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• W4382057625 abstract "Introduction: Tricohepatoenteric syndrome (THES) is a complex condition that confers a poor life prognosis. We describe a conventional multivisceral transplant (MVT) in a patient with THES type II (THES II), who developed multiple infectious complications and graft versus host disease (GVHD). Methods: Clinical, therapeutic and outcome of a MVT in a patient with THES II are analyzed. Results: A 7-year-old male patient who received a first conventional MVT due to the development of intestinal failure associated liver disease related to THES II is described. He had also severe colitis treated with tocilizumab and a humoral immunodeficiency with several studies of cellular immunity without alterations, so it was decided not to perform a hematopoietic stem cell transplant (HSCT) prior to the MVT. Transplant was performed with a non-depleting immunosuppression protocol with basiliximab, tacrolimus, and corticosteroid, with excellent immediate evolution, so he was discharged 46 days after MVT. Three months after MVT, he developed CMV and adenovirus infections, which required 3 admissions and multiple treatments, and the immunosuppression was changed to sirolimus at 5 months post-transplant. At 10 months post-transplant, being autonomous from the digestive point of view, he was admitted for chronic severe GVHD with ocular, cutaneous-mucous, and respiratory involvement. Intravenous corticotherapy at 2 mg/kg/day was iniciated without improvement, so ruxolitinib (2.5 mg/12h orally) was added, with clinical normalization and a decrease in the graft chimerism in peripheral blood from a maximum of 60% to 30%. During the admission, the patient presented a CMV reactivation in the blood which required treatment with foscarnet, and a dysfunction of the intestinal graft secondary to a norovirus infection, with a transient worsening of the GVHD clinical manifestations, probably due to malabsorption of ruxolitinib.13 months post-transplant, he showed a decrease in the three blood cell series, with bone marrow aplasia in a bone marrow biopsy. Ruxolitinib was discontinued because a potential bone marrow toxicity, with a reappearance of the GVHD without improvement of hematological manifestations. The patient died from P. Aeruginosa multiresistant sepsis at 15 months post-transplant. Conclusions: THES is a challenging condition. Normal cellular immunity studies may not rule out immunological abnormalities. Our patient developed complications suggesting of the existence of an underlying cellular immunodeficiency, due to the appearance of multiple infectious complications and a chronic severe GVHD. In tricohepatoenteric syndrome, regardless of the results of the immunological studies, it is necessary to carefully evaluate the possibility of performing an HSCT prior to the MVT." @default.
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• W4382057625 date "2023-06-27" @default.
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• W4382057625 title "127: CHRONIC GRAFT-VERSUS-HOST DISEASE AS THE MAIN COMPLICATION OF MULTIVISCERAL TRANSPLANT IN A PATIENT WITH TRICOHEPATOENTERIC SYNDROME TYPE II" @default.
• W4382057625 doi "https://doi.org/10.1097/01.tp.0000945964.66329.67" @default.
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