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Background: Anogenital distance (AGD) in animals is a sensitive biomarker of fetal endocrine disruption and the associated testicular dysgenesis syndrome (TDS). However, AGD in human infants with cryptorchidism and hypospadias, which are potential manifestations of TDS during childhood, is not clearly described. Objective: Our aim was to compare AGD in boys with cryptorchidism or hypospadias against normative data. Methods: Boys with isolated cryptorchidism (n = 71, age 13.4 ± 5.8 months) or hypospadias (n = 81, age 11.4 ± 6.2 months) were recruited from a tertiary center for measurement of AGD and penile length; they were compared with 487 healthy full-term boys from a birth cohort by deriving age-specific standard deviation scores (SDS). Results: Boys with cryptorchidism were older (p = 0.048) compared with boys with hypospadias. Boys with hypospadias had shorter mean AGD and penile length SDS than healthy boys (both p < 0.0001). Mean AGD and penile length SDS values in boys with cryptorchidism were longer than mean values in boys with hypospadias (both p < 0.01) and shorter than mean values in healthy boys (both p < 0.0001). Mean penile length SDS decreased as the severity of hypospadias increased (ptrend = 0.078). Conclusions: In the study population, AGD and penile length were reduced in boys with hypospadias or cryptorchidism relative to normative data derived from a longitudinal birth cohort. The findings support the use of AGD as a quantitative biomarker to examine the prenatal effects of exposure to endocrine disruptors on the development of the male reproductive tract. Citation: Thankamony A, Lek N, Carroll D, Williams M, Dunger DB, Acerini CL, Ong KK, Hughes IA. 2014. Anogenital distance and penile length in infants with hypospadias or cryptorchidism: comparison with normative data. Environ Health Perspect 122:207–211; http://dx.doi.org/10.1289/ehp.1307178

Objective To review the current management of boys with bilateral anorchia and assess whether surgical exploration is necessary when endocrine investigation indicates absent testicular function. Patients and methods The medical records of 11 boys being managed for bilateral anorchia were reviewed in relation to clinical presentation, pituitary-gonadal function, surgical and histological findings. Results All boys had absence of testicular function based on undetectable levels of serum anti-Mullerian hormone, elevated basal or peak follicle-stimulating hormone and luteinising hormone levels and no testosterone response to human chorionic gonadotrophin stimulation. All boys underwent abdominal exploration, ten of whom showed no macroscopic signs of testis tissue, confirmed histologically in seven. Histology was not available in the remaining three boys. Abnormally small intra-abdominal testes were found bilaterally in one boy. These were sited in the scrotum at orchidopexy but had subsequently atrophied. Endocrine tests confirmed absent testicular function. Conclusion Based on the high degree of concordance between the surgical and histological findings and the results of the endocrine tests, it is suggested that surgery is unnecessary in bilateral anorchia when endocrine tests confirm the absence of functioning testicular tissue.