ABSTRACT
Introduction: Acute fatty liver of pregnancy (AFLP) is a rare condition characterized by maternal liver dysfunction and/or failure during the third trimester or early postpartum period, thought to be caused by defects in fatty acid metabolism during pregnancy. Management includes prompt delivery and supportive care. In the following case, we describe a patient found to have fulminant liver failure after delivery but was unable to undergo liver transplantation due to Coronavirus Disease 2019 (COVID-19) infection. Fortunately, with plasmapheresis, the patient recovered from both fulminant liver failure and COVID-19. Case Description/Methods: A 26-year-old female 38 weeks pregnant with recent diagnosis of COVID-19 infection presented with 3 days of nausea, vomiting, abdominal pain, and polydipsia. Due to non-reassuring fetal heart rate tracings, emergency cesarean section was performed. On postpartum day one, she was noted to be somnolent and jaundiced. Laboratory testing revealed a leukocytosis of 17,800, platelets 168,000, creatinine 3.95 mg/dL, glucose 26 mg/dL, uric acid 8.4 mg/ dL, total bilirubin 8.5 mg/dL, AST 234 U/L, ALT 224 U/L, ALP 466 U/L, PT 57.3 s, and ammonia 69 μmol/L. A chest x-ray showed bibasilar opacities;abdominal ultrasound revealed a mildly fatty liver and ascites. Viral, autoimmune hepatitis, hemochromatosis, alpha-1 antitrypsin deficiency, and Wilson's serologies were unremarkable. Thirteen Swansea criteria were met, thus a diagnosis of AFLP was made. Liver transplantation was initially considered but was high risk due to active COVID-19 infection resulting in respiratory failure requiring intubation. After multidisciplinary discussion, she was started on plasmapheresis. After 6 days of plasmapheresis, liver and renal function improved. She was discharged home on post-partum day 19. Discussion: In the above case, we describe a patient with COVID-19 infection with fulminant liver failure from AFLP. The patient needed a liver transplant due to rapidly worsening liver function. However, due to limited data on the safety of liver transplantation in active COVID-19 infection, it was deemed high-risk. Fortunately, the patient's condition improved with plasmapheresis, and liver transplant was not needed. Plasmapheresis removes toxins, ammonia, and inflammatory cytokines while replacing coagulation factors, albumin, and biologically active substances that are typically produced by liver cells. The therapy has also been reported to be an effective salvage treatment for severe cases of COVID-19.
ABSTRACT
Introduction: Immune checkpoint inhibitors (ICI) are commonly associated with gastrointestinal adverse events including colitis. We present a case of severe immune checkpoint inhibitor colitis managed with vedolizumab after failing standard treatment with steroids and infliximab. Case Description/Methods: A 75-year-old male with metastatic melanoma status post brain tumor resection, radiation therapy and 2 cycles of ipilimumab/nivolumab presented with severe new onset diarrhea 3 weeks after his second cycle of ICI. Given concern for ICI induced colitis, oral prednisone was started with initial improvement of diarrhea. Severe diarrhea recurred with tapering and he was admitted to the hospital. After infectious workup was negative, he received a dose of infliximab for presumed immune-mediated colitis with mild improvement. He was discharged on prednisone 60 mg daily with a rapid 2-week taper before receiving COVID-19 vaccination. He received a second dose of infliximab 2 weeks after the first. However, a week later, he was readmitted to the hospital with grade 3 diarrhea, dehydration, weakness, hypotension, and continued weight loss. Flexible sigmoidoscopy revealed continuous mucosal ulceration with congestion and loss of vascular markings from anus to sigmoid colon (Figure 1). Biopsies showed moderate to severe active colitis with ulceration, increased crypt apoptosis, and crypt dropout-most compatible with immune checkpoint inhibitor colitis. After 1 week of poor response to high dose methylprednisolone, he was given 2 doses of vedolizumab 2 weeks apart which lead to a complete resolution of his GI symptoms. Four months later, he remains asymptomatic with a negative PET scan off vedolizumab and ICI therapy. Discussion: Immune checkpoint inhibitors (ICI) revolutionized therapeutics for malignancy in their enhancement of cytotoxic T cell survival, but the resulting robust immune response engenders adverse gastrointestinal events in 1/3 of patients around 1-2 months after the second or third dose. ICI colitis may result in abscess, perforation, and death, and it is imperative to undergo early endoscopy with biopsy plus initiation of steroids or immunotherapy, especially in severe cases. Our patient presented with grade 3 diarrhea and severe immune-mediated colitis refractory to steroids and infliximab, though vedolizumab was effective in resolution.