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- Title
Bilateral sactosalphinx and congenital adrenal hyperplasia: case report on two rare conditions in two virgin girls.
- Authors
Scarpa, Maria-Grazia; Iaquinto, Marianna; Codrich, Daniela; Schleef, Jürgen
- Abstract
Background: 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. Case presentation: 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. Conclusions: 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.
- Subjects
ADRENOGENITAL syndrome; THERAPEUTICS; VAGINAL diseases; PATIENT aftercare; PERITONITIS; ULTRASONIC imaging; FALLOPIAN tube diseases; CONVALESCENCE; MAGNETIC resonance imaging; MENARCHE; DISEASE relapse; ACUTE abdomen; LAPAROSCOPY; POSTOPERATIVE period; SALPINGECTOMY; ABDOMINAL pain; RARE diseases
- Publication
Italian Journal of Pediatrics, 2021, Vol 47, Issue 1, p1
- ISSN
1720-8424
- Publication type
Article
- DOI
10.1186/s13052-021-01089-2