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Sexual and reproductive health outcomes in females with cloacal malformations and other anorectal malformations

  • Author Footnotes
    x HA and MEK contributed equally to this project and share co-first authorship
    Hira Ahmad
    Footnotes
    x HA and MEK contributed equally to this project and share co-first authorship
    Affiliations
    Department of Pediatric Colorectal and Pelvic Reconstructive Surgery, Nationwide Children's Hospital, Columbus, OH
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  • Author Footnotes
    x HA and MEK contributed equally to this project and share co-first authorship
    Maria E. Knaus
    Footnotes
    x HA and MEK contributed equally to this project and share co-first authorship
    Affiliations
    Department of Pediatric Colorectal and Pelvic Reconstructive Surgery, Nationwide Children's Hospital, Columbus, OH
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  • Alessandra C. Gasior
    Affiliations
    Department of Pediatric Colorectal and Pelvic Reconstructive Surgery, Nationwide Children's Hospital, Columbus, OH

    Center for Colorectal Surgery, The Ohio State University, Columbus, OH
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  • Alberta Negri Jimenez
    Affiliations
    Department of Pediatric Colorectal and Pelvic Reconstructive Surgery, Nationwide Children's Hospital, Columbus, OH
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  • Paige T. Spieth
    Affiliations
    Department of Pediatric Colorectal and Pelvic Reconstructive Surgery, Nationwide Children's Hospital, Columbus, OH
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  • Shruthi Srinivas
    Affiliations
    Department of Pediatric Colorectal and Pelvic Reconstructive Surgery, Nationwide Children's Hospital, Columbus, OH
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  • Joseph R. Stanek
    Affiliations
    Biostatistics Resource at Nationwide Children's Hospital, Columbus, OH
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  • Marc A. Levitt
    Affiliations
    Department of Pediatric Colorectal and Pelvic Reconstructive Surgery, Nationwide Children's Hospital, Columbus, OH
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  • Richard J. Wood
    Affiliations
    Department of Pediatric Colorectal and Pelvic Reconstructive Surgery, Nationwide Children's Hospital, Columbus, OH
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  • Geri D. Hewitt
    Affiliations
    Department of Pediatric Colorectal and Pelvic Reconstructive Surgery, Nationwide Children's Hospital, Columbus, OH

    Pediatric & Adolescent Gynecology & Obstetrics, Nationwide Children's Hospital, Columbus, OH
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  • Kate A. McCracken
    Correspondence
    Corresponding Author: Kate A. McCracken, 700 Children's Drive, FOB 6B, Columbus, OH 43205, Phone: (614) 722-2250, Fax: (614) 355-6229.
    Affiliations
    Department of Pediatric Colorectal and Pelvic Reconstructive Surgery, Nationwide Children's Hospital, Columbus, OH

    Pediatric & Adolescent Gynecology & Obstetrics, Nationwide Children's Hospital, Columbus, OH
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  • Author Footnotes
    x HA and MEK contributed equally to this project and share co-first authorship
Published:October 26, 2022DOI:https://doi.org/10.1016/j.jpag.2022.10.008

      Abstract

      Study Objective

      To improve our understanding of reproductive health and sexual function in women with cloacal malformations and other anorectal malformations (ARM).

      Methods

      An observational cross-sectional survey was administered to females assigned at birth aged 12 to 55 with ARM and cloacal malformations cared for at our institution. Data included age of thelarche/menarche and questions on body image, gynecologic anatomy, sexual function, and pregnancy.

      Results

      Twenty-one patients responded in the ARM group and 30 in the cloacal malformation group. There were no differences in median age of thelarche/menarche in patients with ARM (11/12.5 years) compared to patients with cloacal malformation (11/12 years). Patients with ARM were more likely to have native vaginal tissue than those with cloacal malformations (n=18, 82% versus n=12, 40%. p=0.03). There were no differences between groups regarding concerns about dyspareunia and functionality of their vagina (p>0.05). Forty-two percent of patients with cloacal malformations and 30% of patients with ARM reported having been sexually active. Two patients with cloacal malformations and two with ARM reported having been pregnant. Patients with cloacal malformation reported a lower quality of life score (80.4) compared to those with ARM (87.0) (difference >4.5).

      Conclusions

      Patients with a cloacal malformation were less likely to have native vaginal tissue and reported a lower quality of life than those with ARM. Despite this, patients with a cloacal malformation had similar reproductive health and sexual function compared to patients with ARM. Our results reinforce the need for comprehensive sexual and reproductive healthcare for all women with ARM.

      Keywords

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      References

        • Levitt MA
        • Peña A.
        Anorectal malformations.
        Orphanet Journal of Rare Diseases. 2007; 2: 1-13https://doi.org/10.1186/1750-1172-2-33/COMMENTS
        • Pradhan S
        • Vilanova-Sanchez A
        • McCracken KA
        • et al.
        The Mullerian Black Box: Predicting and defining Mullerian anatomy in patients with cloacal abnormalities and the need for longitudinal assessment.
        J Pediatr Surg. 2018; 53: 2164-2169https://doi.org/10.1016/J.JPEDSURG.2018.05.009
        • Wood RJ
        • Reck-Burneo CA
        • Vilanova-Sanchez A
        • Levitt MA.
        Organizing the care of a patient with a cloacal malformation: Key steps and decision making for pre-, intra-, and post-operative repair.
        Semin Pediatr Surg. 2020; 29https://doi.org/10.1016/J.SEMPEDSURG.2020.150988
        • Lawson A
        • Svetanoff WJ
        • Lopez J
        • et al.
        54. Evaluation and Management Considerations of Gynecologic Issues in Adolescents with Anorectal Malformations.
        Journal of Pediatric and Adolescent Gynecology. 2021; 34: 260https://doi.org/10.1016/J.JPAG.2021.02.058
        • Breech L.
        Gynecologic concerns in patients with anorectal malformations.
        Seminars in Pediatric Surgery. 2010; 19: 139-145https://doi.org/10.1053/J.SEMPEDSURG.2009.11.019
        • Schmidt D
        • Winter S
        • Jenetzky E
        • Zwink N
        • Schmiedeke E
        • Maerzheuser S.
        Sexual function in adults with anorectal malformation: psychosocial adaptation. German Network for Congenital Uro-REctal Malformations (CURE-Net).
        Pediatr Surg Int. 2012; 28: 789-792https://doi.org/10.1007/S00383-012-3119-1
        • Vilanova-Sanchez A
        • McCracken K
        • Halleran DR
        • et al.
        Obstetrical Outcomes in Adult Patients Born with Complex Anorectal Malformations and Cloacal Anomalies: A Literature Review.
        J Pediatr Adolesc Gynecol. 2019; 32: 7-14https://doi.org/10.1016/J.JPAG.2018.10.002
        • Warne SA
        • Wilcox DT
        • Creighton S
        • Ransley PG.
        Long-Term Gynecological Outcome of Patients with Persistent Cloaca.
        The Journal of Urology. 2003; 170: 1493-1496https://doi.org/10.1097/01.JU.0000086702.87930.C2
      1. Rosen R, Brown C, Heiman J, et al. The Female Sexual Function Index (FSFI): A Multidimensional Self-Report Instrument for the Assessment of Female Sexual Function. 101080/009262300278597. 2011;26(2):191-205. doi:10.1080/009262300278597

      2. Varni JW, Burwinkle TM. The PedsQLTM as a patient-reported outcome in children and adolescents with Attention-Deficit/Hyperactivity Disorder: A population-based study. Health and Quality of Life Outcomes. 2006;4. doi:10.1186/1477-7525-4-26

        • Varni JW
        • Limbers CA.
        The PedsQL 4.0 Generic Core Scales Young Adult Version: feasibility, reliability and validity in a university student population.
        J Health Psychol. 2009; 14: 611-622https://doi.org/10.1177/1359105309103580
        • Desai AD
        • Zhou C
        • Stanford S
        • Haaland W
        • Varni JW
        • Mangione-Smith RM.
        Validity and responsiveness of the pediatric quality of life inventory (PedsQL) 4.0 generic core scales in the pediatric inpatient setting.
        JAMA Pediatr. 2014; 168: 1114-1121https://doi.org/10.1001/JAMAPEDIATRICS.2014.1600
        • van den Hondel D
        • Sloots CEJ
        • Bolt JM
        • Wijnen RMH
        • de Blaauw I
        • Ijsselstijn H.
        Psychosexual Well-Being after Childhood Surgery for Anorectal Malformation or Hirschsprung's Disease.
        J Sex Med. 2015; 12: 1616-1625https://doi.org/10.1111/JSM.12886
      3. Teitler JO. Trends in Youth Sexual Initiation and Fertility in Developed Countries: 1960-1995: http://dx.doi.org/101177/000271620258000106. 2016;580(MAR.):134-152. doi:10.1177/000271620258000106

        • Rosenthal SL
        • Weiss TW
        • Zimet GD
        • Ma L
        • Good MB
        • Vichnin MD.
        Predictors of HPV vaccine uptake among women aged 19–26: Importance of a physician's recommendation.
        Vaccine. 2011; 29: 890-895https://doi.org/10.1016/J.VACCINE.2009.12.063
        • Mueller GC
        • Hussain HK
        • Smith YR
        • et al.
        Müllerian duct anomalies: comparison of MRI diagnosis and clinical diagnosis.
        AJR Am J Roentgenol. 2007; 189: 1294-1302https://doi.org/10.2214/AJR.07.2494
        • Hilton G
        • Mihm F
        • Butwick A.
        Anesthetic management of a parturient with VACTERL association undergoing Cesarean delivery.
        Canadian journal of anaesthesia = Journal canadien d'anesthesie. 2013; 60: 570-576https://doi.org/10.1007/S12630-013-9919-5
        • Bouman MB
        • van Zeijl MCT
        • Buncamper ME
        • Meijerink WJHJ
        • van Bodegraven AA
        • Mullender MG.
        Intestinal Vaginoplasty Revisited: A Review of Surgical Techniques, Complications, and Sexual Function.
        The Journal of Sexual Medicine. 2014; 11: 1835-1847https://doi.org/10.1111/JSM.12538