{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,2,24]],"date-time":"2026-02-24T21:46:34Z","timestamp":1771969594011,"version":"3.50.1"},"reference-count":14,"publisher":"Springer Science and Business Media LLC","issue":"1","license":[{"start":{"date-parts":[[2021,6,29]],"date-time":"2021-06-29T00:00:00Z","timestamp":1624924800000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"},{"start":{"date-parts":[[2021,6,29]],"date-time":"2021-06-29T00:00:00Z","timestamp":1624924800000},"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":["Ital J Pediatr"],"published-print":{"date-parts":[[2021,12]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:sec>\n                <jats:title>Background<\/jats:title>\n                <jats:p>Sactosalpinx means a collection of fluid (serum, blood or pus) in the fallopian tube. CAH (Congenital Adrenal Hyperplasia) is a typical 46XX DSD (Disorder of Sex Development) due to a steroidogenic enzymatic defect. Both conditions are rare and can lead to reduced fertility rate.<\/jats:p>\n              <\/jats:sec><jats:sec>\n                <jats:title>Case presentation<\/jats:title>\n                <jats:p>We describe two post-menarche virgin girls with CAH who were hospitalized for acute abdomen due to laparoscopically confirmed sactosalpinx. Case 1 recovered after conservative management, case 2 after a second-look and bilateral salpingectomy. The first case consisted of right sactosalpinx and previous peritonitis reported; the second one of bilateral symptomatic pyosalpinx and previous vaginal stenosis. Recurrent abdominal pain persisted at follow-up in Case 1: post-operative MRI (Magnetic Resonance Imaging) showed bilateral hydrosapinx that disappeared at a following ultrasound scan control. Follow-up was uneventful 36 months after surgery in Case 2, except for the surgical revision of the vaginal introitus.<\/jats:p>\n              <\/jats:sec><jats:sec>\n                <jats:title>Conclusions<\/jats:title>\n                <jats:p>CAH-sactosalpinx association is a very rare but not negligible event. We suggest a conservative approach for sactosalpinx if tubal and\/or ovary torsion can be excluded. Pyosalpinx is more challenging to treat, but during pediatric age we suggest starting with a conservative approach, especially in patients with CAH who have a potential low fertility rate. Careful gynecological follow-up after menarche is recommended to rule out any further causes of infertility.<\/jats:p>\n              <\/jats:sec>","DOI":"10.1186\/s13052-021-01089-2","type":"journal-article","created":{"date-parts":[[2021,6,29]],"date-time":"2021-06-29T13:02:46Z","timestamp":1624971766000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":3,"title":["Bilateral sactosalphinx and congenital adrenal hyperplasia: case report on two rare conditions in two virgin girls"],"prefix":"10.1186","volume":"47","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-4421-8194","authenticated-orcid":false,"given":"Maria-Grazia","family":"Scarpa","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Marianna","family":"Iaquinto","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Daniela","family":"Codrich","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"J\u00fcrgen","family":"Schleef","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"297","published-online":{"date-parts":[[2021,6,29]]},"reference":[{"key":"1089_CR1","doi-asserted-by":"publisher","first-page":"191520","DOI":"10.1155\/2010\/191520","volume":"2010","author":"SA Stout","year":"2010","unstructured":"Stout SA, Litvak M, Robbins NM, Sandberg DE. Congenital adrenal hyperplasia: classification of studies employing psychological endpoints. Int J Pediatr Endocrinol. 2010;2010:191520. https:\/\/doi.org\/10.1155\/2010\/191520.","journal-title":"Int J Pediatr Endocrinol"},{"key":"1089_CR2","first-page":"231","volume":"3","author":"A Prader","year":"1954","unstructured":"Prader A. Der genitalbefund beim pseudohermaphroditismus feminus des kongenitalen androgenotalen syndroms. Helvetica Pediatr Acta. 1954;3:231\u201348.","journal-title":"Helvetica Pediatr Acta"},{"issue":"6 Pt 2","key":"1089_CR3","doi-asserted-by":"publisher","first-page":"2347","DOI":"10.1097\/00005392-200106001-00032","volume":"165","author":"R Jenak","year":"2001","unstructured":"Jenak R, Ludwikowski B, Gonzalez R. Total urogenital sinus mobilization: a modified perineal approach for feminizing genitoplasty and urogenital sinus repair. J Urol. 2001;165(6 Pt 2):2347\u20139. https:\/\/doi.org\/10.1097\/00005392-200106001-00032.","journal-title":"J Urol"},{"issue":"4","key":"1089_CR4","doi-asserted-by":"publisher","first-page":"351","DOI":"10.1016\/j.jpurol.2006.04.002","volume":"2","author":"RC Rink","year":"2006","unstructured":"Rink RC, Metcalfe PD, Kaefer MA, Casale AJ, Meldrum KK, Cain MP. Partial urogenital mobilization: a limited proximal dissection. J Pediatr Urol. 2006;2(4):351\u20136. https:\/\/doi.org\/10.1016\/j.jpurol.2006.04.002.","journal-title":"J Pediatr Urol"},{"issue":"5","key":"1089_CR5","doi-asserted-by":"publisher","first-page":"2199","DOI":"10.1016\/j.juro.2006.07.063","volume":"176","author":"LHP Braga","year":"2006","unstructured":"Braga LHP, Lorenzo AJ, Tatsuo ES, Silva IN, Pippi Salle JL. Prospective evaluation of feminizing Genitoplasty using partial urogenital sinus mobilization for congenital adrenal hyperplasia. Urol. 2006;176(5):2199\u2013204. https:\/\/doi.org\/10.1016\/j.juro.2006.07.063.","journal-title":"Urol"},{"issue":"3","key":"1089_CR6","doi-asserted-by":"publisher","first-page":"1024","DOI":"10.1016\/j.juro.2011.10.162","volume":"187","author":"JL Pippi-Salle","year":"2012","unstructured":"Pippi-Salle JL, Lorenzo AJ, Jesus LE, Leslie B, et al. Surgical treatment of high urogenital sinuses using the anterior sagittal transrectal approach: a useful strategy to optimize exposure and outcomes. J Urol. 2012;187(3):1024\u201331. https:\/\/doi.org\/10.1016\/j.juro.2011.10.162.","journal-title":"J Urol"},{"issue":"5","key":"1089_CR7","doi-asserted-by":"publisher","first-page":"1180","DOI":"10.1016\/j.urology.2012.01.009","volume":"79","author":"J Fuchs","year":"2012","unstructured":"Fuchs J, Warmann SW, Seitz G, Schafer J, Schroder M, Obermayr F. Laparoscopically assisted vaginal pull-through for high urogenital sinus: a new surgical technique. Urology. 2012;79(5):1180\u20133. https:\/\/doi.org\/10.1016\/j.urology.2012.01.009.","journal-title":"Urology"},{"key":"1089_CR8","doi-asserted-by":"publisher","first-page":"407","DOI":"10.1016\/j.jpurol.2018.03.020","volume":"14","author":"EJ Lisieux","year":"2018","unstructured":"Lisieux EJ. Feminizing genitoplasties: where are we now? J Pediatr Urol. 2018;14:407\u201315.","journal-title":"J Pediatr Urol"},{"issue":"2","key":"1089_CR9","doi-asserted-by":"publisher","first-page":"495","DOI":"10.1148\/rg.292085070","volume":"29","author":"M Kim","year":"2009","unstructured":"Kim M, Sung ER, Soon NO, et al. MR imaging findings of Hydrosalpinx: a comprehensive review. Radiographics. 2009;29(2):495\u2013507. https:\/\/doi.org\/10.1148\/rg.292085070.","journal-title":"Radiographics"},{"issue":"12","key":"1089_CR10","doi-asserted-by":"publisher","first-page":"1330","DOI":"10.1007\/s00247-008-1034-3","volume":"38","author":"L Merlini","year":"2008","unstructured":"Merlini L, Anooshiravani M, Vunda A, Borzani I, Napolitano M, Hanquinet S. Noninflammatory fallopian tube pathology in children. Pediatr Radiol. 2008;38(12):1330\u20137. https:\/\/doi.org\/10.1007\/s00247-008-1034-3.","journal-title":"Pediatr Radiol"},{"issue":"6","key":"1089_CR11","doi-asserted-by":"publisher","first-page":"e203","DOI":"10.1016\/j.jpag.2015.06.009","volume":"28","author":"M Demma","year":"2015","unstructured":"Demma M, Cabral I, Siqueira LM. Hydrosalpinx in Postmenarchal nonsexually active girls: a review of 6 cases in a Children\u2019s hospital. J Pediatr Adolesc Gynecol. 2015;28(6):e203\u20137. https:\/\/doi.org\/10.1016\/j.jpag.2015.06.009.","journal-title":"J Pediatr Adolesc Gynecol"},{"key":"1089_CR12","doi-asserted-by":"publisher","first-page":"40","DOI":"10.1016\/j.ejogrb.2015.09.042","volume":"195","author":"Z Kazmi","year":"2015","unstructured":"Kazmi Z, Gupta S. Best practise in management of pediatric and adolescent hydrosalpinges: a systematic review. Eur J Obstet Gynecol Reprod Biol. 2015;195:40\u201351. https:\/\/doi.org\/10.1016\/j.ejogrb.2015.09.042.","journal-title":"Eur J Obstet Gynecol Reprod Biol"},{"issue":"1","key":"1089_CR13","doi-asserted-by":"publisher","first-page":"6","DOI":"10.1016\/j.jpag.2014.01.106","volume":"28","author":"G \u00d6cal","year":"2015","unstructured":"\u00d6cal G, Berberoglu M, Siklar Z, et al. Clinical review of 95 patients with 46,XX disorders of sex development based on the new Chicago classification. J Pediatr Adolesc Gynecol. 2015;28(1):6\u201311. https:\/\/doi.org\/10.1016\/j.jpag.2014.01.106.","journal-title":"J Pediatr Adolesc Gynecol"},{"issue":"7","key":"1089_CR14","doi-asserted-by":"publisher","first-page":"1425","DOI":"10.1016\/j.jpedsurg.2011.01.033","volume":"46","author":"SA Boukaidi","year":"2011","unstructured":"Boukaidi SA, Delotte J, Steyaert H, Valla JS, Sattonet C, Bouaziz J, et al. Thirteen cases of isolated tubal torsions associated with hydrosalpinx in children and adolescent, proposal for conservative management: retrospective review and literature survey. J Pediatr Surg. 2011;46(7):1425\u2013143. https:\/\/doi.org\/10.1016\/j.jpedsurg.2011.01.033.","journal-title":"J Pediatr Surg"}],"container-title":["Italian Journal of Pediatrics"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1186\/s13052-021-01089-2.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/article\/10.1186\/s13052-021-01089-2\/fulltext.html","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1186\/s13052-021-01089-2.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2021,6,29]],"date-time":"2021-06-29T13:12:15Z","timestamp":1624972335000},"score":1,"resource":{"primary":{"URL":"https:\/\/ijponline.biomedcentral.com\/articles\/10.1186\/s13052-021-01089-2"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2021,6,29]]},"references-count":14,"journal-issue":{"issue":"1","published-print":{"date-parts":[[2021,12]]}},"alternative-id":["1089"],"URL":"https:\/\/doi.org\/10.1186\/s13052-021-01089-2","relation":{},"ISSN":["1824-7288"],"issn-type":[{"value":"1824-7288","type":"electronic"}],"subject":[],"published":{"date-parts":[[2021,6,29]]},"assertion":[{"value":"3 February 2021","order":1,"name":"received","label":"Received","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"9 May 2021","order":2,"name":"accepted","label":"Accepted","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"29 June 2021","order":3,"name":"first_online","label":"First Online","group":{"name":"ArticleHistory","label":"Article History"}},{"order":1,"name":"Ethics","group":{"name":"EthicsHeading","label":"Declarations"}},{"value":"Data collection was allowed by written informed consent of patients (now of legal age). Patient\u2019s data were de-identified. Given the anonymous collection of data, ethical approval was not necessary.","order":2,"name":"Ethics","group":{"name":"EthicsHeading","label":"Ethics approval and consent to participate"}},{"value":"Written consent was obtained from patients (now of legal age).","order":3,"name":"Ethics","group":{"name":"EthicsHeading","label":"Consent for publication"}},{"value":"The authors declare that they have no conflict of interest to declare.","order":4,"name":"Ethics","group":{"name":"EthicsHeading","label":"Competing interests"}}],"article-number":"141"}}