Response to the Letter to the Editor on Congenital Spigelian Hernia and Undescended Testis

We appreciate the letter by Mirilas and thank him for his valuable comments. However, we disagree with his interpretations in many ways. We will do our best to answer them point by point.

Durham and Ricketts[1] had reported 24 cases of Spigelian hernia (SH) in their study;16 of the patients were males and 8 were females. Twelve of these male patients had SH and undescended testis (UT) together. However, in our study there were 15 cases that had SH and UT together until 2006.[2] Thus, we think that Durham and Ricketts[1] might have missed three cases in their study.

Pul and Pul[3] and Al-Salem[4] each had reported two cases in their studies. However, Moles Morenilla et al[5] calculated the total number of these patients as two instead of four. Hence, two cases have been overlooked. Also, they have forgotten to add the case of a 4-month-old baby that had been reported by O'Sullivan et al.[6] Therefore, the number of patients who had UT and SH together should have been 19 instead of 16 in their studies.[5] By adding the 5 new cases reported in our study, we come to a sum of 24.[2]

Moles Morenilla et al[5] reported 40 cases with SH in 33 children (bilateral in 7), and 26 of them were males. It is just a coincidence that the number of cases reviewed in our study[2] and Moles Morenilla et al[5] are the same. In our study, all patients were males.[2] Sixteen spontaneous SH cases not accompanied by UT have been determined from 1955 to the present. They were reported by Komura et al[7] in 1994, Hurwitt and Borow[8] in 1955, Bertelsen[9] in 1966, Graivier and Alfieri[10] in 1970, Constantino et al[11] in 1974, Houlihan[12] in 1976, Graivier et al[13] in 1988, Rao et al[14] in 1992, Mehta et al[15] in 1992, Losanoff et al[16] in 2002, and Christianakis et al[17] in 2009, with one apiece; Wright[18] in 1994 with three; and Vaos et al[19] in 2005 with two. By adding the 16 cases that had only SH to the number of cases with UT and SH (24) reviewed in our study,[2] we come to a sum of 40 cases. Therefore the 60% (24/40) that is seen in only male cases is completely true.

We agree with Mirilas about the chaos in ratios. We think that it will be a better approach to calculate the ratio of SH only in male congenital cases (not including traumatic and postoperative ones). In our study, 24 cases of SH were detected in 40 male children and the ratio was 60%.[2]

Of course, the best way to calculate the real ratio of SH and UT is to include all cases in the world. But we think that Mirilas will also agree with us on reality that it is quite difficult to establish the real ratio.

SH is rarely seen in children and, we think that probably, the vast majority of the cases has been reported so far. Therefore, the approximate number of cases seems close to the true number of cases. Because our study was based on the published articles, we do not claim that this ratio is certainly correct.

We do not agree with the author about the statement that if a significant association is not shown, the coexistence of the two anomalies can be just random and all these association measures are meaningless. If we accept that the togetherness of SH and UT is just random, we should disregard most of the syndromes that include multiple anomalies. For example, in Wolfram syndrome (diabetes insipidus, diabetes mellitus, optic atrophy, and deafness), we donot know the exact ratio of each component of the syndrome.

We agree with the idea that the anomalies have a known or presumed “common cause'' while forming the syndrome. In our report, we discussed the causal connection of SH and UT and we think that our statement “the findings seems to be independent from each other but appear as a single disease when they coexist” does not contradict with this idea.

Exploration of the inguinal canal and letting down of the testicles via inguinal canal are the first steps in the surgery of UT. Therefore, the exploration of inguinal canal has been done in our patients as a part of the surgery. But, inguinal canal remnants have been seen in none of our cases. Whereas, as the pediatric surgeons, we all know that during isolated intra-abdominal UT surgery (without SH), at least the remnants of inguinal canal are seen after the exploration of inguinal canal.

To not find the inguinal canal in UT cases associated with SH shows us that there is no embryologic relationship between UT and SH and also that they are not related parts of serial component of development. Therefore, coexistence of these two conditions should be accepted as a syndrome rather than a sequence.

• References

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• 2 Bilici S, Güneş M, Göksu M, Melek M, Pirinçci N. Undescended testis accompanying congenital Spigelian hernia: is it a reason, a result, or a new syndrome?. Eur J Pediatr Surg 2012; 22 (2) 157-161
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