- Email: [email protected]
Mature Testicular Teratoma in Children: Multifaceted Tumors on Ultrasound
Pediatric Urology Mature Testicular Teratoma in Children: Multifaceted Tumors on Ultrasound Matias Epifanio, Marilisa Baldissera, Fernando G. Esteban, and Matteo Baldisserotto OBJECTIVE MATERIALS AND METHODS
RESULTS
CONCLUSION
To describe the different ultrasound (US) findings of mature testicular hamartomas in children. This is a retrospective study from January 2000 to July 2012 that reviewed the clinical, laboratory, and US findings of 7 children with a pathologic diagnosis of mature teratoma of the testis. All patients in the study received surgery (tumorectomy or orchiectomy), and the material was examined by the institution’s Pathology Department. The US findings were varied and distinct. As to the lesions’ consistency, they were cystic (n: 1), multicystic (n: 2), solid-cystic (n: 1), solid containing larger or smaller calcifications (n: 2), and focal calcification (n: 1). With regard to total circulation, all the solid lesions had few vessels in the interior of the lesions. Testicular teratoma has a very variable US appearance and can simulate that of other lesions. It can be single, multiseptated, small, or large. It can contain diffuse or localized calcifications. The testicle can have an increased or a normal volume. UROLOGY 83: 195e198, 2014. 2014 Elsevier Inc.
T
esticular teratoma is the most common histologic type in children and is responsible for approximately 50% of prepubescent tumors. Although some investigators consider yolk sac tumors to be more common in children, in a series of recent studies, they represent only approximately 10%-18% of cases.1-8 The teratoma consists of different germination beds of the endoderm, mesoderm, and ectoderm. These tumors can occur in the neonatal period, and the average age of presentation is 18 months. Yolk sac tumors usually occur before the age of 2 years.3 It can appear as an increase in alpha-1-fetoprotein. The prognosis can be improved when it occurs during the first year of life. To date, few studies have specifically examined the ultrasound (US) findings for mature teratoma. Most these studies only report isolated cases together with a literature review. Therefore, there is a need to document the US presentation of mature teratoma in children. Thus, the objective of our study was to review the US findings of cases seen by our institution during a period of 12 years.
MATERIALS AND METHODS This was a retrospective study from January 2000 to July 2012 that reviewed the clinical, laboratory, and US findings of 7 Financial Disclosure: The authors declare that they have no relevant financial interests. From the Department of Urology, Pontifıcia Universidade Catolica do Rio Grande do Sul, School of Medicine, Hospital São Lucas, Porto Alegre, Brazil; the Department of Pediatrics, Pontifıcia Universidade Catolica do Rio Grande do Sul, Hospital São Lucas, Porto Alegre, Brazil; the Department of Urology, Universdad de Buenos Aires, Buenos Aires, Argentina; and the Department of Radiology and Pediatrics, Pontifıcia Universidade Catolica do Rio Grande do Sul, School of Medicine, Hospital São Lucas, Porto Alegre, Brazil Reprint requests: Matteo Baldisserotto, M.D., Ph.D., Hospital Sao Lucas, Pontifícia Universidade do Rio Grande do Sul, Ipiranga Av., 6690, Porto Alegre, 90.160-090, Brazil. E-mail: [email protected] Submitted: December 27, 2012, accepted (with revisions): July 16, 2013
ª 2014 Elsevier Inc. All Rights Reserved
children with a pathologic diagnosis of mature teratoma of the testis. Our institution is a general hospital that provides tertiary care to an adult and pediatric population in a region of approximately 1,500,000 people. The US findings retrieved from images in the archives of the x-ray service were reviewed, as were the radiological reports. Some examinations were retrieved from the picture archiving and communication system (PACS), whereas older cases were collected from film images. Clinical, surgical, and pathologic data were collected from the institution’s medical archive. The study was approved by the medical ethics committee, which released the informed consent of the patients’ parents or guardians. The US examinations were performed in general because of an increase in scrotal volume and a palpable testicular nodule. Neonatal or general pediatricians initially performed the scrotal physical examinations. Afterward, a pediatric surgeon, who ordered laboratory and imaging examinations, examined the patients. The laboratory investigations included serum alphafetoprotein levels. All patients underwent US of the scrotum and abdomen. The US examinations were performed by HDI 5000 (ATLePhilips Medical Systems, Best, The Netherlands) with 12-5 MHz linear probe, HD 11 (Philips Medical Systems, Best, The Netherlands) with 12-3 MHz linear probe, Acuson 128 XP 10 (Acuson, Mountain View, CA) with linear probe 7.5 MHz. The US examinations of cases in the last 5 years were performed using HD11 equipment, whereas examinations before this period were performed using Acuson XP10 and HDI 5000. Two medical radiologists from the Ultrasound Department with long experience in pediatric x-rays performed the examinations. All patients in the study received surgery (tumorectomy or orchiectomy), and the material was examined by the institution’s Pathology Department.
RESULTS During this period, we found 7 cases of mature teratoma. The age band varied from newborns (3 cases) to the age 0090-4295/14/$36.00 http://dx.doi.org/10.1016/j.urology.2013.07.046
195
Table 1. The relationship between the clinical and ultrasound findings Case
Age
Case 1 (Figure 1)
Neonate
Case 2 (Figure 2)
Neonate
Case 3 (Figure 3)
4 years and 3 months
Case 4 (Supplementary Figure 1) Case 5 (Supplementary Figure 2) Case 6 (Supplementary Figure 3) Case 7 (Supplementary Figure 4)
1 month Neonate 1 month 3 months
Signs
US with Doppler Findings
Increase in the scrotal Multicystic lesion volume Volumetric increase in the Large cystic lesion with thin scrotum septation Palpable nodule Solid cystic lesion containing microcalcifications Volumetric testicular Solid-cystic lesion increase Increase in testicular volume Cystic lesion with irregular wall Increase in the volume of Multiple small calcifications the scrotum hydrocele Palpable nodule Gross calcification
Dimensions 1.0 0.7 cm 3.3 1.8 cm 2.7 1.9 cm 2.1 1.6 cm 0.5 0.5 cm 2.6 1.5 cm 1.5 1.1 cm
US, ultrasound.
of 4 years. The alphafetoprotein levels were as expected for age in all patients. The clinical and US findings are described in Table 1. The US findings were varied and distinct. As to the lesions’ consistency in US, they were multicystic (Fig. 1), cystic with thin septation (Fig. 2), solid cystic (Fig. 3, Supplementary Figure 1), cystic lesion with irregular wall (Supplementary Figure 2), solid containing coarse calcifications, some coalescing with hydrocele (Supplementary Figure 3), and with a focal calcification (Supplementary Figure 4). At Color Doppler, all the solid lesions had few vessels in the interior each of the masses. All patients received surgery, with 3 having a tumorectomy and 4 an orchiectomy. In all cases, the definitive histopathologic diagnosis was mature testicular teratoma.
COMMENT Although testicular teratoma is the most common children’s tumor, only a few studies describe its presentations on US examinations in reports on the basis of a small number or isolated cases. In many situations, the lesion is detected on a prenatal US examination.9 In other cases, the mass is detected during the neonatal period, whereas in yet other cases, it is detected even later because of a volumetric increase in the scrotum and/or the presence of hard nodule when palpating the testicle.10,11 In these cases, the examination indicated should be diagnostic investigation by US. This study of a population affected by neoplasia was in agreement with what other investigators have observed.3 As far as we know, this study is the first one with a higher number of cases of teratomas studied by US. All cases studied showed up on a single US. In 4 cases, the tumors had a cystic appearance, although with different US characteristics. In one of these cases, the lesion was cystic and multiseptated and located in the interpolar region of the testicle. In this case, we had to make a differential diagnosis of cystic tubular ectasia, which can prevent as various cystic structures in the interior of the testicle.12 One aspect of cystic tubular ecstasia that can 196
Figure 1. A neonate boy with a palpable nodule in the testicle. Color Doppler ultrasound shows an avascular cystic lesion, with slightly irregular walls, measuring 0.5 cm. (Color version available online.)
assist in a differential diagnosis is that its cysts might have a tubular shape and can occur in the testicular mediastinum. The second cystic tumor occurred in a newborn, and the image simulated a simple testicular cyst. In this case, the diagnosis was only defined by histopathologic examination, as its appearance simulates a simple testicular cyst.13 The third cystic lesion presented as a multiseptated voluminous cyst. This appearance can be confused with the appearance of a multiseptated encysted hydrocele, making it difficult to characterize its intra- or extratesticular location. The fourth lesion was of the solid-cystic type, a finding observed in malignant lesions. In 3 cases, there were calcifications. In 2 cases, the calcifications were diffuse. In one of these, the calcifications were larger and amorphous, associated with a voluminous hydrocele. This appearance can occur in testicular granulatomous lesions, although they are rare in this age group.14 In another case, the calcifications had an appearance of microlithiasis, although the testicle had a heterogeneous texture and an increased volume, an appearance that might suggest malignancy. The appearance of UROLOGY 83 (1), 2014
felt it essential not to exclude these cases. Another limitation was that the study was not a prospective one, so the cases could not be studied within a methodology correlating pathologic, clinical findings, and US.
CONCLUSION Testicular teratoma has a very variable US appearance and can simulate other lesions. It can be single, multiseptated, small, or large. It can contain diffuse or localized calcifications. The testicle can have an increased or a normal volume. Nevertheless, these appearances collectively, when observed in boys during their first years of life, suggest a diagnosis of testicular teratoma. References
Figure 2. A neonate with an important volumetric increase in the scrotum. Gray-scale ultrasound on the transversal view shows a large multiseptated cystic lesion, dislocating and elongating this testicle (T) and mimicking an encysted hydrocele.
Figure 3. A 4-year-old boy was presenting a swollen and hard testicle. Gray-scale ultrasound on the transversal view revealed a heterogeneous solid cystic mass containing microcalcifications.
microlithiasis in teratoma had already been observed in other cases.15 The US Doppler detected few vessels in the interior of the lesion. In the next case, the teratoma had a focal calcified lesion, making it difficult to define its intraor extratesticular location by the US examination. This appearance mimicked free calcification in the scrotum (scrotolith or scrotal pearl). However, this change has been documented in older boys and has been related to the testicular appendix that comes off and calcifies.16 The present study has some limitations because it is retrospective and involved very ancient cases. One of these is the quality of the oldest cases because the examinations were performed on previous generations of equipment. However, because of the rarity of this neoplasia and the small number of cases accumulated, we UROLOGY 83 (1), 2014
1. Ross JH, Kay R. Prepubertal testis tumors. Rev Urol. 2004;6:11-18. 2. Thomas JC, Ross JH, Kay R. Stromal testis tumors in children: a report from the prepubertal testis tumor registry. J Urol. 2001;166:2338-2340. 3. Metcalfe PD, Farivar-Mohseni H, Farhat W, et al. Pediatric testicular tumors: contemporary incidence and efficacy of testicular preserving surgery. J Urol. 2003;170:2412-2416. 4. Hisamatsu E, Takagi S, Nakagawa Y, et al. Prepubertal testicular tumors: a 20-year experience with 40 cases. Int J Urol. 2010;17:956-959. 5. Belman AB, King LR, Kramer SA. Clinical Pediatric Urology. 4th ed. Edition. London: Martin Dunitz Ltd; 2004. 6. Oottamasathien S, Thomas JC, Adams MC, et al. Testicular tumours in children: a single-institutional experience. BJU Int. 2007 May;99:1123-1126. 7. Taskinen S, Fagerholm R, Aronniemi J, et al. Testicular tumors in children and adolescents. J Pediatr Urol. 2008;4:134-137. 8. Pohl HG, Shukla AR, Metcalf PD, et al. Prepubertal testis tumors: actual prevalence rate of histological types. J Urol. 2004;172(6 Pt 1): 2370-2372. 9. Pramanik DD, Bhatnagar V, Subbarao KC, et al. Antenatally detected mature teratoma in an undescended testis. Eur J Pediatr Surg. 2011;21:209-210. 10. Lin CH, Clark JY. Testicular teratoma presenting as a transilluminating scrotal mass. Urology. 2006;67:1290.e3-1290.e5. 11. Barth RA, Teele RL, Colodny A, et al. Asymptomatic scrotal masses in children. Radiology. 1984;152:65-68. 12. Mac New HG, Terry NE, Fowler CL. Cystic dysplasia of the rete testis. J Pediatr Surg. 2008;43:768-770. 13. Honjo O, Uemura S, Murakami I. Simple testicular cyst in infants: a case report and review of the literature. Eur J Pediatr Surg. 2001 Dec;11:425-427. 14. Bushby LH, Miller FN, Rosairo S, et al. Scrotal calcification: ultrasound appearances, distribution and aetiology. Br J Radiol. 2002;75:283-288. 15. Horowitz MB, Abiri MM. US case of the day. Benign cystic teratoma and testicular microlithiasis. Radiographics. 1997;17:793-796. 16. Aso C, Enrıquez G, Fite M, et al. Gray-scale and color Doppler sonography of scrotal disorders in children: an update. Radiographics. 2005;25:1197-1214.
APPENDIX SUPPLEMENTARY DATA
Supplementary data associated with this article can be found, in the online version, at http://dx.doi.org/10.1016/j.urology. 2013.07.046.
EDITORIAL COMMENT Testis tumors in children are rare, with an incidence of 0.5-2.0 per 100, 000.1 Treatment of testis tumors in children has 197
evolved from radical orchiectomy, as is done in adults and adolescents, to testis sparing surgeries in most children with benign testis lesions found before puberty. Although the Prepubertal Testis Tumor Registry found that yolk sac tumors are the most frequently reported pediatric testis tumor,2 other investigators have found that teratoma tumors are actually more common in their facilities pediatric tumor occurrence.3-6 The incidence of teratoma, generally mature teratoma, is 39%-48% in these series. Evaluation of children before surgical treatment will give the urologic surgeon a better idea on whether the testis might be salvaged with tumor enucleation or excision. Preoperative examination of the testes with ultrasound imaging was discussed in some of the other series but no particular descriptions were given on typical ultrasound findings in testes with teratoma.4-6 The authors describe ultrasound findings in children with mature teratoma testis tumors as commonly having varying degrees of cystic elements, calcifications, and rare vascularity in the mass. All children were also evaluated with alpha-fetoprotein (AFP) serum tumor marker assessment as is recommended to assure that the testis tumor is not malignant with yolk sac elements. Although this series was small with only 7 cases, all the cases were found to have mature teratoma on final pathology. It is reasonable to deduce that mature teratoma with endodermal mesodermal and ectodermal tissue derivatives would have cystic and calcified elements found on ultrasound according to its composition. It is worthwhile noting that although mature teratoma can be treated by testis sparing surgery in most instances, immature teratoma should not be considered or treated the same as mature teratoma. Although there have not been any reported cases of metastasis or recurrence in children younger than 12 years with mature teratoma, that has been a reported case of metastatic immature teratoma with neuroectodermal elements.7 This tumor represents <10% of tumors reported to the Pediatric Oncology Group/Children’s Cancer Group. It was recommended to completely remove this tumor and not to perform testis sparing surgery.
198
All testis tumors, regardless of ultrasound findings, should at a minimum have an AFP serum marker test and surgical excision with an inguinal approach, vascular control with a tourniquet, and excisional biopsy with frozen section pathologic assessment of the mass performed with a nonmalignant assessment of the testis tissue done before consideration for testis sparing surgery. If there is an abnormal elevation of the AFP or any tissue components suspicious for malignancy, the entire testis should be removed. The authors have given us some reassuring ultrasound element assessments found in mature teratoma which help in planning a testis surgical procedure for the patient, barring elevated AFP or tissue findings not consistent with a benign testis tumor in childhood. Irene M. McAleer, M.D., J.D., M.B.A., Department of Urology, University of California, School of Medicine, Irvine, CA
References 1. Ross JH. Prepubertal testicular tumors. Urology. 2009;74:94-99. 2. Ross JH, Rybicki L, Kay R. Clinical behavior and a contemporary management algorithm for prepubertal testis tumors: a summary of the Prepubertal Testis Tumor Registry. J Urol. 2002;168:1675-1679. 3. Pohl HG, Shukla AR, Metcalf PD, et al. Prepubertal testis tumors: actual prevalence rate of histological types. J Urol. 2004;172:23702372. 4. Taskinen S, Fagerholm R, Aronniemi J, et al. Testicular tumors in children and adolescents. J Ped Urol. 2008;4:134-137. 5. Metcalf PD, Farivar-Mohseni H, Farhat W, et al. Pediatric testicular tumors: contemporary incidence and efficacy of testicular preserving surgery. J Urol. 2003;170:2412-2416. 6. Valla J. Testis-sparing surgery for benign testicular tumors in children. J Urol. 2001;165:2280-2283. 7. Kato K, Ijiri R, Tanaka Y, et al. Testicular immature teratoma with primitive neuroectodermal tumor in early childhood. J Urol. 2000; 164:2068-2069.
http://dx.doi.org/10.1016/j.urology.2013.07.051 UROLOGY 83: 197e198, 2014. 2014 Elsevier Inc.
UROLOGY 83 (1), 2014
RESULTS
CONCLUSION
To describe the different ultrasound (US) findings of mature testicular hamartomas in children. This is a retrospective study from January 2000 to July 2012 that reviewed the clinical, laboratory, and US findings of 7 children with a pathologic diagnosis of mature teratoma of the testis. All patients in the study received surgery (tumorectomy or orchiectomy), and the material was examined by the institution’s Pathology Department. The US findings were varied and distinct. As to the lesions’ consistency, they were cystic (n: 1), multicystic (n: 2), solid-cystic (n: 1), solid containing larger or smaller calcifications (n: 2), and focal calcification (n: 1). With regard to total circulation, all the solid lesions had few vessels in the interior of the lesions. Testicular teratoma has a very variable US appearance and can simulate that of other lesions. It can be single, multiseptated, small, or large. It can contain diffuse or localized calcifications. The testicle can have an increased or a normal volume. UROLOGY 83: 195e198, 2014. 2014 Elsevier Inc.
T
esticular teratoma is the most common histologic type in children and is responsible for approximately 50% of prepubescent tumors. Although some investigators consider yolk sac tumors to be more common in children, in a series of recent studies, they represent only approximately 10%-18% of cases.1-8 The teratoma consists of different germination beds of the endoderm, mesoderm, and ectoderm. These tumors can occur in the neonatal period, and the average age of presentation is 18 months. Yolk sac tumors usually occur before the age of 2 years.3 It can appear as an increase in alpha-1-fetoprotein. The prognosis can be improved when it occurs during the first year of life. To date, few studies have specifically examined the ultrasound (US) findings for mature teratoma. Most these studies only report isolated cases together with a literature review. Therefore, there is a need to document the US presentation of mature teratoma in children. Thus, the objective of our study was to review the US findings of cases seen by our institution during a period of 12 years.
MATERIALS AND METHODS This was a retrospective study from January 2000 to July 2012 that reviewed the clinical, laboratory, and US findings of 7 Financial Disclosure: The authors declare that they have no relevant financial interests. From the Department of Urology, Pontifıcia Universidade Catolica do Rio Grande do Sul, School of Medicine, Hospital São Lucas, Porto Alegre, Brazil; the Department of Pediatrics, Pontifıcia Universidade Catolica do Rio Grande do Sul, Hospital São Lucas, Porto Alegre, Brazil; the Department of Urology, Universdad de Buenos Aires, Buenos Aires, Argentina; and the Department of Radiology and Pediatrics, Pontifıcia Universidade Catolica do Rio Grande do Sul, School of Medicine, Hospital São Lucas, Porto Alegre, Brazil Reprint requests: Matteo Baldisserotto, M.D., Ph.D., Hospital Sao Lucas, Pontifícia Universidade do Rio Grande do Sul, Ipiranga Av., 6690, Porto Alegre, 90.160-090, Brazil. E-mail: [email protected] Submitted: December 27, 2012, accepted (with revisions): July 16, 2013
ª 2014 Elsevier Inc. All Rights Reserved
children with a pathologic diagnosis of mature teratoma of the testis. Our institution is a general hospital that provides tertiary care to an adult and pediatric population in a region of approximately 1,500,000 people. The US findings retrieved from images in the archives of the x-ray service were reviewed, as were the radiological reports. Some examinations were retrieved from the picture archiving and communication system (PACS), whereas older cases were collected from film images. Clinical, surgical, and pathologic data were collected from the institution’s medical archive. The study was approved by the medical ethics committee, which released the informed consent of the patients’ parents or guardians. The US examinations were performed in general because of an increase in scrotal volume and a palpable testicular nodule. Neonatal or general pediatricians initially performed the scrotal physical examinations. Afterward, a pediatric surgeon, who ordered laboratory and imaging examinations, examined the patients. The laboratory investigations included serum alphafetoprotein levels. All patients underwent US of the scrotum and abdomen. The US examinations were performed by HDI 5000 (ATLePhilips Medical Systems, Best, The Netherlands) with 12-5 MHz linear probe, HD 11 (Philips Medical Systems, Best, The Netherlands) with 12-3 MHz linear probe, Acuson 128 XP 10 (Acuson, Mountain View, CA) with linear probe 7.5 MHz. The US examinations of cases in the last 5 years were performed using HD11 equipment, whereas examinations before this period were performed using Acuson XP10 and HDI 5000. Two medical radiologists from the Ultrasound Department with long experience in pediatric x-rays performed the examinations. All patients in the study received surgery (tumorectomy or orchiectomy), and the material was examined by the institution’s Pathology Department.
RESULTS During this period, we found 7 cases of mature teratoma. The age band varied from newborns (3 cases) to the age 0090-4295/14/$36.00 http://dx.doi.org/10.1016/j.urology.2013.07.046
195
Table 1. The relationship between the clinical and ultrasound findings Case
Age
Case 1 (Figure 1)
Neonate
Case 2 (Figure 2)
Neonate
Case 3 (Figure 3)
4 years and 3 months
Case 4 (Supplementary Figure 1) Case 5 (Supplementary Figure 2) Case 6 (Supplementary Figure 3) Case 7 (Supplementary Figure 4)
1 month Neonate 1 month 3 months
Signs
US with Doppler Findings
Increase in the scrotal Multicystic lesion volume Volumetric increase in the Large cystic lesion with thin scrotum septation Palpable nodule Solid cystic lesion containing microcalcifications Volumetric testicular Solid-cystic lesion increase Increase in testicular volume Cystic lesion with irregular wall Increase in the volume of Multiple small calcifications the scrotum hydrocele Palpable nodule Gross calcification
Dimensions 1.0 0.7 cm 3.3 1.8 cm 2.7 1.9 cm 2.1 1.6 cm 0.5 0.5 cm 2.6 1.5 cm 1.5 1.1 cm
US, ultrasound.
of 4 years. The alphafetoprotein levels were as expected for age in all patients. The clinical and US findings are described in Table 1. The US findings were varied and distinct. As to the lesions’ consistency in US, they were multicystic (Fig. 1), cystic with thin septation (Fig. 2), solid cystic (Fig. 3, Supplementary Figure 1), cystic lesion with irregular wall (Supplementary Figure 2), solid containing coarse calcifications, some coalescing with hydrocele (Supplementary Figure 3), and with a focal calcification (Supplementary Figure 4). At Color Doppler, all the solid lesions had few vessels in the interior each of the masses. All patients received surgery, with 3 having a tumorectomy and 4 an orchiectomy. In all cases, the definitive histopathologic diagnosis was mature testicular teratoma.
COMMENT Although testicular teratoma is the most common children’s tumor, only a few studies describe its presentations on US examinations in reports on the basis of a small number or isolated cases. In many situations, the lesion is detected on a prenatal US examination.9 In other cases, the mass is detected during the neonatal period, whereas in yet other cases, it is detected even later because of a volumetric increase in the scrotum and/or the presence of hard nodule when palpating the testicle.10,11 In these cases, the examination indicated should be diagnostic investigation by US. This study of a population affected by neoplasia was in agreement with what other investigators have observed.3 As far as we know, this study is the first one with a higher number of cases of teratomas studied by US. All cases studied showed up on a single US. In 4 cases, the tumors had a cystic appearance, although with different US characteristics. In one of these cases, the lesion was cystic and multiseptated and located in the interpolar region of the testicle. In this case, we had to make a differential diagnosis of cystic tubular ectasia, which can prevent as various cystic structures in the interior of the testicle.12 One aspect of cystic tubular ecstasia that can 196
Figure 1. A neonate boy with a palpable nodule in the testicle. Color Doppler ultrasound shows an avascular cystic lesion, with slightly irregular walls, measuring 0.5 cm. (Color version available online.)
assist in a differential diagnosis is that its cysts might have a tubular shape and can occur in the testicular mediastinum. The second cystic tumor occurred in a newborn, and the image simulated a simple testicular cyst. In this case, the diagnosis was only defined by histopathologic examination, as its appearance simulates a simple testicular cyst.13 The third cystic lesion presented as a multiseptated voluminous cyst. This appearance can be confused with the appearance of a multiseptated encysted hydrocele, making it difficult to characterize its intra- or extratesticular location. The fourth lesion was of the solid-cystic type, a finding observed in malignant lesions. In 3 cases, there were calcifications. In 2 cases, the calcifications were diffuse. In one of these, the calcifications were larger and amorphous, associated with a voluminous hydrocele. This appearance can occur in testicular granulatomous lesions, although they are rare in this age group.14 In another case, the calcifications had an appearance of microlithiasis, although the testicle had a heterogeneous texture and an increased volume, an appearance that might suggest malignancy. The appearance of UROLOGY 83 (1), 2014
felt it essential not to exclude these cases. Another limitation was that the study was not a prospective one, so the cases could not be studied within a methodology correlating pathologic, clinical findings, and US.
CONCLUSION Testicular teratoma has a very variable US appearance and can simulate other lesions. It can be single, multiseptated, small, or large. It can contain diffuse or localized calcifications. The testicle can have an increased or a normal volume. Nevertheless, these appearances collectively, when observed in boys during their first years of life, suggest a diagnosis of testicular teratoma. References
Figure 2. A neonate with an important volumetric increase in the scrotum. Gray-scale ultrasound on the transversal view shows a large multiseptated cystic lesion, dislocating and elongating this testicle (T) and mimicking an encysted hydrocele.
Figure 3. A 4-year-old boy was presenting a swollen and hard testicle. Gray-scale ultrasound on the transversal view revealed a heterogeneous solid cystic mass containing microcalcifications.
microlithiasis in teratoma had already been observed in other cases.15 The US Doppler detected few vessels in the interior of the lesion. In the next case, the teratoma had a focal calcified lesion, making it difficult to define its intraor extratesticular location by the US examination. This appearance mimicked free calcification in the scrotum (scrotolith or scrotal pearl). However, this change has been documented in older boys and has been related to the testicular appendix that comes off and calcifies.16 The present study has some limitations because it is retrospective and involved very ancient cases. One of these is the quality of the oldest cases because the examinations were performed on previous generations of equipment. However, because of the rarity of this neoplasia and the small number of cases accumulated, we UROLOGY 83 (1), 2014
1. Ross JH, Kay R. Prepubertal testis tumors. Rev Urol. 2004;6:11-18. 2. Thomas JC, Ross JH, Kay R. Stromal testis tumors in children: a report from the prepubertal testis tumor registry. J Urol. 2001;166:2338-2340. 3. Metcalfe PD, Farivar-Mohseni H, Farhat W, et al. Pediatric testicular tumors: contemporary incidence and efficacy of testicular preserving surgery. J Urol. 2003;170:2412-2416. 4. Hisamatsu E, Takagi S, Nakagawa Y, et al. Prepubertal testicular tumors: a 20-year experience with 40 cases. Int J Urol. 2010;17:956-959. 5. Belman AB, King LR, Kramer SA. Clinical Pediatric Urology. 4th ed. Edition. London: Martin Dunitz Ltd; 2004. 6. Oottamasathien S, Thomas JC, Adams MC, et al. Testicular tumours in children: a single-institutional experience. BJU Int. 2007 May;99:1123-1126. 7. Taskinen S, Fagerholm R, Aronniemi J, et al. Testicular tumors in children and adolescents. J Pediatr Urol. 2008;4:134-137. 8. Pohl HG, Shukla AR, Metcalf PD, et al. Prepubertal testis tumors: actual prevalence rate of histological types. J Urol. 2004;172(6 Pt 1): 2370-2372. 9. Pramanik DD, Bhatnagar V, Subbarao KC, et al. Antenatally detected mature teratoma in an undescended testis. Eur J Pediatr Surg. 2011;21:209-210. 10. Lin CH, Clark JY. Testicular teratoma presenting as a transilluminating scrotal mass. Urology. 2006;67:1290.e3-1290.e5. 11. Barth RA, Teele RL, Colodny A, et al. Asymptomatic scrotal masses in children. Radiology. 1984;152:65-68. 12. Mac New HG, Terry NE, Fowler CL. Cystic dysplasia of the rete testis. J Pediatr Surg. 2008;43:768-770. 13. Honjo O, Uemura S, Murakami I. Simple testicular cyst in infants: a case report and review of the literature. Eur J Pediatr Surg. 2001 Dec;11:425-427. 14. Bushby LH, Miller FN, Rosairo S, et al. Scrotal calcification: ultrasound appearances, distribution and aetiology. Br J Radiol. 2002;75:283-288. 15. Horowitz MB, Abiri MM. US case of the day. Benign cystic teratoma and testicular microlithiasis. Radiographics. 1997;17:793-796. 16. Aso C, Enrıquez G, Fite M, et al. Gray-scale and color Doppler sonography of scrotal disorders in children: an update. Radiographics. 2005;25:1197-1214.
APPENDIX SUPPLEMENTARY DATA
Supplementary data associated with this article can be found, in the online version, at http://dx.doi.org/10.1016/j.urology. 2013.07.046.
EDITORIAL COMMENT Testis tumors in children are rare, with an incidence of 0.5-2.0 per 100, 000.1 Treatment of testis tumors in children has 197
evolved from radical orchiectomy, as is done in adults and adolescents, to testis sparing surgeries in most children with benign testis lesions found before puberty. Although the Prepubertal Testis Tumor Registry found that yolk sac tumors are the most frequently reported pediatric testis tumor,2 other investigators have found that teratoma tumors are actually more common in their facilities pediatric tumor occurrence.3-6 The incidence of teratoma, generally mature teratoma, is 39%-48% in these series. Evaluation of children before surgical treatment will give the urologic surgeon a better idea on whether the testis might be salvaged with tumor enucleation or excision. Preoperative examination of the testes with ultrasound imaging was discussed in some of the other series but no particular descriptions were given on typical ultrasound findings in testes with teratoma.4-6 The authors describe ultrasound findings in children with mature teratoma testis tumors as commonly having varying degrees of cystic elements, calcifications, and rare vascularity in the mass. All children were also evaluated with alpha-fetoprotein (AFP) serum tumor marker assessment as is recommended to assure that the testis tumor is not malignant with yolk sac elements. Although this series was small with only 7 cases, all the cases were found to have mature teratoma on final pathology. It is reasonable to deduce that mature teratoma with endodermal mesodermal and ectodermal tissue derivatives would have cystic and calcified elements found on ultrasound according to its composition. It is worthwhile noting that although mature teratoma can be treated by testis sparing surgery in most instances, immature teratoma should not be considered or treated the same as mature teratoma. Although there have not been any reported cases of metastasis or recurrence in children younger than 12 years with mature teratoma, that has been a reported case of metastatic immature teratoma with neuroectodermal elements.7 This tumor represents <10% of tumors reported to the Pediatric Oncology Group/Children’s Cancer Group. It was recommended to completely remove this tumor and not to perform testis sparing surgery.
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All testis tumors, regardless of ultrasound findings, should at a minimum have an AFP serum marker test and surgical excision with an inguinal approach, vascular control with a tourniquet, and excisional biopsy with frozen section pathologic assessment of the mass performed with a nonmalignant assessment of the testis tissue done before consideration for testis sparing surgery. If there is an abnormal elevation of the AFP or any tissue components suspicious for malignancy, the entire testis should be removed. The authors have given us some reassuring ultrasound element assessments found in mature teratoma which help in planning a testis surgical procedure for the patient, barring elevated AFP or tissue findings not consistent with a benign testis tumor in childhood. Irene M. McAleer, M.D., J.D., M.B.A., Department of Urology, University of California, School of Medicine, Irvine, CA
References 1. Ross JH. Prepubertal testicular tumors. Urology. 2009;74:94-99. 2. Ross JH, Rybicki L, Kay R. Clinical behavior and a contemporary management algorithm for prepubertal testis tumors: a summary of the Prepubertal Testis Tumor Registry. J Urol. 2002;168:1675-1679. 3. Pohl HG, Shukla AR, Metcalf PD, et al. Prepubertal testis tumors: actual prevalence rate of histological types. J Urol. 2004;172:23702372. 4. Taskinen S, Fagerholm R, Aronniemi J, et al. Testicular tumors in children and adolescents. J Ped Urol. 2008;4:134-137. 5. Metcalf PD, Farivar-Mohseni H, Farhat W, et al. Pediatric testicular tumors: contemporary incidence and efficacy of testicular preserving surgery. J Urol. 2003;170:2412-2416. 6. Valla J. Testis-sparing surgery for benign testicular tumors in children. J Urol. 2001;165:2280-2283. 7. Kato K, Ijiri R, Tanaka Y, et al. Testicular immature teratoma with primitive neuroectodermal tumor in early childhood. J Urol. 2000; 164:2068-2069.
http://dx.doi.org/10.1016/j.urology.2013.07.051 UROLOGY 83: 197e198, 2014. 2014 Elsevier Inc.
UROLOGY 83 (1), 2014