Penile Enhancement Using Biodegradable Scaffolds Covered with Platelet-rich Plasma-Fibrin Glue, Mesenchymal Stem Cells for Micropenis.

PURPOSE: Evaluation of preliminary cosmetic and functional outcomes of biodegradable scaffolds covered with platelet-rich plasma in penile girth augmentation. MATERIALS AND METHODS: Between June 2016 and June 2018, 36 males who had a mean age of 28.91 years (range 20 - 48 years) with micropenis underwent this procedure. A mixture of platelets-fibrin glue and mesenchymal cells obtained from dermal fat tissue were prepared. Then the mixture was seeded on the pretreated tube-shaped poly lactic-co-glycolic acid scaffold and underwent a whole day of incubation. Following penile degloving, scaffolds were surgically implanted within the interface region of dartos and Buck's fascia. The 5-point Likert scoring scale was used to evaluate the patients' satisfaction with surgery. RESULTS: Patients followed up for 6-12 (8 ± 2.86) months. The penile length in an erected state before surgery was 6.5 - 12.5 cm (9.08 ± 1.6) which enhanced to 7 - 14 cm (10.59 ± 1.71) after surgery (P < .0001). The penile girth before and after surgery were 8.49 ± 1.53 and 10.91 ± 1.96 cm, respectively (P < .0001). An augment in penile length and girth of 1.5 and 2.6 cm were achieved, respectively. Patients appraised surgical intervention on a rating of one to five. The highest possible score (5) was assigned by 27 %, 33 % expressed a very good mark (4), and 19 % gave a good mark (3). CONCLUSION: Covering the scaffold with a mixture of Platelets-Fibrin glue and mesenchymal cells seems a safe and feasible method for penile reconstruction surgery. More studies should be done to determine the effect of platelets- fibrin glue and mesenchymal cells for treating micropenis.

Initial clinical and molecular investigation of 20q13.33 microdeletion with 17q25.3/14q32.31q32.33 microduplication in Chinese pediatric patients.

BACKGROUND: Limited research has been conducted regarding the elucidation of genotype-phenotype correlations within the 20q13.33 region. The genotype-phenotype association of 20q13.33 microdeletion remains inadequately understood. In the present study, two novel cases of 20q13.33 microdeletion were introduced, with the objective of enhancing understanding of the genotype-phenotype relationship. METHODS: Two unrelated patients with various abnormal clinical phenotypes from Fujian province Southeast China were enrolled in the present study. Karyotype analysis and chromosomal microarray analysis (CMA) were performed to investigate chromosomal abnormalities and copy number variants. RESULTS: The results of high-resolution G-banding karyotype analysis elicited a 46,XY,der(20)add(20)(q13.3) in Patient 1. This patient exhibited various clinical manifestations, such as global developmental delay, intellectual disability, seizures, and other congenital diseases. Subsequently, a 1.0-Mb deletion was identified in the 20q13.33 region alongside a 5.2-Mb duplication in the 14q32.31q32.33 region. In Patient 2, CMA results revealed a 1.8-Mb deletion in the 20q13.33 region with a 4.8-Mb duplication of 17q25.3. The patient exhibited additional abnormal clinical features, including micropenis, congenital heart disease, and a distinctive crying pattern characterized by a crooked mouth. CONCLUSION: In the present study, for the first time, an investigation was conducted into two novel cases of 20q13.33 microdeletion with microduplications in the 17q25.3 and 14q32.31q32.33 regions in the Chinese population. The presence of micropenis may be attributed to the 20q13.33 microdeletion, potentially expanding the phenotypic spectrum associated with this deletion.

Evaluation of Z-plasty versus Heineke-Mikulicz scrotoplasty in the management of penoscrotal web in pediatric age group.

BACKGROUND: The penoscrotal web may be congenital or acquired following excessive ventral skin removal during circumcision. Several surgical techniques were described for the treatment of congenital webbed penis without a clear comparison between their outcomes. This prospective study aimed at comparing the surgical results of Z-scrotoplasty and Heineke-Mikulicz scrotoplasty in the treatment of congenital webbed penis in uncircumcised pediatric patients. METHODS: Our study included 40 uncircumcised patients who were divided randomly into two groups; Group A included 20 patients who were treated by Z-scrotoplasty and Group B included the other 20 patients who were treated by Heineke-Mikulicz scrotoplasty. All patients were circumcised at the end of the procedure. RESULTS: The surgical outcome was good without a significant difference between the two groups in 36 patients. Recurrent webbing developed in one patient of Group A and in three patients of Group B (FE p = 0.605) The only significant difference between the two groups was the operative duration which was shorter in Group B than in Group A (P < 0.001*). CONCLUSIONS: Treatment of congenital penoscrotal web in the pediatric age group could be done with either Z-scrotoplasty or Heineke-Mikulicz scrotoplasty with satisfactory results, however, without significant difference in the surgical outcomes. TRIAL REGISTRATION: • Registration Number: ClinicalTrials.gov ID: NCT05817760. • Registration release date: April 5, 2023.

Classes and predictors of reversal in male patients with congenital hypogonadotropic hypogonadism: a cross-sectional study of six international referral centres.

BACKGROUND: Although some male patients with congenital hypogonadotropic hypogonadism (CHH) undergo spontaneous reversal following treatment, predictors of reversal remain elusive. We aimed to assemble the largest cohort of male patients with CHH reversal to date and identify distinct classes of reversal. METHODS: This multicentre cross-sectional study was conducted in six international CHH referral centres in Brazil, Finland, France, Italy, the UK, and the USA. Adult men with CHH (ie, absent or incomplete spontaneous puberty by age 18 years, low serum testosterone concentrations, and no identifiable cause of hypothalamic-pituitary-gonadal [HPG] axis dysfunction) were eligible for inclusion. CHH reversal was defined as spontaneous recovery of HPG axis function off treatment. Centres provided common data elements on patient phenotype, clinical assessment, and genetics using a structured, harmonised data collection form developed by COST Action BM1105. Latent class mixture modelling (LCMM) was applied to establish whether at least two distinct classes of reversal could be identified and differentially predicted, and results were compared with a cohort of patients without CHH reversal to identify potential predictors of reversal. The primary outcome was the presence of at least two distinct classes of reversal. FINDINGS: A total of 87 male patients with CHH reversal and 108 without CHH reversal were included in the analyses. LCMM identified two distinct reversal classes (75 [86%] in class 1 and 12 [14%] in class 2) on the basis of mean testicular volume, micropenis, and serum follicle-stimulating hormone (FSH) concentration. Classification probabilities were robust (0·998 for class 1 and 0·838 for class 2) and modelling uncertainty was low (entropy 0·90). Compared with class 1, patients in class 2 had significantly larger testicular volume (p<0·0001), no micropenis, and higher serum FSH concentrations (p=0·041), consistent with the Pasqualini syndrome (fertile eunuch) subtype of CHH. Patients without CHH reversal were more likely to have anosmia (p=0·016), cryptorchidism (p=0·0012), complete absence of puberty (testicular volume <4 cm³; p=0·0016), and two or more rare genetic variants (ie, oligogenicity; p=0·0001). Among patients who underwent genetic testing, no patients (of 75) with CHH reversal had a rare pathogenic ANOS1 variant compared with ten (11%) of 95 patients without CHH reversal. Individuals with CHH reversal had a significantly higher rate of rare variants in GNRHR than did those without reversal (nine [12%] of 75 vs three [3%] of 95; p=0·025). INTERPRETATION: Applying LCMM to a large cohort of male patients with CHH reversal uncovered two distinct classes of reversal. Genetic investigation combined with careful clinical phenotyping could help surveillance of reversal after withdrawing treatment, representing the first tailored management approach for male patients with this rare endocrine disorder. FUNDING: National Institutes of Health National Center for Advancing Translational Sciences; Ministry of Health, Rome, Italy; Ministry of University, Rome, Italy; National Institutes of Health Eunice Kennedy Shriver National Institute of Child Health and Human Development; and the Josiah Macy Jr Foundation. TRANSLATION: For the Italian translation of the abstract see Supplementary Materials section.

Surgical correction of congenital megaprepuce: a pilot study.

OBJECTIVE: Congenital megaprepuce (CMP) is a rare penile deformity that usually requires surgical correction. This study was performed to examine the efficacy of the modified Sugita procedure for repairing CMP in pediatric patients. METHODS: We retrospectively analyzed the clinical data of pediatric patients with CMP treated by a surgeon using the modified Sugita procedure in our hospital from January 2019 to April 2021. RESULTS: Twenty patients were enrolled, and their median age at surgery was 70.5 months (range, 60-96 months). All surgeries were successful, and no complications occurred during the operation. The postoperative foreskin had moderate edema in five patients, and soaking in 10% hypertonic saline resulted in disappearance of the edema within 4 to 8 weeks. The follow-up duration was 6 to 20 months (median, 10 months). No other complications occurred, such as dehiscence or hematoma. CONCLUSIONS: The modified Sugita procedure for correction of CMP produces excellent cosmesis and a low complication rate. Our study indicates that the modified Sugita procedure is a safe and feasible treatment option.

Individualized care for patients with intersex (differences of sex development): Diagnosis and treatment of aphallia.

This review discusses issues and concerns in the management of aphallia, updating status of a post-pubertal individual who required further surgery after having initial surgery for aphallia as an infant. Through this case, which discusses an 18-year-old young adult who had penile agenesis, who desired further phalloplasty involving glanuloplasty and implantation of an erectile device, we highlight the importance of periodic evaluation and close follow up. Surgery during infancy or early childhood to create a penis is important for gender development in a boy, especially if there were functional testes during fetal life, even if this surgery would only be the first stage. There is a strong probability of subsequent surgery after initial phalloplasty before puberty, even with the use of currently refined techniques. Here we discuss the changing techniques that document the ongoing, continued refinement of these procedures, highlighting that further outcome data are needed to identify ways to further optimize current techniques.

An epidemiologic study of penoscrotal transposition by maternal characteristics using data from the Texas birth defects registry.

BACKGROUND: Penoscrotal transposition (PST) is an uncommon urogenital malformation in which the penis is mal-positioned to be inferior to the scrotum. The purpose of this study was to explore PST risk by maternal characteristics and to describe co-occurring congenital abnormalities in the Texas Birth Defects Registry (TBDR). METHODS: We conducted a population-based descriptive study examining occurrence of PST in the TBDR between 1999 and 2019. The primary outcome variable was PST diagnosis during infancy. Descriptive variables included maternal age, education, and race/ethnicity. Prevalence ratios (PRs) were calculated within each maternal variable category using Poisson regression. Counts and percentages of cases with select co-occurring congenital abnormalities were also calculated. RESULTS: Overall, 251 infants had PST, providing a prevalence of 0.61/10,000 live male births (95% CI: 0.53-0.68). PST prevalence was significantly lower among infants of mothers who had lower educational attainment (high school), who were younger (<25 vs. 25-34), and who were Hispanic (vs. non-Hispanic White) and was significantly higher among older mothers (35+ vs. 25-39). Hypospadias was the most common co-occurring genitourinary anomaly, affecting close to 70% of cases. CONCLUSIONS: To our knowledge, this is the first investigation exploring the prevalence of PST in a population-based birth defects registry. Our findings help to understand the risk for PST among select maternal demographic characteristics and may assist in generating hypotheses about the underlying etiology of this condition for future work.

Shaeer's corporal rotation IV: length-preserving correction of congenital ventral penile curvature.

BACKGROUND: For cases with severe penile curvature, the loss in length with shortening techniques or the loss in rigidity with incision grafting can compromise the results, hence the advent of Shaeer's corporal rotation III technique, which corrects the most severe degrees of ventral penile curvature without loss in length, though with a certain degree of narrowing. AIM: We sought to describe Shaeer's corporal rotation IV, aiming at correction of moderate-to-severe ventral penile curvature with minimal shortening, minimal narrowing, and minimal mobilization of the neurovascular bundle, among other improvements such as using thicker suture material to decease recurrence while inverting the surgical knots. METHODS: Forty-two patients with congenital ventral penile curvature were selected for the study with a curve ranging from 60° to 90°. Patients were randomized into 1 of the 2 groups: Shaeer's corporal rotation III (SCR-III) and SCR-IV. In SCR-IV, rotation is used to correct the main brunt of curvature, up to 70% to 80% of the curve. Plication is used to correct the residual curvature up to 100% straightness. This keeps the rotation points fewer and closer to the midline, thereby minimizing narrowing and mobilization of the neurovascular bundle. OUTCOMES: Both groups were compared with regard to intraoperative erection angle, length, and girth, before and after rotation, as well as subjectively reported postoperative recurrence, penile sensitivity, satisfaction, and IIEF. RESULTS: The postcorrection angle was zero for all cases in the 2 groups. Dorsal length decreased by 3% in the SCR-IV group compared with 0.5% in the SCR-III group (2.5% difference). The difference in circumference between the narrowest and widest points was 2% in the SCR-IV group vs 9.3% in the SCR-III group (7.3% difference). The average operative time was 19.2% shorter with SCR-IV. Girth asymmetry was reported in 1 (4.8%) of 21 patients in the SCR-IV group compared with 15 (71.4%) of 21 in the SCR-III group. Partial hyposensitivity of the penis was reported in 9.5% in the SCR-IV group compared with 19% in the SCR-III group. CLINICAL IMPLICATIONS: SCR-IV is an improvement over former versions of the technique, with higher patient satisfaction. STRENGTHS AND LIMITATIONS: A strength of the study is the long follow-up period. Limitations include being a single-center study and reliance on patient reporting to evaluate recurrence and satisfaction. CONCLUSION: The SCR-IV technique corrects moderate and severe degrees of congenital ventral penile curvature, with little or no compromise in penile length, girth, or sensitivity.

The factors that cause penile shortening after plication surgery in patients with congenital penile curvature: [[es]]Los factores causantes del acortamiento de pene después de la cirugía con plicatura en pacientes con incurvación peneana congénita.

OBJECTIVE: Congenital penile curvature is defined as the non-straightness of the penis without any urethral or penile pathology. We aimed to evaluate the factors that cause penile shortening after plication surgery in patients with congenital penile curvature. METHODS: Between November 2010 and December 2020, we retrospectively reviewed patients with CPC undergoing tunica albuginea plication surgery. Before the procedure, patients' age, curvature location and degrees, as well as penile length were recorded. After the treatment, penile lengths were measured and recorded again. Early and late period results were recorded. RESULTS: Plication surgery was performed in 130 patients. The median age was 24 years. Seventy-six patients had ventral curvature, 22 had dorsal curvature, 32 had lateral curvature. Average shortening of penile length in patients with curvature below 30° was: ventral 8-16 mm, dorsal 6-13 mm, lateral 5-12 mm. Patients with curvatures above 30° were: ventral 12-22 mm, dorsal 8-20 mm, lateral 2-12 mm. CONCLUSION: Penile length shortening after plication is inevitable. Curvature degree and direction are factors affecting penile length after surgery. Therefore, patients and relatives should be informed in more detail about this complication.

Spectrum and classification of megameatus anomalies.

OBJECTIVE: To define and classify megameatus anomalies, the parameters of a considerable number of cases were investigated and compared with those of normal children. METHODS: A total of 1150 normal babies were examined during routine nonmedical circumcision, and another 750 boys referred with hypospadias were examined during the previous 3 years. All patients were evaluated and assessed for the size, location, and configuration of the urinary meatus, and penile length and girth were measured. Children with normal size and location of the meatus were considered control group A, and 42 cases of different forms of megameatus were considered group B. Other penoscrotal, urinary, and general anomalies were examined and investigated accordingly. All data were analyzed by the SPSS 9.0.1 statistical package and compared by paired t tests. RESULTS: Forty-two uncircumcised patients aged from 1 month to 4 years (mean 18 months) were diagnosed with a urinary meatus that engrossed the whole ventral or dorsal aspects of the glans, exceeding half the width of the glans or penile girth with the complete vanishing of the glans closure in most cases. Megameatus is usually associated with the abnormal meatal position as hypospadiac, orthotopic, or epispadic. Additionally, megameatus may be associated with a normally intact or deficient prepuce. Consequently, we had four categories of megameatus, and the intact prepuce orthotopic megameatus subcategory has not been described before. Megameatus was also detected with deficient prepuce, and this was considered a hypospadiac variant. CONCLUSION: Megameatus is diagnosed precisely with penile biometry and is classified into 4 groups: hypospadiac, epispadic, and orthotopic or central, either with or without intact prepuce. This classification is applicable for expansion to other centers.

Pediatric urologists' confidence and accuracy in estimating penile curvature.

BACKGROUND: Assessment and management of congenital penile curvature (PC) can be variable. Methods for correction of PC usually are dependent on degree of PC which is reliant on how degree is assessed. We sought to assess the confidence and accuracy of measuring PC and hence management using case-based examples. METHODS: A survey was emailed to members of the Societies for Pediatric Urology. Demographic information, management strategies for PC, and self-reported confidence in measuring PC were assessed. A Likert scale measured self-confidence. Case scenarios were used to assess ability to measure PC and methods of correction. The cases consisted of three computer-generated penis model images with arc-type ventral curvature and one image of lateral curvature in an infant. RESULTS: The response rate was 30% (108/355). The mean confidence score was 3.6 ± 0.8 (3-fairly confident; 4-very confident). In clinic, 89% of urologists used eyeball estimates to assess PC; 5% used both eyeball and goniometer. In the operating room, 71% used eyeball estimates, 8% used goniometer, and 16% used both. If sole decision-maker, urologists recommend surgical correction of PC over observation at median 30° (IQR 21-30°). At a median of 45°, there was a shift in corrective surgical preference from dorsal plication (DP) (IQR 30-54°) to ventral lengthening (IQR 34-60°). Urologists underestimated PC degree for all cases (summary table). For all cases, there was no association between years in practice or confidence level on estimated PC degree. In case 1, only 24% of urologists would correct a mean estimate of 23° PC; those who would correct had a higher mean PC estimate vs those who would not (28° vs 21°, p < 0.001). Case 2 and 4 had similar estimations and correction methods. In case 2, those who chose VL had a higher mean PC estimate vs those who did not (43° vs 37°, p < 0.01), but no estimate difference was seen for DP (p = 0.52). In case 4 with lateral PC, those who chose DP had a higher mean PC estimate vs those who did not (41° vs 33°, p = 0.049). Yet in case 3, there was no difference in PC estimate in urologists who chose VL vs not (57° vs 53°, p = 0.16). CONCLUSIONS: A uniform underestimation of PC existed despite self-reported confidence in the ability to measure PC. An increasing willingness to perform surgical correction was demonstrated with a shift towards VL for ventral curvature and less so for lateral curvature as PC worsens.

Scrotal flap phalloplasty as temporary neophallus in infants and children with penile agenesis: Multi-institutional experience and long-term follow-up.

INTRODUCTION: Aphallia is a rare congenital disorder pertaining to genotypic males. Early surgical creation of a neophallus is recommended to reinforce the child's male gender-identity, favoring proper psychosexual development. Modern microsurgical techniques used to create a neophallus in adults are not recommended in children due to the invasiveness and complexity of the procedures, along with high complication rates. Scrotal flap phalloplasty is a simple and reproducible technique to create a temporary neophallus in prepubertal boys with aphallia. OBJECTIVE: We present a multi-institutional experience, ten years after the initial description of the scrotal flap phalloplasty (SFP) technique, in which a flap from the well-developed scrotum is used to build a temporary neophallus, without obvious scars in patients with aphallia. STUDY DESIGN: The records of surgical neophalloplasty for aphallia patients from 4 centers between 2011 and 2021 were reviewed. All patients had at least one year follow-up to assess for short and long-term complications. Age at initial operation, associated anomalies, and other related surgical procedures were analyzed. RESULTS: The post-operative aesthetic result in all patients was satisfactory and has been maintained in the long-term follow-up, with all patients presenting a cylindrical structure resembling an uncircumcised penis, without evidence of significant contraction or loss of length. (Summary Figure) DISCUSSION: Non-microsurgical neophalloplasty techniques in patients with penile agenesis are temporary procedures that help to establish the body image and preserve the psychosexual development of the patient with aphallia. These techniques do not involve tissue transplant from a distant region, and are simpler to perform, with less scarring at the donor sites. Due to significant donor scars and considerable morbidity and complexity associated with the definitive phalloplasty techniques, we created a simple, reproducible and straightforward procedure to serve as a temporary neophallus in young boys with aphallia. As affected patients usually have a well-formed scrotum with normal and orthotopic testicles, it is the ideal donor site for a temporary neo-phallus in childhood. Furthermore, other donor sites are preserved for a definitive phalloplasty. There are limitations to this study, as quality of life could not be assessed and psychological or gender-identity investigations have not been carried out. None of these children have reached puberty, and hence decision and outcomes of definitive neophallus reconstruction has not been considered to date. CONCLUSION: Scrotal flap phalloplasty is a minimally invasive, simple and reproducible technique used to create a temporary neophallus in boys with aphallia, while waiting for definitive reconstructive surgery after puberty.

Fasciocutaneous foreskin twin flaps in hypospadias repair: Reconstructing a functional penis with freestyle design.

BACKGROUND: Hypospadias is the most common congenital urological deformity in males; however, there is still no perfect solution or surgical method for one-stage operations to fix proximal hypospadias with ventral penile curvature (VPC). We developed a fasciocutaneous foreskin twin flap technique that can provide freestyle flap designs in hypospadias repair. METHODS: A retrospective study including 26 patients was performed between January 2017 and June 2020. The surgical procedures included VPC correction, skin flap design, transfer and reconstruction. The urethra and shaft were reconstructed by twin flaps in one stage. All patients were followed up until 12 months. RESULTS: All VPC cases were corrected after the operation. The fistula rate was 11.5% (3/26). No meatal stenosis, urethral diverticulum, or stricture was observed. A total of 92.3% (24/26) of patients were satisfied with their penis appearance. CONCLUSIONS: The fasciocutaneous foreskin twin flaps provide freestyle designs in hypospadias repair, which can achieve a one-stage operation for proximal hypospadias with low fistula rates.

Quantification of the Androgen and Estrogen Receptors in the Penile Tissues of Hypospadias in Comparison with Normal Children.

INTRODUCTION: Hypospadias is a common congenital abnormality typified by a proximally placed ectopic urethral meatus along the ventral surface of the penis. Androgen receptor (AR) and estrogen receptor (ER) expression in the hypospadias tissues may be altered in hypospadias. METHODOLOGY: We evaluated by immunohistochemistry the AR and ER expression in 75 tissues from hypospadias repair, and compared this expression to that of tissue from 75 patients undergoing circumcision. We also compared the intensity of AR and ER expression between different severities of hypospadias. RESULTS: AR quantitative grading score decreased with severity of hypospadias, while the ER score increased as the hypospadias worsened, which was statistically significant (p-value <0.05). CONCLUSION: The penile tissue AR expression is decreased and ER expression is increased with increasing severity of hypospadias.

Severe penile torsion of 180 degrees in an adult patient: a uro-radiological case report.

Penile torsion is the abnormal three-dimensional twisting of penile corporal bodies. It can be classified as mild, moderate, or severe, depending on the degree of torsion. Severe penile torsion (>90°) is a very rare condition, with an estimated incidence of 0.4%-1% among all penile torsion cases. Our patient was a 37-year-old man complaining of a 2-year history of lower urinary tract symptoms. These symptoms appeared after the patient sustained an iatrogenic injury during Foley catheter insertion. Physical examination incidentally revealed an obvious counterclockwise penile rotation of 180°. Several theories have been proposed to explain the etiology of penile torsion, including theories based on genetic factors, abnormal urethral development, and abnormal attachment of the dartos fascia to the skin. Penile torsion may be associated with other penile anomalies, including chordee, hypospadias, and epispadias; however, it is often detected as an isolated finding. Clinical examination is sufficient to confirm its diagnosis without the need for further imaging. While no standardized procedure has been indicated for all penile torsion cases, the severity of torsion and the presence of other anomalies determine the most suitable procedure. No reports on the imaging features of penile torsion (irrespective of the degree of torsion) are available. We present the first such report on the imaging features, including advanced magnetic resonance imaging findings, of a 180° penile torsion in an adult patient.

CONGENITAL SCAPHOID MEGALOURETHRA: REVISITING A RARE ANOMALY.

Megalourethra is an infrequent malformation of the anterior urethra that is caused by the lack of corpus sponigosum and in severe cases is accompanied by the lack of corpora cavernosa as well. We report a five-year-old boy presented to us with scaphoid variety of megalourethra having complaints of ballooning of urethra during voiding. He was investigated with urethrogram and cystoscopy and subsequently repaired with Nesbitt Longitudinal Reduction Urethroplasty. He had smooth recovery postoperatively with normal voiding stream on follow up.

Buried penis secondary to ectopic scrotum. / Pene oculto secundario a ectopia escrotal.

INTRODUCTION: Ectopic scrotum is a rare clinical entity, potentially associated with other congenital abnormalities. We present the case of a patient with buried penis secondary to ectopic scrotum. The surgical technique was described, and a literature review was carried out. CLINICAL CASE: 1-year-old patient with ectopic right hemiscrotum and the testes within the scrotal sac. A double Z-plasty was performed with two flaps - one above the penis, surrounding the ectopic scrotum, and the other one at the scrotum to modify the bifid scrotum. The upper flap was rotated downwards, which allowed ectopy to be repaired, and the lower flap was used to repair bifidity. No postoperative complications were recorded. Follow-time was 6 months, with good final cosmetic results. DISCUSSION: Ectopic scrotum is an infrequent congenital malformation. Cutaneous rotation flaps with Z-plasties are a valid treatment option, with good long-term cosmetic results.

INTRODUCCION: La ectopia escrotal constituye una entidad clínica rara, que puede asociar otras anomalías congénitas. Presentamos el caso de un paciente con un pene oculto secundario a una ectopia escrotal, con descripción de la técnica quirúrgica y revisión de la literatura. CASO CLINICO: Paciente de un año de vida que presentaba un hemiescroto derecho ectópico con testes en bolsa. Se diseñó una doble Z-plastia con realización de dos colgajos, uno suprapeneano rodeando el escroto ectópico y otro escrotal para modificar el escroto bífido. El colgajo superior se rotó hacia abajo corrigiendo la ectopia y el colgajo inferior corregió la bifidez. No se produjeron complicaciones posoperatorias. El tiempo de seguimiento fue de seis meses con buen aspecto estético final. COMENTARIOS: El escroto ectópico es una malformación congénita infrecuente. Los colgajos de rotación cutáneos con Z-plastias son una opción válida de tratamiento con buenos resultados estéticos a largo plazo.