Zahedi Bita, Wallace Zachary S, Côté Maya McEwen, Yu Elaine W
Endocrine Unit, Massachusetts General Hospital, Boston, MA 02114, USA.
Rheumatology Department, Massachusetts General Hospital, Boston, MA 02114, USA.
JCEM Case Rep. 2023 Jul 14;1(4):luad085. doi: 10.1210/jcemcr/luad085. eCollection 2023 Jul.
We report a case of isolated cutaneous small vessel vasculitis (SVV) occurring after zoledronic acid (Zol) infusion in a 58-year-old postmenopausal woman with a history of sleeve gastrectomy. This was the patient's first exposure to a bisphosphonate medication. Within minutes of the Zol infusion, she developed an episode of diffuse watery diarrhea. Although the diarrheal symptoms resolved quickly, she experienced nonsteroidal anti-inflammatory drug-responsive generalized myalgias and skin tenderness in her abdomen and extremities within a few hours. These symptoms progressed in severity over the next 5 days, and she developed nonblanching, palpable purpura extending from the ankles to the knees. Prior to Zol, labs showed sufficient 25-hydroxyvitamin D and calcium as well as normal renal and liver function. On day 10, laboratory tests revealed aspartate transaminase twice and alanine transaminase thrice the upper limit of normal. The patient was diagnosed with cutaneous SVV, with a timeline highly suggestive of an idiosyncratic reaction to Zol. She was successfully treated with a prednisone taper. No prior cases of Zol-induced cutaneous vasculitis have been reported, although there are a handful of reported cases of giant cell arteritis and urticarial vasculitis after bisphosphonate therapy. Clinicians should be aware that isolated cutaneous SVV may be a rare complication of Zol.
我们报告了一例58岁绝经后女性在接受唑来膦酸(Zol)输注后发生孤立性皮肤小血管血管炎(SVV)的病例,该患者有袖状胃切除术病史。这是该患者首次接触双膦酸盐药物。在输注Zol的几分钟内,她出现了一次弥漫性水样腹泻。尽管腹泻症状很快缓解,但在数小时内她出现了非甾体抗炎药反应性的全身性肌痛以及腹部和四肢皮肤触痛。这些症状在接下来的5天里严重程度不断进展,并且她出现了从脚踝延伸至膝盖的非压之褪色的可触及紫癜。在使用Zol之前,实验室检查显示25-羟维生素D和钙水平充足,肾功能和肝功能正常。在第10天,实验室检查显示天冬氨酸转氨酶是正常上限的两倍,丙氨酸转氨酶是正常上限的三倍。该患者被诊断为皮肤SVV,时间线高度提示对Zol的特异反应。她通过逐渐减量使用泼尼松成功治愈。尽管有少数双膦酸盐治疗后发生巨细胞动脉炎和荨麻疹性血管炎的病例报告,但此前尚无Zol诱导的皮肤血管炎病例报告。临床医生应意识到孤立性皮肤SVV可能是Zol的一种罕见并发症。