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. 2024 Sep 18;123:110317. doi: 10.1016/j.ijscr.2024.110317

Multiseptate gallbladder in a pediatric pateint: A case report and review of literature

Ahmed Amgad a,b,c,⁎, Sarah Magdy Abdelmohsen d, Ahmed K Ali e, Tarek Abdelazeem Sabra e
PMCID: PMC11437745  PMID: 39303484

Abstract

Introduction

Multiseptate gallbladder (MSG) is an uncommon congenital condition characterized by internal septa dividing the gallbladder into multiple compartments. This condition poses unique challenges in diagnosis and management due to its rarity and varied clinical presentations.

Case presentation

A 4-year-old female presented with a three-month history of colicky abdominal pain. Imaging studies confirmed MSG with gallbladder sludge but no stones. Despite normal initial lab results, the patient underwent a laparoscopic cholecystectomy due to persistent symptoms, resulting in a full recovery with no complications over six months.

Discussion

The etiology of MSG is unclear, with several embryological theories proposed. Diagnosis relies on imaging techniques like ultrasound and MRI. A literature review of 37 pediatric cases shows a higher prevalence in females and varied clinical presentations. Management includes conservative observation or surgical intervention, with cholecystectomy proving effective for symptomatic cases.

Conclusion

Multiseptate gallbladder is a rare congenital anomaly in pediatric patients, often requiring imaging techniques like MRCP for accurate diagnosis. While many cases remain asymptomatic and can be managed conservatively, surgical intervention is beneficial for those with persistent symptoms. Early diagnosis and tailored management lead to favorable outcomes.

Keywords: Multiseptate gallbladder, Cholecystectomy, Congenital anomaly, Pediatrics, Case report

Highlights

  • •

    MSG A rare congenital anomaly with septa dividing the gallbladder into compartments.

  • •

    MRCP is preferred for non-invasive visualization of the biliary tree over ERCP and other imaging methods.

  • •

    MSG management ranges from conservative observation to surgery, depending on symptoms' severity.

  • •

    Thorough assessment and individualized treatment are vital for optimal outcomes and complication prevention.

1. Introduction

Multiseptate gallbladder (MSG) is a rare congenital anomaly characterized by the presence of multiple internal septa dividing the gallbladder lumen into numerous compartments of varying sizes [1,2]. The first pediatric case was described by Haslam et al. in 1966 [3]. MSG poses distinct clinical challenges because its presentation can range widely, often mimicking more common biliary conditions. This variability contributes to its clinical relevance, as MSG may cause significant biliary symptoms or remain completely asymptomatic, making it difficult to establish a clear diagnostic pathway.

In pediatric populations most cases of MSG are discovered incidentally during imaging studies for unrelated conditions [4]. However, some patients may present with symptoms such as right upper quadrant pain, which often mimics more common biliary pathologies. The clinical presentation can range from asymptomatic to recurrent episodes of biliary colic, with potential complications including biliary sludge, cholelithiasis, and choledochal cysts [[5], [6], [7]].

Herein, we present the case of a 4-year-old female patient with a multiseptate gallbladder, who experienced three months of colicky abdominal pain, followed by a comprehensive review of the available literature on diagnosis and appropriate therapeutic management which clarifies the uncertainties in managing MSG, particularly regarding the decision of whether to operate or to follow a conservative approach with regular follow-up. This manuscript was prepared following the SCARE guidelines [8].

2. Case presentation

A 4-year-old female patient presented with a history of colicky abdominal pain persisting for three months. The pain was intermittent, associated with crying attacks and significant discomfort. The patient had no significant past medical history. Her physical examination revealed tenderness on superficial palpation in the right hypochondrium. There were no changes in skin color or sclera, no pruritus, and no fever.

Initial laboratory tests showed normal liver function tests and a mild elevation in white blood cell count (WBC = 11,000/μL). Ultrasound imaging of the abdomen revealed a septated gallbladder with evidence of sludge, with no stones or pericholecystic collection. An MRI of the abdomen confirmed the diagnosis, showing a clearly septated gallbladder with no other anomalies.

Given the persistent nature of the symptoms and the imaging findings, a decision by the senior surgeon was made to proceed with a laparoscopic cholecystectomy. The operation was performed under general anesthesia with the patient in a supine position and pneumoperitoneum maintained at 7 mmHg. The laparoscopic cholecystectomy was executed using a four-port technique: a 5 mm umbilical port with a 30° camera axis for visualization, a 3 mm port in the right upper quadrant for dissection and retraction, a 3 mm port in the left upper quadrant, and a 3 mm port positioned midway between the umbilicus and the left costal margin. A Maryland dissector was used to isolate the cystic artery, which was then clipped with two clips on the proximal side and one clip on the distal side to ensure secure ligation. Following this, the critical view of safety was achieved by clearing the hepatocystic triangle of all fat and fibrous tissue and ensuring that only two structures, the cystic duct and the cystic artery, were seen entering the gallbladder. The lower third of the gallbladder was also separated from the liver to expose the cystic plate. Subsequently, the cystic duct was isolated and clipped. The cystic duct was then doubly clipped and divided. Finally, the gallbladder was carefully dissected with hook diathermy and removed without intraoperative complications (Fig. 1, Fig. 2).

Fig. 1.

Fig. 1

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Gross appearance of the gallbladder post-excision. It is elongated with visible internal septations, creating multiple compartments within the lumen.

Fig. 2.

Fig. 2

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Dissection of the multiseptate gallbladder with a closer view of the internal septations being separated. The septa are shown to be delicate, semi-translucent, and dividing the gallbladder into multiple distinct chambers.

The patient was discharged the day after surgery in stable condition with post-operative instructions, including adherence to a low-fat diet to reduce biliary strain and careful monitoring for signs of infection or complications, such as fever, jaundice, or worsening abdominal pain. The patient was also advised to avoid strenuous activities for at least four weeks to support healing. During the six-month follow-up period, the patient underwent regular evaluations to check for any post-operative complications, such as bile leakage, wound infection, or recurrent symptoms. The follow-up period was uneventful, and the patient made a full recovery, exhibiting normal growth and activity levels with no biliary complaints.

3. Discussion

The etiology of multiseptate gallbladder remains unclear, with several embryological theories proposed to explain its formation. One prominent hypothesis suggests that MSG results from incomplete cavitation of the embryonic gallbladder bud, potentially leading to the absence of a muscular layer in the septa observed in many cases [1,3,[9], [10], [11]]. Another explanation, the “wrinkling theory,” posits that invaginations and fusions occur during the gallbladder's development, creating multiple septa [12]. Similarly, the “Phrygian cap theory” suggests that rapid growth of the gallbladder during its solid stage causes wrinkling and kinking due to spatial constraints, which are supported by the presence of muscle fibers within the septa [12,13].

Beyond embryological explanations, recent research has suggested potential genetic factors in the development of MSG. Familial cases and associations with other congenital anomalies, such as pancreaticobiliary maljunction, imply a possible genetic predisposition [9,14]. Additionally, studies have identified the presence of MSG alongside other biliary or gastrointestinal abnormalities, suggesting shared genetic or developmental pathways [10,11].

MSG in pediatric patients is often associated with a range of congenital anomalies affecting the hepatobiliary and pancreatic systems. Commonly linked conditions include choledochal cysts, which can cause biliary obstruction and increase malignancy risk, and pancreaticobiliary maljunction (PBM), predisposing patients to biliary reflux and pancreatitis [5,[14], [15], [16]]. Other associated anomalies include ectopic pancreatic tissue, biliary atresia, biliary sludge, lithiasis, and, less frequently, gallbladder duplication [6,7,16,17]. Additionally, MSG has been observed with congenital heart defects as ventricular septal defects (VSD) and patent ductus arteriosus (PDA), suggesting it may be part of a broader congenital syndrome [18]. These associations underscore the importance of comprehensive diagnostic evaluations to identify coexisting conditions and guide effective management.

Analyzing MSG alongside other biliary anomalies, such as choledochal cysts and biliary atresia, highlights both its distinct and overlapping clinical features [19,20]. Like choledochal cysts, MSG can present with biliary obstruction, pain, and an increased risk of complications such as cholangitis and, potentially, malignancy [19]. However, unlike choledochal cysts, which often require early surgical intervention due to their malignant potential, MSG may present without such risk, allowing for more conservative management in certain cases [21]. Similarly, while biliary atresia typically presents with severe jaundice and requires prompt surgical correction, MSG may exhibit a more variable and often milder clinical course, with some patients remaining asymptomatic [20]. This distinction suggests that while MSG shares some complications common to other biliary anomalies, it may not always necessitate the same aggressive treatment approaches.

To gain a deeper insight into this uncommon condition among pediatric patients, we conducted a systematic literature search using PubMed and Scopus. The structure of the review was based on PRISMA guidelines [22]. The search term used was “multiseptate gallbladder.” Our search identified a total of 137 articles, which were then screened for duplication and relevance. During the first screening, duplicates and articles focusing on adult patients were excluded. Both English and non-English articles were considered, and we included pediatric patients up to the age of 18. After the screening and validation process, we included 30 articles in the literature review, summarizing 37 cases [2,3,[5], [6], [7],9,[15], [16], [17],[23], [24], [25], [26], [27], [28], [29], [30], [31], [32], [33], [34], [35], [36], [37], [38], [39], [40], [41], [42]]. To illustrate our search and data extraction methods, a PRISMA flow chart was formulated (Fig. 3). The summarized details of these cases are presented in Table 1.

Fig. 3.

Fig. 3

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A PRISMA flow chart of our search through databases.

Table 1.

Cases of pediatric multiseptate gallbladder reported in the literature.

Author Year Age (year) Sex Biliary symptoms Duration of symptoms Associated anamoly Investigations Managment
Haslam [3] 1966 15 1/2 F Yes 4 Years None Cholecystogram Cholecystectomy
Pery [5] 1985 8 F Yes 4.5 years Choledochal cyst US, intravenous cholangiography Choledochoduodenostomy
Fremond [23] 1989 13 F Yes Not mentioned None US, cholecystogram Cholecystectomy
Adear [24] 1990 12 F No – None US Conservative
Strauss [25] 1993 16 M Yes Not mentioned None US N/A
9 F Yes Not mentioned None US, cholecystogram, CT N/A
3 M No – None US N/A
Tan [15] 1993 14 F Yes 1 year Choledochal cyst US, ERCP Cholecystectomy and Hepatojejunostomy
Choulot [26] 1996 9 M No – None US Conservative
Kong [27] 1996 9 M Yes 3 days None US, CT Conservative
Saddik [28] 1998 10 M No – None US Conservative
Dalgic [29] 2003 11 F Yes Not mentioned None US Cholecystectomy
Kocakoc [30] 2003 9 M Yes 3 Years None US, MRI, MRCP Conservative
Erdogmus [6] 2004 10 F Yes Not mentioned Cholelithiasis US Cholecystectomy
12 M Yes Not mentioned None US Cholecystectomy
Bahadir [16] 2006 15 Days M Yes 12 days Ectopic pancreas/choledochal cyst US Total excision of cyst with Roux-en-Y anastomosis
Demirpolat [31] 2010 5 F Yes 2 Years None US Conservative
Herliczek [32] 2011 11 M No – None US N/A
Wanaguru [9] 2011 9 months F No – None US, MRCP, HIDA scan Conservative
López [33] 2012 1 week F No – None US, MRCP Conservative
Aydin [34] 2013 10 M Yes 3 h None US, MRI, MRCP Conservative
Geremia [7] 2013 10 M Yes Not mentioned biliary sludge US Conservative
Öztorun [35] 2017 1 M Yes Not mentioned None US, MRCP Cholecystectomy
Vazquez [36] 2017 15 months M Yes Not mentioned None US, MRCP, HIDA scan Conservative
Sabra [42] 2017 12 M Yes 6 months None US, MRCP Cholecystectomy
Bertozzi [37] 2019 7 F Yes 2 years None US, MRCP Cholecystectomy
Mendola [17] 2019 3 F Yes Not mentioned Cholelithiasis US, MRCP Cholecystectomy
Hsieh [38] 2021 14 F Yes 6 months None US, CT, MRCP Cholecystectomy
Zahid [39] 2021 10 F Yes Not mentioned None US Conservative
Gazagne [2] 2022 7 days N/A Yes Not mentioned None US Conservative
1 month N/A No – None US Conservative
4 F No – None US Conservative
5 N/A No – None US Conservative
Oyachi [14] 2022 5 F No – Pancreaticobiliary maljunction (PBM) without biliary dilatation US, MRCP Cholecystectomy
González [40] 2023 2 F No – None US Conservative
Faraj [41] 2024 5 F No – None US Conservative
Present case 2024 4 F Yes 3 months None US, MRI Cholecystectomy
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*US: Ultrasound *CT: Computed Tomography *MRI: Magnetic Resonance Imaging *MRCP: Magnetic Resonance Cholangiopancreatography.

*ERCP: Endoscopic Retrograde Cholangiopancreatography *HIDA scan: Hepatobiliary Iminodiacetic Acid scan *N/A: Not Available.

3.1. Demographics

Out of the 37 cases, 20 cases (54 %) were female, 14 cases (38 %) were male, and 3 cases (8 %) were not known (N/A), resulting in a female-to-male ratio of approximately 1.43:1 among the cases with known gender. The ages of the patients ranged from 1 week to 16 years. Among the pediatric cases, the median age was 10 years, with the youngest being a 15-day-old male and the oldest a 16-year-old female. These demographics indicate a slightly higher prevalence of biliary symptoms in females compared to males.

3.2. Clinical presentation

Biliary symptoms were defined as either right upper quadrant pain, epigastric pain, fever, nausea, vomiting, or jaundice. Individuals with “recurrent abdominal pain” and/or “abdominal pain” were sorted into the group that did not have biliary symptoms. Among the 37 cases, 54 % (n = 20) reported right upper quadrant pain, making it the most common symptom. This was followed by nausea and vomiting, which were present in 22 % (n = 8) of the cases, and jaundice, observed in 11 % (n = 4) of the cases. Fever was the least common symptom, occurring in only 5 % (n = 2) of the cases.

The exact cause of pain in these cases remains unclear, but it is generally believed that sluggish bile flow and increased pressure within the bile ducts contribute to the discomfort [38]. Evidence for this includes the delayed bile movement observed in biliary manometry and scintigraphy tests [43].

The duration of symptoms varied widely, with some cases presenting emergency symptoms lasting from a few hours to several years. Although MSG is typically not linked with cancer, it can be complicated by conditions like choledochal cysts or abnormal connections in the pancreaticobiliary duct, which may heighten the risk of cancerous changes [44,45]. Hence, it is essential to conduct a thorough assessment of any associated ductal anomalies.

3.3. Investigative techniques

All cases were initially diagnosed using ultrasound (US), which showed the gallbladder with multiple intercommunicating compartments, fine echogenic bands, and no acoustic shadowing. Differential diagnoses included desquamated gallbladder mucosa, polypoid cholesterolosis, hydatid cysts, and acute hepatitis [27,[46], [47], [48], [49]]. In certain situations, further imaging methods such as endoscopic ultrasound, magnetic resonance imaging (MRI), magnetic resonance cholangiopancreatography (MRCP), computed tomography (CT), and endoscopic retrograde cholangiopancreatography (ERCP) were utilized to confirm the diagnosis and provide detailed visualization of the biliary tracts.

Although oral cholecystography (OCC) was used in the past, it has largely been replaced by these modern imaging techniques. Additionally, hepatobiliary iminodiacetic acid scans and biliary manometry with scintigraphy were employed to evaluate bile excretion functions. MRCP has become more favored over ERCP due to its non-invasive nature, lower radiation exposure, and ability to simultaneously identify biliary and pancreatic pathologies, which aids in treatment planning [45]. However, adjustments should be made according to patient's specific needs, as these diagnostic tools are essential for accurate diagnosis and effective treatment planning.

3.4. Management and treatment

Out of the 37 cases, 15 patients (41 %) underwent surgical treatment, primarily cholecystectomy, due to persistent biliary symptoms. Non-surgical management was chosen for 18 cases (49 %), particularly when symptoms were not severe or were self-limiting. For example, a 5-year-old female with pancreaticobiliary maljunction without biliary dilatation underwent cholecystectomy, while a 10-year-old male with biliary sludge was managed through observation.

The outcomes for both surgical and conservative management were generally similar, suggesting that the decision for surgery was primarily based on the severity of symptoms and the presence of associated anomalies, with follow-up deemed sufficient in asymptomatic cases. The prognosis largely depended on these factors, with surgical intervention often leading to symptom resolution when indicated.

4. Conclusion

To summarize, Multiseptate Gallbladder is a rare congenital anomaly predominantly presenting with biliary symptoms in pediatric patients, although asymptomatic cases are also common. Diagnosis primarily relies on imaging techniques, with MRCP favored for its non-invasive nature and detailed visualization of the biliary anatomy. The variability in clinical presentations and the potential for associated biliary anomalies, including those with higher malignancy risks, necessitate thorough diagnostic assessments. Management strategies vary, with cholecystectomy reserved for symptomatic cases, while asymptomatic patients can be managed with regular follow-ups.

Our review of 37 pediatric cases indicates that while some patients remain asymptomatic, those with persistent symptoms benefit significantly from surgical intervention. Overall, the prognosis is favorable, particularly with prompt and accurate diagnosis and appropriate management.

Consent for publication

Written informed consent was obtained from the patient's parents for publication of this case. A copy of the written consent is available for review by the Editor-in- Chief of this journal on request.

Ethical approval

It is a case report, and there is no need for ethical approval from Aswan University Ethical Committee as it does not contain any personal information.

Funding

None.

Author contribution

Conceptualization: AAM & SMA.

Methodology: AAM.

Formal Analysis: AAM.

Investigation: SMA.

Resources: AKA & SMA.

Writing – Original Draft: AAM.

Writing – Review & Editing: AAM & SMA.

Supervision: TAS & SMA & AKA.

Guarantor

Sarah Magdy Abdelmohsen.

Research registration number

There is no registration.

Conflict of interest statement

The authors declare that they have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this paper.

Contributor Information

Ahmed Amgad, Email: ahmed30204010101734@med.helwan.edu.eg.

Sarah Magdy Abdelmohsen, Email: sara.magdy@aswu.edu.eg.

Ahmed K. Ali, Email: Ahmedk.ali@aun.edu.eg.

Tarek Abdelazeem Sabra, Email: tareksabra@aun.edu.eg.

Availability of data and material

Available when the editor requests.

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