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Testis-Sparing Surgery for Non-Palpable Leydig Cell Tumors in Prepubertal Children

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Testis-Sparing Surgery for Non-Palpable Leydig Cell Tumors in Prepubertal Children Case Report Testis-Sparing Surgery for Non-Palpable Leydig Cell Tumors in Prepubertal Children Vassilis Lambropoulos 1,* , Antonios Theodorakopoulos 2, Vasileios Mouravas 1, Elissavet Pazarli 3, Dimitrios Godosis 1, Chrysostomos Kepertis 1 , Kleanthis Anastasiadis 1 and Ioannis Spyridakis 1 1 2nd Paediatric Surgery Department, Aristotle University of Thessaloniki, “Papageorgiou” General Hospital, 56403 Thessaloniki, Greece; [email protected] (V.M.); [email protected] (D.G.); [email protected] (C.K.); [email protected] (K.A.); [email protected] (I.S.) 2 Radiology Department, “Papageorgiou” General Hospital, 56403 Thessaloniki, Greece; [email protected] 3 Pathology Department, “Papageorgiou” General Hospital, 56403 Thessaloniki, Greece; [email protected] * Correspondence: [email protected]; Tel.: +30-6977224420 Received: 17 January 2020; Accepted: 16 March 2020; Published: 26 October 2020 Abstract: Leydig cell tumor (LCT) is an infrequent stromal neoplasm of the testis with an incidence of less than 3% of all gonadal tumors in the general male population. Only 25% is found in prepubertal children, where Leydig cell tumors are always reported benign. The hospital records of two prepubertal male children, who underwent organ sparing surgery for testicular LCT the last five years, were retrospectively reviewed. In both of them, the lesion was incidentally found during a scrotal ultrasonography for testicular pain. The diagnosis of a benign LCT was based on the pre-operative physical examination and imaging (Ultrasound-US, Magnetic Resonance Imaging-MRI) as well as the negative tumor markers. A testicle-sparing procedure was decided and the pathologic examination of the surgical specimen confirmed the diagnosis. No tumor recurrence was noted on follow-up. Testis-sparing surgery provides the possibility of complete excision of such lesions and should be considered as the treatment of choice. Keywords: Leydig cell tumor; testis; children; organ sparing surgery 1. Introduction Leydig cell tumor (LCT) is an infrequent stromal neoplasm of the testis with an incidence of less than 3% of all gonadal tumors in the general male population [1,2]. In children of prepubertal age, the incidence increases up to 9% of all primary testicular masses [3]. In the adult population, 10% exhibits malignant behavior in contrast to the childhood where Leydig cell tumors are always reported benign [4–6]. Usually it occurs in adulthood and only 25% is found in prepubertal children [7]. In most cases, it is an incidental finding during a scrotal sonographic evaluation for other reasons, since in childhood is usually presented as a small sized, non-palpable intratesticular lesion. If hormonally active, they secrete a variety of hormones which may give an onset of endocrine signs, usually before the development of a palpable mass [8]. On scrotal ultrasound (US), these lesions appear as hypoechogenic masses [9]. Contrast-enhanced Magnetic Resonance Imaging (MRI) is essential for the differential diagnosis [10]. Tumor markers such as alpha-fetoprotein (AFP), beta human chorionic gonadotropin (bhCG) and lactate dehydrogenase (LDH) are usually within normal values, in small non palpable lesions in children. Testis-sparing surgery should be considered as the treatment of choice for such cases [6]. Pediatr. Rep. 2020, 12, 86–92; doi:10.3390/pediatric12030020 www.mdpi.com/journal/pediatrrep Pediatr. Rep. 2020, 12 87 Pediatr. Rep. 2020, 12, FOR PEER REVIEW 2 2. Case Case Reports Reports The hospital records of two prepubertal male children who underwent organ sparing surgery forfor testicular LCT the last five five years were retrospectively reviewed. Their Their age age was was 10 10 and 11 years old, respectively, and both of them underwent a scrotal US exam due to testicular pain, which could not be be related related to to any any pathology pathology on on clinical clinical examination, examination, as the as thefirst first child child was wascomplaining complaining about about pain onpain the on ipsilateral the ipsilateral side of side the of lesion the lesion and the and latter the latterto the tocontralateral the contralateral side. Physical side. Physical examination examination of the externalof the external genitalia genitalia in both in patients both patients revealed revealed normal normal testis testisin position, in position, size and size consistency. and consistency. No palpableNo palpable masses masses were were noted. noted. The testicular The testicular sonographic sonographic findings findings revealed revealed a hypoechoic a hypoechoic mass with mass a diameterwith a diameter of 3 and of 33.4 and mm, 3.4 mm,respectively. respectively. In both In both cases, cases, there there was was a aclear clear demarcation demarcation from from the surrounding normal normal testicular testicular tissue, tissue, and and on on Color Color Doppler Doppler evaluation evaluation they they both both presented presented a rim a rim of marginalof marginal blood blood flow. flow. No Nointratesticular intratesticular calcifications calcifications were were noted noted (Figure (Figure 1). Due1). to Due the to nodule’s the nodule’s small dimensions,small dimensions, the position the position coordinates coordinates of the lesion of the were lesion determined were determined in three inaxes, three transverse, axes, transverse, sagittal andsagittal coronal and coronalso that the so that surgeon the surgeon could have could a have dissection a dissection plane planecorresponding corresponding to the toUS the findings. US findings. Our patientsOur patients underwent underwent a scrotal a scrotal MRI in MRI order in orderto evaluate to evaluate the intratesticular the intratesticular mass and mass contribute and contribute to the surgicalto the surgical treatment. treatment. The lesions The were lesions well were circumscribed well circumscribed isointense isointense on T1‐WI, onhypointense T1-WI, hypointense on T2‐W1 and on demonstratedT2-W1 and demonstrated intense enhancement intense enhancement after intravenous after administration intravenous administration of contrast material of contrast (Figure material 2). A preoperative(Figure2). A preoperative diagnostic orientation diagnostic orientationtowards LCT towards was LCTmade. was Both made. males Both were males evaluated were evaluated by a pediatricby a pediatric endocrinologist endocrinologist and andno nohormonal hormonal or ordevelopmental developmental abnormalities abnormalities were were found. found. Tumor Tumor markers were within normal range. No No intra intra-abdominal‐abdominal or or inguinal inguinal lymph lymph nodes nodes were were found on US exam and chest chest X X-rays‐rays were were normal. normal. The The surgeon’s surgeon’s choice choice of of testicle testicle sparing sparing surgery surgery had had the the parents’ parents’ consent in both cases. Figure 1. 1. UltrasoundUltrasound (US) (US) images, images, in in B B ( (AA)) and and color color Doppler Doppler mode mode ( (BB),), depicting depicting a a hypoechoic hypoechoic lesion lesion with intense vascularization due to a prominent peripheral arterial vessel. Pediatr. Rep. 2020, 12 88 Pediatr. Rep. 2020, 12, FOR PEER REVIEW 3 Figure 2. (A(A) )Magnetic Magnetic resonance resonance imaging imaging (MRI) (MRI) depicting depicting the the lesion lesion as isointense as isointense on T1 on‐WI, T1-WI, (B) hypointense(B) hypointense on onT2‐ T2-WI,WI, (C) ( Cand) and with with intense intense enhancement enhancement after after administration administration of of endovenous contrast material. Through a standardstandard inguinalinguinal incision, incision, the the spermatic spermatic cord cord was was isolated isolated and and clamped clamped at at the the level level of ofthe the internal internal inguinal inguinal ring, ring, using using a soft a soft vascular vascular clamp. clamp. The The affected affected testis testis was was then then delivered delivered into into the woundthe wound and placedand placed into a separateinto a separate operative operative field in order field to in avoid order tumor to avoid spillage. tumor The gubernaculumspillage. The gubernaculumtestis was sectioned, testis andwas the sectioned, tunica albuginea and the tunica was opened albuginea longitudinally, was opened having longitudinally, in mind a dissectionhaving in planemind a corresponding dissection plane to thecorresponding US findings. to Thethe US lesion findings. was easily The lesion recognized was easily due to recognized its characteristic due to itsmacroscopic characteristic appearance macroscopic as a yellow appearance to soft as brown a yellow mass, to well soft demarcated brown mass, from well the demarcated surrounding from normal the surroundingtesticular tissue. normal The noduletesticular as welltissue. as aThe 2–5 nodule mm rim as of well normal as a tissue 2–5 mm around rim wereof normal excised tissue and around sent for pathologicwere excised examination. and sent for The pathologic tunica albuginea examination. was repaired The tunica using albuginea a 6.0 absorbable was repaired running using suture a and 6.0 absorbablethe vascular running clamp was suture removed and the (Figure vascular3). The clamp ischemia was timeremoved was recorded (Figure 3). and The did ischemia not exceed time 30 minwas (28recorded and 22 and min, did respectively). not exceed 30 The min organ (28 and was 22 re-inserted min, respectively). into the scrotum. The organ On was discharge, re‐inserted both patientsinto the werescrotum. evaluated
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