Rare involvement in Behcet's disease at once: Bilateral axillary artery aneurysms and microaneurysms in the gastrointestinal vasculature

A 33-year-old man with Behcet's disease (BD) was hospitalized due to profuse lower gastrointestinal bleeding. One year prior to admission, he was operated on for bilateral axillary aneurysms sequentially at an external center, complicated by bleeding, recurrence of aneurysms, and wound infection (Figure 1A,B). Three months later, a diagnosis of BD was made based on recurrent oral ulceration, genital ulceration, and vascular involvement at an external center. He was treated with a combination of cyclophosphamide (CYC) and methylprednisolone (with a taper) for the last 5 months. At current admission, colonoscopy was unable to demonstrate the source of bleeding due to active hemorrhage during examination but showed multiple ulcerations in the terminal ileum. Conventional angiography examination revealed multiple microaneurysms in the distal parts of the mesenteric arterial systems, but no active bleeding (Figure 1C,D). Diagnosis of polyarteritis nodosa was ruled out based on the absence of other compatible symptoms. The patient was considered to have BD with arterial and gastrointestinal involvement, which was unresponsive to CYC (a total of 4 cycles). We started the infliximab infusion in line with high-dose methylprednisolone and performed blood transfusions as needed. He was discharged 2 weeks after his hospitalization. BD is a unique type of systemic vasculitis involving both the arterial and/or venous systems. Despite the rarity and general expectation of unilateral aneurysmal development in BD,1 a previous case report described bilateral axillary aneurysm.2 It is well-described that surgical exploration of aneurysms in BD before immunosuppression may worsen the outcome in several aspects as in our case: recurrence of aneurysms in surgical site, leading to decrease in the efficacy of induction treatment and infection risk. On the other hand, the unique part of our case is that we present bilateral axillary artery aneurysms and microaneurysms in the gastrointestinal vasculature simultaneously. Management of arterial aneurysms in BD may sometimes be challenging even with cyclophosphamide. Infliximab has been exponentially used during recent years with promising well-response in arterial involvement.3 Reşit Yıldırım, Döndü Üsküdar Cansu and Cengiz Korkmaz have prepared the manuscript. Burcu Ceren Uludoğan and Berat Acu provided information regarding images and treatment data. The final version of the manuscript has been evaluated and accepted by all authors. All authors declare no conflict of interest. Written informed consent has been obtained from the patient to access and collect data from the medical record to be used in scientific publications. Data sharing not applicable to this article as no datasets were generated or analysed during the current study.