Paediatrics (RCH) - Research Publications
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ItemAudit of referrals for concern regarding labial appearance at the Royal Children's Hospital: 2000-2012(WILEY, 2018-04)AIM: To audit the clinical features and outcomes for all patients referred to our centre with concerns regarding labial appearance. METHODS: Young females referred to a paediatric/adolescent gynaecology tertiary centre between 2000 and 2012 with concerns regarding their labial appearance were retrospectively identified. Adolescents presenting with anomalies were excluded. Retrospective chart review was undertaken to identify reasons for referral, patient characteristics, outcome of referral and concurrent health problems. RESULTS: In total, 46 females presenting with concerns about labial appearance were identified. Five were excluded. Median age of the study population was 14.5 years (range 5-21 years). Only four (9.8%) underwent surgery after a minimum of five consultations each, with mental health review in three of four cases prior to surgery. None of the 41 patients had documented abnormal labia; however, 6 patients had asymmetry, and 3 had a labial width of >5 cm. Of mothers, 24% (n = 10) raised the initial concern regarding labial appearance to a physician, of whom, 50% of patients had a comorbid condition. In total, 70.7% initially reported interference with daily activities, and 87.8% were reassured following discussion. CONCLUSIONS: With appropriate education and counselling, the majority of girls with concerns regarding labial appearance can be managed without surgery. Overall, our data support current international policy that female cosmetic genital surgery not be performed in mature minors unless there are specific indications. More research about characteristics of patients referred with labial concerns, definition of labial size and long-term satisfaction of conservative versus surgical methods is necessary to determine the best approach.
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ItemFertility Preservation in Children and Adolescents With Cancer: Pilot of a Decision Aid for Parents of Children and Adolescents With Cancer(JMIR Publications, 2018)Background: Future infertility is a significant concern for survivors of childhood and adolescent cancer. Children and adolescents may have the opportunity to undergo fertility preservation (FP) procedures (which preserve gonadal tissue or gametes for future use) prior to the cancer treatment. However, the decision is very complex, as it is often made by parents as proxy decision makers at the time of cancer diagnosis, and is time-sensitive (needing to occur before the cancer treatment begins). Furthermore, FP procedures in children and adolescents are experimental and cannot guarantee future fertility. An uninformed decision may result in future decision regret. Objective: This study aimed to assess the acceptability, usability, and feasibility of a Web-based FP decision aid (DA) in parents of children and adolescents with cancer and clinicians. Fertility knowledge and decision regret were compared in families who reviewed the DA compared with those who did not. Methods: The Web-based DA was developed according to the International Patient Decision Aid Standards. A cross-sectional study of parents of patients with cancer, who discussed fertility, and clinicians at a tertiary children’s hospital was undertaken. The acceptability, usability, and feasibility of the DA were assessed using a pre-post survey design. Measures included the validated Decision Regret Scale, a purpose-designed fertility-related knowledge scale, questions regarding satisfaction with the DA, and open-ended responses for additional feedback. Furthermore, clinicians involved in FP were also invited to review the DA. Results: We enrolled 34 parents and 11 clinicians in this study. Participants who reviewed the DA (15 parents and 11 clinicians) expressed satisfaction with its content and functionality. Parents reported an improved understanding of cancer treatments, infertility, and FP procedures and did not report greater decision regret after DA review. Most parents (13/15, 86%) would recommend the DA to other parents. All clinicians had a consensus that this was a valid and relevant information source for all involved in fertility care. Conclusions: It is an international standard of care to discuss the impact of cancer treatment on fertility before cancer treatment. This is the first fertility DA for parents of children and adolescents with cancer and is found to be relevant and acceptable by parents and clinicians. This DA has the potential to help support parents to make informed fertility-related decisions for their children and adolescents. However, future research is needed to assess the impact of the DA on prospective decision making.
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ItemThe Association Between Unwanted Sexual Experiences and Early-Onset Cervical Cancer and Precancer by Age 25: A Case-Control Study(MARY ANN LIEBERT, INC, 2017-07)BACKGROUND: We examined the association between unwanted sexual experiences and cervical cancer, cervical intraepithelial neoplasia 3, adenocarcinoma in situ, diagnosed ≤25 years of age. METHODS: A case-control study of women ≤55 years who attended gynecological hospitals in Australia between 1983 and 2007. Cases were ≤25 years when diagnosed with disease, control group 1 were "older women" >25 years at diagnosis; control group 2 were "well women" ≤25 years attending preventive health clinics. A self-administered postal survey was utilized. The main outcome measures were prevalence of childhood sexual abuse (<16 years) and unwanted adolescent sexual experiences (between 16 and 18 years) in cases compared to controls. RESULTS: Of 400 contactable subjects, 251 participated (62.8%). Prevalence of childhood sexual abuse in cases (26.6% [25/94]) was similar to other groups. Prevalence of childhood genital contact abuse in cases with cervical cancer was 45.5% [5/11], compared to older women (20% [10/50], p = 0.08) and well women (13.8% [8/58], p = 0.01), and was marginally more common compared to well women when adjusted for other lifestyle factors (odds ratio [OR]: 4.7 [1.0-22.6], p = 0.05). Prevalence of unwanted adolescent sexual experiences in cases was 28.9% [33/114]. Prevalence of adolescent penile-genital contact experiences in cervical cancer cases was 46.7% [7/15], compared to older women (9.4%, [6/64], p < 0.001) and well women (13.7%, [10/73], p = 0.003), and was more common compared to well women when adjusted for lifestyle (OR: 5.9 [1.4-24.9], p = 0.02) and sexual health risk factors (OR: 5.6 [1.4-22.1] p = 0.01). CONCLUSIONS: Unwanted sexual experiences with genital contact were a risk factor for invasive cervical cancer ≤25 years, likely due to a complex interplay of biological and environmental factors.
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ItemLiterature Review of Benign Mullerian Papilloma Contrasted With Vaginal Rhabdomyosarcoma(ELSEVIER SCIENCE INC, 2016-08)STUDY OBJECTIVES: Benign müllerian papillomas of the genital tract are rare and, hence, can be mistaken for vaginal rhabdomyosarcoma on initial clinical review. This review of the literature will consolidate the previous cases of müllerian papilloma reported and looks for clues to differentiate the 2 entities. DESIGN AND SETTING: We provide a case report and literature review, with patients from a pediatric adolescent gynecology clinic in a tertiary center. METHODS: We conducted a search of English-language publications from 1951 (the first case report) until January 2014 by using the search words "Müllerian papilloma" and "prepubertal bleeding." References from previous published reports were also obtained for completeness. MAIN OUTCOME: Literature review of benign müllerian papilloma. RESULTS: Since 1951, 56 cases of müllerian papilloma were reported, including 4 cases at our institution. Comorbid conditions were found in 31.5% of cases (with 3 cases associated with mesenchymal tumors). The average length of time from onset of symptoms (primarily vaginal bleeding) to diagnosis was 6.7 months (range, 1 day to 3 years), with only 1 case diagnosed incidentally. Median age of presentation was 5 years (range, 1 day to 52 years). Most cases were localized and resected with ease. Histology reveals complex papillary lesions without cytologic atypia. CONCLUSION: Benign müllerian papilloma is distinguished from the more significant diagnosis of vaginal rhabdomyosarcoma by initial length of vaginal bleeding at presentation, lack of vaginal wall extension, ease of resection, and histopathology. This is compared with vaginal rhabdomyosarcoma which commonly exhibits both localized and distant spread.
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ItemNo Preview AvailableCurrent concepts in bone and reproductive health in adolescents with anorexia nervosa(WILEY, 2008-02)Anorexia nervosa (AN) initiates an adaptive response at the level of the hypothalamus, which results in a complex interplay involving most elements of the neuroendocrine axis. Consequences of onset of disease in adolescence include amenorrhoea, pubertal arrest with potential loss of target height, and osteoporosis with reduced capacity for future attainment of peak bone mass. With recovery, delay in restoration of menses is common. Hormonal therapies for restoration of bone mineral density (BMD) in adolescents have shown limited efficacy. This review will discuss the reproductive endocrine effects of AN in adolescence, and discuss new investigative tools for monitoring restoration of reproductive function and BMD in this population.
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ItemNo Preview AvailableA short report on current fertility preservation strategies for boys(WILEY, 2017-09)BACKGROUND: Advances in cancer treatment have led to improved long-term survival after childhood cancer, but often at a price of impaired future fertility. Fertility preservation (FP) in male children and early adolescents poses unique challenges as efficacy is unproven. OBJECTIVES: To describe characteristics of testicular tissue cryopreservation (TTCP) specimens taken from paediatric and adolescent patients, stratified by age, and prior chemotherapy, if any, and to demonstrate evidence for germ cells. MATERIALS AND METHODS: Retrospective review of gonadal biopsies and clinical records of patients consented into the Royal Children's Hospital FP programme between 1987 and 2015. Tissue was sliced into blocks, with one section sent for histopathology prior to cryopreservation. In boys ≥12 years where spermatogenesis could be expected, a portion of tissue was disaggregated completely to look for mature sperm and if found, additional tissue was dissected and the resulting suspension frozen. RESULTS: Testicular tissue cryopreservation specimens in 44 males (0.3-16.8 years) provided an average of 7.8 slices per patient. All the specimens were taken at the same time as another necessary surgical procedure, under one general anaesthesic. There was only one complication of scrotal wound dehiscence. Seven of the forty-four (15.9%) patients had chemotherapy prior to testicular biopsy, while the rest were chemotherapy naïve. Five of these were prepubertal, and two were pubertal patients. Eleven subjects had tissue dissected with mature sperm found in eight. Of these eight patients where sperm were found, all were pubertal with testicular size of more than 10 mL and showing histological evidence of spermatogenesis. No histologic specimen demonstrated any malignant cells. CONCLUSIONS: Testicular tissue cryopreservation can be performed in young patients without delay, preferably prior to cancer treatment. As testicular tissue contains germ cells from which haploid spermatozoa are ultimately derived, future technologies may allow their utilization for fertility in humans. This may be the only hope for biological offspring in some patients undergoing fertility compromising treatment. Retrieval of mature sperm from some pubertal patients, however, offers realistic hope to these patients of future fertility.
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ItemNo Preview AvailableA Clinical Decision Support System to Assist Pediatric Oncofertility: A Short Report(MARY ANN LIEBERT, INC, 2018-08)PURPOSE: Fertility preservation discussions with pediatric and adolescent cancer patients can be difficult for clinicians. This study describes the acceptability of a fertility clinician decision support system (CDSS). METHODS: A cross-sectional study of clinicians at The Royal Children's Hospital, Melbourne. Participants were trained on CDSS purpose, contents, and use. A survey captured the perceived benefits and weaknesses of the CDSS. RESULTS: Thirty-nine clinicians participated. Over 90% felt the CDSS aims and format were clear, and understood the components. Over 80% felt it would enable adherence to clinical pathways, policy, and standards of care. CONCLUSIONS: The CDSS provided significant perceived benefits to oncofertility care.
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ItemPaternal Understanding of Menstrual Concerns in Young Women(ELSEVIER SCIENCE INC, 2018-10)STUDY OBJECTIVE: No studies have specifically considered paternal understanding of menstruation. This study aimed to establish the degree of understanding of fathers of adolescent girls with menstrual symptoms relative to mothers. DESIGN AND SETTING: This was a cross-sectional survey-based study. Adolescent patients attending an outpatient gynecology clinic for dysmenorrhea and/or heavy menstrual bleeding and their parents were invited to complete surveys. PARTICIPANTS: Sixty surveys were completed (24 of 40 daughters, 20 of 40 mothers, 16 of 40 fathers). INTERVENTIONS AND MAIN OUTCOME MEASURES: Surveys aimed to test parents' understanding of menstrual symptoms and potential medications, as well as fathers' concerns with their daughters' health. RESULTS: The fathers' knowledge of menstrual symptoms was poorer than mothers, although most knew heavy menstrual bleeding (15/16, 94%) and mood swings (14/16, 87%). Many parents answered "don't know" or did not answer questions about potential consequences of medications, although parents were clearly concerned about side effects. Most fathers (13/16, 81%) were open to discussing menstrual concerns with daughters; however, only 54% (13/24) of daughters were open to such discussions. Of fathers, 81% (13/16) were sympathetic/concerned, 56% (9/16) felt helpless, and 13% (2/16) were frustrated when daughters were in pain. When asked about effects, 88% (14/16) of fathers (79% [15/20] of mothers) were worried about their daughter's welfare and 63% (10/16) (55% [11/20] of mothers) about schooling. CONCLUSION: We present, to our knowledge, the first insight into fathers' knowledge of their daughters' menstrual health. Overall, parents have an incomplete picture of menstrual symptoms. Even in this cohort, which could be expected to be well informed because of their daughters' attendance at a tertiary hospital, it is clear that further knowledge would assist them caring for their daughters.
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ItemFertility in Turner syndrome(WILEY, 2013-11)There is increasing interest in fertility and use of assisted reproductive technologies for women with Turner syndrome (TS). Current parenting options include adoption, surrogacy, and spontaneous and assisted reproduction. For women with TS, specific risks of pregnancy include higher than usual rates of spontaneous abortion, foetal anomaly, maternal morbidity and mortality. Heterologous fertility assistance using oocytes from related or unrelated donors is an established technique for women with TS. Homologous fertility preservation includes cryopreservation of the patient's own gametes prior to the progressive ovarian atresia known to occur: preserving either mature oocytes or ovarian tissue containing primordial follicles. Mature oocyte cryopreservation requires ovarian stimulation and can be performed only in postpubertal individuals, when few women with TS have viable oocytes. Ovarian tissue cryopreservation, however, can be performed in younger girls prior to ovarian atresia - over 30 pregnancies have resulted using this technique, however, none in women with TS. We recommend consideration of homologous fertility preservation techniques in children only within specialized centres, with informed consent using protocols approved by a research or clinical ethics board. It is essential that further research is performed to improve maternal and foetal outcomes for women with TS.
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ItemThe prevalence of childhood sexual abuse and adolescent unwanted sexual contact among boys and girls living in Victoria, Australia(PERGAMON-ELSEVIER SCIENCE LTD, 2010-05)OBJECTIVES: Childhood sexual abuse (CSA) is associated with both short- and long-term adverse mental and physical health consequences, yet there remains considerable controversy about the prevalence of CSA in the general population. There is also little prospective data on unwanted sexual contact (USC) collected during adolescence. METHODS: Data from a 10-year cohort study of a nationally representative sample of students aged 14-15 years in Victoria, Australia from 1992 to 2003 was used. CSA prior to age 16 was assessed retrospectively at age 24 years using a 6-item validated questionnaire. USC was assessed prospectively via questionnaire at 3 time points during adolescence. Multiple imputation was used to handle missing data. RESULTS: One thousand nine hundred forty-three of 2032 eligible adolescents participated in at least one wave of the study. One thousand seven hundred forty-five (812 males and 933 females) provided sufficient information to allow for multiple imputation and inclusion in the main analysis. The prevalence of any CSA was substantially higher among girls [17%, 95% confidence interval (CI): 14-20%] than boys (7%, 95% CI: 3-10%), as was the prevalence of USC reported during adolescence (14%, 95% CI 11-16%, versus 6%, 95% CI: 4-8% respectively). CONCLUSIONS: These findings highlight the high prevalence of childhood sexual abuse and unwanted sexual contact among girls as well as boys. PRACTICE IMPLICATIONS: In order to accurately inform early recognition, intervention and education programs for individuals with a history of CSA the frequency of sexual abuse must first be precisely quantified. Developing more standardized approaches will be important in order to improve our understanding of the extent of this problem.