Uemura Yoshiki, Tsuboi Norihiko, Nakagawa Satoshi
Department of Critical Care Medicine, National Center for Child Health and Development, Tokyo, JPN.
Cureus. 2024 Aug 13;16(8):e66774. doi: 10.7759/cureus.66774. eCollection 2024 Aug.
The patient was a six-year-old boy with a history of musculocontractural Ehlers-Danlos syndrome (mcEDS). He presented to the emergency department after falling on the road the day before admission, which led to an increase in subcutaneous hematoma in his left lower leg and brief syncope. Initial blood tests revealed a decreased hemoglobin level of 8.1 g/dL (normal range: 14 g/dL). Contrast-enhanced CT showed a massive subcutaneous and intermuscular hematoma in the left thigh. He was diagnosed with hemorrhagic shock due to this extensive hemorrhage and was admitted to the ICU. The affected area was elevated, and hemostasis was achieved through compression. The swelling gradually improved, and he was discharged from the hospital on day 13 after admission. EDS is a systemic condition caused by genetic mutations affecting collagen and collagen-modifying enzymes. mcEDS is an extremely rare variant with a recently identified causative gene, characterized by abnormal connective tissue development and progressive fragility. Giant subcutaneous hematomas resulting from tissue fragility are serious complications of this disease, often occurring with minor trauma and sometimes leading to gradual hemorrhagic shock. Desmopressin nasal drops can be effective in preventing such hematomas. It is crucial to consider the risk of hemorrhagic shock from subcutaneous hemorrhage in patients with mcEDS, especially when repeated subcutaneous hematomas of unknown origin are observed.
该患者是一名6岁男孩,有肌肉挛缩型埃勒斯-当洛综合征(mcEDS)病史。入院前一天他在路上摔倒后被送往急诊科,摔倒导致他左小腿皮下血肿增大并出现短暂晕厥。初步血液检查显示血红蛋白水平降至8.1 g/dL(正常范围:14 g/dL)。增强CT显示左大腿有巨大的皮下和肌间血肿。由于这次广泛出血,他被诊断为失血性休克,并被收入重症监护病房。将患侧抬高,并通过压迫止血。肿胀逐渐好转,入院后第13天他出院了。埃勒斯-当洛综合征是一种由影响胶原蛋白和胶原蛋白修饰酶的基因突变引起的全身性疾病。mcEDS是一种极其罕见的变异型,其致病基因最近才被确定,其特征是结缔组织发育异常和进行性脆弱。由组织脆弱导致的巨大皮下血肿是这种疾病的严重并发症,常因轻微创伤而发生,有时会导致逐渐性失血性休克。去氨加压素滴鼻剂对预防此类血肿可能有效。对于mcEDS患者,考虑皮下出血导致失血性休克的风险至关重要,尤其是当观察到不明原因的反复皮下血肿时。