Abstract
Cystic dysplasia of the testis is characterized by the presence of multiple cysts within the testicular parenchyma. It is a rare benign tumor. It is often accompanied by kidney malformations. There is no consensus on treatment. We report here the case of testicular dysplasia revealed by a torsion of the spermatic cord in an adult. The diagnosis of cystic dysplasia of the testis was made intraoperatively and confirmed by pathology. An orchiectomy was performed. Serum testicular cancer markers were normal postoperatively.
1. Introduction
Cystic dysplasia of the testis is characterized by the presence of multiple cysts within the testicular parenchyma.1 It is a rare benign tumor.2 It is often accompanied by kidney malformations.3 Although there is no consensus on treatment, the exceptional context of spermatic cord torsion justifies orchiectomy. We report the case of testicular dysplasia revealed by a torsion of the spermatic cord in an adult.
2. Observation
This is a case of 43-year-old patient with a history of inguinal hernia repair indicated for a right inguinoscrotal hernia 10 years previously. He was referred to the emergency department for a right testicular pain associated with scrotal swelling that occurred approximately a week before admission. Testicular pain was intermittent and was accompanied by vomiting at its paroxysm. Initial examination showed a large right bursa tender at palpation without ascension. Abdomen was soft, without palpable mass and the rest of the examination was unremarkable. Scrotal Doppler ultrasound showed twisting of the right spermatic cord with the presence (Fig. 1) with a right testicle increased in size and presenting several cystic nests (Fig. 2). The left epididymis was the site of several cysts at the head and body with a left testicle of homogeneous appearance. Surgical exploration of the right scrotum showed a septate vaginal hydrocele with serous fluid and multiple cysts deforming the testicle (Fig. 3) and involving the epididymis and the spermatic cord. The testicle was ischemic. The cord had two turns of turns. The surgical procedure consisted of an orchiectomy with primary ligation of the cord via the scrotal route. The postoperative course was simple. Serum testicular cancer markers were at normal level. Pathological examination of the surgical specimen showed simple cysts with almost non-existent and non-functional testicular parenchyma.
Fig. 1.
Turns of the spermatic cord at ultrasound examination.
Fig. 2.
Testicular cysts at ultrasound examination.
Fig. 3.
Per-operative image of polycystic testis.
3. Discussion
Cystic dysplasia of the testis, also called cystic dysplasia of the rete testis, is a rare or even exceptional condition.4 It is believed to result from a malformation occurring early during intrauterine development but its pathophysiology is not completely understood.5 The discovery is most often made in childhood as shown by the study of Ngaiet al.6 However, we report here a case in an adult. This can be explained by the relatively difficult access to healthcare. These same phenomena explains the long delay between the start of the symptoms and the consultation.7
The large bursa can be a circumstance for the discovery of polycystic dysplasia.3 Testicular tumors can present in the form of a torsion of the spermatic cord as shown by Uguz et al.8 and Tepeneau and al..9 However, torsion of the spermatic cord revealing cystic dysplasia of the testis is an exceptional situation. In case of clinical doubt, Color Doppler Ultrasound is the examination of choice to confirm the diagnosis of torsion of the spermatic cord.10 For the diagnosis of cystic dysplasia of the testicle, ultrasound and MRI are of great benefit.3 The pathological examination confirms the diagnosis.
The management of testicular dysplasia is not the subject of any consensus to date.5 Given the tumoral appearance of the testicle on surgical exploration, we opted for an orchiectomy with primary ligation of the cord.
4. Conclusion
Cystic dysplasia of the testis is a rare pathology. Its diagnosis in a context of spermatic cord torsion in adults is exceptional.
CRediT authorship contribution statement
Sidy Mohamed Abdoulaye Fall: Methodology. Abdourahmane Ndong: Writing – review & editing. Ibrahima Faye: Investigation.
References
- 1.Glantz L., Hansen K., Caldamone A., Medeiros L.J. Cystic dysplasia of the testis. Hum Pathol. 1993;24(10):1142–1145. doi: 10.1016/0046-8177(93)90197-o. [DOI] [PubMed] [Google Scholar]
- 2.Camassei F.D., Francalanci P., Ferro F., Capozza N., Boldrini R. Cystic dysplasia of the rete testis: report of two cases and review of the literature. Pediatr Dev Pathol Off J Soc Pediatr Pathol Paediatr Pathol Soc. 2002;5(2):206–210. doi: 10.1007/s10024001-0112-4. [DOI] [PubMed] [Google Scholar]
- 3.Landry J.L., Dodat H., Pelizzo G., et al. Dysplasie kystique du rete testis et agénésie rénale ipsilatérale chez l’enfant. Arch Pédiatrie. 1 avr. 1999;6(4):416–420. doi: 10.1016/s0929-693x(99)80223-1. [DOI] [PubMed] [Google Scholar]
- 4.Contini G., Frediani S., Pardi V., Diomedi-Camassei F., Inserra A. Cystic dysplasia of the rete testis: case report and systematic review of the literature. Front Pediatr. 2022;10 doi: 10.3389/fped.2022.898038. [DOI] [PMC free article] [PubMed] [Google Scholar]
- 5.Helman T., Epelman M., Ellsworth P. Cystic dysplasia of the rete testis: does pathophysiology guide management? Urology. juill. 2020;141:150–153. doi: 10.1016/j.urology.2020.03.026. [DOI] [PubMed] [Google Scholar]
- 6.Ngai R.L., Yeung B.K., Tsui W.M., Cheng F.Y. Cystic dysplasia of the testis associated with ipsilateral renal agenesis and high anorectal anomalies. J Pediatr Surg. mai. 1998;33(5):787–788. doi: 10.1016/s0022-3468(98)90222-6. [DOI] [PubMed] [Google Scholar]
- 7.Sarr A., Fall B., Mouss B., et al. Aspects diagnostiques et thérapeutiques de la torsion du cordon spermatique au CHU Aristide-Le-Dantec de Dakar. Basic Clin Androl. sept. 2010;20(3):203–208. [Google Scholar]
- 8.Uguz S., Yilmaz S., Guragac A., Topuz B., Aydur E. Association of torsion with testicular cancer: a retrospective study. Clin Genitourin Cancer. 1 févr. 2016;14(1):e55–e57. doi: 10.1016/j.clgc.2015.09.014. [DOI] [PubMed] [Google Scholar]
- 9.Tepeneu N.F., Krafka K., Meglic S., Rogatsch H., Fasching G. Testicular cavernous hemangioma associated with testicular torsion – case report and review of literature. Int J Surg Case Rep. 1 janv. 2018;49:247–250. doi: 10.1016/j.ijscr.2018.06.019. [DOI] [PMC free article] [PubMed] [Google Scholar]
- 10.Mongiat-Artus P. Torsion du cordon spermatique et des annexes testiculaires. Ann Urol. 1 févr. 2004;38(1):25–34. [PubMed] [Google Scholar]