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Rare disease
CASE REPORT
Novel management of vaginal chronic graft-versus-host disease causing haematometra and haematocolpos
  1. Alice Buchan1,2,
  2. Melissa A Merideth3,4,
  3. Richard W Childs5,
  4. Pamela Stratton6,7
  1. 1Institute of Genetic Medicine, Newcastle University, Newcastle Upon Tyne, UK
  2. 2Gynaecology, Newcastle Upon Tyne Hospitals NHS Foundation Trust, Newcastle Upon Tyne, UK
  3. 3Medical Genetics Branch, National Human Genome Research Institute, National Institutes of Health, Bethesda, Maryland, USA
  4. 4Intramural Office of Rare Diseases, Office of the Director, National Institutes of Health, Bethesda, Maryland, USA
  5. 5Hematology Branch, National Heart Lung and Blood Institute, National Institutes of Health, Bethesda, Maryland, USA
  6. 6Eunice Kennedy Shriver National Institute of Child Health and Human Development, National Institutes of Health, Bethesda, Maryland, USA
  7. 7National Institute of Neurological Disorders and Stroke, National Institutes of Health, Bethesda, Maryland, USA
  1. Correspondence to Dr Pamela Stratton, strattop{at}mail.nih.gov

Summary

Genital chronic graft-versus-host disease (GVHD) in women posthaematopoietic cell transplantation may cause vaginal mucosal sclerosis. Human papillomavirus (HPV) reactivation, also common post-transplantation, limits local immunosuppressive, but not oestrogen treatment. A 36-year-old nulliparous woman developed coincidental genital chronic GVHD and HPV 22 months after transplant for aplastic anaemia. Topical immunosuppression for GVHD led to an eruption of warts successfully treated with laser surgery and cone biopsy. She maintained normal ovarian function and used extended cycle combined hormonal contraception. A vaginal oestrogen ring used continuously limited most scarring for 8 years. Progressive apical vaginal scarring obstructed menstrual flow leading to haematocolpos and haematometra. Normal anatomy was restored with a cruciate incision in the cervicovaginal scar performed during menses. When HPV disease limits use of topical immunosuppression in women with vaginal GVHD, the local scar-reducing effect of a vaginal oestrogen ring is limited, and surgery may be needed and can be successful in treating haematocolpos.This study was registered in ClinicalTrials.gov with trial registration number of NCT00003838.

  • obstetrics, gynaecology and fertility
  • obstetrics and gynaecology
  • contraception
  • haematology (incl blood transfusion)

https://doi.org/10.1136/bcr-2017-222720

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    Footnotes

    • Contributors All authors (AB, MM, RC and PS) contributed to the planning and writing of the manuscript. MM, RC and PS were directly involved in the medical care of the patient.

    • Funding The authors have not declared a specific grant for this research from any funding agency in the public, commercial or not-for-profit sectors.

    • Competing interests None declared.

    • Patient consent Obtained.

    • Provenance and peer review Not commissioned; externally peer reviewed.