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      Multiseptate gallbladder coexisting with pancreaticobiliary maljunction treated by laparoscopic cholecystectomy: report of a pediatric case

      case-report

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          Abstract

          Background

          A multiseptate gallbladder is a very rare congenital malformation in which the lumen is divided into variously sized multiseptal compartments. The pathogenesis and natural history of this disease remain uncertain. We herein describe a pediatric case of a multiseptate gallbladder with pancreaticobiliary maljunction (PBM), which was treated by laparoscopic cholecystectomy.

          Case presentation

          A 5-year-old girl was referred to our hospital, because a multiseptate gallbladder had been incidentally detected on abdominal ultrasonography when she presented for transient abdominal pain. Ultrasonography showed hyperechoic septa throughout the lumen of the gallbladder, giving it a honeycomb appearance. The atrophied gallbladder had weak or no contractility. Magnetic resonance cholangiopancreatography performed to detect other coexisting biliary disorders revealed PBM without dilatation of the common bile duct. Although physical examination and laboratory tests revealed no abnormalities, we performed laparoscopic cholecystectomy to prevent cholecystitis and reduce the risk of cancer secondary to the PBM.

          Conclusions

          In recent pediatric case reports, the indication and timing of cholecystectomy has tended to be determined by the patient’s symptoms and the presence of biliary complications. In the present case, however, the combination of a multiseptate gallbladder and PBM may become problematic in the future. Surgical treatment without delay was appropriate even in this pediatric patient.

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          Most cited references18

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          Japanese clinical practice guidelines for pancreaticobiliary maljunction.

          There have been no clinical guidelines for the management of pancreaticobiliary maljunction (PBM). The Japanese Study Group on Pancreaticobiliary Maljunction (JSPBM) has proposed to establish clinical practice guidelines on how to deal with PBM, with the support of the Japan Biliary Association (JBA). Because the body of evidence-based literature is relatively small, we decided to create guidelines based on the consensus of experts, using the medical literature for reference. A total of 46 clinical questions (CQs) were considered by the members of the editorial committee responsible for the guidelines. The CQs covered distinct aspects of PBM: (1) Concepts and Pathophysiology (10 CQs); (2) Diagnosis (10 CQs); (3) Pancreatobiliary complications (9 CQs); and (4) Treatments and prognosis (17 CQs). Statements and comments for each CQ were prepared by the guidelines committee members and collaborating partners. The CQs were completed after review by members of the editorial committee, meetings of this committee, public comments on the homepages of the JSPBM and the JBA, public hearings, and assessment and approval by the guidelines evaluation board. PBM includes cases where the bile duct is dilated (PBM with biliary dilatation) and those in which it is not (PBM without biliary dilatation). In these guidelines, PBM with biliary dilatation is defined as being identical to congenital biliary dilatation of Todani type I (except for type Ib) and type IV-A, both of which are accompanied by PBM in almost all cases. These guidelines are created to provide assistance in the clinical practice of PBM management; their contents focus on clinical utility, and they include general information on PBM to make this disease more widely recognized.
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            Multiseptate gallbladder. A case report.

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              Multiseptate hypoplastic gallbladder.

              A case is presented of multiseptate hypoplastic gallbladder, which is an extremely rare combination of two congenital anomalies. The patient was diagnosed as having cholecystitis and was relieved of her symptoms following cholecystectomy, even though the specimen did not show evidence of inflammation or calculi. The significance of this is emphasized by reviewing the 5 authenticated cases of multiseptate gallbladder reported in the literature. Awareness of this condition should avoid misinterpretation of cholecystograms.
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                Author and article information

                Contributors
                oyachi-ampw@ych.pref.yamanashi.jp
                numano-bdch@ych.pref.yamanashi.jp
                koizumi72ac@gmail.com
                a-takano0902@ych.pref.yamanashi.jp
                hiroshishibusawa0721@gmail.com
                Journal
                Surg Case Rep
                Surg Case Rep
                Surgical Case Reports
                Springer Berlin Heidelberg (Berlin/Heidelberg )
                2198-7793
                21 January 2022
                21 January 2022
                December 2022
                : 8
                : 16
                Affiliations
                [1 ]GRID grid.417333.1, ISNI 0000 0004 0377 4044, Department of Pediatric Surgery, , Yamanashi Prefectural Central Hospital, ; 1-1-1 Kofu, Yamanashi, 409-8506 Japan
                [2 ]GRID grid.417333.1, ISNI 0000 0004 0377 4044, Department of Surgery, , Yamanashi Prefectural Central Hospital, ; Kofu, Japan
                [3 ]Department of Pediatrics, Yamanashi Kosei Hospital, Yamanashi, Japan
                Author information
                http://orcid.org/0000-0002-3412-586X
                Article
                1370
                10.1186/s40792-022-01370-4
                8782956
                35061125
                300738e9-4398-4f14-b197-b6dc72eb57b9
                © The Author(s) 2022

                Open AccessThis article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article's Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article's Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http://creativecommons.org/licenses/by/4.0/.

                History
                : 2 October 2021
                : 12 January 2022
                Categories
                Case Report
                Custom metadata
                © The Author(s) 2022

                multiseptate gallbladder,pancreaticobiliary maljunction,cholecystectomy,ultrasonography,pediatrics

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