Pokharel Sunil, Singh Sukhraj, Shaukat Muhammad Saad
Department of Internal Medicine, Albany Medical Center, Albany, NY, USA.
Am J Case Rep. 2018 Sep 7;19:1071-1073. doi: 10.12659/AJCR.909718.
BACKGROUND This is a very interesting case of Granulomatosis with Polyangiitis (GPA) presenting with hand ischemia which rapidly evolved into dry gangrene from the involvement of digital arteries. GPA usually affects the small and medium sized blood vessels. GPA affecting muscular artery causing limb ischemia is a rare manifestation. CASE REPORT A 72 years old Caucasian female was sent to our hospital for the evaluation of left hand pain and bluish discoloration, which she noticed for few days. Physical examination was notable for bluish discoloration of left 2nd to 5th fingers, which later evolved, into dry gangrene at the tips. Angiogram revealed ischemia with no evidence of thrombosis. Administration of intra-arterial nitroglycerin resulted in improved blood flow through the radial artery. Labs revealed ESR of 142 mm/hr. C-ANCA titer was 5120, (normal <20) and antiproteinase 3 was 1117 (normal <20) consistent with GPA. CONCLUSIONS GPA usually affects the small and medium sized blood vessels. This case highlights an unusual presentation of GPA manifesting as critical limb ischemia. Given the rarity of this circumstance, a high index of suspicion is necessary in order to initiate proper treatment and limb salvaging intervention.
这是一例非常有趣的肉芽肿性多血管炎(GPA)病例,表现为手部缺血,由于指动脉受累迅速发展为干性坏疽。GPA通常累及中小血管。GPA累及肌性动脉导致肢体缺血是一种罕见的表现。
一名72岁的白种女性因左手疼痛和皮肤发蓝数日被送至我院评估。体格检查发现左手第2至5指皮肤发蓝,随后指尖发展为干性坏疽。血管造影显示缺血,无血栓形成迹象。动脉内给予硝酸甘油后桡动脉血流改善。实验室检查显示血沉为142mm/hr。C-ANCA滴度为5120(正常<20),抗蛋白酶3为1117(正常<20),符合GPA。
GPA通常累及中小血管。本病例突出了GPA表现为严重肢体缺血的不寻常表现。鉴于这种情况罕见,为启动适当治疗和肢体挽救干预,必须保持高度怀疑。