Systematic Reviews
Copyright ©The Author(s) 2016. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Pediatr. May 8, 2016; 5(2): 223-227
Published online May 8, 2016. doi: 10.5409/wjcp.v5.i2.223
Adenomyomatosis of the gallbladder in childhood: A systematic review of the literature and an additional case report
Filippo Parolini, Giuseppe Indolfi, Miguel Garcia Magne, Marianna Salemme, Maurizio Cheli, Giovanni Boroni, Daniele Alberti
Filippo Parolini, Miguel Garcia Magne, Giovanni Boroni, Daniele Alberti, Department of Paediatric Surgery, “Spedali Civili” Children’s Hospital, 25123 Brescia, Italy
Giuseppe Indolfi, Department of Paediatrics, “Meyer” Children’s University Hospital, 50139 Florence, Italy
Marianna Salemme, Department of Pathology, “Spedali Civili” Hospital, 25123 Brescia, Italy
Maurizio Cheli, Department of Paediatric Surgery, “Papa Giovanni XXIII” Hospital, 24127 Bergamo, Italy
Daniele Alberti, Clinical and Experimental Sciences, University of Brescia, 25123 Brescia, Italy
Author contributions: Parolini F, Indolfi G, Magne MG, Salemme M, Cheli M, Boroni G and Alberti D contributed equally to the work; Parolini F and Indolfi G conceptualized and designed the review together with Magne MG; Parolini F, Cheli M and Boroni G carried out the analysis; Parolini F, Boroni G and Alberti D drafted the initial manuscript; all authors reviewed and approved the final manuscript as submitted.
Conflict-of-interest statement: We hereby declare that the following information relevant to this article are true to the best of our knowledge: The above mentioned manuscript has not been published, accepted for publication or under editorial review for publication elsewhere and it won’t be submitted to any other journal while under consideration for publication in your Journal; We have no financial relationship relevant to this article to disclose; There isn’t any conflict of interest relevant to this article; All authors participated in the concept and design, analysis and interpretation of data, drafting and revising the manuscript, and they have approved the manuscript as submitted.
Data sharing statement: No additional data are available.
Open-Access: This article is an open-access article which was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution Non Commercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See:
Correspondence to: Filippo Parolini, MD, Department of Paediatric Surgery, “Spedali Civili” Children’s Hospital, Piazzale Spedali Civili 1, 25123 Brescia, Italy.
Telephone: +39-030-3996201 Fax: +39-030-3996154
Received: January 29, 2016
Peer-review started: January 30, 2016
First decision: February 29, 2016
Revised: March 4, 2016
Accepted: March 24, 2016
Article in press: March 25, 2016
Published online: May 8, 2016

AIM: To investigate the diagnostic and therapeutic assessment in children with adenomyomatosis of the gallbladder (AMG).

METHODS: AMG is a degenerative disease characterized by a proliferation of the mucosal epithelium which deeply invaginates and extends into the thickened muscular layer of the gallbladder, causing intramural diverticula. Although AMG is found in up to 5% of cholecystectomy specimens in adult populations, this condition in childhood is extremely uncommon. Authors provide a detailed systematic review of the pediatric literature according to PRISMA guidelines, focusing on diagnostic and therapeutic assessment. An additional case of AMG is also presented.

RESULTS: Five studies were finally enclosed, encompassing 5 children with AMG. Analysis was extended to our additional 11-year-old patient, who presented diffuse AMG and pancreatic acinar metaplasia of the gallbladder mucosa and was successfully managed with laparoscopic cholecystectomy. Mean age at presentation was 7.2 years. Unspecific abdominal pain was the commonest symptom. Abdominal ultrasound was performed on all patients, with a diagnostic accuracy of 100%. Five patients underwent cholecystectomy, and at follow-up were asymptomatic. In the remaining patient, completely asymptomatic at diagnosis, a conservative approach with monthly monitoring via ultrasonography was undertaken.

CONCLUSION: Considering the remote but possible degeneration leading to cancer and the feasibility of laparoscopic cholecystectomy even in small children, evidence suggests that elective laparoscopic cholecystectomy represent the treatment of choice. Pre-operative evaluation of the extrahepatic biliary tree anatomy with cholangio-MRI is strongly recommended.

Keywords: Adenomyomatosis, Children, Gallbladder, Laparoscopy, Ultrasound

Core tip: Adenomyomatosis of the gallbladder (AMG) in childhood is an extremely rare condition, with only few cases reported so far. We provided a detailed systematic review on diagnostic and therapeutic assessment of children with AMG.