local infections that can invade any tissues, organs or systems, ranging from the most common skeletal system to the rare cardiovascular system, the latter of which refers to endocarditis, myocarditis, pericarditis, and arteritis. Although rare, cardiovascular complications are the main cause of death regarding brucellosis.
Among variable cardiovascular involvement, vasculitis, especially concurrent with aortic and splanchnic artery involvements, has been reportedly limited as a clinical manifestation of Brucellosis. Herrick et al. reported 34 cases of Brucella infection leading to vascular involvement in which the most frequent site of involvement was the aorta, with 23 cases (68%) followed by the upper extremity blood vessels and the lower extremity blood vessels [1]. In the last few decades, the literatures reported suggested that brucellosis vascular involvement mainly arose in the aorta. A summary by Cascio demonstrated that a total of 46 cases complicated with aortic involvement had been investigated, including 18 cases of ascending aorta involvement, 30 cases descending aorta or abdominal aorta, of pseudoaneurysm in 6 cases and an ulcerative process in 2 cases [2]. Among many of these cases, vascular involvement led to increased mortality. Of this patient, aorta CTA exhibited multiple arteritis in aorta and its branches, accompanied by small aortic ulcers. Additionally, with the large inflammatory mass around the aorta the patient was also encompassed with considerable risk of adverse events.
The diagnosis of brucellosis with vascular involvement remains challenging. For those patients who had abnormal imaging findings on vascular involvement as the main manifestation and relatively simple clinical symptoms without fever, we were first required to determine whether it was vascular disease or vasculitis. This patient was an elderly woman with a long-term history of smoking and drinking, with imaging results showing the large arteries had atherosclerosis with plaque formation, on a basis of atherosclerosis vascular disease was supposed to be taken into consideration, while it can generally never result in elevated inflammatory markers such as hsCRP. Therefore, single vascular disease was hard to explain the whole story. According to the 2012 classification criteria for vasculitis, it was divided into 7 categories [3]. Immune-related vasculitis and vasculitis caused by other infections such as tuberculosis were also excluded after carefully examination. By chance, this patient had a positive result in blood culture after a series of screenings. The vasculitis attributed to brucellosis belonged to seventh category, that was, secondary vasculitis associated with infection etiology. This case also highlighted the importance of complete collection of clinical data, thus having the opportunity to discover the truth about the disease from the clues. Interestingly, while the high titers of rheumatoid arthritis related to autoantibodies existed, the patient had no symptoms of joints. Combined its above positive antibodies with the patient’s long-term history of smoking, it was considered that this was associated with smoking, the association and complicated pathogenesis of which had been discussed before [4, [5].
The main principles to identify antibiotics regiments for Brucellae are as follows: first, with the capability of entering into macrophages and keep active in the acidic intracellular environment; second, available to be applied for a longer period of time; last but not least, at least two antimicrobial drugs are preferred [6]. Derived from expert recommendation, comparative randomized and nonrandomized clinical trials, meta-analyses, observational studies, the current approaches are developed. In practical clinical work, the specific antibacterial drug treatment protocol, however, requires integrate multifactorial elements, including organ involvement, clinical severity, scope of application, drug symptoms, and so on. Regarding the course of treatment, there is yet another issue with a great controversy. Compared with other focal infection, aortic involvement is suggested to maintain targeted antibiotic therapy for longer length. Nevertheless, relapse frequently occurs even in adequate treated patients.
In addition to drug treatment, many literatures have reported surgical treatment. The appropriate timing of surgery is supposed to be carefully weighed. For cardiovascular involvement, indications for surgery include impending rupture of aneurysm, hemodynamic instability, and severe limb ischemia, which are similar to those used to treat fungal aneurysms and other intravascular infections [1]. Most patients undergo surgery immediately when infection is discovered.
Of this patient, she had multiple vasculitis changes in its aorta and was at high risk of rupture of the mass around the aorta at any time. As with previous cases, vascular rupture caused by aortic vasculitis was not uncommon and aortic vasculitis tended to cause increased morbidity even when receiving medication therapy [7, [8, [9]. Based on focal infection of crucial involvement, the patient underwent triple antimicrobial therapy and was slightly adjusted due to drug intolerance. Given that ceftriaxone was viewed as a promising candidate for combination therapy in specific situations of Brucella infection, therefore, alternative therapy considered it as prior choice. On the other hand, we were always ready for emergency surgery even after the initial start of antibacterial drugs. Fortunately, everything went smoothly during antibiotic therapy. We became greatly relieved that the mass around her aorta had dramatically shrunk after months of antibrucellar treatment, which also meant that the risk of rupture was significantly reduced compared with that of the beginning of admission to the ward. However, aortic CTA uncovered that aortic ulcers and arterial lumen stenosis failed to reach improved. It is necessary to complete the aortic MRA examination to assess whether there are vascular inflammation signals in the next follow-up, which will facilitate us to comprehensively evaluate how to deal with anti-inflammatory drugs of corticosteroids and immunosuppressive agents to treat secondary vasculitis. Hence, more long-term follow-up was indispensable for this patient.
A detailed knowledge of patients’ medical history always merits to be fully appreciated, followed by early diagnosis, timely utilization of appropriate drugs and surgical treatment, thus hopefully decreasing the morbidity rate, improving the prognosis, and reducing the social economic burden.