Mutsago Tonderai, Kazzazi Danny, Ibrahim Yahya, Kazzazi Fawz, Patel Hasu, Pafitanis Georgios
Barts Health NHS Trust, London, UK.
University College London, London, UK.
Hosp Pharm. 2024 Jun;59(3):276-281. doi: 10.1177/00185787231224064. Epub 2024 Jan 28.
Purple glove syndrome (PGS) is a rare condition characterized by limb edema, discoloration, and pain associated with intravenous and oral phenytoin administration. The pathophysiology is poorly understood, and there is no established treatment. Simple cases have previously been managed with hyaluronidase subcutaneous injections, with more severe cases resulting in compartment syndrome, debridement, or even amputation. In this case report, a 2-year-old boy with status epilepticus developed PGS after receiving intravenous phenytoin via a cannula on the dorsum of the right hand. The patient was successfully managed by locally infiltrating subcutaneous hyaluronidase diffusely to the affected area, titrating its dose to effect, rather than aiming to adhere to any specific dosing limitation. The child was reviewed daily by the Plastic Surgery team until being discharged, and focal lesions began to demarcate after 48 hours, with epidermal loss but no deeper trauma. The epidermis peeled within one month, with healthy underlying skin found underlying when followed up in clinic. This case illustrates that subcutaneous administration of hyaluronidase and titrating to effect provides an effective and safe treatment for treating distal cases of early PGS in children.
紫手套综合征(PGS)是一种罕见病症,其特征为肢体水肿、变色以及与静脉内和口服苯妥英钠给药相关的疼痛。其病理生理学尚不清楚,且尚无既定的治疗方法。此前,简单病例通过皮下注射透明质酸酶进行处理,更严重的病例则导致骨筋膜室综合征、清创术,甚至截肢。在本病例报告中,一名患有癫痫持续状态的2岁男孩在通过右手背的套管接受静脉内苯妥英钠治疗后发生了PGS。通过将皮下透明质酸酶广泛地局部浸润到受影响区域,根据效果调整剂量,而非遵循任何特定的剂量限制,成功地对该患者进行了治疗。整形外科团队每天对该患儿进行检查,直至其出院,48小时后局灶性病变开始分界,出现表皮脱落,但无更深层创伤。表皮在一个月内脱落,门诊随访时发现其下方的皮肤健康。本病例表明,皮下注射透明质酸酶并根据效果调整剂量为治疗儿童早期PGS的远端病例提供了一种有效且安全的治疗方法。