Another case of testicle duplication with contralateral agenesis.

To the Editor: We read with interest the recent case report on the rare occurrence of unilateral testicular duplication with contralateral testicular agenesis or anorchism. (1) In the introduction of this case, a sentence reads "We have found only one similar case reported in the literature." In the conclusion, the last sentence reads "We report the second case of this association." This association is indeed rare; however, we are reporting another case not mentioned by the authors that followed the earlier report of Kalani and Purohit (2) but preceded the case presented above by Leung et al. (1)

The authors did not identify their search criteria to substantiate claiming their case as being only the second one reported in the literature. Using the PubMed database, we found another case (3) by using the search phrase "testicle duplication and agenesis" or "testicle duplication" alone. Also, in MEDLINE (1966 to November Week 2 2003), we found the same case in the intersection of two search sets using the keywords "testicle" and "duplication." According to the English abstract, a 7-year-old boy was found, during surgery to correct cryptorchidism, to have duplication of the left testicle with right testicular agenesis. This finding is similar to that reported by Kalani and Purohit, but contrasts with the patient reported by Leung et al, who had a duplicated right testicle and absent left testicle. A translation of the report revealed the larger testicle measured 3 X 3 cm and the smaller one measured 2 X 2 cm. Both testicles were located in the region of the external or superficial inguinal ring; each testicle possessed its own appendix and epididymis. They reportedly shared the same spermatic cord, which branched proximal to the duplicate testicles. Exploration during surgery revealed no right testicle in the scrotum, right inguinal canal, retroperitoneum, or in an ectopic location.

This case is another rare example of testicular duplication with contralateral anorchism. Also, it serves to remind authors to be cautious in making claims of priority unless a thorough and meticulous literature search clearly substantiates the claim; albeit, the title of the case we mention only emphasizes the testicular duplication and not the contralateral anorchism, so this association would not be appreciated unless noticed in the English abstract or a translation of the text. In any case, the Uniform Requirements for Manuscripts Submitted to Biomedical Journals (4) espoused by the International Committee of Medical Journal Editors recommend that authors avoid claiming priority; however, it would probably be advisable to mention in the manuscript the search strategy used to make such a determination of "firstness" or, in this case, "secondness." Few cases are actually unique, (5) and erroneous claims of priority can be a source of embarrassment to the authors of such cases as well as causing the chronology of the scientific or medical record to be confused.

References

1. Leung AKC, Wong AL, Kao CP. Duplication of the testis with contralateral anorchism. South Med J 2003;96:809-810.

2. Kalani BP, Purohit AD. Duplication of the testis. J Pediatr Surg 1972;7:73.

3. Porras-Ramirez G, Di Lauro Delgado L, Vergara Rodriguez J. Duplication of the left testicle and cryptorchidism (report of a case) [in Spanish]. Bol Med Hosp Infant Mex 1980;37:429-434.

4. International Committee of Medical Journal Editors. Uniform requirements for manuscripts submitted to biomedical journals. PRHSJ 2000;19:321-329.

5. Partridge C. Editorial: Writing case reports and short reports. Physiother Res Int 2003;8:iii-iv.

James Bradley Summers, MS, MD

Department of Radiology

University of South Alabama 

Mobile, AL

Joseph Kaminski, MD

Department of Radiology

Medical College of Georgia 

Augusta, GA