Dual Cancer: Papillary and Signet Ring Cell Carcinoma, Arising in a Patient Operated for Choledochal Cyst: an Unusual Hi
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CASE REPORT
Dual Cancer: Papillary and Signet Ring Cell Carcinoma, Arising in a Patient Operated for Choledochal Cyst: an Unusual Histological Finding Aruna Prabhu 1 & Andreas Brandl 2 & Wakama Satoshi 3 & Shouzou Sako 3,4 & Haruaki Ishibashi 3 & Akiyoshi Mizumoto 5 & Nobuyuki Takao 5 & Masumi Ichinose 5 & Shunsuke Motoi 5 & Yang Liu 3 & Kazuo Nishihara 3,4,5 & Yutaka Yonemura 3,4,5,6 Received: 10 July 2019 / Accepted: 7 May 2020 # Indian Association of Surgical Oncology 2020
Introduction Choledochal cysts are cystic dilatations of parts of the biliary ducts, most commonly involving the extrahepatic bile ducts with varying amounts of intrahepatic involvement. Incidence is higher in Asian countries (1 in 1000) with two-thirds of all cases reported in Japan, as compared with the western countries having a relatively low incidence [1, 2]. Though choledochal cysts are known to be a disorder of childhood and infancy, 25–40% of patients are initially diagnosed in adulthood [3]. There is a female predominance with a male:female ratio of 1:3 to 1:4 [4]. Choledochal cysts are recognized to be a premalignant condition with an estimated incidence of malignancy, which is 20 times higher compared with healthy population. In general, malignancies occur in patients with choledochal cysts at a younger age, 10–
* Yutaka Yonemura [email protected] 1
Department of Surgical Oncology, Thangam Cancer Center, Namakkal, Tamil Nadu, India
2
Digestive Unit, Champalimaud Foundation, Lisbon, Portugal
3
Department of Regional Cancer therapy, Peritoneal Surface Malignancy Center, Kishiwada Tokushukai Hospital, 4 Chome-27-1 Kamoricho,, Kishiwada, Osaka 596-004, Japan
4
Japanese/Asian School of Peritoneal, Surface Oncology, Kyoto, Japan
5
Department of Regional Cancer therapy, Peritoneal Surface Malignancy Center, Kusatsu General Hospital, Kusatsu, Shiga, Japan
6
NPO Organization to Support Peritoneal Dissemination Treatment, Peritoneal Metastasis Center, Kishiwada, Japan
15 years earlier, as compared with the general population [5, 6]. The risk of malignancy increases with increasing age; it is 23% at 20–30 years and can increase up to 75% at 70–80 years [7]. Hence, the recommended treatment, even in asymptomatic patients, includes a complete excision of the choledochal cyst with Roux-en-Y hepatico-jejunostomy, in most cases. The incidence of malignant transformation after cyst excision, though is significantly reduced, is still approximately 9.9%, warranting a life-long follow-up [8]. The most common histological types are adenocarcinoma (73–84%), anaplastic carcinoma (10%), undifferentiated carcinoma (5–7%), and squamous cell carcinoma (5%) [9, 10]. Rare type like botryoid sarcoma has also been reported [11]. There has been only one reported case of signet ring cell carcinoma arising 25 years after choledochal cyst excision [12]. We hereby report a unique histopathological finding of a dual cancer, papillary as well as signet ring cell carcinoma, arising 14 months after choledochal cyst excision along with right hepatect
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