Original Article - Pediatric Urology Korean J Urol 2015;56:318-323. http://dx.doi.org/10.4111/kju.2015.56.4.318 pISSN 2005-6737 • eISSN 2005-6745
Changes of calcific density in pediatric patients with testicular microlithiasis Bumjin Lim, Sang Hoon Song, Geehyun Song1, Kun Suk Kim Department of Urology, Asan Medical Center, University of Ulsan College of Medicine, Seoul, 1Department of Urology, Kangwon National University Hospital, Chuncheon, Korea
Purpose: Testicular microlithiasis (TM) is a relatively rare clinical entity of controversial significance characterized by the existence of hydroxyapatite microliths located in the seminiferous tubules. The aim of this study was to observe the natural course of changes in the calcific density of pediatric TM. Materials and Methods: We included a total of 23 TM patients undergoing scrotal ultrasound (US) on at least two occasions from July 1997 to August 2014. We retrospectively analyzed the patient characteristics, clinical manifestations, specific pathological features, and clinical outcomes. We measured the calcified area and compared the calcific density between the initial and final USs. Results: The mean age at diagnosis was 11.3±4.6 years, and the follow-up period was 79.1±38.8 months (range, 25.4–152.9 months). During the follow-up period, no patients developed testicular cancer. Calcific density on US was increased in the last versus the initial US, but not to a statistically significant degree (3.74%±6.0% vs. 3.06%±4.38%, respectively, p=0.147). When we defined groups with increased and decreased calcification, we found that diffuse TM was categorized into the increased group to a greater degree than focal TM (10/20 vs. 4/23, respectively, p=0.049). In addition, five of eight cases of cryptorchidism (including two cases of bilateral cryptorchidism) were categorized in the increased calcification group. Conclusions: Diffuse TM and cryptorchidism tend to increase calcific density. Close observation is therefore recommended for cases of TM combined with cryptorchidism and cases of diffuse TM. Keywords: Cryptorchidism; Gonadoblastoma; Testicular microlithiasis This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/by-nc/3.0) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.
INTRODUCTION The ultrasound (US) appearance of testicular microli thiasis (TM) was first described by Doherty et al. [1] in 1987. These microcalcifications can be identified on US as punctate, nonshadowing, echogenic foci [2]. The prevalence of TM has been reported to be between 2.4%–5.6% [3-5]. The prevalence of TM in symptomatic Korean men was found to be 6.0% with significant co-occurrence of TM, testicular
cancer, and infertility by Yee et al. [6] Although the cause-and-effect relationships are unclear, TM has been seen in patients with cryptorchidism, varicoce les, infertility, testicular torsion, Klinefelter syndrome, pul monary alveolar microlithiasis, neurofibromatosis, acquired immunodeficiency syndrome, intratubular germ cell neo plasia, and most importantly, primary testicular neoplasms [7]. Several authors have also reported the association of TM with testicular cancer. Ikinger et al. [8] reported that
Received: 2 February, 2015 • Accepted: 25 February, 2015 Corresponding Author: Kun Suk Kim Department of Urology, Asan Medical Center, University of Ulsan College of Medicine, 88 Olympic-ro 43-gil, Songpa-gu, Seoul 138-736, Korea TEL: +82-2-3010-3736, FAX: +82-2-477-8928, E-mail: [email protected] ⓒ The Korean Urological Association, 2015
318
www.kjurology.org
Pediatric testicular microlithiasis 74% of testes with tumors had associated ipsilateral TM on radiological inspection; whereas, only 8% of testicular spec im ens with benign conditions had microcalcifications. Although Chen et al. [9] reported that there was a significant difference in the rate of malignancy in males with TM compared with those without TM, the question remains whether TM independently increases the risk of testicular malignancy in Taiwanese Men. Ikinger et al. [8], after reporting an association between TM and testicular tumor specimens, suggested in 1982 that radiographic studies be incorporated into diagnosing TM because of the perceived risk for testicular cancer in testicles with microlithiasis. However, Ganem et al. [10] performed US follow-up in 9 of 22 patients with TM for a mean of 32 months without any newly developing tumors being diagnosed. Bennett et al. [11] and Skyrme et al. [12] reported similar results; therefore, a regular scrotal US is controversial in asymptomatic TM patients. The current recommendations, including those of the European Association of Urology, are that the presence of microlithiasis alone is not an indication for a regular scrotal US in the absence of other risk factors (size
Changes of calcific density in pediatric patients with testicular microlithiasis Bumjin Lim, Sang Hoon Song, Geehyun Song1, Kun Suk Kim Department of Urology, Asan Medical Center, University of Ulsan College of Medicine, Seoul, 1Department of Urology, Kangwon National University Hospital, Chuncheon, Korea
Purpose: Testicular microlithiasis (TM) is a relatively rare clinical entity of controversial significance characterized by the existence of hydroxyapatite microliths located in the seminiferous tubules. The aim of this study was to observe the natural course of changes in the calcific density of pediatric TM. Materials and Methods: We included a total of 23 TM patients undergoing scrotal ultrasound (US) on at least two occasions from July 1997 to August 2014. We retrospectively analyzed the patient characteristics, clinical manifestations, specific pathological features, and clinical outcomes. We measured the calcified area and compared the calcific density between the initial and final USs. Results: The mean age at diagnosis was 11.3±4.6 years, and the follow-up period was 79.1±38.8 months (range, 25.4–152.9 months). During the follow-up period, no patients developed testicular cancer. Calcific density on US was increased in the last versus the initial US, but not to a statistically significant degree (3.74%±6.0% vs. 3.06%±4.38%, respectively, p=0.147). When we defined groups with increased and decreased calcification, we found that diffuse TM was categorized into the increased group to a greater degree than focal TM (10/20 vs. 4/23, respectively, p=0.049). In addition, five of eight cases of cryptorchidism (including two cases of bilateral cryptorchidism) were categorized in the increased calcification group. Conclusions: Diffuse TM and cryptorchidism tend to increase calcific density. Close observation is therefore recommended for cases of TM combined with cryptorchidism and cases of diffuse TM. Keywords: Cryptorchidism; Gonadoblastoma; Testicular microlithiasis This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/by-nc/3.0) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.
INTRODUCTION The ultrasound (US) appearance of testicular microli thiasis (TM) was first described by Doherty et al. [1] in 1987. These microcalcifications can be identified on US as punctate, nonshadowing, echogenic foci [2]. The prevalence of TM has been reported to be between 2.4%–5.6% [3-5]. The prevalence of TM in symptomatic Korean men was found to be 6.0% with significant co-occurrence of TM, testicular
cancer, and infertility by Yee et al. [6] Although the cause-and-effect relationships are unclear, TM has been seen in patients with cryptorchidism, varicoce les, infertility, testicular torsion, Klinefelter syndrome, pul monary alveolar microlithiasis, neurofibromatosis, acquired immunodeficiency syndrome, intratubular germ cell neo plasia, and most importantly, primary testicular neoplasms [7]. Several authors have also reported the association of TM with testicular cancer. Ikinger et al. [8] reported that
Received: 2 February, 2015 • Accepted: 25 February, 2015 Corresponding Author: Kun Suk Kim Department of Urology, Asan Medical Center, University of Ulsan College of Medicine, 88 Olympic-ro 43-gil, Songpa-gu, Seoul 138-736, Korea TEL: +82-2-3010-3736, FAX: +82-2-477-8928, E-mail: [email protected] ⓒ The Korean Urological Association, 2015
318
www.kjurology.org
Pediatric testicular microlithiasis 74% of testes with tumors had associated ipsilateral TM on radiological inspection; whereas, only 8% of testicular spec im ens with benign conditions had microcalcifications. Although Chen et al. [9] reported that there was a significant difference in the rate of malignancy in males with TM compared with those without TM, the question remains whether TM independently increases the risk of testicular malignancy in Taiwanese Men. Ikinger et al. [8], after reporting an association between TM and testicular tumor specimens, suggested in 1982 that radiographic studies be incorporated into diagnosing TM because of the perceived risk for testicular cancer in testicles with microlithiasis. However, Ganem et al. [10] performed US follow-up in 9 of 22 patients with TM for a mean of 32 months without any newly developing tumors being diagnosed. Bennett et al. [11] and Skyrme et al. [12] reported similar results; therefore, a regular scrotal US is controversial in asymptomatic TM patients. The current recommendations, including those of the European Association of Urology, are that the presence of microlithiasis alone is not an indication for a regular scrotal US in the absence of other risk factors (size
