{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,3]],"date-time":"2026-07-03T01:45:23Z","timestamp":1783043123384,"version":"3.54.6"},"reference-count":12,"publisher":"Wiley","issue":"6","license":[{"start":{"date-parts":[[2021,6,14]],"date-time":"2021-06-14T00:00:00Z","timestamp":1623628800000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Pediatric Transplantation"],"published-print":{"date-parts":[[2021,9]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:sec><jats:title>Background<\/jats:title><jats:p>While operational tolerance has been previously described in isolated intestinal transplant, reports of this phenomenon in combined liver\u2010intestine transplant are lacking.<\/jats:p><\/jats:sec><jats:sec><jats:title>Case Description<\/jats:title><jats:p>We detail a unique case of a patient who received a composite allograft including liver, pancreas, and small bowel due to short gut syndrome secondary to gastroschisis complicated by volvulus. The indication for transplantation was permanent dependence on total parenteral nutrition, end\u2010stage liver disease, recurrent sepsis, and persistent stomal variceal hemorrhage. The patient developed severe graft\u2010versus\u2010host disease with grade 3\u00a0skin involvement, ophthalmic, and pulmonary involvement with 53% donor T\u2010cell chimerism. She required aggressive therapy including high\u2010dose methylprednisolone, rituximab, cyclophosphamide, and alemtuzumab. Due to infection concerns following depletion of her lymphocytes, immunosuppression was discontinued with close surveillance of her allograft. Nearly 10\u00a0years later, the patient has continued off all immunosuppression without evidence of rejection or graft dysfunction and demonstrates immunocompetence with normal functional immune assays and development of appropriate live vaccination titers.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusion<\/jats:title><jats:p>This report of operational tolerance following pediatric composite liver\u2010pancreas\u2010intestine transplantation provides evidence that the complex immunologic balance in intestinal transplantation may on rare occasions favor immunosuppression reduction or even discontinuation. Future trials of immunosuppression minimization in this population may be warranted.<\/jats:p><\/jats:sec>","DOI":"10.1111\/petr.14069","type":"journal-article","created":{"date-parts":[[2021,6,14]],"date-time":"2021-06-14T14:05:37Z","timestamp":1623679537000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":6,"title":["Operational tolerance after pediatric composite liver\u2010pancreas\u2010intestine transplantation following severe graft\u2010versus\u2010host disease"],"prefix":"10.1111","volume":"25","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-7291-7561","authenticated-orcid":false,"given":"Julie","family":"Osborn","sequence":"first","affiliation":[{"name":"Division of Gastroenterology, Hepatology and Nutrition Cincinnati Children's Hospital Medical Center Cincinnati OH USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jaimie D.","family":"Nathan","sequence":"additional","affiliation":[{"name":"Division of Pediatric General and Thoracic Surgery Cincinnati Children\u2019s Hospital Medical Center Cincinnati OH USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Gregory","family":"Tiao","sequence":"additional","affiliation":[{"name":"Division of Pediatric General and Thoracic Surgery Cincinnati Children\u2019s Hospital Medical Center Cincinnati OH USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Maria","family":"Alonso","sequence":"additional","affiliation":[{"name":"Division of Pediatric General and Thoracic Surgery Cincinnati Children\u2019s Hospital Medical Center Cincinnati OH USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Samuel","family":"Kocoshis","sequence":"additional","affiliation":[{"name":"Division of Gastroenterology, Hepatology and Nutrition Cincinnati Children's Hospital Medical Center Cincinnati OH USA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2021,6,14]]},"reference":[{"key":"e_1_2_10_2_1","doi-asserted-by":"publisher","DOI":"10.1097\/MOT.0000000000000231"},{"key":"e_1_2_10_3_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jpedsurg.2011.04.016"},{"key":"e_1_2_10_4_1","doi-asserted-by":"publisher","DOI":"10.1586\/14737159.2016.1139455"},{"key":"e_1_2_10_5_1","doi-asserted-by":"publisher","DOI":"10.1111\/petr.13580"},{"key":"e_1_2_10_6_1","doi-asserted-by":"publisher","DOI":"10.1111\/ajt.16224"},{"key":"e_1_2_10_7_1","doi-asserted-by":"publisher","DOI":"10.1111\/ajt.13325"},{"key":"e_1_2_10_8_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.iac.2009.10.001"},{"key":"e_1_2_10_9_1","doi-asserted-by":"publisher","DOI":"10.1097\/TP.0000000000002711"},{"key":"e_1_2_10_10_1","doi-asserted-by":"publisher","DOI":"10.1097\/01.tp.0000219235.97036.9c"},{"key":"e_1_2_10_11_1","doi-asserted-by":"publisher","DOI":"10.1097\/01.TP.0000071362.99021.D9"},{"key":"e_1_2_10_12_1","doi-asserted-by":"publisher","DOI":"10.1097\/01.tp.0000174341.49741.8f"},{"key":"e_1_2_10_13_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jpedsurg.2011.04.016"}],"container-title":["Pediatric Transplantation"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/petr.14069","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/full-xml\/10.1111\/petr.14069","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/petr.14069","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,8,29]],"date-time":"2023-08-29T15:30:32Z","timestamp":1693323032000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1111\/petr.14069"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2021,6,14]]},"references-count":12,"journal-issue":{"issue":"6","published-print":{"date-parts":[[2021,9]]}},"alternative-id":["10.1111\/petr.14069"],"URL":"https:\/\/doi.org\/10.1111\/petr.14069","archive":["Portico"],"relation":{},"ISSN":["1397-3142","1399-3046"],"issn-type":[{"value":"1397-3142","type":"print"},{"value":"1399-3046","type":"electronic"}],"subject":[],"published":{"date-parts":[[2021,6,14]]},"assertion":[{"value":"2021-03-05","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2021-05-24","order":1,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2021-06-14","order":2,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}],"article-number":"e14069"}}