Von Willebrand's disease and angiodysplasia treated with thalidomide

Hirri, H. M.; Green, P. J.; Lindsay, James O.

doi:10.1111/j.1365-2516.2006.01210.x

Cited by 34 publications

(31 citation statements)

References 7 publications

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“…Oestrogen and progesterone have been investigated in a number of non-VWD studies but when compared to control groups were ineffective and are no longer recommended [8]. A dramatic response to thalidomide in a patient with type 2B VWD who failed treatment with octreotide, DDAVP, rFVIIa and VWF concentrate was reported by Hirri et al [14]. Bowers and colleagues reported the successful use of intravenous octreotide in two patients who failed hormonal, steroidal, immunoglobulin and VWF concentrate treatment [13].…”

Section: Discussionmentioning

confidence: 99%

The natural history of occult or angiodysplastic gastrointestinal bleeding in von Willebrand disease

et al. 2014

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Recurrent gastrointestinal bleeding is one of the most challenging complications encountered in the management of patients with von Willebrand disease (VWD). The commonest cause is angiodysplasia, but often no cause is identified due to the difficulty in making the diagnosis. The optimal treatment to prevent recurrences remains unknown. We performed a retrospective study of VWD patients with occult or angiodysplastic bleeding within the setting of the von Willebrand Disease Prophylaxis Network (VWD PN) to describe diagnostic and treatment strategies. Centres participating in the VWD PN recruited subjects under their care with a history of congenital VWD and gastrointestinal (GI) bleeding due to angiodysplasia, or cases in which the cause was not identified despite investigation. Patients with acquired von Willebrand syndrome or those for whom the GI bleeding was due to another cause were excluded. Forty-eight patients from 18 centres in 10 countries were recruited. Seven individuals had a family history of GI bleeding and all VWD types except 2N were represented. Angiodysplasia was confirmed in 38%, with video capsule endoscopy and GI tract endoscopies being the most common methods of making the diagnosis. Recurrent GI bleeding in VWD is associated with significant morbidity and required hospital admission on up to 30 occasions. Patients were treated with multiple pharmacological agents with prophylactic von Willebrand factor concentrate being the most efficient in preventing recurrence of the GI bleeding. The diagnosis and treatment of recurrent GI bleeding in congenital VWD remains challenging and is associated with significant morbidity. Prophylactic treatment with von Willebrand factor concentrate was the most effective method of preventing recurrent bleeding but its efficacy remains to be confirmed in a prospective study.

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Section: Discussionmentioning

confidence: 99%

The natural history of occult or angiodysplastic gastrointestinal bleeding in von Willebrand disease

et al. 2014

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“…16 Available data on its use in this setting derives mostly from small studies and case reports of patients mainly with vWD and GI bleeding due to angiodysplasia. 5,7,17-19 Only one randomized controlled trial of thalidomide has been reported. 5 This trial enrolled 10 patients with gastrointestinal vascular malformations randomized to receive either 100 mg of thalidomide daily or 400 mg of iron daily.…”

Section: Discussionmentioning

confidence: 99%

Thalidomide for the Treatment of Gastrointestinal Bleeding Due to Angiodysplasia in a Patient with Glanzmann’s Thrombasthenia

et al. 2017

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Angiodysplasia is a frequent cause of persistent gastrointestinal (GI) hemorrhage in elderly patients. Although GI bleeding isn’t the most common manifestation in patients with bleeding disorders, when present, it represents a challenging complication. We describe a 62-year-old patient with Glanzmann’s thrombasthenia, who used thalidomide for severe and recurrent GI bleeding. For 6 months, the patient experienced temporary control of GI bleeding with thalidomide in a daily oral dose of 100 mg. The anti-angiogenic effects of thalidomide have recently been explored by several groups, particularly in the management of bleeding from angiodysplasia, including cases with von Willebrand disease. Here, we review the relevant descriptions of the use of thalidomide in this situation, and also discuss potential reasons why we observed only a temporary control of the GI bleeding in our patient, such as the use of low-dose regimen due to limitations posed by thalidomide side effects.

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“…Nosotros comenzamos con los análogos de la somatostatina, a las dosis recomendadas (100 µg/12 h s.c.) durante cuatro semanas sin obtener resultado. A la vista de los casos publicados en el tratamiento con talidomida (3)(4)(5)(6)(7)(8), cuyo éxito parece derivar de su efecto antiangiogénico (3,9), decidimos intentar dicha terapia. Hasta el momento, el resultado ha sido más que satisfactorio, con control de su sintomatología, "ahorro" de ingresos hospitalarios y transfusiones sanguíneas y sin efectos secundarios mayores.…”

Section: Discussionunclassified

Hemorragia invalidante y recidivante por angiodisplasia intestinal: tratamiento con talidomida

Colsa

Medina

Marín³

et al. 2008

Rev. esp. enferm. dig.

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Von Willebrand's disease and angiodysplasia treated with thalidomide

Cited by 34 publications

References 7 publications

The natural history of occult or angiodysplastic gastrointestinal bleeding in von Willebrand disease

The natural history of occult or angiodysplastic gastrointestinal bleeding in von Willebrand disease

Thalidomide for the Treatment of Gastrointestinal Bleeding Due to Angiodysplasia in a Patient with Glanzmann’s Thrombasthenia

Hemorragia invalidante y recidivante por angiodisplasia intestinal: tratamiento con talidomida

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