{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,12]],"date-time":"2026-07-12T06:09:59Z","timestamp":1783836599547,"version":"3.55.0"},"update-to":[{"DOI":"10.1371\/journal.pntd.0011612","type":"new_version","label":"New version","source":"publisher","updated":{"date-parts":[[2023,9,15]],"date-time":"2023-09-15T00:00:00Z","timestamp":1694736000000}}],"reference-count":113,"publisher":"Public Library of Science (PLoS)","issue":"9","license":[{"start":{"date-parts":[[2023,9,5]],"date-time":"2023-09-05T00:00:00Z","timestamp":1693872000000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"name":"Horizon 2020 Research and Innovation programme","award":["773830"],"award-info":[{"award-number":["773830"]}]},{"DOI":"10.13039\/501100003196","name":"Italian Ministry of Health","doi-asserted-by":"crossref","id":[{"id":"10.13039\/501100003196","id-type":"DOI","asserted-by":"crossref"}]}],"content-domain":{"domain":["www.plosntds.org"],"crossmark-restriction":false},"short-container-title":["PLoS Negl Trop Dis"],"abstract":"<jats:sec id=\"sec001\"><jats:title>Background<\/jats:title><jats:p>Human cystic echinococcosis (CE) is a zoonotic parasitic infection caused by the larval stage of the species belonging to the<jats:italic>Echinococcus granulosus sensu lato<\/jats:italic>(<jats:italic>s<\/jats:italic>.<jats:italic>l<\/jats:italic>.) complex. Parasitic cysts causing human CE are mainly localized in the liver and in the lungs. In a smaller number of cases, larvae may establish in any organ or tissue, including the central nervous system (CNS). Cerebral CE (CCE) is rare but poses serious clinical challenges.<\/jats:p><\/jats:sec><jats:sec id=\"sec002\"><jats:title>Methods<\/jats:title><jats:p>This study presents a case of CCE in a child living in the countryside near Rome (Italy), along with a comparative molecular analysis of the isolated cyst specimens from the patient and sheep of local farms. We also systematically searched the literature to summarize the most relevant epidemiological and clinical aspects of this uncommon localization.<\/jats:p><\/jats:sec><jats:sec id=\"sec003\"><jats:title>Findings<\/jats:title><jats:p>The comparative molecular analysis confirmed that the infection was caused by<jats:italic>E<\/jats:italic>.<jats:italic>granulosus sensu stricto<\/jats:italic>(<jats:italic>s<\/jats:italic>.<jats:italic>s<\/jats:italic>.) (G3 genotype), and most likely acquired in the family farm.<\/jats:p><jats:p>The literature search identified 2,238 cases of CCE. In 80.51% of cases, brain was the only localization and single CCE cysts were present in 84.07% of cases. Mean patients\u2019 age was 20 years and 70.46% were children. Cyst rupture was reported in 12.96% and recurrence of CCE after treatment in 9.61% of cases. Permanent disability was reported in 7.86% of cases, while death occurred in 6.21%. In case series reporting all CE localization, CCE represented 1.5% of all CE cases. In the few reports that identified at molecular level the CCE cyst,<jats:italic>E<\/jats:italic>.<jats:italic>granulosus s<\/jats:italic>.<jats:italic>s<\/jats:italic>. was found in 40% and<jats:italic>E<\/jats:italic>.<jats:italic>canadensis<\/jats:italic>in 60% of cases.<\/jats:p><\/jats:sec><jats:sec id=\"sec004\"><jats:title>Conclusions<\/jats:title><jats:p>We report a rare case of CCE and evidenced the probable local origin of infection. The proportions of CE cases with uncommon localizations and with high impact on patients\u2019 lives have been globally neglected and should be included in the computation of the global burden of CE.<\/jats:p><\/jats:sec>","DOI":"10.1371\/journal.pntd.0011612","type":"journal-article","created":{"date-parts":[[2023,9,5]],"date-time":"2023-09-05T17:28:47Z","timestamp":1693934927000},"page":"e0011612","update-policy":"https:\/\/doi.org\/10.1371\/journal.pntd.corrections_policy","source":"Crossref","is-referenced-by-count":24,"title":["Primary cerebral cystic echinococcosis in a child from Roman countryside: Source attribution and scoping review of cases from the literature"],"prefix":"10.1371","volume":"17","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-9183-7591","authenticated-orcid":true,"given":"Adriano","family":"Casulli","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Stefania","family":"Pane","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Franco","family":"Randi","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Paola","family":"Scaramozzino","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Andrea","family":"Carvelli","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Carlo Efisio","family":"Marras","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Andrea","family":"Carai","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Azzurra","family":"Santoro","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Federica","family":"Santolamazza","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Francesca","family":"Tamarozzi","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lorenza","family":"Putignani","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"340","published-online":{"date-parts":[[2023,9,5]]},"reference":[{"key":"pntd.0011612.ref001","author":"World Health Organization","year":"2021","journal-title":"Ending the neglect to attain the Sustainable Development Goals: a road map for neglected tropical diseases 2021\u20132030"},{"key":"pntd.0011612.ref002","doi-asserted-by":"crossref","first-page":"e0009373","DOI":"10.1371\/journal.pntd.0009373","volume":"15","author":"A. 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