Laminated intestinal calculi – A rare complication of Crohn’s … · 2019. 8. 1. · Laminated...

Laminated intestinal calculi –A rare complication of Crohn’s

diseaseHUGH J FREEMAN MD, ANDREW SEAL MD, DAVID LI MD

INTESTINAL FOREIGN BODIES MAYbe either exogenous or endogenous(1). Endogenous foreign bodies may beprimary and formed within the intesti-nal tract (eg, enteroliths), or secondaryand enter the gastrointestinal tractfrom adjacent structures (eg, gall-stones). Enteroliths may be further sub-divided into a false type formed fromclumping of insoluble ingested materialor by inspissation of bowel contents(eg, phytobezoars, trichobezoars, feco-liths), or a true type formed from pre-cipitation of substances present in thelumen (eg, bile or mineral salts, such ascalcium) (1).

Laminated radiopaque intestinalcalculi detected during the course ofclinical evaluation may be associatedwith congenital (eg, Meckel’s diver-ticulum, ileal duplication cyst) or ac-quired conditions (eg, anastomotic orpostinflammatory strictures, such asCrohn’s disease of the small intestine).in both, local stasis of the intestinalcontents is critical to stone develop-ment (1).

Although small intestinal stricturesare extremely common in Crohn’s dis-ease, enterolithiasis is very rare, beingrecorded to date in only 14 patients

BRIEF COMMUNICATION

HJ FREEMAN, A SEAL, D LI. Laminated intestinal calculi – A rare complica-tion of Crohn’s disease. Can J Gastroenterol 1995;9(1):13-16. A 59-year-oldmale with a 36-year history of Crohn’s disease and repeated resections for smallintestinal strictures developed anemia and symptoms of an intermittent partialbowel obstruction. Barium studies showed recurrent small intestinal strictures aswell as filling defects in a dilated loop proximal to a stenosed segment. Subse-quent abdominal films and a computed tomographic study suggested laminatedradiopaque calculi with peripheral calcification in the dilated small intestinalloop. Resection of the strictured segment confirmed the presence of intestinalenterolithiasis.

Key Words: Abdominal calcification, Crohn’s disease, Enteric stones, Enterolithiasis,Inflammatory bowel disease, Small intestinal strictures

Calculs intestinaux lamellaires : rare complication de lamaladie de Crohn

RÉSUMÉ : Un homme de 59 ans atteint depuis 36 ans d’une maladie de Crohnet ayant subi de multiples résections intestinales pour strictures a développé del’anémie et des symptômes d’obstruction intestinale intermittente. Les épreuvesbarytées ont révélé des strictures récurrentes au niveau de l’intestin grêle, demême que des anomalies de remplissage dans une anse dilatée avoisinant un seg-ment sténosé. Les clichés abdominaux subséquents et un examen tomographiqueont suggéré la présence de calculs lamellaires radio-opaques, avec calcificationpériphérique dans l’anse dilatée du grêle. La résection du segment porteur destrictures a confirmé la présence d’entérolithiase intestinale.

Departments of Medicine (Gastroenterology), Surgery and Radiology, University of BritishColumbia, Vancouver, British Columbia

Correspondence and reprints: Dr Hugh Freeman, ACU F-137, Gastroenterology, UniversityHospital (UBC Site), 2211 Wesbrook Mall, Vancouver, British ColumbiaV6R 3V8. Telephone (604) 822-7216

Received for publication March 27, 1994. Accepted May 27, 1994

CAN J GASTROENTEROL VOL 9 NO 1 JANUARY/FEBRUARY 1995 13

(2-10). Detection is important, how-ever, since intestinal calculi may be as-sociated with obstruction, bleeding,refractory anemia and, in rare in-stances, perforation.

CASE PRESENTATIONA 51-year-old male was initially re-

ferred for assessment of Crohn’s diseasein November 1984. A diagnosis ofCrohn’s disease involving the small in-testine was first established in 1956 inthe United States followed by repeated

admissions to a teaching hospital inMontreal for abdominal pain andbloody diarrhea over a two-year period.A 20 cm small bowel resection with anileocolic anastomosis was done in 1959followed by a second resection for re-current ileal disease in 1969. Pathologi-cal review revealed characteristicfeatures of Crohn’s disease with granu-lomatous inflammation. In 1970, bar-ium radiographic studies demonstratedrecurrent small bowel disease withstricture formation. The patient was

treated with sulfasalazine. Although heremained virtually asymptomatic, bar-ium studies of the upper and lower gas-trointestinal tract in 1974 revealed ajejunal stricture with small bowel dila-tion proximal to this narrowed seg-ment.

In 1984, abdominal pain recurred.Laboratory studies, including a hemo-globin, white blood cell and plateletcounts, liver chemistry tests, sedimen-tation rate, serum iron, folic acid andvitamin B12, were normal. Urinalysiswas normal. Colonoscopy and biopsieswere normal. Barium studies revealedsimilar changes to 1974; in addition,thickening and nodularity of the mu-cosa in the descending duodenum werenow evident. Sulfasalazine treatmentwas continued and the patient’s symp-toms appeared to resolve with alactose-free diet. Clinical evaluationsand blood tests done semi-annuallyover the next eight years were normal.In 1990, abdominal pain recurred tem-porarily; barium studies now showed anormal duodenum as well as previouslydefined strictures but no new radiologi-cal findings and, following this study,his symptoms spontaneously resolved.

In May 1992, the patient was re-evaluated because of recurrent and se-vere abdominal pain. Laboratory stud-ies revealed (normal range in brackets):hemoglobin, 126 g/L (140 to 180),mean cell volume, 76 fL (82 to 98),

Figure 1) Barium radiograph of the small in-testine, May 1992, showing small bowel stric-ture with dilated loop of small intestine; withinthe dilated small bowel loop, a filling defect isseen

Figure 2) Delayed film showing a persistentcollection of barium in a dilated small bowelloop 24 h later with two persisting filling defects

Figure 3) Abdominal film in September 1992showing two intestinal calculi with peripherallamination

Figure 4) Computed tomographic image from September 1992 showing multiple thickened small in-testine with a single widely dilated intestinal loop containing the two calculi

14 CAN J GASTROENTEROL VOL 9 NO 1 JANUARY/FEBRUARY 1995

FREEMAN et al

white blood cell count, 7.3x109/L (4.0to 11.0), serum ferritin, 16 �g/L (18 to250), serum iron, 6 �mol/L (7 to 23)and serum iron saturation 0.09 (0.20 to0.55). A barium study revealed a mark-edly dilated loop of small bowel proxi-mal to a stricture; in the dilated loop, afilling defect was seen (Figure 1). In de-layed films done 24 h later, residualbarium was seen in the dilated loop;two radiolucent and mobile filling de-fects were clearly seen (Figure 2). Fol-lowing treatment with oral5-aminosalicylic acid his pain subsided.

In September 1992, pain recurred inthe patient’s right upper quadrant withdevelopment of erythema and swellingover the right rectus sheath. Within24 h a small amount of purulent drain-age was seen from an apparent fistula.Except for a hemoglobin level of 112g/L, other laboratory studies, includinga white blood cell count, were normal.An abdominal film revealed two struc-tures with faint peripheral calcification(Figure 3). An abdominal computedtomography scan showed a thickenedright rectus muscle with an irregular3 cm fluid collection typical of an ab-scess. A dilated segment of small bowelwas present with two peripherally cal-cified structures (Figure 4). After ini-tiation of parenteral nutrition andintravenous antibiotics, a fistulogramdemonstrated communication with the

small intestine; again, the two fillingdefects were seen (Figure 5). After fail-ure of the fistula to heal with conserva-tive management, a small intestinalresection was done including the en-terocutaneous fistula. The resected seg-ment of small bowel revealedcharacteristic features of Crohn’s dis-ease with two enteroliths (Figure 6).

DISCUSSIONPrimary enterolithiasis or intestinal

calculi are rare but may be a clue to oc-cult intestinal disease and must be dif-ferentiated from secondary calculi thatoriginate elsewhere and subsequentlyenter or erode into the gastrointestinaltract, such as gallstones. Formation ofintestinal calculi requires the properchemical milieu within the lumen ofthe intestine (1,8,10) as well as condi-tions that lead to local stasis, as may beobserved with congenital abnormali-ties such as a Meckel’s diverticulum ora duplication cyst of the distal ileum.Occasionally, acquired causes of intes-tinal stasis may be responsible, includ-ing postoperative or postinflammatorystrictures such as occurs in Crohn’s dis-ease. Although our patient had long-standing Crohn’s disease, the initial de-tection of intestinal calculi (eg, withabdominal films) in a new patientshould lead to further studies includingbarium radiographs to exclude unrcog-

nized intestinal disease, includingCrohn’s disease. Enteroliths are alwaysclinically significant because they mayrepresent the first clue to an occultsmall bowel diverticulum or a stenosinglesion. Misinterpretation as biliary orurinary tract calculi or as innocuousconcretions in the peritoneal orextraperitoneal spaces could result ininappropriate treatment (1).

To date, there have been 14 patientsreported in the literature with Crohn’sdisease and a clinical course that hasbeen complicated with intestinal cal-culi (1-10). Usually, but not exclu-sively, these are detected in the smallintestine. Single or multiple calculiwith smooth or faceted borders havebeen described, usually, as in our pa-tient, in a chronically dilated smallbowel loop proximal to a stricture. Insome patients, intestinal calculi mayhave been primarily responsible for theobstructing symptoms along withchronic or recurrent blood loss and irondeficiency anemia that has been refrac-tory or difficult to manage (10). In veryrare patients with Crohn’s disease andintestinal calculi, the presentation hasbeen dramatic with perforation (5);thus, while rare, the potentially seriousoutcome of intestinal stones deservesemphasis. In all patients described withCrohn’s disease and intestinal calculi,the average duration between the

Figure 6) Resected small intestine showing typical features of Crohn’s disease with intestinal calculi

Figure 5) Fistulogram demonstrating commu-nication of the fistulous tract with the small in-testine containing two filling defects

CAN J GASTROENTEROL VOL 9 NO 1 JANUARY/FEBRUARY 1995 15

Enteroliths and Crohn’s disease

clinical onset of Crohn’s disease andthe detection of enterolithiasis wasprolonged, usually over a decade, rangefive to 33 years (10). In our patient,who had calculi detected in 1992,the Crohn’s disease was first estab-lished elsewhere 36 years earlier in1956.

Although stasis appears to be a criti-cal factor in the pathogenesis of thesestones, their chemical composition isbelieved to depend on the site of forma-

tion along the length of the small intes-tine as well as the pH of the intestinalchyme at the location of the stones.The relatively lower pH of the duode-num and proximal jejunum apparentlyfavours deposition of bile acids thattend to be radiolucent on plain ab-dominal radiographs. In contrast, ilealstones usually contain mineral saltsthat tend to precipitate in the more al-kaline environment of the distal smallintestine. These calculi tend to be

laminated and more radiopaque; a vari-ety of minerals such as calcium carbon-ate, calcium oxalate and, rarely,magnesium or even barium sulphatemay be present (1,8,10). The possiblerole that dietary factors, medicationssuch as antacids or investigative bar-ium studies may play in stone develop-ment is not known.

REFERENCES1. Atwell JD, Pollock AV. Intestinal

calculi. Br J Surg 1960;47:367-74.2. Katz I, Fischer RM. Enteroliths

complicating regional enteritis.A report of two cases. Am JRoentgenol 1957;78:653-61.

3. Brettner A, Euphrat EJ. Radio-logical significance of primaryenterolithiasis. Radiology1970;94:283-8.

4. Johnson CK, Levagood FB. Regionalenteritis: enteroliths causing

intermittent obstruction. Arizona Med1975;32:94-6.

5. Zeit RM. Enterolithiasis associatedwith ileal perforation in Crohn’sdisease. Am J Gastroenterol1979;72:662-4.

6. Javors BR, Bryk D. Enterolithiasis:a report of four cases. GastrointestRadiol 1983;8:359-62.

7. Schut JM, Mallens WMC. Calcifiedenteroliths in regional enteritis. DiagnImaging Clin Med 1986;55:146-50.

8. Paige ML, Ghahremani GG, Brosnan

JJ. Laminated radiopaque enteroliths:diagnostic clues to intestinal pathology.Am J Gastroenterol 1987;82:432-7.

9. Harvey IC, White HJO. Stones,Crohn’s disease and Meckel’sdiverticulum. Br Med J 1976;2:1107.

10. Yuan JG, Sachar DB, Koganei K,Greenstein AJ. Enterolithiasis,refractory anemia, and strictures ofCrohn’s disease. J Clin Gastroenterol1994;18:105-8.

16 CAN J GASTROENTEROL VOL 9 NO 1 JANUARY/FEBRUARY 1995

FREEMAN et al

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