RESUMO
Aberrant male germline development can lead to the formation of seminoma, a testicular germ cell tumor. Seminomas are biologically similar to primordial germ cells (PGCs) and many bear an isochromosome 12p [i(12p)] with two additional copies of the short arm of chromosome 12. By mapping seminoma transcriptomes and open chromatin landscape onto a normal human male germline trajectory, we find that seminoma resembles premigratory/migratory PGCs; however, it exhibits enhanced germline and pluripotency programs and upregulation of genes involved in apoptosis, angiogenesis, and MAPK/ERK pathways. Using pluripotent stem cell-derived PGCs from Pallister-Killian syndrome patients mosaic for i(12p), we model seminoma and identify gene dosage effects that may contribute to transformation. As murine seminoma models do not exist, our analyses provide critical insights into genetic, cellular, and signaling programs driving seminoma transformation, and the in vitro platform developed herein permits evaluation of additional signals required for seminoma tumorigenesis.
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Epigênese Genética , Células Germinativas , Seminoma , Neoplasias Testiculares , Humanos , Seminoma/genética , Seminoma/patologia , Seminoma/metabolismo , Masculino , Células Germinativas/metabolismo , Neoplasias Testiculares/genética , Neoplasias Testiculares/patologia , Neoplasias Testiculares/metabolismo , Transcrição Gênica , Regulação Neoplásica da Expressão Gênica , Transcriptoma/genéticaRESUMO
There is a broad range of variant phenotypes that can occur within the bladder exstrophy and epispadias complex spectrum. Accurate prenatal detection helps prepare families and to coordinate subspecialty resources. Here, we present the case of a patient with prenatally diagnosed patient with covered cloacal exstrophy variant along with four additional cases illustrating the nonlinear spectrum from isolated epispadias to cloacal exstrophy. Given the rarity of these variants overall and of each subtype within the spectrum, there is a need for long-term multi-institutional outcomes data to improve detection, characterization, and prognostication for these patients.
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Malformações Anorretais , Extrofia Vesical , Epispadia , Gravidez , Feminino , Humanos , Epispadia/diagnóstico , Epispadia/cirurgia , Extrofia Vesical/diagnóstico , Extrofia Vesical/cirurgiaRESUMO
INTRODUCTION: RENAL Nephrometry is a complexity score validated in adults with renal tumors and describes the likelihood of complication after partial nephrectomy (PN). Utilization in pediatrics has been limited. Thus, our goal is to quantify inter-rater agreement as well as determine how scores correlate with outcomes. We hypothesize that the RENAL Nephrometry Score is reproducible in children with renal tumors and is related to perioperative and post-operative complications. METHODS: All pediatric patients who underwent PN for a renal mass from 2006 to 2019 were identified. Patient data, operative details, and outcomes were aggregated. Pre-operative CT/MR imaging was anonymized and scored by 2 pediatric radiologists and 2 pediatric urologists using RENAL Nephrometry metrics. Statistical analysis utilized Fleiss' kappa and the intraclass correlation coefficient (ICC). Comparative analyses were performed based on Nephrometry Score <9 and ≥ 9. RESULTS: 28 patients undergoing 33 PN were identified. Median age at surgery was 3.2 years (IQR 1.8-4.0). There is moderate-good agreement across scorers on the domains of RENAL Nephrometry Score, with the lowest agreement noted for anterior vs posterior tumors. Comparing patients with scores <9 and ≥ 9, there was increased operative time (357 vs 267 min, p = 0.003) and LOS for those with a higher score, but no difference in the incidence of 30-day complications. CONCLUSION: RENAL Nephrometry Score is an easily reproducible complexity score for renal tumors in pediatric patients. Higher scores are associated with increased length of stay and estimated blood loss but not complications. Reporting of nephrometry scores in future publications on pediatric renal tumors should become standard in the literature.
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Neoplasias Renais , Rim , Adulto , Humanos , Criança , Lactente , Pré-Escolar , Rim/diagnóstico por imagem , Rim/cirurgia , Rim/patologia , Neoplasias Renais/cirurgia , Neoplasias Renais/patologia , Nefrectomia/métodos , Projetos de Pesquisa , Néfrons/cirurgia , Néfrons/patologia , Estudos Retrospectivos , Resultado do TratamentoRESUMO
OBJECTIVE: Differences/disorders of sex development (DSDs) are rare, congenital conditions involving discordance between chromosomes, gonads, and phenotypic sex and are often diagnosed in infancy. A key subset of parents of children newly diagnosed with a DSD experience clinically elevated distress. The present study examines the relationship between perinatal factors (i.e., gestational age, delivery method) and trajectories of parental adjustment. METHODS: Parent participants (mothers = 37; fathers = 27) completed measures at baseline, 6- and 12-month follow-up. Multilevel linear regression controlled for clustering of the data at three levels (i.e., time point, parent, and family) and examined the relationship between perinatal factors and trajectories of depressive and anxious symptoms. Two-way interactions between perinatal factors and parent type were evaluated. RESULTS: Overall depressive and anxious symptoms decreased over time. There were significant interactions between gestational age and parent type for depressive and anxious symptoms, with younger gestational age having a stronger negative effect on mothers vs. fathers. There was a significant interaction between time and gestational age for depressive symptoms, with 36 weeks' gestational age demonstrating a higher overall trajectory of depressive symptoms across time compared to 38 and 40 weeks. Findings for the delivery method were not significant. CONCLUSIONS: Findings uniquely demonstrated younger gestational age was associated with increased depressive symptoms, particularly for mothers compared to fathers. Thus, a more premature birth may predispose parents of infants with DSD to distress. Psychosocial providers should contextualize early diagnosis-related discussions within stressful birth experiences when providing support.
Assuntos
Mães , Pais , Feminino , Lactente , Criança , Gravidez , Humanos , Masculino , Pais/psicologia , Mães/psicologia , Idade Gestacional , Desenvolvimento Sexual , Genitália , Pai/psicologia , Depressão/psicologiaRESUMO
INTRODUCTION: The use of caudal anesthesia at the time of hypospadias repair remains controversial as some prior studies have reported increased postoperative complication rates associated with caudal nerve block. However, these results have been called into question due to confounding factors and heterogeneous study groups. OBJECTIVE: Given the importance of identifying true risk factors associated with increased hypospadias complication rate, we examined our experience with caudal anesthesia limiting our analysis to distal repairs. We hypothesized that caudal anesthesia would not be associated with increased postoperative complications. STUDY DESIGN: We retrospectively reviewed our institutional hypospadias database from June 2007 to January 2021. All boys who underwent single-stage distal hypospadias repair with either caudal or penile block with minimum 1 month follow up were included. Records were reviewed to determine the type of local anesthesia, type of hypospadias repair, all complications, and time to complication. Association between any complication and local anesthesia type was evaluated by univariate and multivariate logistic regression analysis controlling for age at surgery and type of repair. A sub-analysis was performed for complications occurring ≤30 days. RESULTS: Overall, 1008 boys, 832 (82.5%) who received caudal and 176 (17.5%) penile block, were included. Median age at surgery was 8.1 months and median follow up was 13 months. Overall complication rate was 16.4% with 13.8% of patients requiring repeat operation. Median time to complication was 10.59 months and was significantly shorter in the caudal group (8.45 vs. 25.2 months). Caudal anesthesia was associated with higher likelihood of complication on univariate analysis; however, this was not true on multivariate analysis when controlling for age and type of repair. Caudal anesthesia was not associated with increased likelihood of complication within 30 days. DISCUSSION: Since the association between caudal anesthesia and hypospadias complications was first suggested, several studies have tried to answer this question with variable results. Our findings add to the evidence that there is no association between caudal anesthesia and increased hypospadias complications in either the short or long term. The major strengths of our study are a large, homogenous study population, robust follow up and inclusion of data from 14 surgeons over 14 years. Limitations include the study's retrospective nature as well as lack of standardized follow up protocol throughout the study period. CONCLUSIONS: After controlling for possible confounders, caudal nerve block was not associated with increased risk of postoperative complications following distal hypospadias repair.
Assuntos
Anestesia Caudal , Hipospadia , Masculino , Humanos , Lactente , Hipospadia/cirurgia , Hipospadia/etiologia , Estudos Retrospectivos , Uretra , Complicações Pós-Operatórias/epidemiologia , Complicações Pós-Operatórias/etiologia , Anestesia Caudal/efeitos adversos , Resultado do TratamentoRESUMO
INTRODUCTION & OBJECTIVE: The opioid crisis has raised concerns for long-term sequela of routine administration of opioids to patients, particularly in the pediatric population. Nonsteroidal anti-inflammatory drug use is limited in hypospadias surgery due to concerns for post-operative bleeding, particularly with ketorolac. We hypothesize that ketorolac administration at the time of hypospadias repair is not associated with increased bleeding or immediate adverse events. METHODS: A retrospective single institution study included all patients undergoing hypospadias surgery from 2018 to 2021. Outcomes measured include peri-operative ketorolac administration, opioid prescriptions, and unplanned encounters (i.e., emergency department or office visits). Comparative statistics using non-parametric and binary/categorical tests and a logistic regression were performed. RESULTS: 1044 patients were included, among whom there were 562 distal, 278 proximal and 204 hypospadias complication repairs. Ketorolac was administered to 396 (37.9%) patients and its utilization increased during the study period [Summary Figure]. Patients receiving ketorolac were older (p = 0.002) and were prescribed opioids less often after surgery (2.0% vs 5.2%, p = 0.009). There was no difference in unplanned encounters across repair types (p = 0.1). Multivariate logistic regression showed ketorolac use was not associated with an increased likelihood of an unplanned encounter. DISCUSSION: The use of NSAIDs post-operatively has traditionally been limited due to concerns about bleeding risks, however the present study displayed no significant increases in unplanned patient encounters either in the ED or outpatient clinic after ketorolac administration. Our study has several limitations including its retrospective and single-institutional design, difficulties of pain assessment in pediatric population, and possibility of under estimation of unplanned encounters due to limited access to patients' records outside of our institution. CONCLUSIONS: The use of ketorolac is not associated with an increase in unplanned encounters in children undergoing hypospadias repair. It should be considered a safe agent for perioperative analgesia to decrease opioid utilization. Further studies will evaluate long-term surgical outcomes in children receiving ketorolac after hypospadias repair.
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Hipospadia , Cetorolaco , Masculino , Humanos , Criança , Cetorolaco/efeitos adversos , Hipospadia/cirurgia , Hipospadia/induzido quimicamente , Analgésicos Opioides/uso terapêutico , Estudos Retrospectivos , Anti-Inflamatórios não Esteroides/efeitos adversos , Dor Pós-Operatória/tratamento farmacológicoRESUMO
CHD7 disorder is a multiple congenital anomaly syndrome with a highly variable phenotypic spectrum, and includes CHARGE syndrome. Internal and external genital phenotypes frequently seen in CHD7 disorder include cryptorchidism and micropenis in males, and vaginal hypoplasia in females, both thought to be secondary to hypogonadotropic hypogonadism. Here, we report 14 deeply phenotyped individuals with known CHD7 variants (9 pathogenic/likely pathogenic and 5 VOUS) and a range of reproductive and endocrine phenotypes. Reproductive organ anomalies were observed in 8 of 14 individuals and were more commonly noted in males (7/7), most of whom presented with micropenis and/or cryptorchidism. Kallmann syndrome was commonly observed among adolescents and adults with CHD7 variants. Remarkably, one 46,XY individual presented with ambiguous genitalia, cryptorchidism with Müllerian structures including uterus, vagina and fallopian tubes, and one 46,XX female patient presented with absent vagina, uterus and ovaries. These cases expand the genital and reproductive phenotype of CHD7 disorder to include two individuals with genital/gonadal atypia (ambiguous genitalia), and one with Müllerian aplasia.
Assuntos
Síndrome CHARGE , Criptorquidismo , Transtornos do Desenvolvimento Sexual , Humanos , Masculino , Feminino , Fenótipo , Síndrome CHARGE/genética , Transtornos do Desenvolvimento Sexual/genética , Genitália , DNA Helicases/genética , Proteínas de Ligação a DNA/genéticaRESUMO
OBJECTIVE: To describe the development and implementation of a process for creating accurate Pediatric genitourinary 3D modeling and printing with multiphase postcontrast imaging for surgical planning. MATERIALS AND METHODS: Additive manufacturing and 3D model present opportunities to support clinical planning, this manuscript's specific process and considerations for creating pediatric genitourinary 3D modeling to support urology. The process for creating the 3D models and prints covers 3 key aspects from image acquisition, imaging review and selection, and segmentation and modification (as needed). Each step is outlined with the key roles and procedures. RESULTS: The described case had digital and printed models prepared with references to the optimized imaging sequence for 3D modeling of Pediatric genitourinary. Case shared include complex genitourinary reconstruction and Kideny with Wilms tumors. CONCLUSION: The processes described have become a standard of practice for complex kidney tumors and exstrophy planning. The team continues to work on ever-changing improvements to make the best possible models to support clinical and surgical planning.
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Neoplasias Renais , Urologia , Humanos , Criança , Impressão Tridimensional , Neoplasias Renais/diagnóstico por imagem , Neoplasias Renais/cirurgia , Sistema Urogenital , Urologia/métodos , Imageamento Tridimensional/métodos , Modelos AnatômicosRESUMO
There is a broad differential diagnosis for a pre-pubertal child presenting with a scrotal mass including both benign and malignant etiologies. Lipoblastomas are rare benign neoplasms originating from fat cells that occur most commonly on the trunk or extremities of young children. There have been less than 20 cases of scrotal lipoblastomas reported in the literature, with the majority occurring in children less than 3 years of age. Here we present a unique case of an 18-month male presenting with a paratesticular mass found to be a lipoblastoma on final pathology.
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Lipoblastoma , Criança , Pré-Escolar , Diagnóstico Diferencial , Humanos , Lipoblastoma/diagnóstico , Lipoblastoma/patologia , Lipoblastoma/cirurgia , Masculino , Escroto/patologiaRESUMO
PURPOSE: Testosterone (T) administration prior to hypospadias surgery to increase glans size remains controversial. Understanding T's effect on glans width (GW) is essential to understanding its potential impact on surgical outcomes. We hypothesized that preoperative T in prepubertal boys significantly increases GW at the time of hypospadias surgery. MATERIALS AND METHODS: Our single institutional database was queried to identify patients who underwent hypospadias surgery from 2016 to 2020, in which data for T administration and GW were available. Descriptive, nonparametric and categorical statistics were performed as indicated. RESULTS: A total of 579 patients were eligible for analysis. Median age at surgery was 0.9 years (IQR 0.6-1.6). A total of 247/579 patients (42.7%) received T. The median GW at surgery was 15 mm (IQR 13-17). When comparing patients who had T administered to those who did not, we found a significant difference in GW at surgery (16 mm vs 14 mm, p <0.001). The median change in GW from the office to surgery was 4 mm for those receiving T vs 0 mm for those not receiving T (p <0.001). We identified a greater change in GW from preoperative to intraoperative measurements in patients who received 2 doses of T vs 1 dose (4 mm vs 2 mm, p <0.001). A histogram plot revealed the distribution of GW change at surgery. CONCLUSIONS: In our prospectively collected cohort of patients undergoing hypospadias surgery, we were able to quantitate the change in GW from preoperative T. Two doses of T resulted in a significant increase in GW vs 1 dose.
Assuntos
Hipospadia , Procedimentos de Cirurgia Plástica , Androgênios , Feminino , Humanos , Hipospadia/cirurgia , Lactente , Masculino , Procedimentos de Cirurgia Plástica/efeitos adversos , Testosterona , Resultado do Tratamento , Uretra/cirurgia , Procedimentos Cirúrgicos Urológicos Masculinos/métodosRESUMO
Radical nephrectomy combined with contemporary chemotherapeutic and radiation therapy protocols has drastically improved outcomes for children with Wilms tumor. Patients with bilateral disease and a syndrome predisposing to tumor development have necessitated the use of nephron-sparing surgery in select cases. Success in managing these patients has increased the indication for partial nephrectomy, although current guidelines for unilateral Wilms tumor are limited. Given that children are being cured with increasing success, recent focus has shifted to long-term health outcomes in addition to tumor treatment. Specifically, renal function has an impact on long-term cardiovascular health and events. Adult outcomes with partial nephrectomy provide a guideline for a paradigm shift in the management of children with Wilms tumor, particularly with advances in imaging and adjuvant therapy. The data are limited for children undergoing partial nephrectomy for unilateral Wilms tumor and outcomes for larger tumors will need to be studied closely in future trials. Increased utilization of neoadjuvant chemotherapy could further expand the number of patients eligible for partial nephrectomy.
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Neoplasias Renais , Tumor de Wilms , Criança , Humanos , Neoplasias Renais/cirurgia , Neoplasias Renais/patologia , Néfrons/cirurgia , Néfrons/patologia , Tumor de Wilms/cirurgia , Tumor de Wilms/patologia , Terapia Combinada , Nefrectomia/métodosRESUMO
Introduction: Osteopenia and osteoporosis have been reported in adults with Complete Androgen Insensitivity Syndrome (CAIS). Little is known about changes in bone mineral density (BMD) in adolescents with CAIS and whether it is affected by early gonadectomy. Body composition data have not been reported. Methods: Single-center, retrospective study of CAIS adolescents who underwent dual-energy x-ray absorptiometry (DXA) (Hologic, Horizon A). Body composition is presented as lean and fat mass indices (LMI, FMI). Z-scores for lumbar spine areal BMD (LBMD), total body less head (TBLH), bone mineral content (BMC), LMI, and FMI were calculated using female normative data. Results are expressed as median and min, max. Results: Six females with genetically confirmed CAIS were identified-one with intact gonads and five with history of gonadectomy at 2-11 months. In the subject with intact gonads, LBMD-Z and TBLH BMC-Z were -1.56 and -1.26, respectively, at age 16 years. Among those with gonadectomy, LBMD-Z was -1.8 (-3.59 to 0.49) at age 15.6 years (12-16.8) and decreased in all three subjects who had longitudinal follow-up despite hormone replacement therapy (HRT). Adherence to HRT was intermittent. LMI-Z and FMI-Z were 0.1 (-1.39 to 0.7) and 1.0 (0.22 to 1.49), respectively. Conclusions: These limited data indicate that adolescents with CAIS have bone mass deficit. Further studies are needed to understand the extent of BMD abnormalities and the effect of gonadectomy, especially early in childhood, and to establish the optimal HRT regimen for bone accrual. Data on lean mass are reassuring.
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Síndrome de Resistência a Andrógenos/complicações , Composição Corporal/fisiologia , Doenças Ósseas Metabólicas/etiologia , Absorciometria de Fóton , Adolescente , Síndrome de Resistência a Andrógenos/metabolismo , Síndrome de Resistência a Andrógenos/patologia , Síndrome de Resistência a Andrógenos/cirurgia , Densidade Óssea , Doenças Ósseas Metabólicas/metabolismo , Doenças Ósseas Metabólicas/patologia , Castração , Feminino , Humanos , Peso Corporal Ideal/fisiologia , Lactente , Masculino , Músculos/patologia , Tamanho do Órgão , Estudos RetrospectivosRESUMO
INTRODUCTION: Widespread utilization of telemedicine in our practice to date has been limited to the evaluation of certain post-surgical patients. The COVID-19 pandemic acutely stressed our established system and required us to enhance our utilization of telemedicine. We hypothesized that expansion of telemedicine to new and follow up patient visits for pediatric urology could be done effectively in a way that satisfied patient and parental expectations. MATERIALS AND METHODS: Using a pre-COVID-19 established telemedicine program based in our electronic medical record (EMR), patients and providers transitioned to primarily virtual visits when clinically appropriate. Guidelines were formulated to direct patient scheduling, provider and staff education was provided, including a process map designed for multiple providers to complete video visits (VV), and the EMR was redesigned to incorporate telehealth terminology. The number of VV per provider was recorded using the electronic medical record, and patient reported outcomes (PRO) were measured using a standardized questionnaire. RESULTS: A total of 631 VV met inclusion criteria during the period of May 2018-April 2020. This included 334 follow up, 172 new, and 125 postoperative visits. The median age of patients at time of visit was 7 years (IQR 2-12 years), median visit time was 20 min (IQR 15-30 min), and the median travel distance saved by performing a VV was 12.2 miles (IQR 6.3-26.8 miles). Diagnoses were varied and included the entire breadth of a standard pediatric urology practice. The PRO questionnaire was completed for 325 of those patient visits. Families reported a high overall satisfaction with the video visits (median score of 10 out of 10) and felt that the visit met their child's medical needs. 90% stated that they would strongly recommend a telehealth visit to other families. Patients and parents reported benefits of VV including decreased travel costs and less time taken off from work and school. CONCLUSION: The EMR enabled nimble redirection of clinical care in the setting of a global pandemic. The enhanced use of telemedicine has proved to be an alternative method to provide care for pediatric urology patients. Families indicate a high degree of satisfaction with this technology in addition to significant time and cost savings. Telemedicine should remain a key aspect of medical care and expanded from post-operative visits to new patient and follow up visits, even as we return to our normal practices as the pandemic restrictions soften.
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COVID-19 , Telemedicina , Urologia , Criança , Pré-Escolar , Redução de Custos , Humanos , Pacientes Ambulatoriais , Pandemias , Satisfação do Paciente , SARS-CoV-2RESUMO
INTRODUCTION: Differences of sex development (DSD) are congenital conditions in which there is atypical chromosomal, gonadal and/or phenotypic sex. A phenotype of severe genital atypia in patients raised as male is a relatively rare occurrence and standards for management are lacking. Decision making for early surgical planning in these rare cases includes, but is not limited to, degree of atypia, location of testes, and presence of MÏllerian remnants. In this study we describe surgical approaches and short-term outcomes for masculinizing genitoplasty in moderate to severe genital atypia in young patients raised male, for whom parents opted for early surgery. METHODS: This NIH-sponsored study is an ongoing, observational, multicenter investigation assessing medical, surgical and psychological outcomes in children and their parents affected by atypical genitalia due to DSD. Participants were prospectively enrolled from 12 children's hospitals across the United States that specialize in DSD care. Criteria for child enrollment were a Quigley score of 3-6 in those with a 46, XY or 45,X/46, XY chromosome complement, age <3 years with no previous genitoplasty; patients were included independent of whether genitoplasty was performed. Cosmesis was graded according to a 4-point Likert scale and complications per the Clavian-Dindo classification. RESULTS: Of the 31 participants, 30 underwent hypospadias repair and 1 patient did not undergo a genitoplasty procedure. The majority of participants (22) received a staged hypospadias repair. Seventeen complications were identified in 12 of the 31 children (41%) at 12 months of follow up. Glans dehiscence and urethrocutaneous fistula were the most common complications. Orchiopexy was performed in 14 (44%) and streak gonads were removed in 4 (13%) participants. Both parents and surgeons reported improved cosmesis after surgery when compared to baseline. CONCLUSION: Genitoplasty was chosen by parents for the majority of children eligible for study. No single surgical approach for masculinizing moderate to severe genital ambiguity in young patients with 46, XY or 45,X/46, XY DSD was adopted by all surgeons. Complications occurred in 41% of those who underwent genitoplasty for severe hypospadias. Overall, appearance of the genitals, as determined by parents and surgeons, improved following genitoplasty. Outcomes of early genitoplasty are needed to guide families when making decisions about such procedures for their young children.
Assuntos
Transtornos do Desenvolvimento Sexual , Procedimentos de Cirurgia Plástica , Criança , Pré-Escolar , Transtornos do Desenvolvimento Sexual/cirurgia , Feminino , Genitália/cirurgia , Humanos , Masculino , Estudos Prospectivos , Desenvolvimento SexualRESUMO
An identifiable genetic malformation or predisposition syndrome is present in 18% of Wilms tumor cases. Given this, children with conditions associated with a greater than 1% risk of developing Wilms tumor are recommended to have regular surveillance imaging with renal ultrasound until age 7. Seven years is the recommended screening duration because 95% of cases will occur by this age. We present a case of a child with isolated hemihypertrophy, associated with 5% risk of Wilms tumor, who presented with a tumor after the recommended screening, at age 9, brining into question the age cutoffs currently used.
Assuntos
Hiperplasia/diagnóstico , Neoplasias Renais/diagnóstico por imagem , Tumor de Wilms/diagnóstico por imagem , Dor Abdominal/etiologia , Criança , Detecção Precoce de Câncer , Feminino , Humanos , Lactente , Neoplasias Renais/complicações , Imageamento por Ressonância Magnética , Vômito/etiologia , Tumor de Wilms/complicaçõesRESUMO
PURPOSE: The risk factors for future infertility in adolescents with varicocele are controversial, and little is known about the association between hormone levels and semen parameters. Semen analysis is likely the closest marker of fertility but may be difficult to obtain in some boys secondary to personal, familial or religious reasons. Identifying other clinical surrogates for abnormal semen parameters may offer an alternative for assessing varicocele severity in these boys. We hypothesized that hormone levels and total testicular volume are predictive of abnormal total motile sperm count. MATERIALS AND METHODS: We retrospectively reviewed Tanner 5 boys with palpable left varicoceles who underwent a semen analysis and had serum hormone levels tested (luteinizing hormone, follicle-stimulating hormone, inhibin B, anti-müllerian hormone and/or total testosterone) within a 6-month period. Total testicular volume was also calculated. Abnormal total motile sperm count was defined as <9 million sperm per ejaculate. RESULTS: A total of 78 boys (median age 17.2 years, IQR 16.5-18.0) were included. Luteinizing hormone, anti-müllerian hormone and total testosterone were not correlated with any semen analysis parameter. There was a negative correlation between follicle-stimulating hormone and total motile sperm count (ρ -0.35, p=0.004) and positive correlation between inhibin B and total motile sperm count (ρ 0.50, p <0.001). Total testicular volume was significantly positively correlated with total motile sperm count (ρ 0.35, p=0.01). ROC analyses revealed an optimal follicle-stimulating hormone cutoff of 2.9, an optimal inhibin B cutoff of 204 and an optimal total testicular volume cutoff of 34.4 cc to predict abnormal total motile sperm count. CONCLUSIONS: Total motile sperm count is inversely associated with follicle-stimulating hormone levels, and directly associated with inhibin B levels and total testicular volume. Optimized cutoffs for serum follicle-stimulating hormone, inhibin B and total testicular volume may prove to be reasonable surrogates for total motile sperm count in boys who defer semen analysis for personal or religious/cultural reasons.