Severe gastrointestinal bleeding during pregnancy in a case of blue rubber bleb nevus syndrome

Blue rubber bleb nevus syndrome (BRBNS) is a rare disorder characterized by distinctive cutaneous and gastrointestinal venous malformations that usually cause massive or occult gastrointestinal hemorrhage and iron deficiency anemia secondary to the bleeding episodes. Only two reports of BRBNS in pregnant women can be found, according to the MEDLINE search. Both cases did not describe severe complications during pregnancy. This is the first case report of BRBNS with severe intestinal hemorrhage in a pregnant woman. The woman had been symptom-free for 5 years before the pregnancy and remained symptom-free for 2 years after delivery. This suggests that pregnancy induced the hemangiomas of the gastrointestinal tract to bleed. Celiotomy was needed to deal with massive gastrointestinal bleeding. We propose that women with BRBNS should be checked for systemic hemangiomas before pregnancy. However, it is difficult to check the small intestine thoroughly for hemangioma. Therefore, adequate attention needs to be paid to possible gastrointestinal bleeding when caring for patients with this condition. Our experience suggests that when dealing with pregnant women with BRBNS, it is essential to bear in mind the possibility that pregnancy may trigger the symptoms of hemangioma to become manifest, possibly causing life-threatening massive bleeding from the digestive tract.