The Obstructed Hemivagina, Ipsilateral Renal Anomaly, and Uterine Didelphys Triad and the Subsequent Manifestation of Cervical Aplasia

Abstract Study Objective To compare a case series of the obstructed hemivagina, ipsilateral renal anomaly and uterine didelphys triad with the literature, with a focus on a subset of patients with cervical aplasia. Design, Setting, and Participants A retrospective case series was conducted of all pa...

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Bibliographic Details
Published in:Journal of pediatric & adolescent gynecology 2014-12, Vol.27 (6), p.375-378
Main Authors: Sabdia, S., MBBS, DRANZCOG, Sutton, B., MBBS, FRANZCR, Dip Fetal Medicine, Kimble, R.M.N., MBBS, FRANZCOG
Format: Article
Language:English
Subjects:
16p11.2
Absent kidney
Adolescent
Cervical aplasia
Cervix Uteri - abnormalities
Cervix Uteri - surgery
Child
Child, Preschool
Female
Humans
Hysterectomy
Infant
Infant, Newborn
Kidney - abnormalities
Magnetic Resonance Imaging
Obstetrics and Gynecology
Obstructed hemivagina
Pediatrics
Retrospective Studies
Urogenital Abnormalities - diagnosis
Urogenital Abnormalities - surgery
Uterine didelphys
Uterus - abnormalities
Uterus - surgery
Vagina - abnormalities
Vagina - surgery
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Summary:Abstract Study Objective To compare a case series of the obstructed hemivagina, ipsilateral renal anomaly and uterine didelphys triad with the literature, with a focus on a subset of patients with cervical aplasia. Design, Setting, and Participants A retrospective case series was conducted of all patients with the triad managed between 2005-2013 at a tertiary center for adolescent gynecology. Results Similarities in this cohort, compared to the literature, included heterogeneity of presentation, presence of endometriosis, and asymmetry of affected side. Notable differences included 1 patient with a 16p11.2 microdeletion and 2 patients with subsequent unilateral cervical aplasia. All patients underwent magnetic resonance imaging for diagnosis. Vaginal septum division was performed in 8 cases and excision in 1 case. Both cases with cervical aplasia ultimately underwent hemi-hysterectomy, and highlight the implications of this rare variant in regards to its existence, limitations of magnetic resonance imaging in this context, and suggestions for improvement in diagnosis and management. Conclusion The complexity of these cases, especially the evolving manifestation of cervical aplasia postoperatively, illustrates the need to recognize limitations in imaging and divergence in definitive management.
ISSN:1083-3188
1873-4332
DOI:10.1016/j.jpag.2014.02.001