Malignant peripheral nerve sheath tumors (MPNSTs) are rare soft tissue sarcomas that arise from peripheral nerve fibers and are derived from Schwann cells, perineural cells, or fibroblasts. MPNST is ...an aggressive neoplasm in which local recurrence is common and complete excision of the mass should be the goal of surgery. We report a case of MPNST involving the penis in a 14-month-old boy. This is only the second reported case of penile MPNST without evidence of neurofibromatosis 1 and the first of which to occur in a patient this young.
To identify patient factors associated with a clinically significant improvement in semen parameters among infertile men treated with the aromatase inhibitor anastrozole.
Multi-institutional ...retrospective cohort study.
Two Tertiary Academic Medical Centers.
A total of 90 infertile men treated at 2 tertiary academic medical centers who met inclusion criteria and obtained pretreatment and posttreatment semen analyses.
Prescription of anastrozole (median 3 mg/wk).
Upgrade in the World Health Organization sperm concentration category (WHO-SCC). Univariate logistic regression, multivariable logistic regression, and partitioning analyses were performed to identify statistically significant patient factors capable of predicting treatment response.
With anastrozole treatment, 46% (n = 41/90) of men responded favorably with a WHO-SCC upgrade, and 12% (n = 11/90) experienced a downgrade. Responders exhibited lower pretreatment levels of luteinizing hormone (LH, 4.7 vs. 8.3 IU/L) and follicle-stimulating hormone (4.7 vs. 6.7 IU/mL), higher pretreatment levels of testosterone (T, 356 vs. 265 ng/dL), and similar baseline level of estradiol (E2, 73% vs. 70% with detectible level). Baseline semen parameters differed, with anastrozole responders demonstrating higher baseline semen concentration (3.6 vs. 0.3 M/mL) and higher total motile sperm counts (3.7 vs. 0.1 M). Anastrozole therapy converted 29% (n = 26/90) of the cohort to normozoospermia and enabled intrauterine insemination access in 31% (n = 20/64) of previously ineligible patients. Interestingly, neither body mass index nor the baseline E2 level or E2-T ratio was associated with WHO-SCC upgrade. Multivariable logistic regression revealed the T-LH ratio (odds ratio: 1.02, 95% confidence interval: 1.00–1.03) and baseline nonazoospermia (odds ratio: 9.4, 95% confidence interval: 1.1–78.9) to be statistically significant predictors of WHO-SCC upgrade (area under receiver operating characteristic curve: 0.77). The final user-friendly partitioning model consisting of the T-LH ratio ≥100 and baseline non-azoospermia was 98% sensitive and 33% specific for WHO-SCC upgrades (area under the curve: 0.77).
Anastrozole therapy decreases serum E2 levels, increases serum gonadotropins, and clinically improves semen parameters in half of men with idiopathic infertility. Nonazoospermic infertile men with T-LH ratios ≥100 are likely to benefit from anastrozole treatment irrespective of baseline E2 level or E2-T ratio. Men with azoospermia rarely respond to anastrozole and should be counseled on alternative treatments.
La testosterona y la hormona luteinizante predicen la mejoría en los parámetros seminales en hombres infértiles tratados con anastrazol
Identificar los factores de los pacientes asociados con una mejoría clínicamente significativa en los parámetros del semen entre hombres infértiles tratados con el inhibidor de la aromatasa anastrazol.
Estudio de cohorte retrospectivo multi institucional.
Dos centros médicos académicos terciarios.
Un total de 90 hombres infértiles tratados en 2 centros médicos académicos terciarios quienes cumplieron con los criterios de inclusión y obtuvieron análisis de semen previo y post tratamiento.
Prescripción de anastrazol (mediana 3 mg/semana).
Aumento en la categoría de concentración de semen de la Organización Mundial de la Salud (OMS-SCC). Se realizaron análisis de regresión logística univariada, regresión logística multivariable y partición para identificar factores estadísticamente significativos de los pacientes capaces de predecir la respuesta al tratamiento.
Con el tratamiento con anastrazol, el 46 % (n = 41/90) de los hombres respondieron favorablemente con un aumento en OMS-SCC y el 12 % (n = 11/90) experimentó una disminución. Los que respondieron exhibieron niveles previos al tratamiento más bajos de hormona luteinizante (LH, 4,7 versus 8,3 UI/L) y hormona folículo estimulante (4,7 versus 6,7 UI/mL), niveles más altos de testosterona previo al tratamiento (T, 356 versus 265 ng/dL) y nivel basal similar de estradiol (E2, 73% vs. 70% con nivel detectable). Los parámetros basales del semen difirieron, los que respondieron al anastrazol demostraron mayor concentración inicial de semen (3,6 frente a 0,3 M/ml) y mayores recuentos totales de espermatozoides móviles (3,7 frente a 0,1 M). La terapia con anastrazol convirtió el 29% (n = 26/90) de la cohorte a normozoospermia y permitió el acceso a la inseminación intrauterina en el 31% (n = 20/64) de pacientes previamente no elegibles. De manera interesante, ni el índice de masa corporal ni el nivel inicial de E2 o la tasa E2-T se asociaron con cambios en OMS-SCC. La regresión logística multivariable reveló que la relación T-LH (odds ratio: 1,02, intervalo de confianza del 95 %: 1,00–1,03) y la no azoospermia inicial (odds ratio: 9,4; intervalo de confianza del 95%: 1,1–78,9) son predictores estadísticamente significativos de la mejora del WHOSCC (área bajo la curva característica operativa del receptor: 0,77). El modelo de partición final fácil de usar que consta de la relación T-LH R100 y la no azoospermia inicial fueron 98 % sensibles y 33 % específicas para las mejoras de WHO-SCC (área bajo la curva:0,77).
La terapia con anastrazol disminuye los niveles séricos de E2, aumenta las gonadotropinas séricas y mejora clínicamente los parámetros seminales en la mitad de los hombres con infertilidad idiopática. Los hombres infértiles no azoospérmicos con ratios T-LH R100 probablemente se beneficien del tratamiento con anastrazol independientemente del nivel inicial de E2 o la relación E2-T. Los hombres con azoospermia rara vez responden al anastrazol y se les debe aconsejar tratamientos alternativos.
We aimed to identify predictor variables associated with pituitary abnormalities in hypogonadal men with mild hyperprolactinemia. We also sought to develop a decision-making aid to select patients ...for evaluation with pituitary magnetic resonance imaging.
We retrospectively examined men with mild hyperprolactinemia (15.1-50.0 ng/ml) who presented with symptoms of hypogonadism and underwent pituitary magnetic resonance imaging. Demographics, laboratory values and clinical data were obtained from the electronic medical record. Selected predictor variables were included in multivariable logistic regression and partitioning models. Cost avoidance analysis was performed on models achieving sensitivities ≥90%.
A total of 141 men were included in the study, of whom 40 (28%) displayed abnormalities on pituitary magnetic resonance imaging. Patients with pituitary abnormalities exhibited higher prolactin (p=0.01), lower testosterone (p=0.0001) and lower luteinizing hormone (p=0.03) levels than those with normal anatomy, as well as higher prolactin-to-testosterone ratios (p <0.0001) and lower luteinizing hormone-to-follicle-stimulating hormone ratios (p=0.0001). These serological variables were identified as the best performing predictor variables. The partition incorporating a prolactin-to-testosterone ratio cutoff of 0.10 and prolactin cutoff of 25 ng/ml achieved 90% sensitivity and 48% specificity, and reduced diagnostic expenses by 28%.
Hypogonadal men presenting with mild hyperprolactinemia and pituitary abnormalities declare themselves via endocrine studies routinely ordered to evaluate these conditions. The prolactin-to-testosterone ratio is the best independent predictor of finding a pituitary abnormality on magnetic resonance imaging, although sensitivity improves by referencing additional serological parameters. Significant cost avoidance may result from screening this population prior to ordering pituitary magnetic resonance imaging.
In men with impaired semen parameters, empiric medical therapies such as clomiphene citrate, a selective estrogen receptor modulator (SERM), and anastrozole, a selective aromatase inhibitor, are ...often employed. The effects of jointly administering these agents on semen parameters are not well understood. Here, we describe the findings of our multi-center, retrospective cohort study of men with idiopathic primary or secondary infertility. Twenty-one men were treated with combination therapy (anastrozole and clomiphene) and 69 men were treated with monotherapy (anastrozole). Patients with pre-treatment normozoospermia and recent or current exogenous testosterone therapy were excluded. Baseline and post-treatment semen and sex hormone parameters were compared among groups. The median follow-up duration was 91 days interquartile range (IQR), 64-117 days. Following treatment, 43% of men in the combination therapy group demonstrated normozoospermia, compared to 25% in the monotherapy group. Furthermore, men in the combined group demonstrated marked improvements in total motile sperm count (TMSC) 11.3
2.1 million (M), P=0.03. There were no significant differences in hormone levels among the two groups following treatment. Combination therapy with clomiphene citrate and anastrozole was associated with modest benefits in post-treatment semen parameters, when compared to anastrozole monotherapy. These benefits may contribute to improvements in pregnancy outcomes with less invasive assisted reproductive technologies, such as intrauterine insemination (IUI). Future investigations with larger sample sizes and prospective study designs are necessary.
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