Successful treatment with thalidomide of a patient with congenital factor V deficiency and factor V inhibitor with recurrent gastrointestinal bleeding from small bowel arteriovenous malformations

Thalidomide in angiodysplasia‐related bleeding

“…Seven patients in separate case reports, but none in the randomised controlled trial, had increased or recurrence of bleeding on stopping thalidomide after completing treatment courses of between 4 and 12 months . Thalidomide was re‐initiated in four of these patients with good effect …”

“…We performed a literature review to identify all English‐language reports of the use of thalidomide in angiodysplasia‐related gastrointestinal bleeding finding 19 relevant publications. The number of patients from these reports totalled 70, drawing from one randomised controlled trial with 28 patients on the thalidomide arm, 29 patients from five case series and 13 single‐case reports . Of these patients, six had bleeding diatheses – type 2 von Willebrand disease ( n = 3), factor V deficiency ( n = 1) and anticoagulation ( n = 2) while five had hereditary haemorrhagic telangiectasia .…”

Management of bleeding in severe factor V deficiency with a factor V inhibitor

“…FV inhibitor occurs in around 3% of such patients after treatment with fresh frozen plasma (FFP) . In such a case, FFP, Platelet Concentrate (PC), recombinant activated Factor VII (rFVIIa) and/or tranexamic acid can be used as treatment, although there is a large variability in their clinical efficacy , and no guidelines are available for the treatment of life‐threatening bleeding. The use of corticosteroid, intravenous immunoglobulin or chemotherapy (i.e.…”