Post-transplant gastric antral vascular ectasia after intra-venous busulfan regimen

Abstract

Gastric antral vascular ectasia (GAVE) is an angiodysplastic disorder that causes gastric bleeding. GAVE can develop as a complication of hematopoietic stem cell transplantation (HSCT-GAVE), and it has been suggested that it may be associated with oral administration of busulfan. We report two cases of HSCT-GAVE after a conditioning regimen containing intra-venous busulfan (ivBu), not oral busulfan. The first case, a 42-year-old woman with blastic plasmacytoid dendritic cell neoplasm, underwent second allogeneic HSCT with conditioning regimen consisting of cyclophosphamide (120 mg/kg) and ivBu (12.8 mg/kg). HSCT-GAVE developed on day 84 post-transplant, and argon plasma coagulation (APC) was performed successfully. The second case, a 60-year-old woman with acute myelogenous leukemia, underwent allogeneic HSCT with the conditioning regimen consisting of ivBu (12.8 mg/kg) and fludarabine (150 mg/kg). She developed melena and was diagnosed with GAVE by endoscopy on day 145 post-transplant. Although complete hemostasis was not achieved despite four administrations of APCs, the melena spontaneously terminated on day 235 post-transplant. To our knowledge, this is the first report describing HSCT-GAVE after ivBU-based HSCT. Although there is no established therapy for HSCT-GAVE, APC may be an option for HSCT-GAVE.

Gastric Antral Vascular Ectasia: Trends of Hospitalizations, Biodemographic Characteristics, and Outcomes With Watermelon Stomach

“…In our study population, a female predominance was noted which was in line with current literature, but there is no established pathophysiologic cause for this female predominance [ 18 , 19 ]. However, it is worth noting that studies investigating the use of antineoplastic drugs in women with gastric adenocarcinoma and gastric stromal tumors have reported the presence of GAVE [ 20 , 21 ]. This suggests that there may be a neoplastic or drug-induced pathophysiology for GAVE in these women [ 20 , 21 ].…”

HSCT-GAVE as a Manifestation of Chronic Graft versus Host Disease: A Case Report and Review of the Existing Literature

“…Several other cases have been reported, all of which included an oral busulfan-containing conditioning regimen [ 9 – 11 ]. In 2013, Fukuda et al presented 2 cases of HSCT-GAVE developed after IV busulfan-containing regimens (IV Bu/cyclophosphamide and IV Bu/cytosine arabinoside) [ 12 ]. These were the first two cases of HSCT-GAVE after a regimen that did not contain PO busulfan.…”

“…Though there could be no direct gastric antral mucosal contact in these cases, the authors stated that busulfan might still be implicated in the pathogenesis. Instead of direct contact to the mucosal membrane, they implicated “increased portal vein pressure caused by sinusoidal obstruction under the influence of busulfan, [which] induces ectasia of antral vein” [ 12 ]. Neither patient had signs of sinusoidal obstruction.…”

“…HSCT-GAVE was thought to have a connection to direct gastric mucosal contact with busulfan and the development of vascular ectasia from this exposure. However, the report of two patients who developed GAVE after HSCT with IV busulfan as part of their conditioning regimen shows that direct mucosal contact is not necessary [ 12 ]. Additionally, Hirayama described the cooccurrence of GAVE and protein losing enteropathy against a background of chronic GVHD [ 13 ].…”

Gastric antral vascular ectasia: a case report

“…Sy. GAVE may also be developed as a complication after haematopoetic stem cell transplantation or after per oral or intravenous application of busulfan (3). An average of 30% of Sy GAVE cases is associated with liver cirrhosis (4).…”