An unusual cause of gastrointestinal bleeding in a hemodialysis patient

Abstract

Iron deficiency caused by gastrointestinal (GI) bleeding is a common cause of anemia in hemodialysis patient. Herein, we report the case of an 89-year-old hemodialysis patient who presented with acute anemia and melena. Endoscopy found gastritis and diverticulosis without active bleeding. A capsule endoscopy (CE) was then performed and revealed multiple vascular lesions that lead to the diagnosis of Blue Rubber Bleb Nevus syndrome (BRBNS). This rare disease is associated with multiple venous malformations in the skin and the GI tract, usually observed in children. The patient developed 4 months latter 2 skin lesions compatible with BRBNS. Treatment included blood transfusion and intravenous iron supplementation. We reported an unusual presentation of venous malformation BRBS which differs from angioectasia, in an adult hemodialysis patient. Overall prognosis is good.

Blue Rubber Bleb Nevus Syndrome With Multiple Cavernoma-Like Lesions on MRI: A Familial Case Report and Literature Review

“…In our cases, the genetic suspicion might involve an autosomal dominant inheritance pattern, most commonly seen in chromosome 9 (6, 7). In addition, in 2017, new somatic double (cis) mutations were discovered in the TEK gene encoding for TIE2, also known as an angiopoietin-1 receptor expressed in the endothelial cells, which is critical for endothelial-cell to smooth-muscle-cell communication in venous morphogenesis (3,8). These variations in patterns of inheritance may influence the clinical heterogeneity of our cases.…”

“…They can be further classified as high or low flow lesions. Spontaneous bleeding is rare, although it may be caused by trauma (3,4). Cutaneous angiomas are found in up to 93% of patients with Bean's syndrome, with GI hemangiomas in 76%, CNS lesions in 13%, liver lesions in 11%, and diverse muscles in 11% (5).…”

Blue rubber bleb nevus syndrome: an unusual cause of intestinal bleeding in the elderly

“…Blue rubber bleb nevus syndrome (or Bean syndrome) is a rare cause of gastrointestinal bleeding. Only three cases have been reported in the elderly 1 2 3 .…”

“…Blue rubber bleb nevus syndrome (or Bean syndrome) is a rare cause of gastrointestinal bleeding. Only three cases have been reported in the elderly [1][2][3]. A 70-year-old man was hospitalized for recurrent and severe digestive bleeding for one month.…”