Operations for choledochal cysts: A 25-year experience at a tertiary care center in India

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Dear Editor, Choledochal cysts (CDCs) are reported primarily in children, with only 20% present in adulthood. Our retrospective review of prospectively maintained data of surgical treatment in 86 adult patients with CDC for operative procedure and outcomes aims to show that surgical treatment in adults has added complexity due to coexisting hepatobiliary disease. The mean age was 38 years with a male-to-female ratio of 2:3.5. Most cysts were Todani types I and IVa (Fig. 1). Only 4.6% presented with the classic triad (pain, jaundice, and abdominal lump), and 70% had complications at the time of presentation, commonest being choledocho/cystolithiasis (Table 1). Seven patients had malignancy at presentation, with five having gallbladder carcinoma. Complete excision of the cyst was possible in 69.7%. Hepatectomy and radical cholecystectomy were required in eight patients. Eight patients had severe postoperative complications with no 30-day mortality. Regression analysis for factors associated with severe postoperative complications and mortality showed presenting complaints and previous surgery did not show significant associations with major complications (Clavien-Dindo grade III and above). The presence of malignancy (preoperative/intraoperative) showed an increased but not statistically significant odds of major complications (odds ratio [OR] = 3.11, 95% confidence interval [CI] = 0.40–17.02, P = 0.212 in the univariate model; OR = 1.76, 95% CI = 0.15–16.02, P = 0.623 in the multivariate model). Complete excision (without additional procedures such as hepatectomies, etc) of the cyst was associated with lower odds of major complications, but this association was not statistically significant (OR = 0.38, 95% CI = 0.09–1.37, P = 0.150 in the univariate model; OR = 0.41, 95% CI = 0.08–1.85, P = 0.245 in the multivariate model). No specific patient factor showed statistically significant association with mortality in 69 patients followed up for a median duration of 6 years. Four developed jaundice, and redo hepaticojejunostomy was needed in three. Two developed malignancy during follow-up (one cholangiocarcinoma, one pancreatic adenocarcinoma). Choledochal cysts are congenital anomalies that rarely present in adults. The classic clinical presentation is rare in adults,[1] and only 4.6% of the patients in our study had the full triad, so high index of suspicion is required to diagnose these cases. Adult CDCs are more likely to present with complications, coexistent hepatobiliary malignancy, and prior inadequate surgery.[2–4] In our study, 70% of patients presented with complications, with more than a quarter having had suboptimal surgery previously. Contrary to studies,[5] we found a higher incidence of gallbladder cancer, with CDC representing 55% of those who developed malignancies that may be due to additive carcinogenic effect of CDC in an already at-risk population. These patients require good-quality cross-sectional imaging and additional hepatobiliary surgery to deal with complications and malignancy. Complete surgical excision and bilioenteric drainage are advocated in view of biliary carcinogenesis and prevention of other complications such as recurrent cholangitis and pancreatitis, and when performed at experienced centers, the subsequent outcomes are good. The operative treatment of CDCs is safe and has long-lasting benefit. Ethical statement This study was conducted in accordance with the Declaration of Helsinki and was approved by our institute (approval number: EC/09/23/2369). The patient consent was waived by the IRB. Table 1 - Patient Characteristics and Presenting Features Including Previous Surgeries Demographic Characteristics n = 86, n (%) Age 18–70 y 86 Male/female 30/56 (35/65) Type of cyst (Todani) I 60 (69.7) II 2 (2.3) III 2 (2.3) IVa 18 (20.9) IVb 2 (2.3) V 1 (1.1) VI 1 (1.1) Symptoms Abdominal pain 77 (89.5) Fever (cholangitis) 32 (37.2) Vomiting 17 (19.7) Jaundice 20 (23.2) Palpable lump 8 (9.3) Classic triad (pain, lump, jaundice) 4 (4.6) Type of complications Stone in common bile duct/cyst 22 (25.5) Cholelithiasis 16 (18.6) Hepatolithiasis 3 (3.4) Chronic pancreatitis 6 (6.9) Acute pancreatitis 2 (2.3) Portal hypertension 2 (2.3) Mucocele gallbladder 2 (2.3) Biliary ascariasis 1 (1.1) Pseudocyst of pancreas 1 (1.1) Gallbladder polyp 1 (1.1) Malignancy 5 (5.8) Previous procedures Cholecystectomy only 20 (23.2) Cystojejunostomy 1 (1.1) Cholecystectomy with choledochoduodenostomy 1 (1.1) Cholecystectomy with choledochojejunostomy 4 (4.6) Figure 1: MRCP image of a type I cyst with cystolithiasis.

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