Literature DB >> 35199719

Undiagnosed Case of Klippel-Trenaunnay Syndrome Presenting as Extensive Heterotrophic Ossification and Flexion Deformity of Right Lower Limb Requiring Amputation : A Case Report.

Kushal Gautam1, Sangharsha Thapa2, Anu Radha Twayana2, Lokendra Chhantyal3, Puskar Poudel4, Avinash Kc5, Swati Chand6.   

Abstract

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.

Entities:  

Keywords:  flexion deformity; heterotrophic ossification; Klippel -Trenanunay syndrome; vascular malformation.

Mesh:

Year:  2021        PMID: 35199719      PMCID: PMC9107906          DOI: 10.31729/jnma.6932

Source DB:  PubMed          Journal:  JNMA J Nepal Med Assoc        ISSN: 0028-2715            Impact factor:   0.556


  12 in total

1.  The Klippel-Trenaunay syndrome: clinical, radiological and haemodynamic features and management.

Authors:  P A Baskerville; J S Ackroyd; M Lea Thomas; N L Browse
Journal:  Br J Surg       Date:  1985-03       Impact factor: 6.939

2.  Pulmonary thromboembolism associated with Klippel-Trenaunay syndrome.

Authors:  Erin E Huiras; Cheryl J Barnes; Lawrence F Eichenfield; Andrew N Pelech; Beth A Drolet
Journal:  Pediatrics       Date:  2005-09-15       Impact factor: 7.124

3.  Klippel-Trénaunay syndrome: spectrum and management.

Authors:  A G Jacob; D J Driscoll; W J Shaughnessy; A W Stanson; R P Clay; P Gloviczki
Journal:  Mayo Clin Proc       Date:  1998-01       Impact factor: 7.616

4.  Dominant inheritance and intra-familial variations in the association of Sturge-Weber and Klippel-Trenaunay-Weber syndromes.

Authors:  José Maria Pereira de Godoy; Agnes Cristina Fett-Conte
Journal:  Indian J Hum Genet       Date:  2010-01

Review 5.  Vascular malformations. Part II: associated syndromes.

Authors:  Maria C Garzon; Jennifer T Huang; Odile Enjolras; Ilona J Frieden
Journal:  J Am Acad Dermatol       Date:  2007-04       Impact factor: 11.527

6.  Genetic aspects of the Klippel-Trenaunay syndrome.

Authors:  G E Aelvoet; P G Jorens; L M Roelen
Journal:  Br J Dermatol       Date:  1992-06       Impact factor: 9.302

Review 7.  Klippel-Trenaunay Syndrome.

Authors:  Philip R John
Journal:  Tech Vasc Interv Radiol       Date:  2019-09-23

8.  Overlap of sturge-weber syndrome and klippel-trenaunay syndrome.

Authors:  Radheshyam Purkait; Tryambak Samanta; Tapankumar Sinhamahapatra; Mridula Chatterjee
Journal:  Indian J Dermatol       Date:  2011-11       Impact factor: 1.494

9.  An atypical case of Klippel-Trénaunay syndrome presenting with crossed-bilateral limb hypertrophy and postaxial polydactyly: a case report.

Authors:  Rawan M Al-Najjar; Rafael Fonseca
Journal:  BMC Pediatr       Date:  2019-04-06       Impact factor: 2.125

10.  Klippel-Trenaunay Syndrome Causing Life-Threatening GI Bleeding: A Case Report and Review of the Literature.

Authors:  Salih Samo; Muhammed Sherid; Husein Husein; Samian Sulaiman; Margaret Yungbluth; John A Vainder
Journal:  Case Rep Gastrointest Med       Date:  2013-06-03
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