{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,30]],"date-time":"2026-10-30T09:44:31Z","timestamp":1793353471228,"version":"4.5.0"},"reference-count":9,"publisher":"Springer Science and Business Media LLC","issue":"1","license":[{"start":{"date-parts":[[2024,5,21]],"date-time":"2024-05-21T00:00:00Z","timestamp":1716249600000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"},{"start":{"date-parts":[[2024,5,21]],"date-time":"2024-05-21T00:00:00Z","timestamp":1716249600000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"}],"content-domain":{"domain":["link.springer.com"],"crossmark-restriction":false},"short-container-title":["BMC Ophthalmol"],"abstract":"<jats:title>Abstract<\/jats:title><jats:sec>\n                <jats:title>Background<\/jats:title>\n                <jats:p>Only seven cases of ocular <jats:italic>Spiroplasma<\/jats:italic> infection have been reported to date, all presenting as congenital cataracts with concomitant intraocular inflammation. We describe the first case of <jats:italic>Spiroplasma<\/jats:italic> infection initially presenting as a corneal infiltrate.<\/jats:p>\n              <\/jats:sec><jats:sec>\n                <jats:title>Case presentation<\/jats:title>\n                <jats:p>A 1-month-old girl was referred for a corneal infiltrate in the left eye. She presented in our hospital with unilateral keratouveitis. Examination showed a stromal corneal infiltrate and dense white keratic precipitates in the left eye. Herpetic keratouveitis was suspected and intravenous acyclovir therapy was initiated. Two weeks later, the inflammation in the left eye persisted and was also noticed in the right eye. Acute angle-closure glaucoma and a cataract with dilated iris vessels extending onto the anterior lens capsule developed in the left eye. The inflammation resolved after treatment with azithromycin. Iridectomy, synechiolysis and lensectomy were performed. Bacterial metagenomic sequencing (<jats:italic>16\u00a0S rRNA<\/jats:italic>) and transmission electron microscopy revealed <jats:italic>Spiroplasma ixodetis<\/jats:italic> species in lens aspirates and biopsy. Consequently, a diagnosis of bilateral <jats:italic>Spiroplasma<\/jats:italic> uveitis was made.<\/jats:p>\n              <\/jats:sec><jats:sec>\n                <jats:title>Conclusions<\/jats:title>\n                <jats:p>In cases of congenital cataract with concomitant intraocular inflammation, <jats:italic>Spiroplasma<\/jats:italic> infection should be considered. The purpose of this case report is to raise awareness of congenital <jats:italic>Spiroplasma<\/jats:italic> infection as a cause of severe keratouveitis, cataract and angle-closure glaucoma in newborns. Performing molecular testing on lens aspirates is essential to confirm diagnosis. Systemic macrolides are suggested as the mainstay of treatment.<\/jats:p>\n              <\/jats:sec>","DOI":"10.1186\/s12886-024-03480-z","type":"journal-article","created":{"date-parts":[[2024,5,21]],"date-time":"2024-05-21T08:01:31Z","timestamp":1716278491000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":7,"title":["Spiroplasma infection as a cause of severe congenital keratouveitis, cataract and glaucoma"],"prefix":"10.1186","volume":"24","author":[{"given":"Helena","family":"Van Haecke","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Dimitri","family":"Roels","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Fanny","family":"Nerinckx","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Heidi","family":"Schaballie","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Petra","family":"Schelstraete","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Linos","family":"Vandekerckhove","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jolien","family":"Van Cleemput","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Wim","family":"Van den Broeck","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Liesbeth","family":"Couck","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hannelore","family":"Hamerlinck","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Stien","family":"Vandendriessche","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jerina","family":"Boelens","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Inge","family":"Joniau","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2024,5,21]]},"reference":[{"key":"3480_CR1","doi-asserted-by":"publisher","first-page":"589279","DOI":"10.3389\/fmicb.2020.589279","volume":"11","author":"S Harne","year":"2020","unstructured":"Harne S, Gayathri P, B\u00e9ven L. Exploring Spiroplasma Biology: opportunities and challenges. Front Microbiol. 2020;11:589279.","journal-title":"Front Microbiol"},{"issue":"2","key":"3480_CR2","doi-asserted-by":"publisher","first-page":"719","DOI":"10.1128\/JCM.02841-14","volume":"53","author":"A Aquilino","year":"2015","unstructured":"Aquilino A, Masi\u00e1 M, L\u00f3pez P, Galiana AJ, Tovar J, Andr\u00e9s M, et al. First human systemic infection caused by Spiroplasma. J Clin Microbiol. 2015;53(2):719\u201321.","journal-title":"J Clin Microbiol"},{"issue":"12","key":"3480_CR3","doi-asserted-by":"publisher","first-page":"2382","DOI":"10.3201\/eid2412.180567","volume":"24","author":"N Etienne","year":"2018","unstructured":"Etienne N, Bret L, Le Brun C, Lecuyer H, Moraly J, Lanternier F, et al. Disseminated Spiroplasma apis infection in patient with agammaglobulinemia, France. Emerg Infect Dis. 2018;24(12):2382\u20136. https:\/\/doi.org\/10.3201\/eid2412.180567","journal-title":"Emerg Infect Dis"},{"issue":"9","key":"3480_CR4","doi-asserted-by":"publisher","first-page":"2511","DOI":"10.1111\/ajt.13254","volume":"15","author":"NJ Mueller","year":"2015","unstructured":"Mueller NJ, Tini GM, Weber A, Gaspert A, Husmann L, Bloemberg G, et al. Hepatitis from Spiroplasma sp. in an immunocompromised patient. Am J Transpl. 2015;15(9):2511\u20136. https:\/\/doi.org\/10.1111\/ajt.13254","journal-title":"Am J Transpl"},{"issue":"8","key":"3480_CR5","doi-asserted-by":"publisher","first-page":"1681","DOI":"10.3201\/eid2808.212524","volume":"28","author":"J Eimer","year":"2022","unstructured":"Eimer J, Fernstr\u00f6m L, Rohl\u00e9n L, Grankvist A, Loo K, Nyman E, et al. Spiroplasma ixodetis infections in Immunocompetent and immunosuppressed patients after Tick exposure, Sweden. Emerg Infect Dis. 2022;28(8):1681\u20135. https:\/\/doi.org\/10.3201\/eid2808.212524","journal-title":"Emerg Infect Dis"},{"issue":"5","key":"3480_CR6","doi-asserted-by":"publisher","first-page":"348","DOI":"10.1007\/s00417-002-0453-3","volume":"240","author":"B Lorenz","year":"2002","unstructured":"Lorenz B, Schroeder J, Reischl U. First evidence of an endogenous Spiroplasma sp. infection in humans manifesting as unilateral cataract associated with anterior uveitis in a premature baby. Graefes Arch Clin Exp Ophthalmol. 2002;240(5):348\u201353.","journal-title":"Graefes Arch Clin Exp Ophthalmol"},{"issue":"2","key":"3480_CR7","doi-asserted-by":"publisher","first-page":"340","DOI":"10.3201\/eid2602.191097","volume":"26","author":"A Matet","year":"2020","unstructured":"Matet A, Le Fl\u00e8che-Mat\u00e9os A, Doz F, Dureau P, Cassoux N. Ocular Spiroplasma ixodetis in newborns, France. Emerg Infect Dis. 2020;26(2):340\u20134.","journal-title":"Emerg Infect Dis"},{"key":"3480_CR8","doi-asserted-by":"publisher","first-page":"434","DOI":"10.1186\/s12886-021-02201-0","volume":"21","author":"N Farassat","year":"2021","unstructured":"Farassat N, Reich M, Serr A, K\u00fcchlin S, Erwemi M, Auw-H\u00e4drich C, et al. Spiroplasma species as a rare cause of congenital cataract and uveitis: a case series. BMC Ophthalmol. 2021;21:434. https:\/\/doi.org\/10.1186\/s12886-021-02201-0","journal-title":"BMC Ophthalmol"},{"issue":"3","key":"3480_CR9","first-page":"255","volume":"130","author":"GC Martin","year":"2023","unstructured":"Martin GC, Denier M, Le Fleche-Mateos A. Spiroplasma-induced Uveitis Mimicking Congenital Glaucoma Newborn. 2023;130(3):255.","journal-title":"Spiroplasma-induced Uveitis Mimicking Congenital Glaucoma Newborn"}],"container-title":["BMC Ophthalmology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1186\/s12886-024-03480-z.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/article\/10.1186\/s12886-024-03480-z\/fulltext.html","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1186\/s12886-024-03480-z.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2024,5,21]],"date-time":"2024-05-21T08:08:50Z","timestamp":1716278930000},"score":1,"resource":{"primary":{"URL":"https:\/\/bmcophthalmol.biomedcentral.com\/articles\/10.1186\/s12886-024-03480-z"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2024,5,21]]},"references-count":9,"journal-issue":{"issue":"1","published-online":{"date-parts":[[2024,12]]}},"alternative-id":["3480"],"URL":"https:\/\/doi.org\/10.1186\/s12886-024-03480-z","relation":{},"ISSN":["1471-2415"],"issn-type":[{"value":"1471-2415","type":"electronic"}],"subject":[],"published":{"date-parts":[[2024,5,21]]},"assertion":[{"value":"13 February 2024","order":1,"name":"received","label":"Received","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"9 May 2024","order":2,"name":"accepted","label":"Accepted","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"21 May 2024","order":3,"name":"first_online","label":"First Online","group":{"name":"ArticleHistory","label":"Article History"}},{"order":1,"name":"Ethics","group":{"name":"EthicsHeading","label":"Declarations"}},{"value":"Not applicable.","order":2,"name":"Ethics","group":{"name":"EthicsHeading","label":"Ethics approval and consent to participate"}},{"value":"Written informed consent was obtained from the parents of the patient for publication of this case report.","order":3,"name":"Ethics","group":{"name":"EthicsHeading","label":"Consent for publication"}},{"value":"The authors have no competing interests.","order":4,"name":"Ethics","group":{"name":"EthicsHeading","label":"Competing interests"}}],"article-number":"217"}}