Clin Transplant Res.  2024 Mar;38(1):63-69. 10.4285/kjt.23.0064.

Hepatic artery vasospasm masquerading as early hepatic artery thrombosis in progressive familial intrahepatic cholestasis 3: a case report

Affiliations
  • 1Department of General Surgery, Postgraduate Institute of Medical Education and Research (PGIMER), Chandigarh, India

Abstract

Post-liver transplant (LT) hepatic artery vasospasm is a vascular complication that is not well recognized and its incidence is not known. As a possible sequela to vasospasm, hepatic artery thrombosis is the second major cause of allograft failure after primary nonfunction and its reported incidence is 2.9% in adults and 8.3% in pediatric LT. Lacuna in knowledge regarding early hepatic artery vasospasm post-LT makes it a difficult condition to diagnose and treat, as the initial ischemic insult on graft can have devastating consequences. We report a case of pediatric progressive familial intrahepatic cholestasis type 3-related chronic liver disease who underwent cadaveric orthotopic LT and postoperatively developed fever, worsening hypotension, and elevated liver enzymes with an absence of arterial flow in intrahepatic branches on Doppler ultrasound. Suspecting early hepatic artery thrombosis, the patient was re-explored and the graft hepatic artery was found to be in a state of vasospasm. Following the infusion of intra-arterial papaverine, urokinase, and intravenous nicorandil, there was an improvement in blood flow. The patient responded well and was discharged on postoperative day 23 with normal liver enzymes.

Keyword

Thrombosis; Vasospasm; Liver transplantation; Liver dysfunction; Surgery

Figure

  • Fig. 1 Pedigree chart depicting the inheritance of progressive familial intrahepatic cholestasis type 3 in our patient.

  • Fig. 2 Postoperative trend of liver enzyme levels. The black arrows indicate re-exploration. AST, aspartate aminotransferase; ALT, alanine transaminase; ALP, alkaline phosphatase; PREOP, preoperative; POD, postoperative day.

  • Fig. 3 Postoperative trend of bilirubin. The black arrows indicate re-exploration. PREOP, preoperative; POD, postoperative day.

  • Fig. 4 (A) H&E staining (×400) showing multiple nodules arranged in a typical jigsaw-puzzle-pattern surrounded by fibrosis. The fibrous septa are predominantly porto-portal, have a central area of collagenization and lymphocyte-rich inflammatory infiltrate, and peripheral area of edema giving rise to a "halo" effect (arrows). (B) CK7 immunostaining (×400) showing exuberant ductular reaction without any ductopenia and ductular metaplasia along with copper retention, ductular cholestasis, and maintained reticulin pattern (arrows).


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