Document Detail

Multiseptate gallbladder in an asymptomatic child.
Jump to Full Text
MedLine Citation:
PMID:  22606419     Owner:  NLM     Status:  PubMed-not-MEDLINE    
Abstract/OtherAbstract:
A one-year-old child being investigated for urinary tract infection was diagnosed with a multiseptate gallbladder. The patient remains asymptomatic, and investigations demonstrate no associated anomalies. Forty-three cases, including 13 cases in children were identified in the literature. Their presentation and management were reviewed.
Authors:
Dylan Wanaguru; Ashish Jiwane; Andrew S Day; Susan Adams
Related Documents :
22813829 - Classical cloacal exstrophy with intravesical phallus.
22752799 - Reviewing for clinical orthopaedics and related research.
22785449 - Tracheo-innominate artery fistula: two case reports and a clinical review.
9819949 - Psychogenic parasitosis. a case series and literature review.
22307189 - Posttraumatic pulmonary pseudocysts as a manifestation of child abuse.
22319719 - "ancient" schwannoma of hypopharynx: a case report with review of literature.
10442689 - Mononeuropathy of the medial branch of the deep peroneal nerve in a scuba diver.
8684299 - Ignored occupational risks of asbestos mesotheliomas.
20920939 - Occult middle fossa encephaloceles in patients with temporal lobe epilepsy.
Publication Detail:
Type:  Journal Article     Date:  2011-09-15
Journal Detail:
Title:  Case reports in gastrointestinal medicine     Volume:  2011     ISSN:  2090-6536     ISO Abbreviation:  Case Rep Gastrointest Med     Publication Date:  2011  
Date Detail:
Created Date:  2012-05-18     Completed Date:  2012-08-23     Revised Date:  2013-02-28    
Medline Journal Info:
Nlm Unique ID:  101580185     Medline TA:  Case Rep Gastrointest Med     Country:  United States    
Other Details:
Languages:  eng     Pagination:  470658     Citation Subset:  -    
Affiliation:
Department of Paediatric Surgery, Sydney Children's Hospital, University of New South Wales, High Street, Randwick, Sydney, NSW 2031, Australia.
Export Citation:
APA/MLA Format     Download EndNote     Download BibTex
MeSH Terms
Descriptor/Qualifier:
Comments/Corrections

From MEDLINE®/PubMed®, a database of the U.S. National Library of Medicine

Full Text
Journal Information
Journal ID (nlm-ta): Case Rep Gastrointest Med
Journal ID (iso-abbrev): Case Rep Gastrointest Med
Journal ID (publisher-id): CRIM.GM
ISSN: 2090-6528
ISSN: 2090-6536
Publisher: Hindawi Publishing Corporation
Article Information
Download PDF
Copyright © 2011 Dylan Wanaguru et al.
open-access:
Received Day: 3 Month: 7 Year: 2011
Accepted Day: 21 Month: 7 Year: 2011
Print publication date: Year: 2011
Electronic publication date: Day: 15 Month: 9 Year: 2011
Volume: 2011E-location ID: 470658
ID: 3350280
PubMed Id: 22606419
DOI: 10.1155/2011/470658

Multiseptate Gallbladder in an Asymptomatic Child
Dylan Wanaguru1*
Ashish Jiwane1
Andrew S. Day2, 3I3
Susan Adams1
1Department of Paediatric Surgery, Sydney Children's Hospital, University of New South Wales, High Street, Randwick, Sydney, NSW 2031, Australia
2Department of Gastroenterology, University of New South Wales, High Street, Randwick, NSW 2031, Australia
3Sydney Children's Hospital and School of Women's and Children's Health, University of New South Wales, High Street, Randwick, NSW 2031, Australia
Correspondence: *Dylan Wanaguru: d.wanaguru@yahoo.com.au
[other] Academic Editors: F.-Y. Chang, K. Haruma, and A. J. Lembo

1. Introduction

Multiseptate gallbladder (MSG) is a rare congenital anomaly with less than 50 cases described in the English literature. Of these, 13 are in paediatric patients [19]. We report a case of MSG in a one-year-old child. We review the data from the published literature to consider the most appropriate management of symptomatic and asymptomatic children, including whether or not cholecystectomy is indicated.


2. Case Report

A nine-month-old, previously well, female infant presented with an acute episode of vomiting and was diagnosed with an Escherichia coli urinary tract infection (UTI). Renal tract ultrasound was normal, but the gallbladder (GB) was incidentally noted to have “multiple thin smooth septa, giving a honeycomb appearance,” consistent with MSG (Figure 1). No other biliary tract abnormality was noted. The UTI was treated, and the child subsequently remained asymptomatic.

Repeat ultrasound six months later showed the images were unaltered. Liver function tests were normal apart from a raised alkaline phosphatase 1632 U/L (age appropriate normal range 80–450). Magnetic resonance cholangiopancreaticography (MRCP) confirmed the diagnosis of MSG, and excluded intra- and extrahepatic biliary and pancreatic anomalies. Nuclear medicine HIDA (hepatobiliary imino-diacetic acid) scan revealed no evidence of obstruction to bile flow at any level of the biliary tree.


3. Discussion

Multiseptate gallbladder was first described in 1963 by Simon and Tandon [10]. It is characterised by multiple thin septations within the gallbladder lumen, giving a honeycomb-like appearance. Simon and Tandon [10] proposed that this was due to incomplete vacuolisation of the developing gallbladder bud. Bhagavan et al. [11] have suggested that MSG may be a result of the solid embryonic GB growing faster than its bed and investing peritoneum, causing aberrant bends and kinks. The same authors also postulate that a variation in the wrinkling, lobulation, and clefting of the gallbladder (seen in cat and guinea pig embryos) may result in multiseptation [11].

Including the current case, 44 cases are described in the English literature, with a male-to-female ratio of almost 1 : 2. Overall the mean age at diagnosis of these individuals was 28.6 years (range from 15 days to 70 years). Thirty of these cases were in adults [1036]. Thirteen have been reported in children: eight being female (Table 1) [19]. Most children were diagnosed in mid-to-late childhood (mean age 9.4 years) although one was detected at 15 days of age.

Biliary symptoms such as right upper quadrant pain, nausea and vomiting are the most common complaints in this condition, with 31 of the 44 cases presenting in this manner. Only three of the 44 cases were associated with cholelithiasis [8, 12, 13], and one was associated with acute acalculous cholecystitis [14], but none of these were in children. The presence of an associated biliary tract abnormality is an important consideration in the assessment of MSG. One case in a 46-year-old woman was associated with anomalous pancreaticobiliary ductal union [15]. The three reported cases found to have associated choledochal cysts were all in children (23% of paediatric cases). These children presented with jaundice [2, 9] and a combination of fever, nausea, and abdominal discomfort [6]. Seven of the remaining 10 children presented with biliary symptoms [1, 3, 5, 7, 8]. The other three children (including the current case) were asymptomatic with no biliary tract anomaly [4, 5]. The incidence of asymptomatic MSG in the community is unknown, so it is not possible to comment on the likelihood of symptoms developing in these children.

There is no reported association between uncomplicated MSG and malignancy; however, there is a known link between biliary tract anomalies and cholangiocarcinoma. The incidence of malignancy in choledochal cyst is reported between 10% and 30%, and anomalous arrangement of the pancreaticobiliary duct is considered to be a high-risk factor for biliary tract malignancy [16, 17]. Consequently, four of the reported cases of MSG (all adults) with such associated abnormalities have an increased long-term risk of malignancy.

The majority of reported cases were diagnosed on ultrasound. Differential diagnosis includes desquamated gallbladder mucosa, polypoid cholesterolosis, hydatid cyst and acute hepatitis [18, 19]. Kocakoc et al. [7] first reported the use of MRCP to noninvasively define biliary and pancreatic pathology in MSG. MRCP is useful in confirming the diagnosis and delineating any associated biliary tree pathology and eliminates the potential complications associated with endoscopic cholangiopancreaticography (ERCP).

Saimura et al. [19] conducted biliary manometry and scintigraphy on a 30-year-old man with epigastric pain and MSG. Impairment of bile flow into and out of the gallbladder was demonstrated, supporting a biliary origin of the patient's pain. In the same study, Saimura and colleagues went on to reproduce typical biliary colic in the patient by injection of Cerulein to stimulate gallbladder contraction.

In symptomatic patients diagnosed with MSG, cholecystectomy provides relief of symptoms [21]. The three children with associated choledochal cyst were successfully treated with excision of the extrahepatic biliary tree combined with hepatojejunostomy or choledochoduodenostomy. In the 12 reported cases of asymptomatic and uncomplicated MSG, including the three paediatric cases, management has been nonoperative with regular followup.


4. Conclusion

MSG is a rare biliary anomaly that may be diagnosed in the first decades of life. Associated biliary tract anomalies should be excluded, particularly when the diagnosis is made in childhood. Cholelithiasis is rarely associated with MSG, and has never been reported in a child. Investigation with ultrasound, MRCP, and HIDA scan is recommended. In the absence of symptoms attributable to the MSG, or an associated biliary tract anomaly, nonoperative management in children and regular followup is reasonable. Symptomatic uncomplicated MSG is successfully treated with cholecystectomy.


References
1. Haslam RH,Gayler BW,Ebert PA. Multiseptate gallbladder. A cause of recurrent abdominal pain in childhoodAmerican Journal of Diseases of ChildrenYear: 196611266006035928828
2. Pery M,Kaftori JK,Marvan H. Ultrasonographic appearance of multiseptate gallbladder: report of a case with coexisting choledochal cystJournal of Clinical UltrasoundYear: 19851385705733934221
3. Fremond B,Stasik C,Jouan H,et al. The multiseptate gallbladder. A rare malformation of the biliary tractChirurgie PediatriqueYear: 19893062922942698288
4. Adear H,Barki Y. Multiseptate gallbladder in a child: incidental diagnosis on sonographyPediatric RadiologyYear: 1990203p. 192
5. Strauss S,Starinsky R,Alon Z. Partial multiseptate gallbladder: sonographic appearanceJournal of Ultrasound in MedicineYear: 19931242012038497025
6. Tan CEL,Howard ER,Driver M,Murray-Lyon IM. Non-communicating multiseptate gall bladder and choledochal cyst: a case report and review of publicationsGutYear: 19933468538568314522
7. Kocakoc E,Kiris A,Alkan A,Bozgeyik Z,Sen Y,Ozdemir H. Multiseptate gallbladder in a child with chronic abdominal pain: ultrasonography, magnetic resonance imaging and magnetic resonance cholangiography findingsEuropean Journal of Radiology ExtraYear: 20034712225
8. Erdogmus B,Yazici B,Ozdere BA,Akcan Y. Clinical and ultrasonographical findings in patients with multiseptate gallbladderTohoku Journal of Experimental MedicineYear: 2004204321521915502421
9. Bahadir B,Ozdamar SO,Gun BD,Bektas S,Numanoglu KV,Kuzey GM. Ectopic pancreas associated with choledochal cyst and multiseptate gallbladderPediatric and Developmental PathologyYear: 20069431231516944990
10. Simon M,Tandon BN. Multiseptate gallbladder. A case reportRadiologyYear: 196380848613977484
11. Bhagavan BS,Amin PB,Land AS,Weinberg T. Multiseptate gallbladder. Embryogenetic hypothesesArchives of pathologyYear: 19708943823855435679
12. Croce EJ. The multiseptate gallbladderArchives of SurgeryYear: 197310711041054714428
13. Erdogmus B,Yazici B,Safak AA,Ozdere BA. Multiseptate gallbladder with acute acalculous cholecystitisJournal of Clinical UltrasoundYear: 200432842342415372452
14. Yamamoto T,Matsumoto J,Hashiguchi S,Yamaguchi A,Sakoda K,Taki C. Multiseptate gallbladder with anomalous pancreaticobiliary ductal union: a case reportWorld Journal of GastroenterologyYear: 200511386066606816273627
15. Metcalfe MS,Wemyss-Holden SA,Maddern GJ. Management dilemmas with choledochal cystsArchives of SurgeryYear: 2003138333333912611583
16. Ono S,Sakai K,Kimura O,Iwai N. Development of bile duct cancer in a 26-year-old man after resection of infantile choledochal cystJournal of Pediatric SurgeryYear: 2008436E17E1918558159
17. Nakazawa T,Ohara H,Sano H,et al. Multiseptate gallbladder: diagnostic value of MR cholangiography and ultrasonographyAbdominal ImagingYear: 200429669169315185027
18. Türkvatan A,Erden A,Çelik M,Ölçer T. Ectopic hypoplastic and multiseptate gallbladder with coexisting choledochal cyst: evaluation with sonography and magnetic resonance cholangiopancreaticographyJournal of Clinical UltrasoundYear: 2006342889116547984
19. Saimura M,Ichimiya H,Naritomi G,et al. Multiseptate gallbladder: biliary manometry and scintigraphyJournal of GastroenterologyYear: 19963111331368808443
20. Kapoor V,Federle MP,Peterson MS,Coll DA. Long-term sonographic follow-up of stable imaging findings of multiseptate gallbladderJournal of Ultrasound in MedicineYear: 200221667768012054306
21. Bigg RL. Multiseptate gallbladderArchives of SurgeryYear: 196488350150214088284
22. Shaw RB,Donato CA,Douglas DD. Multiseptate gallbladder diagnosed during pregnancyAmerican SurgeonYear: 197541128188221239232
23. Jena PK,Hardie RA,Hobsley M. Multiseptate hypoplastic gallbladderBritish Journal of SurgeryYear: 1977643192193890264
24. Okuda K,Nakajima M,Nakayama M,Nomura F. Multiseptate gallbladder. Report of a case with a review of literatureActa Hepato-GastroenterologicaYear: 19792617075425806
25. Toombs BD,Foucar E,Rowlands BJ,Strax R. Multiseptate gallbladderSouthern Medical JournalYear: 19827556106127079823
26. Oliva IO,Moran MR,Sanchez F,Alonso AG. Multiseptate gallbladderInternational SurgeryYear: 198570183844019093
27. Lev-Toaff AS,Friedman AC,Rindsberg SN. Multiseptate gallbladder: incidental diagnosis on sonographyAmerican Journal of RoentgenologyYear: 19871486111911203554932
28. Isomoto I,Matsunaga N,Ochi M,et al. Multiseptate gallbladder: computed tomographic appearanceRadiation MedicineYear: 19908255572217864
29. Vasinrapee P,Linden K,Cook RE. Multiseptate gallbladder demonstrated on Tc-99m hepatobiliary imagingClinical Nuclear MedicineYear: 1990154p. 272
30. Hahm KB,Yim DS,Kang JK,Park IS. Cholangiographic appearance of multiseptate gallbladder: case report and a review of the literatureJournal of GastroenterologyYear: 19942956656688000519
31. Naritomi G,Kimura H,Konomi H,et al. Multiseptate gallbladder as a cause of biliary painAmerican Journal of GastroenterologyYear: 19948910189118927942690
32. Paciorek ML,Lackner D,Daly C,Sekas G. A unique presentation of multiseptate gallbladderDigestive Diseases and SciencesYear: 19974212251925239440630
33. Saddik D. Multiseptate gall-bladder: incidental diagnosis on ultrasoundAustralasian RadiologyYear: 19984243743769833380
34. Miwa W,Toyama K,Kitamura Y,et al. Multiseptate gallbladder with cholelithiasis diagnosed incidentally in an elderly patientInternal MedicineYear: 200039121054105911197789
35. Kocakoc E,Kiris A,Bozgeyik Z. Multiseptate gallbladder: MR and MR cholangiographic findingsAbdominal ImagingYear: 2005304p. 501
36. Yamasaki A,Chijiiwa K,Jimi SI,et al. Multiseptate gallbladder: report of a caseHepato-GastroenterologyYear: 2008558485986018705283

Article Categories:
  • Case Report


Previous Document:  Retroperitoneal schwannoma: a rare case.
Next Document:  Atypical Clinical and Diagnostic Features in Ménétrier's Disease in a Child.