The aim of this study was to describe genital hair thread tourniquet syndrome (HTTS) and its treatment by pediatric and adolescent health care providers through a systematic literature review. Methods:We performed a systematic literature review on pediatric male and female genital HTTS. Studies were included if they involved genital HTTS in males or females 21 years and younger and were published in English. The main outcomes were body parts involved, symptoms, treatment, anesthetic type, providers involved in diagnosis and management, and implications of delayed or missed diagnosis.Results: There were 38 female cases from 33 publications and 147 male cases from 47 publications . The average age among females and males was 9.1 and 5.1 years, respectively. Among cases involving female patients, 93% of them were premenarchal; patients were circumcised in 90% of reviewed cases of male HTTS. The most commonly involved body parts were clitoris and labia minora in females, and penis and urethra in males. Males most commonly presented with edema and urinary symptoms, whereas females most commonly presented with edema and pain. General anesthesia was used for tourniquet excision in most cases. Male and female genital HTTS were mostly managed by urologists and emergency medicine physicians, respectively. Conclusion:This systematic literature review of more than 150 cases of male and female genital HTTS describes evaluation and management of genital HTTS spanning 7 decades. The main treatment of genital HTTS remains prompt diagnosis and removal of the tourniquet, as well as education on prevention strategies. Delayed diagnosis due to lack of recognition of the HTTS can lead to serious sequelae. Development of national guidelines regarding best practices in management of genital HTTS disseminated to all providers taking care of pediatric and adolescent patients will lead to improved patient care.
<b><i>Background/Purpose:</i></b> Although maternal-fetal surgery to treat fetal anomalies such as spina bifida continues to grow more common, potential health disparities in the field remain relatively unexamined. To address this gap, we identified maternal-fetal surgery studies with the highest level of evidence and analyzed the reporting of participant sociodemographic characteristics and representation of racial and ethnic groups. <b><i>Methods:</i></b> We conducted a systematic review of the scientific literature using biomedical databases. We selected randomized control trials (RCTs) and cohort studies with comparison groups published in English from 1990 to May 5, 2020. We included studies from across the globe that examined the efficacy of fetal surgery for twin-twin transfusion syndrome (TTTS), obstructive uropathy, congenital diaphragmatic hernia (CDH), myelomeningocele (MMC), thoracic lesions, cardiac malformations, or sacrococcygeal teratoma. We determined the frequency of reporting of age, gravidity/parity, race, ethnicity, education level, language spoken, insurance, income level, and relationship status. We identified whether sociodemographic factors were used as inclusion or exclusion criteria. We calculated the racial and ethnic group representation for studies in the USA using the participation-to-prevalence ratio (PPR). <b><i>Results:</i></b> We included 112 studies (10 RCTs, 102 cohort) published from 1990–1999 (8%), 2000–2009 (30%), and 2010–2020 (62%). Most studies were conducted in the USA (47%) or Europe (38%). The median sample size was 58. TTTS was the most common disease group (37% of studies), followed by MMC (23%), and CDH (21%). The most frequently reported sociodemographic variables were maternal age (33%) and gravidity/parity (20%). Race and/or ethnicity was only reported in 12% of studies. Less than 10% of studies reported any other sociodemographic variables. Sociodemographic variables were used as exclusion criteria in 13% of studies. Among studies conducted in the USA, White persons were consistently overrepresented relative to their prevalence in the US disease populations (PPR 1.32–2.11), while Black or African-American, Hispanic or Latino, Asian, American-Indian or Alaska-Native, and Native-Hawaiian or other Pacific Islander persons were consistently underrepresented (PPR 0–0.60). <b><i>Conclusions:</i></b> Sociodemographic reporting quality in maternal-fetal surgery studies is poor and inhibits examination of potential health disparities. Participants enrolled in studies in the USA do not adequately represent the racial and ethnic diversity of the population across disease groups.
Background High anti-Müllerian hormone (AMH) levels and 25-hydroxyvitamin D [25(OH)D] deficiency have been associated with polycystic ovarian syndrome (PCOS) in adult women, and implicated in its pathogenesis. Herein we determined if the level of both AMH and 25(OH)D are altered in adolescent females with clinical features of PCOS. Methods This is a cross-sectional study utilizing a retrospective chart review of 128 patients aged 12–20 referred to an academic adolescent gynecology and endocrinology clinic for an evaluation of suspected PCOS. Unadjusted comparisons of AMH and 25(OH)D distributions between subjects with and without PCOS were performed using the Wilcoxon Rank Sum test. Quantile regression was used to compare the median AMH and 25(OH)D between subject groups; adjusting for race, ethnicity, BMI, insurance type, age, and season when bloodwork was performed. Results Seventy-four subjects were classified as having PCOS by meeting ≥2 of the three Rotterdam diagnostic criteria, and 47 subjects met only one Rotterdam diagnostic criteria, and were used as the comparative non-PCOS group. There were statistically significant unadjusted differences in median levels of AMH and 25(OH)D. In the adjusted analyses, median AMH was significantly higher in the PCOS group compared to the non-PCOS group (+ 2.39 ng/mL, 95% CI 0.43, 4.35, p = 0.018); 25(OH)D was significantly lower in the PCOS group (− 9.01 ng/mL, 95% CI -14.49, − 3.53 p = 0.001). In our sample, adolescents in both groups had insufficient 25(OH)D level (22 ng/mL) and elevated BMI (32.2 kg/m2). Conclusions Adolescents with PCOS display high levels of AMH and low 25(OH)D levels. Since traditional clinical markers of PCOS may be physiologic in adolescents, AMH and 25(OH)D may be used as surrogate markers of PCOS risk in adolescents.
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