Pulmonary arterial hypertension in patients with slow-flow vascular malformations

Arch Dermatol. 2010 Dec;146(12):1347-52. doi: 10.1001/archdermatol.2010.379.

Abstract

Objective: To determine the prevalence of pulmonary arterial hypertension in asymptomatic patients with 2 types of extensive slow-flow vascular malformations: extensive venous malformations or Klippel-Trénaunay syndrome (KTS).

Design: Case-control.

Setting: Multidisciplinary center for vascular anomalies.

Patients: A consecutive sample of 32 patients with slow-flow vascular malformations of at least 15% of the body surface was identified retrospectively and matched by age and sex with 32 healthy controls.

Interventions: Standard 2-dimensional transthoracic Doppler echocardiography. Venous samples were obtained the same day that echocardiography was performed.

Main outcome measures: Pulmonary artery systolic pressure (PASP) was determined. Levels of D-dimer, fibrinogen, and von Willebrand factor (vWF) in plasma were measured.

Results: Patients had a mean (SD) PASP that was significantly higher than that of healthy controls (42.16 [8.49] mm Hg in patients vs 27.69 [6.54] mm Hg in healthy controls; P < .001). No significant differences in PASP were found between patients with KTS and patients with venous malformations (P = .80). We observed significant differences in the mean (SD) levels of vWF between patients and healthy controls (124.41% [52.28%] in patients vs 92.69% [28.92%] in controls; P = .01) and also in levels of D-dimer (1032.99 [1367.0] ng/mL in patients vs 102.97 [29.39] ng/mL in healthy controls; P < .001). There was a moderate positive correlation between levels of vWF and levels of PASP (r = 0.42; P = .001) and a high positive correlation between D-dimer and PASP (r = 0.52; P < .001)

Conclusions: The presence of pulmonary arterial hypertension in patients with extensive slow-flow vascular malformations is not an isolated feature but is relatively frequent. Levels of D-dimer correlate with PASP in these patients.

Publication types

  • Comparative Study

MeSH terms

  • Adolescent
  • Adult
  • Blood Flow Velocity / physiology*
  • Child
  • Child, Preschool
  • Female
  • Fibrin Fibrinogen Degradation Products / metabolism
  • Fibrinogen / metabolism
  • Follow-Up Studies
  • Humans
  • Hypertension, Pulmonary / diagnostic imaging
  • Hypertension, Pulmonary / etiology*
  • Hypertension, Pulmonary / physiopathology
  • Lymphatic Vessels / abnormalities
  • Lymphatic Vessels / diagnostic imaging
  • Lymphatic Vessels / physiopathology*
  • Male
  • Middle Aged
  • Pulmonary Wedge Pressure / physiology*
  • Retrospective Studies
  • Ultrasonography, Doppler
  • Vascular Malformations / blood
  • Vascular Malformations / complications*
  • Vascular Malformations / diagnostic imaging
  • Veins / abnormalities
  • Veins / diagnostic imaging
  • Veins / physiopathology*
  • Young Adult
  • von Willebrand Factor / metabolism

Substances

  • Fibrin Fibrinogen Degradation Products
  • fibrin fragment D
  • von Willebrand Factor
  • Fibrinogen