Pulmonary thromboembolism associated with Klippel-Trenaunay syndrome

被引:65
作者
Huiras, EE
Barnes, CJ
Eichenfield, LF
Pelech, AN
Drolet, BA
机构
[1] Childrens Hosp Wisconsin, Dept Pediat Dermatol, Milwaukee, WI 53226 USA
[2] Childrens Hosp Wisconsin, Dept Pediat Cardiol, Milwaukee, WI 53226 USA
[3] Med Coll Wisconsin, Milwaukee, WI 53226 USA
[4] Childrens Hosp, Dept Pediat & Adolescent Dermatol, San Diego, CA USA
[5] Univ Calif San Diego, San Diego, CA 92103 USA
关键词
Klippel-Trenaunay syndrome; Proteus syndrome; venous malformation; hypercoagulable state; deep venous thrombosis; pulmonary embolism; Kasabach-Merritt syndrome;
D O I
10.1542/peds.2004-1607
中图分类号
R72 [儿科学];
学科分类号
100202 ;
摘要
Klippel-Trenaunay syndrome (KTS) is a rare congenital anomaly characterized by unilateral limb overgrowth, venous varicosities, and capillary malformations (port wine stains) of the affected limb or limbs. Large venous malformations such as those observed in KTS are rare, and many physicians are unfamiliar with the potential complications, which include hypercoagulability, thrombosis, and pulmonary embolism (PE). As a result, patients may suffer from delayed diagnosis of a potentially life-threatening thromboembolic event. We present 2 cases of children with KTS complicated by PE, and we review the English-language literature regarding pathophysiologic features, interventions, and outcomes of PE in the setting of KTS among both pediatric and adult patients, with emphasis on issues relevant to pediatricians.
引用
收藏
页码:E596 / E600
页数:5
相关论文
共 29 条
[1]   Klippel-Trenaunay syndrome with a life-threatening thromboembolic event [J].
Aggarwal, K ;
Jain, VK ;
Gupta, S ;
Aggarwal, HK ;
Sen, J ;
Goyal, V .
JOURNAL OF DERMATOLOGY, 2003, 30 (03) :236-240
[2]   VENOUS THROMBOEMBOLIC COMPLICATIONS (VTE) IN CHILDREN - FIRST ANALYSES OF THE CANADIAN REGISTRY OF VTE [J].
ANDREW, M ;
DAVID, M ;
ADAMS, M ;
ALI, K ;
ANDERSON, R ;
BARNARD, D ;
BERNSTEIN, M ;
BRISSON, L ;
CAIRNEY, B ;
DESAI, D ;
GRANT, R ;
ISRAELS, S ;
JARDINE, L ;
LUKE, B ;
MASSICOTTE, P ;
SILVA, M .
BLOOD, 1994, 83 (05) :1251-1257
[3]  
ARANOFF DM, 1998, SOUTH MED J, V91, P1073
[4]   EVALUATION OF KLIPPEL-TRENAUNAY-SYNDROME WITH RADIONUCLIDE TOTAL-BODY ANGIOGRAPHY A CASE-REPORT [J].
AWAD, AN ;
YANG, DC ;
GIRGIS, M ;
GIOVANNIELLO, J ;
HUANG, M ;
BEBAHANI, M ;
HOLTKAMP, DK .
CLINICAL NUCLEAR MEDICINE, 1992, 17 (11) :866-870
[5]   THE KLIPPEL-TRENAUNAY SYNDROME - CLINICAL, RADIOLOGICAL AND HEMODYNAMIC FEATURES AND MANAGEMENT [J].
BASKERVILLE, PA ;
ACKROYD, JS ;
THOMAS, ML ;
BROWSE, NL .
BRITISH JOURNAL OF SURGERY, 1985, 72 (03) :232-236
[6]  
Bousema MT, 1999, CLIN EXP DERMATOL, V24, P372
[7]  
DAMICO JA, 1977, CLEVELAND CLIN Q, V44, P181, DOI 10.3949/ccjm.44.4.181
[8]   2-YEAR-OLD BOY WITH PROTEUS SYNDROME AND FATAL PULMONARY THROMBOEMBOLISM [J].
EBERHARD, DA .
PEDIATRIC PATHOLOGY, 1994, 14 (05) :771-779
[9]   Extensive pure venous malformations in the upper or lower limb: A review of 27 cases [J].
Enjolras, O ;
Ciabrini, D ;
Mazoyer, E ;
Laurian, C ;
Herbreteau, D .
JOURNAL OF THE AMERICAN ACADEMY OF DERMATOLOGY, 1997, 36 (02) :219-225
[10]   Recurrent pulmonary embolism associated with Klippel-Trenaunay-Weber syndrome [J].
Gianlupi, A ;
Harper, RW ;
Dwyre, DM ;
Marelich, GP .
CHEST, 1999, 115 (04) :1199-1201