Carcinoma Arising in Choledochocele: Is Choledochocele Innocent Bystander or Culprit?

Dear Sir,

We read with interest the article by Ohtsuka et al. [1], entitled “Carcinoma arising in choledochocele.” The prevalence of 27 % for periampullary carcinoma in choledochoceles in their study was much higher than that of 2.5 % previously reported by Ladas et al. [2]. We have already published a study of a large series of choledochoceles (n = 17) treated by endoscopic sphincterotomy (ES) [3]. During long-term follow-up (mean 3 years) of all patients, however, we have never found periampullary cancer in those patients. Therefore, we would like to raise some questions.

The etiology of choledochocele is uncertain, but there may be two distinct forms, i. e., congenital and acquired. We recently encountered one case of choledochocele which appeared to develop secondary to early superficial carcinoma, involving the mucosa around the ampullary orifice (Figures [1], [2]), not extending to the intramural portion of the distal common bile duct (CBD).

Figure 1 Duodenoscopy shows broad whitish granular mucosal change (arrows) surrounding the ampullary orifice. The infundibulotomy site is shown proximal to the lesion

Figure 2 Cholangiographic finding during percutaneous transhepatic cholangioscopic examination demonstrates a choledochocele; the small arrow indicates the tip of the cholangioscope

The muscle of the duodenal wall and the variable muscle fibers of the sphincter of Oddi enforce duodenal window. The sphincter of Oddi system can be divided into three zones (superior, middle, and inferior sphincter), and the middle one, defining the infundibulum, can be easily dilated [4]. Bile stagnation between the obstructed papillary orifice and the duodenal window may result in increased pressure within the intramural portion of the CBD, and eventually it may cause diverticular formation. Because a cryptic ampullary tumor which might contribute to the formation of choledochocele, could be present, the ampulla should be carefully biopsied several days after ES in their study. It is therefore difficult to say whether choledochocele was the culprit, or an innocent bystander in their study.

So far, ES has been generally accepted as a treatment modality for choledochocele. If the figure for the high prevalence (27 %) of periampullary carcinoma in choledochocele found in the study of Ohtsuka et al. is accurate, there should be a change in the treatment strategy for symptomatic choledochocele from ES to operative laparotomy.

In summary, we suggest that the data for carcinoma in choledochocele in the study should be carefully interpreted, and it may be more appropriate for the title of the article by Ohtsuka et al., ”Carcinoma arising in choledochocele,“ to be replaced by ”Carcinoma associated with choledochocele.“

References

• 1 Ohtsuka T, Inoue K, Ohuchida J. et al . Carcinoma arising in choledochocele.  Endoscopy. 2001;  33 614-619
• 2 Ladas S D, Katsogridakis I, Tassios P. et al . Choledochocele, an overlooked diagnosis: Report of 15 cases and review of 56 published reports from 1984 to 1992.  Endoscopy. 1995;  27 233-239
• 3 Kim M H, Myung S J, Lee S K. et al . Ballooning of the papilla during contrast injection: The semaphore of choledochocele.  Gastrointest Endosc. 1998;  48 258-262
• 4 Barraya L, Pujol Soler R, Yvergneaux J P. The region of Oddi’s sphincter: Millimetric anatomy.  Presse Méd. 1971;  79 2527-2534