{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,20]],"date-time":"2026-08-20T15:37:00Z","timestamp":1787240220302,"version":"build-2736575974"},"reference-count":26,"publisher":"SAGE Publications","issue":"3","license":[{"start":{"date-parts":[[2019,9,11]],"date-time":"2019-09-11T00:00:00Z","timestamp":1568160000000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/journals.sagepub.com\/page\/policies\/text-and-data-mining-license"}],"content-domain":{"domain":["journals.sagepub.com"],"crossmark-restriction":true},"short-container-title":["Pediatr Dev Pathol"],"published-print":{"date-parts":[[2020,6]]},"abstract":"<jats:sec>\n                    <jats:title>Background<\/jats:title>\n                    <jats:p>There are only 15 reported hepatic epidermoid cysts; they include patients presenting congenitally through adulthood, with varied speculations about pathogenesis. Aside from recently reported pancytokeratin staining, no other descriptions have included immunohistochemistry. Splenic epidermoid cysts were recently characterized as positive for HBME-1, p63, CEA, CK7 (luminal), and CK19. We interrogate 2 hepatic epidermoid cysts with a broad panel of immunohistochemistry, with the aim of elucidating histogenesis.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>Archives were searched for \u201cliver,\u201d \u201chepatic,\u201d and \u201ccyst.\u201d Hepatic cysts lined by squamous epithelium were included. Clinical records, macroscopic findings, and hematoxylin and eosin and immunohistochemically stained slides were reviewed.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>We identified 2 patients with epidermoid cysts of the liver, first detected on antenatal ultrasound. Both were females and asymptomatic; neither had other congenital abnormalities. Cysts enlarged slowly after birth. Resection was at ages 2 and 6 months, done to avoid potentially more difficult surgery in the future. Cysts were unilocular (4.8\u2009cm) and multilocular (7.0\u2009cm). Both were lined by stratified nonkeratinizing squamous to focally transitional-like epithelium and surrounded by paucicellular fibrous stroma. In the multilocular cyst, hepatocytes and fibrous stroma populated septa. Epithelium was positive for HBME-1, p63, CK19, CEA, Cam5.2, and CK7, negative for EMA, D2-40, WT-1, calretinin, and Ca19-9. Cytogenetic analysis of one showed a normal female karyotype. During the study period, 22 other pediatric liver cysts were diagnosed.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusion<\/jats:title>\n                    <jats:p>Hepatic epidermoid cyst is a distinct entity, rare but nevertheless constituting 8% of pediatric hepatic cysts at our institution. It is characterized by intrauterine onset and growth roughly commensurate with that of the fetus\/infant; it is apparently unsyndromic. It may be unilocular or multilocular. It stains for an array of epithelial markers as well as HBME-1. Strong immunohistochemical overlap with splenic epidermoid cyst points to a shared pathogenesis and detracts from hypotheses that hepatic epidermoid cysts derive from hepatic elements.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1177\/1093526619875237","type":"journal-article","created":{"date-parts":[[2019,9,11]],"date-time":"2019-09-11T20:26:11Z","timestamp":1568233571000},"page":"181-188","update-policy":"https:\/\/doi.org\/10.1177\/sage-journals-update-policy","source":"Crossref","is-referenced-by-count":3,"title":["Congenital Epidermoid Cyst of the Liver: A Rare Entity Characterized by Antenatal Onset, Slow Postnatal Growth, and Consistent Histologic and Immunohistologic Features"],"prefix":"10.1177","volume":"23","author":[{"given":"Matthew K","family":"Morrow","sequence":"first","affiliation":[{"name":"Department of Pathology, Boston Children\u2019s Hospital, Boston, Massachusetts"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Aofei","family":"Li","sequence":"additional","affiliation":[{"name":"Department of Pathology, Boston Children\u2019s Hospital, Boston, Massachusetts"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Antonio R","family":"Perez-Atayde","sequence":"additional","affiliation":[{"name":"Department of Pathology, Boston Children\u2019s Hospital, Boston, Massachusetts"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sara O","family":"Vargas","sequence":"additional","affiliation":[{"name":"Department of Pathology, Boston Children\u2019s Hospital, Boston, Massachusetts"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"179","published-online":{"date-parts":[[2019,9,11]]},"reference":[{"key":"bibr1-1093526619875237","doi-asserted-by":"publisher","DOI":"10.1001\/archsurg.1940.01210020220022"},{"issue":"5","key":"bibr2-1093526619875237","first-page":"607","volume":"103","author":"Dockerty MB","year":"1956","journal-title":"Surg Gynecol Obstet"},{"issue":"5","key":"bibr3-1093526619875237","first-page":"687","volume":"61","author":"Clark DD","year":"1967","journal-title":"Surgery"},{"key":"bibr4-1093526619875237","doi-asserted-by":"publisher","DOI":"10.1016\/S0022-3468(83)80092-X"},{"key":"bibr5-1093526619875237","doi-asserted-by":"publisher","DOI":"10.1007\/BF00497819"},{"key":"bibr6-1093526619875237","doi-asserted-by":"publisher","DOI":"10.1016\/j.jpedsurg.2005.08.038"},{"key":"bibr7-1093526619875237","doi-asserted-by":"publisher","DOI":"10.1016\/j.jpedsurg.2006.10.024"},{"key":"bibr8-1093526619875237","doi-asserted-by":"crossref","unstructured":"Allan M, Asimakidou M, Davenport M. Antenatally-detected liver cysts: causes and characteristics, indications for intervention [published online ahead of print May 1, 2019].\n                      J Pediatr Surg\n                      . doi:10.1016\/j.jpedsurg.2019.03.023.","DOI":"10.1016\/j.jpedsurg.2019.03.023"},{"issue":"5","key":"bibr9-1093526619875237","doi-asserted-by":"crossref","first-page":"247","DOI":"10.1016\/S0210-5705(01)70165-3","volume":"24","author":"Fern\u00e1ndez-Castroagud\u00edn J","year":"2001","journal-title":"Gastroenterol Hepatol"},{"key":"bibr10-1093526619875237","doi-asserted-by":"publisher","DOI":"10.1002\/1097-0142(197611)38:5<2002::AID-CNCR2820380523>3.0.CO;2-9"},{"issue":"4","key":"bibr11-1093526619875237","first-page":"426","volume":"83","author":"Lynch MJ","year":"1988","journal-title":"Am J Gastroenterol"},{"key":"bibr12-1093526619875237","doi-asserted-by":"publisher","DOI":"10.1097\/00004728-199501000-00025"},{"key":"bibr13-1093526619875237","doi-asserted-by":"publisher","DOI":"10.1007\/s10620-006-8049-0"},{"issue":"1","key":"bibr14-1093526619875237","first-page":"53","volume":"3","author":"Lakshmi K","year":"2015","journal-title":"Int J Res Health Sci Int"},{"key":"bibr15-1093526619875237","doi-asserted-by":"publisher","DOI":"10.3109\/15513819509026977"},{"key":"bibr16-1093526619875237","doi-asserted-by":"publisher","DOI":"10.1186\/1471-2407-6-10"},{"key":"bibr17-1093526619875237","doi-asserted-by":"publisher","DOI":"10.1016\/j.humpath.2014.06.016"},{"key":"bibr18-1093526619875237","doi-asserted-by":"publisher","DOI":"10.1038\/modpathol.3800163"},{"key":"bibr19-1093526619875237","unstructured":"Biocare Medical.\n                      HBME-1 Prediluted Monoclonal Antibody\n                      . Pacheco, CA: Biocare Medical. https:\/\/biocare.net\/wp-content\/uploads\/3177.pdf. Published 2016. Accessed December 20, 2018."},{"key":"bibr20-1093526619875237","doi-asserted-by":"publisher","DOI":"10.1097\/PAS.0b013e3182a0218f"},{"issue":"8","key":"bibr21-1093526619875237","doi-asserted-by":"crossref","first-page":"1304","DOI":"10.5858\/2007-131-1304-ICOAGC","volume":"131","author":"Lisovsky M","year":"2007","journal-title":"Arch Pathol Lab Med"},{"key":"bibr22-1093526619875237","doi-asserted-by":"publisher","DOI":"10.1007\/s00428-017-2139-6"},{"key":"bibr23-1093526619875237","doi-asserted-by":"publisher","DOI":"10.1080\/00015458.2013.11680939"},{"key":"bibr24-1093526619875237","doi-asserted-by":"publisher","DOI":"10.1007\/BF00193502"},{"key":"bibr25-1093526619875237","unstructured":"Chisholm K, Puder M, Vargas SO. Splenic cysts in the pediatric population [abstract]. (Society for Pediatric Pathology Spring 2016 Abstracts-Seattle, Washington.)\n                      Pediatr Devel Pathol\n                      . http:\/\/www.pedpath.org\/doi\/pdf\/10.2350\/16-03-1793-MISC.1 Published 2016. Accessed August 28, 2019."},{"key":"bibr26-1093526619875237","doi-asserted-by":"crossref","unstructured":"Vargas SO, Perez-Atayde AR. Letter to the editor. Comment on: Apellaniz-Ruiz M, Segni M, Kettwig M, Gl\u00fcer S, Pelletier D, Nguyen VH, Wagener R, L\u00f3pez C, Muchantef K, Bouron-Dal Soglio D, Sabbaghian N, Wu MK, Zannella S, Fabian MR, Siebert R, Menke J, Priest JR, Foulkes WD. Mesenchymal hamartoma of the liver and DICER1 syndrome.\n                      N Engl J Med\n                      . 2019; 381(6): 586\u2013587.","DOI":"10.1056\/NEJMc1907523"}],"container-title":["Pediatric and Developmental Pathology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/journals.sagepub.com\/doi\/pdf\/10.1177\/1093526619875237","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/journals.sagepub.com\/doi\/full-xml\/10.1177\/1093526619875237","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/journals.sagepub.com\/doi\/pdf\/10.1177\/1093526619875237","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,5,2]],"date-time":"2026-05-02T04:06:06Z","timestamp":1777694766000},"score":1,"resource":{"primary":{"URL":"https:\/\/journals.sagepub.com\/doi\/10.1177\/1093526619875237"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2019,9,11]]},"references-count":26,"journal-issue":{"issue":"3","published-print":{"date-parts":[[2020,6]]}},"alternative-id":["10.1177\/1093526619875237"],"URL":"https:\/\/doi.org\/10.1177\/1093526619875237","relation":{},"ISSN":["1093-5266","1615-5742"],"issn-type":[{"value":"1093-5266","type":"print"},{"value":"1615-5742","type":"electronic"}],"subject":[],"published":{"date-parts":[[2019,9,11]]}}}