{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,7,30]],"date-time":"2025-07-30T16:00:33Z","timestamp":1753891233173,"version":"3.41.2"},"reference-count":19,"publisher":"Frontiers Media SA","license":[{"start":{"date-parts":[[2024,2,14]],"date-time":"2024-02-14T00:00:00Z","timestamp":1707868800000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":["frontiersin.org"],"crossmark-restriction":true},"short-container-title":["Front. Med."],"abstract":"<jats:sec><jats:title>Background<\/jats:title><jats:p>Wilson\u2019s disease (WD) is not an uncommon genetic disease in clinical practice. However, the current WD therapies have limitations. The effectiveness of stem cell therapy in treating WD has yet to be verified, although a few animal studies have shown that stem cell transplantation could partially correct the abnormal metabolic phenotype of WD. In this case report, we present the therapeutic effect of human amniotic fluid containing stem cells in one WD patient.<\/jats:p><\/jats:sec><jats:sec><jats:title>Case presentation<\/jats:title><jats:p>A 22-year-old Chinese woman was diagnosed with WD 1\u2009year ago in 2019. The available drugs were not effective in managing the progressive neuropsychiatric symptoms. We treated the patient with pre-cultured human amniotic fluid containing stem cells. Amniotic fluid was collected from pregnant women who underwent induced labor at a gestational age of 19\u201326\u2009weeks, and then, the fluid was cultured for 2\u2009h to allow stem cell expansion. Cultured amniotic fluid that contained amniotic fluid derived stem cells (AFSC) in the range of approximately 2.8\u20135.5\u2009\u00d7\u200910<jats:sup>4<\/jats:sup>\/ml was administrated by IV infusion at a rate of 50\u201370 drops per minute after filtration with a 300-mu nylon mesh. Before the infusion of amniotic fluid, low-molecular-weight heparin and dexamethasone were successively administrated. The patient received a total of 12 applications of amniotic fluid from different pregnant women, and the treatment interval depended on the availability of amniotic fluid. The neuropsychiatric symptoms gradually improved after the stem cell treatment. Dystonia, which included tremor, chorea, dysphagia, dysarthria, and drooling, almost disappeared after 1.5\u2009years of follow-up. The Unified Wilson\u2019s Disease Rating Scale score of the patient decreased from 72 to 10. Brain magnetic resonance imaging (MRI) showed a reduction in the lesion area and alleviation of damage in the central nervous system, along with a partial recovery of the lesion to the normal condition. The serum ceruloplasmin level was elevated from undetectable to 30.8\u2009mg\/L, and the 24-h urinary copper excretion decreased from 171 to 37\u2009\u03bcg. In addition, amniotic fluid transplantation also alleviates hematopoietic disorders. There were no adverse reactions during or after amniotic fluid administration.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusion<\/jats:title><jats:p>Amniotic fluid administration, through which stem cells were infused, significantly improves the clinical outcomes in the WD patient, and the finding may provide a novel approach for managing WD effectively.<\/jats:p><\/jats:sec>","DOI":"10.3389\/fmed.2024.1297457","type":"journal-article","created":{"date-parts":[[2024,2,14]],"date-time":"2024-02-14T04:25:11Z","timestamp":1707884711000},"update-policy":"https:\/\/doi.org\/10.3389\/crossmark-policy","source":"Crossref","is-referenced-by-count":0,"title":["Case report: Treatment of Wilson\u2019s disease by human amniotic fluid administration"],"prefix":"10.3389","volume":"11","author":[{"given":"Libin","family":"Liang","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Hong","family":"Xin","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Xueyan","family":"Shen","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Yanping","family":"Xu","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Lansen","family":"Zhang","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Dehui","family":"Liu","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Liling","family":"Zhao","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Xinglong","family":"Tong","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"1965","published-online":{"date-parts":[[2024,2,14]]},"reference":[{"key":"ref1","doi-asserted-by":"publisher","first-page":"e0150","DOI":"10.1097\/HC9.0000000000000150","article-title":"Wilson disease: a summary of the updated AASLD practice guidance","volume":"7","author":"Alkhouri","year":"2023","journal-title":"Hepatol Commun"},{"key":"ref2","doi-asserted-by":"publisher","first-page":"417","DOI":"10.1016\/j.ncl.2020.01.005","article-title":"Wilson disease: an overview and approach to management","volume":"38","author":"Mulligan","year":"2020","journal-title":"Neurol Clin"},{"key":"ref3","doi-asserted-by":"publisher","first-page":"211","DOI":"10.1016\/B978-0-444-63625-6.00018-5","article-title":"Symptomatic treatment of neurologic symptoms in Wilson disease","volume":"142","author":"Litwin","year":"2017","journal-title":"Handb Clin Neurol"},{"key":"ref4","doi-asserted-by":"publisher","first-page":"765","DOI":"10.3727\/000000004783983341","article-title":"The potential of bone marrow stem cells to correct liver dysfunction in a mouse model of Wilson\u2019s disease","volume":"13","author":"Allen","year":"2004","journal-title":"Cell Transplant"},{"key":"ref5","doi-asserted-by":"publisher","first-page":"e111425","DOI":"10.1371\/journal.pone.0111425","article-title":"Transplantation of ATP7B-transduced bone marrow mesenchymal stem cells decreases copper overload in rats","volume":"9","author":"Chen","year":"2014","journal-title":"PLoS One"},{"key":"ref6","doi-asserted-by":"publisher","first-page":"712","DOI":"10.2174\/1574888X16666211006111556","article-title":"Stem cells treatment for Wilson disease","volume":"17","author":"Tang","year":"2022","journal-title":"Curr Stem Cell Res Ther"},{"key":"ref7","doi-asserted-by":"publisher","first-page":"7","DOI":"10.2174\/1574888X16666211210143640","article-title":"Amniotic fluid stem cells: what they are and what they can become","volume":"18","author":"Rosner","year":"2023","journal-title":"Curr Stem Cell Res Ther"},{"key":"ref8","doi-asserted-by":"publisher","first-page":"e928047","DOI":"10.12659\/AOT.928047","article-title":"Amniotic fluid transplantation alleviates hematopoietic deficits in experimental rat aplastic Anemia","volume":"26","author":"Liang","year":"2021","journal-title":"Ann Transplant"},{"key":"ref9","doi-asserted-by":"publisher","first-page":"1428","DOI":"10.1002\/hep.32805","article-title":"A multidisciplinary approach to the diagnosis and management of Wilson disease: executive summary of the 2022 practice guidance on Wilson disease from the American Association for the Study of Liver Diseases","volume":"77","author":"Schilsky","year":"2023","journal-title":"Hepatology"},{"key":"ref10","doi-asserted-by":"publisher","first-page":"81","DOI":"10.1111\/nyas.12437","article-title":"Clinical considerations for an effective medical therapy in Wilson\u2019s disease","volume":"1315","author":"Weiss","year":"2014","journal-title":"Ann N Y Acad Sci"},{"key":"ref11","doi-asserted-by":"publisher","first-page":"e38170","DOI":"10.7759\/cureus.38170","article-title":"Neurological-type Wilson disease: epidemiology, clinical manifestations, diagnosis, and management","volume":"15","author":"Kipker","year":"2023","journal-title":"Cureus"},{"key":"ref12","doi-asserted-by":"publisher","first-page":"100389","DOI":"10.1016\/j.jhepr.2021.100389","article-title":"CRISPR-targeted genome editing of human induced pluripotent stem cell-derived hepatocytes for the treatment of Wilson\u2019s disease","volume":"4","author":"Wei","year":"2022","journal-title":"JHEP Rep"},{"key":"ref13","doi-asserted-by":"publisher","first-page":"368","DOI":"10.1016\/S1726-4901(09)70389-2","article-title":"Potential function of amniotic fluid in fetal development\u2014novel insights by comparing the composition of human amniotic fluid with umbilical cord and maternal serum at mid and late gestation","volume":"72","author":"Tong","year":"2009","journal-title":"J Chinese Med Assoc"},{"key":"ref14","doi-asserted-by":"publisher","first-page":"1663","DOI":"10.1002\/stem.2553","article-title":"Concise review: amniotic fluid stem cells: the known, the unknown, and potential regenerative medicine applications","volume":"35","author":"Loukogeorgakis","year":"2017","journal-title":"Stem cells"},{"key":"ref15","doi-asserted-by":"publisher","first-page":"514","DOI":"10.1111\/nan.12341","article-title":"Brain iron accumulation in Wilson disease: a post mortem 7 tesla MRI - histopathological study","volume":"43","author":"Dusek","year":"2017","journal-title":"Neuropathol Appl Neurobiol"},{"key":"ref16","doi-asserted-by":"publisher","first-page":"606","DOI":"10.1016\/j.jcma.2013.07.006","article-title":"Amniotic fluid may act as a transporting pathway for signaling molecules and stem cells during the embryonic development of amniotes","volume":"76","author":"Tong","year":"2013","journal-title":"J Chinese Med Assoc"},{"key":"ref17","doi-asserted-by":"publisher","first-page":"100","DOI":"10.1038\/nbt1274","article-title":"Isolation of amniotic stem cell lines with potential for therapy","volume":"25","author":"De Coppi","year":"2007","journal-title":"Nat Biotechnol"},{"key":"ref18","doi-asserted-by":"publisher","first-page":"965","DOI":"10.14670\/HH-18-607","article-title":"Development of a new treatment for preterm birth complications using amniotic fluid stem cell therapy","volume":"38","author":"Abe","year":"2023","journal-title":"Histol Histopathol"},{"key":"ref19","doi-asserted-by":"publisher","first-page":"57","DOI":"10.2302\/kjm.2017-0019-IR","article-title":"Human amniotic fluid stem cells: therapeutic potential for perinatal patients with intractable neurological disease","volume":"67","author":"Ochiai","year":"2018","journal-title":"Keio J Med"}],"container-title":["Frontiers in Medicine"],"original-title":[],"link":[{"URL":"https:\/\/www.frontiersin.org\/articles\/10.3389\/fmed.2024.1297457\/full","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2024,2,14]],"date-time":"2024-02-14T04:25:13Z","timestamp":1707884713000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.frontiersin.org\/articles\/10.3389\/fmed.2024.1297457\/full"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2024,2,14]]},"references-count":19,"alternative-id":["10.3389\/fmed.2024.1297457"],"URL":"https:\/\/doi.org\/10.3389\/fmed.2024.1297457","relation":{},"ISSN":["2296-858X"],"issn-type":[{"type":"electronic","value":"2296-858X"}],"subject":[],"published":{"date-parts":[[2024,2,14]]},"article-number":"1297457"}}