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O R IGINA L A R T IC L E

Brendan T. Campbell Nathan P. NarasimhanEustace S. Golladay Ronald B. Hirschl

Biliary dyskinesia: a potentially unrecognized causeof abdominal pain in children

Received: 1 August 2003 / Accepted: 27 February 2004 / Published online: 18 August 2004 Springer-Verlag 2004

Abstract Biliary dyskinesia is defined as symptomaticbiliary colic without cholelithiasis, and is diagnosedduring cholescintigraphy by assessing gallbladder emp-tying with cholecystokinin (CCK) stimulation. Unfor-

tunately, gallbladder emptying is not routinely assessedduring cholescintigraphy in pediatric patients. The pur-pose of this review is to assess the effectiveness of cho-lecystectomy in patients with chronic abdominal painand delayed gallbladder emptying and to assess whetherthese findings correlate with the histologic evidence ofchronic cholecystitis. We retrospectively reviewed themedical records of all patients (n=16) at our institutionfrom October 1997 to August 2001 who underwentquantitative cholescintigraphy with CCK stimulationthat demonstrated delayed gallbladder emptying(

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stimulated cholescintigraphy at our institution had theirgallbladder ejection fraction measured using a standardtechnique that is described in detail elsewhere [5]. Dataregarding symptom resolution postoperatively was ob-tained from clinic notes. This study was approved by the

Institutional Research Review Committee at SparrowHealth System (Lansing, Michigan, USA) and theInstitutional Review Board at Hurley Medical Center(Flint, Michigan, USA).

Results

Laparoscopic cholecystectomy was performed in 16patients with chronic abdominal pain who had preop-erative cholescintigraphy. Each of the 16 patients dem-onstrated delayed gallbladder emptying (Table 1; meanejection fraction: 158%, range: 332%). The mean age

was 122 years (range: 817 years). There were 11 girls(69%) and five boys (31%). Presenting symptoms in-cluded abdominal pain (86%), fatty food intolerance(27%), emesis (13%), and diarrhea (13%). Mean dura-tion of abdominal pain before operation was1119 months (range: 2 weeks6 years). There were noconversions to open operation and no intraoperative orpostoperative complications. One patients symptomspersisted postoperatively, but abdominal pain resolvedin all other patients. Histologic evidence of chroniccholecystitis was demonstrated in 86% of surgicalspecimens. All five patients who underwent concurrentappendectomy had normal appendiceal histology.

Discussion

We have demonstrated that most children with chronicabdominal pain and no evidence of cholelithiasis whohave delayed gallbladder emptying on CCK-stimulatedcholescintigraphy have resolution of symptoms follow-ing laparoscopic cholecystectomy. We have also shown acorrelation between these findings and histologic evi-

dence of chronic cholecystitis.Biliary dyskinesia has been a recognized clinical en-

tity in adults for more than 2 decades, and laparoscopiccholecystectomy is now believed by many authors to bea highly effective treatment for this condition [610].Two recent reviews of biliary tract disease in childrenand indications for pediatric cholecystectomy, however,fail to mention biliary dyskinesia [1112]. Biliary dys-kinesia (also called chronic acalculous cholecystitis) hasonly recently received attention in the pediatric surgicalliterature [24].

Our findings corroborate those of the three previ-ously published case series on this subject in children.

The patients with biliary dyskinesia described in thesestudies were between 5 and 18 years old, and the con-dition appears more commonly in females. The non-specific nature of the childrens complaints described inthese studies coupled with the absence of abnormalitieson routine laboratory and radiologic tests usually delaysdiagnosis for nearly a year, and often for significantlylonger.

For the five children who underwent concurrentappendectomy, it is possible that the appendectomy ra-ther than the cholecystectomy, despite normal appen-diceal histology, was responsible for the cessation oftheir symptoms. In the one patient who did not experi-

ence symptom relief following cholecystectomy, limitedfollow-up provided no alternative explanation for theetiology of the abdominal pain.

The small sample size of this study, along with thefact that it is a retrospective case series without acomparison group, limits the quality of the evidence thatit provides [13]. Data provided by case series like thisone and those cited in this study could be improved withmore detailed and thorough postoperative follow-up.Future studies in which children with persistentabdominal pain of unclear etiology are identified earlyand evaluated prospectively will provide valuable insightinto the natural history of biliary dyskinesia. Promising

Table 1 Characteristics of patients undergoing lap cholecystec-tomy for biliary dyskinesia (n=16)

n (%)

Age (mean) 122 yearsGenderMale 5 (31)Female 11 (69)

SymptomsAbdominal pain 14 (86)Fatty food intolerance 4 (27)Emesis 2 (13)Diarrhea 2 (13)

Duration of symptoms (mos) 1119Mean GB ejection fraction (%) 158Complications 0Resolution of symptoms 15 (94)Chronic cholecystitis 12 (86)

Table 2 Literature review ofpediatric biliary dyskinesia caseseries

Study No. of patients

Mean age(years)

Duration ofsymptoms (mos)

Symptomresolution (%)

Histologicchroniccholecystitis(%)

Tsakayannis et al. [2] 12 13 100 100Gollin et al. [3] 29 13 11 79 72Michail et al. [4] 63 12 14 72 80Campbell et al.

(current study)16 12 11 94 86

580

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computer models for improving the diagnostic accuracyof less common causes of abdominal pain in children arebeing developed [14].

Biliary dyskinesia is a rare cause of persistentabdominal pain in children and a diagnosis of exclusion.When routine laboratory and radiologic tests areinconclusive, CCK-stimulated cholescintigraphy shouldbe considered. Children whose studies demonstrateabnormal gallbladder emptying should then be consid-ered candidates for laparoscopic cholecystectomy.

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