Pediatric Vulvovaginal Graft-Versus-Host Disease: A Retrospective Cohort Study and Literature Review

Dowlut‐McElroy, Tazim; Shin, Stephanie; Stepanek, Elizabeth; Jacobsohn, David A.; Gómez-Lobo, Veronica

doi:10.1016/j.jpag.2022.04.005

Cited by 5 publications

(6 citation statements)

References 22 publications

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“…Several studies have also recorded the presence of other organ GVHD in up to 72%–90% of patients, most commonly in the skin, mouth, and eyes 8–10,13,18,23,25,26 . However isolated female genital GVHD was still reported as frequently as 23% 16 .…”

Section: Resultsmentioning

confidence: 99%

“…True incidence is unknown and varies widely between studies due to diverse definitions, biased cohort selection, and practitioners' general lack of awareness of the condition and failure to inquire. 37 We found that some studies reported incidence as low as 1.6% to 5%; 22,26,30 however, they had a high degree of bias because they only included patients who self-reported symptoms. True incidence is far more likely to be between 27% and 66% as demonstrated by prospective studies that performed routine gynecological reviews on all female patients after their allogeneic stem cell transplantation, thus identifying asymptomatic disease and overcoming patient reporting bias.…”

Section: Discussionmentioning

confidence: 96%

“…2,8,12,13 Several studies have also recorded the presence of other organ GVHD in up to 72%-90% of patients, most commonly in the skin, mouth, and eyes. [8][9][10]13,18,23,25,26 However isolated female genital GVHD was still reported as frequently as 23%. 16 It is unclear if there is any causative link between the presence of female genital GVHD and other organ system GVHD manifestations.…”

Section: Clinical Findingsmentioning

confidence: 99%

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Vulvar and Vaginal Graft Versus Host Disease After Allogeneic Stem Cell Transplant—A Systematic Review

Preston

Richards

2023

J Low Genit Tract Dis

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Objective: To conduct a systematic literature search to identify and determine the prevalence, signs and symptoms, and clinical management of vulvar and vaginal graft versus host disease (GVHD).Methods: A systematic literature search of articles from 1993 to August 2022 was performed. Studies were included if full text was available in the English language and provided reports on female subjects with more than four patients. Review articles, conference abstracts, case reports, and case series of less than 5 patients were excluded. Included studies had their reference list searched for further manuscripts. Two authors reviewed the search results and independently identified studies that met the selection criteria and summarized available data.Results: There were 29 studies available in the literature that met the inclusion criteria. There was a high risk of bias within the available literature. The prevalence of vulval and vaginal GVHD varied between 27% and 66% of women after allogeneic stem cell transplant. Other organ GVHD, most commonly the skin, mouth, and eyes, may be present concurrently in these patients, or they may be asymptomatic. Specialist gynecology review, topical estrogen, topical steroids, topical immunosuppression, and vaginal dilatation led to a reduction in complications associated with the condition, and surgery was helpful in some severe refractory cases. These patients remain at higher risk of developing cervical dysplasia, and regular human papillomavirus screening is recommended.Conclusions: Female genital GVHD is a rare phenomenon. Early, coordinated, and regular gynecological reviews after stem cell transplant are essential to reduce the long-term complications.

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Section: Resultsmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 96%

Section: Clinical Findingsmentioning

confidence: 99%

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Vulvar and Vaginal Graft Versus Host Disease After Allogeneic Stem Cell Transplant—A Systematic Review

Preston

Richards

2023

J Low Genit Tract Dis

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“…Thirty-two young females were included, and their full characteristics are presented in Table 1 —three cases by Dowlut-McElroy et al (2022) [ 9 ], four cases by Allen et al (2020) [ 7 ], seventeen cases by Cizek et al (2019) [ 8 ], two cases by Michala et al (2018) [ 19 ], one case by Childress et al (2015) [ 20 ], one case by Choi et al (2009) [ 21 ] and four cases by Stratton et al (2007) [ 11 ]. The age of the patients during the HSCT ranged from 1.4 to 20 years, with a mean of 10.9 (SD: 4.7).…”

Section: Resultsmentioning

confidence: 99%

“…In adult studies, the reported incidence varies to up to 66%, but nonetheless, genital GVHD appears to be an under-reported condition [ 4 , 5 , 6 ]. Three case series of pediatric populations post HSCT by Allen et al [ 7 ], Cizek et al [ 8 ] and, more recently, Dowlut-McElroy et al [ 9 ] reported an incidence of 1.2%, 5.9% and 5%, respectively, though it is mentioned that these percentages are probably not reliable estimates of the true incidence. In another retrospective study, Takahashi et al [ 10 ] did not observe any cases of genital GVHD in a population of 573 children who had undergone HSCT, mentioning again the high possibility of underdiagnosis.…”

Section: Introductionmentioning

confidence: 99%

Genital GVHD in Female Children and Adolescents: A Systematic Review of Case Reports and Case Series

Tsimeki,

Tsimpidakis,

Roidi

et al. 2023

Children

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Genital graft-versus-host disease (GVHD) after hematopoietic stem cell transplantation (HSCT) is an underdiagnosed manifestation of chronic GVHD. Few articles have been published in pediatric populations, and there are no established guidelines for the management of this condition in children. This study aims to provide a systematic literature review of the published studies and cases of genital (vulvovaginal) GVHD in girls and adolescents post HSCT, with a focus on the time of diagnosis and clinical manifestations. The authors searched for English-language articles published after 1990, which included full patient details. Thirty-two cases of female patients under 20 years of age were identified. The median time of diagnosis was 381 days (IQR: 226–730 days), and 83% of patients developed Grade 3 vulvovaginal GVHD. Based on these observations, an early pediatric gynecologic examination of these patients, soon within the first year after HSCT, could be suggested for early diagnosis, treatment initiation and prevention of long-term complications.

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Early Puberty in Girls—What Do We Know in 2022?

Hillard¹

2022

Journal of Pediatric and Adolescent Gynecology

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Pediatric Vulvovaginal Graft-Versus-Host Disease: A Retrospective Cohort Study and Literature Review

Cited by 5 publications

References 22 publications

Vulvar and Vaginal Graft Versus Host Disease After Allogeneic Stem Cell Transplant—A Systematic Review

Vulvar and Vaginal Graft Versus Host Disease After Allogeneic Stem Cell Transplant—A Systematic Review

Genital GVHD in Female Children and Adolescents: A Systematic Review of Case Reports and Case Series

Early Puberty in Girls—What Do We Know in 2022?

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