Department of Paediatric Research, Patan Hospital, Lagankhel, Nepal.
Kathmandu University School of Medical Sciences, Dhulikhel, Kavrepalanchowk, Nepal.
JNMA J Nepal Med Assoc. 2021 Sep 11;59(241):938-941. doi: 10.31729/jnma.6932.
Klippel-Trenaunnay Syndrome is a rare disease characterized by a clinical triad of capillary malformation, soft tissue and bony hypertrophy, and atypical varicosity. This syndrome ranges from asymptomatic disease to life-threatening bleeding, embolism, and deformities. Management includes early diagnosis, prevention, and treatment of complications. We present a case of a 43-year-old male presenting with pain, swelling and deformity of the right leg for 30 years. On examination, diffusely enlarged tender right limb with several dark patchy discolorations, multiple tortuous vessels were found. Right leg X-ray showed heterotrophic ossification and distortion of ankle joint. Due to chronic severe pain, recurrent infection, contracture and flexion deformity of right leg, the patient underwent above knee amputation. This case focuses on the variable presentation and multiple problems faced by patients with Klippel-Trenaununay Syndrome as they get diagnosed late and shows the importance of high index of suspicion for early diagnosis and prevention of complications.
克莱佩特-屈纳综合征是一种罕见疾病,其特征为毛细血管畸形、软组织和骨肥大以及非典型静脉曲张三联征。该综合征的表现范围从无症状疾病到危及生命的出血、栓塞和畸形。治疗包括早期诊断、预防和处理并发症。我们报告了一例 43 岁男性,其 30 年来右下肢出现疼痛、肿胀和畸形。体格检查发现,右下肢弥漫性肿大,触痛,有多处暗斑状变色,可见多个迂曲的血管。右下肢 X 线片显示异位骨化和踝关节畸形。由于慢性严重疼痛、反复感染、右下肢挛缩和屈曲畸形,患者接受了膝关节以上截肢。本病例重点介绍了克莱佩特-屈纳综合征患者的多种表现和面临的多种问题,因为他们往往被误诊,这凸显了提高对该病的早期诊断和预防并发症的认识的重要性。