Soyama Akihiko, Askeyev Baglan, Hara Takanobu, Matsushima Hajime, Adachi Tomohiko, Eguchi Susumu
Department of Surgery, Nagasaki University Graduate School of Biomedical Sciences, Nagasaki, Japan.
Department of Surgery, Nagasaki University Graduate School of Biomedical Sciences, Nagasaki, Japan.
Int J Surg Case Rep. 2024 Dec;125:110606. doi: 10.1016/j.ijscr.2024.110606. Epub 2024 Nov 15.
Hyperacute rejection leading to hepatic necrosis or intrahepatic bile duct stricture in ABO incompatible living-donor liver transplant (ABO-i LDLT) has been reported many times. With the advent of rituximab, the incidence of these complications has decreased significantly. However, consecutive biliary disruption after ABO-i LDLT has rarely been reported.
A female in her 50s with blood type A was admitted to our hospital for ABO-i LDLT due to failure of a graft (refractory ascites [Child-Pugh C(10), MELD 9]) that had been primarily transplanted 20 years ago from her ABO-identical father. Since the living donor was her husband with blood type B, rituximab was administered for ABO-i re-LDLT. After the LDLT, the patient recovered quickly despite bile leakage at the biliary anastomosis. Subsequently, the bile duct of the graft liver was serially disrupted with a bile lake, which required multiple instances of biliary drainage. A liver biopsy was performed and did not show any C4d staining on 195 post-transplant days. The patient ultimately developed sepsis due to cholangitis and expired at 11 months after the re-LDLT and finally C4d was positive on post-mortem biopsy.
Advances in ABO-i LDLT, particularly with rituximab, have reduced complications, but consecutive bile duct disruption remains challenging. Despite positive donor-specific antibody, early rejection markers were absent, suggesting complex mechanisms of complication.
We herein report a rare case as an important observation that may aid in preventing and treating potentially fatal complications after ABO-i LDLT.
超急性排斥反应导致ABO血型不相容的活体肝移植(ABO-i LDLT)出现肝坏死或肝内胆管狭窄的情况已有多次报道。随着利妥昔单抗的出现,这些并发症的发生率已显著降低。然而,ABO-i LDLT术后连续性胆管破裂的情况鲜有报道。
一名50多岁的A型血女性因20年前从与其ABO血型相同的父亲处首次移植的移植物失功(难治性腹水[Child-Pugh C(10),终末期肝病模型评分9])入住我院接受ABO-i LDLT。由于活体供体是其B型血的丈夫,因此在ABO-i再次肝移植中使用了利妥昔单抗。肝移植术后,尽管胆管吻合处有胆汁漏出,患者恢复迅速。随后,移植肝的胆管相继破裂并形成胆汁湖,需要多次进行胆管引流。在移植后195天进行了肝活检,未显示任何C4d染色。患者最终因胆管炎发展为脓毒症,并在再次肝移植后11个月死亡,尸检活检时C4d最终呈阳性。
ABO-i LDLT技术的进步,尤其是利妥昔单抗的应用,减少了并发症,但连续性胆管破裂仍然具有挑战性。尽管存在供体特异性抗体阳性,但早期排斥反应标志物阴性,提示并发症的机制复杂。
我们在此报告一例罕见病例,作为一项重要观察结果,可能有助于预防和治疗ABO-i LDLT术后潜在的致命并发症。