Long-Term Follow-Up of Testicular Microlithiasis in Children and Adolescents: Multicenter Prospective Cohort Study of the Italian Society of Pediatric Urology

Abstract

Introduction Testicular microlithiasis (TM), characterized by the presence of intratubular calcifications in a single or both the gonads, is an uncommon entity with unknown etiology and outcome in pediatric and adolescent age. In this study, the results of a multicenter long-term survey are presented.

Materials and Methods From 11 units of pediatric urology/surgery, patients with TM were identified and yearly, followed up in a 7-year period, adopting a specific database. The recorded items were: age at diagnosis, presenting symptoms/associated abnormalities, ultrasonographic finding, surgery and histology at biopsy, if performed.

Results Out of 85 patients, 81 were evaluated yearly (4 patients lost to follow-up). TM was bilateral in 66.6% of the patients. Associate genital abnormalities were present in 90%, more frequently undescended/retractile testis (23.4%) and varicocele (22.2%). TM remained unchanged at 4.7 years follow-up in 77 patients (93.8%) and was reduced in 4 patients after 1 to 5 years of inguinoscrotal surgery. Orchiectomy was performed in three patients (3.7%), one for severe testicular hypoplasia and two for seminoma (2.5%), respectively, concurrent and metachronous to diagnosis of TM. Tumorectomy with parenchymal sparing surgery was performed in a teratoma associated with TM.

Conclusion TM is a controversial entity, often associated with several inguinogenital features, which rarely can recover. Testicular malignancy, although present in TM, has not proven definitively associated to microliths. Proper counseling, yearly ultrasound, and self-examination are long-term recommended.

Keywords

testicular microlithiasis - microcalcifications - gonadal dysgenesis - seminoma - germ

• References

• 1 Oiye T. Über anscheinend noch nicht beschriebene Steinchen in den menschlichen Hoden. Beitr Path Anat 1928; 80: 479-495
• 2 Blumensaat C. Über einen neuen Befund in Knabenhoden. Virchows Archiv für pathologische Anatomie und Physiologie und für klinische Medizin 1929; 273 (1) 51-61
• 3 Höbarth K, Susani M, Szabo N, Kratzik C. Incidence of testicular microlithiasis. Urology 1992; 40 (5) 464-467
• 4 Aizenstein RI, Hibbeln JF, Sagireddy B, Wilbur AC, O'Neil HK. Klinefelter's syndrome associated with testicular microlithiasis and mediastinal germ-cell neoplasm. J Clin Ultrasound 1997; 25 (9) 508-510
• 5 Pourbagher MA, Pourbagher A, Erol I. Fragile x syndrome associated with testicular microlithiasis in siblings. J Ultrasound Med 2005; 24 (12) 1727-1729
• 6 Vachon L, Fareau GE, Wilson MG, Chan LS. Testicular microlithiasis in patients with Down syndrome. J Pediatr 2006; 149 (2) 233-236
• 7 Chakrabarti S, Marshall I. Another unusual presentation of McCune Albright syndrome with fibrous dysplasia, unilateral testicular enlargement, and testicular microlithiasis. J Pediatr Endocrinol Metab 2010; 23 (5) 513-515
• 8 Smith WS, Brammer HM, Henry M, Frazier H. Testicular microlithiasis: sonographic features with pathologic correlation. AJR Am J Roentgenol 1991; 157 (5) 1003-1004
• 9 Doherty FJ, Mullins TL, Sant GR, Drinkwater MA, Ucci Jr AA. Testicular microlithiasis. A unique sonographic appearance. J Ultrasound Med 1987; 6 (7) 389-392
• 10 Ikinger U, Wurster K, Terwey B, Möhring K. Microcalcifications in testicular malignancy: diagnostic tool in occult tumor?. Urology 1982; 19 (5) 525-528
• 11 Brenner JS, Hergenroeder AC, Kozinetz CA, Kelder SH. Teaching testicular self-examination: education and practices in pediatric residents. Pediatrics 2003; 111 (3) e239-e244
• 12 Rovito MJ, Cavayero C, Leone JE , et al. Interventions promoting testicular self-examination (TSE) performance: A systematic review. Am J Mens Health 2015; 9 (6) 506-518
• 13 Bunge RG, Bradbury JT. Intratubular bodies of the human testis. J Urol 1961; 85: 306-310
• 14 Priebe Jr CJ, Garret R. Testicular calcification in a 4-year-old boy. Pediatrics 1970; 46 (5) 785-788
• 15 Martin B, Tubiana JM. Significance of scrotal calcifications detected by sonography. J Clin Ultrasound 1988; 16 (8) 545-552
• 16 Bennett HF, Middleton WD, Bullock AD, Teefey SA. Testicular microlithiasis: US follow-up. Radiology 2001; 218 (2) 3592-3563
• 17 Halley JB. The growth of Sertoli cell tumors: A possible index of differential gonadotrophin activity in the male. J Urol 1963; 90: 220-229
• 18 Huber R, Weber E, Hedinger C. Structure of so called “ring-like” testicular tubules with intratubular bodies in children. Virchows Arch A Pathol Pathol Anat 1968; 344 (1) 40-46
• 19 Vegni-Talluri M, Bigliardi E, Vanni MG, Tota G. Testicular microliths: their origin and structure. J Urol 1980; 124 (1) 105-107
• 20 Domínguez Hinarejos C, Bonillo García MA, Sangüesa C, Serrano Durbá A, García Ibarra F. Calcificaciones intratesticulares en la edad pediátrica. Actas Urol Esp 2007; 31 (1) 33-37
• 21 Kocaoğlu M, Bozlar U, Bulakbaşi N, Sağlam M, Uçöz T, Somuncu I. Testicular microlithiasis in pediatric age group: ultrasonography findings and literature review. Diagn Interv Radiol 2005; 11 (1) 60-65
• 22 Furness III PD, Husmann DA, Brock III JW , et al. Multi-institutional study of testicular microlithiasis in childhood: a benign or premalignant condition?. J Urol 1998; 160 (3 Pt 2) 1151-1154 , discussion 1178
• 23 Coley BD. Resolving testicular microlithiasis in a 12-year-old boy. J Ultrasound Med 2005; 24 (10) 1445-1448
• 24 Suominen JS, Jawaid WB, Losty PD. Testicular microlithiasis and associated testicular malignancies in childhood: a systematic review. Pediatr Blood Cancer 2015; 62 (3) 385-388
• 25 Tan IB, Ang KK, Ching BC, Mohan C, Toh CK, Tan MH. Testicular microlithiasis predicts concurrent testicular germ cell tumors and intratubular germ cell neoplasia of unclassified type in adults: a meta-analysis and systematic review. Cancer 2010; 116 (19) 4520-4532
• 26 van Casteren NJ, Looijenga LH, Dohle GR. Testicular microlithiasis and carcinoma in situ overview and proposed clinical guideline. Int J Androl 2009; 32 (4) 279-287
• 27 Dagash H, Mackinnon EA. Testicular microlithiasis: what does it mean clinically?. BJU Int 2007; 99 (1) 157-160
• 28 Sakamoto H, Shichizyou T, Saito K , et al. Testicular microlithiasis identified ultrasonographically in Japanese adult patients: prevalence and associated conditions. Urology 2006; 68 (3) 636-641
• 29 Lam DL, Gerscovich EO, Kuo MC, McGahan JP. Testicular microlithiasis: our experience of 10 years. J Ultrasound Med 2007; 26 (7) 867-873
• 30 Frush DP, Kliewer MA, Madden JF. Testicular microlithiasis and subsequent development of metastatic germ cell tumor. AJR Am J Roentgenol 1996; 167 (4) 889-890
• 31 Bennett HF, Middleton WD, Bullock AD, Teefey SA. Testicular microlithiasis: US follow-up. Radiology 2001; 218 (2) 359-363
• 32 Dell'Acqua A, Tomà P, Oddone M, Ciccone MA, Marsili E, Derchi LE. Testicular microlithiasis: US findings in six pediatric cases and literature review. Eur Radiol 1999; 9 (5) 940-944
• 33 Leenen AS, Riebel TW. Testicular microlithiasis in children: sonographic features and clinical implications. Pediatr Radiol 2002; 32 (8) 575-579
• 34 Wang T, Liu L, Luo J, Liu T, Wei A. A meta-analysis of the relationship between testicular microlithiasis and incidence of testicular cancer. Urol J 2015; 12 (2) 2057-2064