Staphylococcal acalculous cholecystitis in a child. (Case Report).

Abstract

Acute acalculous cholecystitis is inflammation of the gallbladder in the absence of gallstones. It usually occurs in critically ill patients and is rare in the pediatric age group. We describe a 12-year-old boy who presented with fever, jaundice, and abdominal pain and was found to have acute acalculous cholecystitis, sacroiliitis, and pelvic osteomyelitis associated with bacteremia as a result of Staphylococcus aureus. Antibiotic therapy without surgical intervention was effective. A high index of suspicion is required to make an early diagnosis and institute appropriate treatment for children with this condition. Although cholecystectomy has been considered the standard therapy, medical treatment alone can be successful.

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A cute acalculous cholecystitis is an inflammatory process of the gallbladder in the absence of gallstones. Disease of the gallbladder is rare in the pediatric age group. It is estimated that 1.3 pediatric cases occur for every 1,000 adult cases. (1) In pediatric patients, 30 to 50% of cholecystitis cases are acalculous, compared with 2 to 17% of cases in adult patients. (2-4) We present the case of a 12-year-old boy who had acute acalculous cholecystitis in association with sacroiliitis and pelvic osteomyelitis as a result of staphylococcal bacteremia.

Discussion

Our patient had acute acalculous cholecystitis, sacroiliitis, and pelvic osteomyelitis associated with S. aureus bacteremia. Acute acalculous cholecystitis is an inflammatory process of the gallbladder in the absence of gallstones. Duncan was the first, in 1844, to describe acute acalculous cholecystitis, and this presentation has been associated with a mortality rate more than twice that of acute calculous cholecystitis. (2,5) Acalculous cholecystitis occurs mostly in critically ill patients. (5,6) Risk factors include hyperalimentation, mechanical ventilation, massive blood transfusions, severe debilitation, severe trauma, and burns. (2-6) Its incidence in the pediatric age group is rare. The exact pathogenesis of acute acalculous cholecystitis is not fully understood; however, gallbladder ischemia and biliary stasis are two probable mechanisms. (2) Acute acalculous cholecystitis may be complicated by gangrene formation and perforation of the gallbladder. (2)

In 1975, Ternberg and Keating (7) reported 7 cases and reviewed another 67 cases of acute acalculous cholecystitis in children that had been reported between 1931 and 1969. The patients' ages ranged from younger than 1 month to 15 years. History of previous illness was present in 45 cases (60%) and included upper respiratory tract infection, diarrhea, scarlet fever, and burns with Pseudomonas sepsis. Right upper-quadrant abdominal pain was present in the majority of patients. Jaundice was noted in one-third of the patients. The most common incorrect diagnosis was appendicitis, which at times led to delays in appropriate management. Thirty-six (49%) of the 74 patients had cholecystectomy, and 25 (34%) had cholecystostomy. Gallbladder bile cultures were available from 31 patients, and they yielded organisms such as Staphylococcus albus, Eseherichia coil, Aerobacter aerogenes, nonhemolytic streptococcus, diphtheroids, Bacillus paratyphosum, Salmonella otonienberg, Pseudomonas and Leptospira interrogans, (8) Salm onella typhi, (9,10) Salmonella virchow, (11) Salmonella newport, (7) Group A streptococcus, (7) S. aureus, (12) Candida albicans, (13) ascariasis, (14) and malaria. (15)

In 1996, Tsakayannis et a1 (4) described 25 children with acalculous cholecystitis who had been treated at Children's Hospital in Boston during a 24-year period. Infection was designated acute if the duration of the child's illness was less than 1 month and chronic if it was longer than 3 months. Thirteen patients who ranged in age from 2 months to 20 years (median age, 14 yr) had acute cholecystitis. Fever, right upper-quadrant pain, and vomiting were documented in all of these patients. Jaundice was present in 38% of these patients, and a right upper-quadrant mass was seen in 23%. Abnormal liver function with elevated bilirubin was noted in 62%. Associated conditions included salmonellosis, cystic fibrosis, end-stage liver disease, hemolytic-uremic syndrome, and leukemia. The majority (9 of 13) of the patients underwent open cholecystectomy and recovered. Three of four critically ill patients were treated with intravenous antibiotics only and fully recovered. Histopathologic studies of nine gallbladder spec imens revealed acute inflammation. Salmonella species grew from two specimens. The remaining 12 children with chronic acalculous cholecystitis had right upper-quadrant pain with nausea and vomiting. All previously had been healthy. Their ages ranged from 7 to 18 years. The results of liver function tests were normal. Ultrasonography showed normal gallbladders in all patients, but 75% had abnormal findings on hydroxy iminodiacetic acid scans or cholecystograms. All underwent cholecystectomy with complete recovery. Chronic inflammation was noted on microscopic examinations of 6 of the 12 gallbladder specimens examined.

Gangrenous acalculous cholecystitis has been reported in association with necrotizing enterocolitis in a 2-month-old premature infant with Staphylococcus epidermidis sepsis. (16) Acute acalculous cholecystitis also has been seen in individuals with acquired immunodeficiency syndrome who were coinfected with cytomegalovirus or Cryptosporidium. (17)

Our patient was a previously healthy 12-year-old boy who had acute acalculous cholecystitis, sacroiliitis, and pelvic osteomyelitis in association with S. aureus bacteremia. Jaundice and abdominal pain resolved after antibiotic therapy, indicating that the acute cholecystitis was the result of inflammation of the gallbladder because of S. aureus infection. Thomas et al (12) described a 19-year-old patient with acute acalculous cholecystitis in association with S. aureus bacteremia. Cholecystectomy revealed a gangrenous gallbladder from which S. aureus was isolated. The patient was treated with flucloxacillin and recovered uneventfully. Our patient did not require any surgical intervention and responded favorably to antibiotic treatment. Although acute acalculous cholecystitis can be diagnosed clinically, imaging studies facilitate early diagnosis and treatment, thereby leading to decreased morbidity and mortality. Sonography and CT have been reported to be both highly sensitive (92 and 100%) and highly specif ic (96 and 100%). (18) Sonography is considered to be as sensitive as cholescintigraphy in making the diagnosis. (18) Cholecystectomy, both percutaneous and transhepatic, has been used successfully and is considered standard therapy. (2,19) As seen in our patient, however, antimicrobial treatment alone can be curative.

Conclusion

Acalculous cholecystitis can occur in the pediatric age group and should be suspected in ill children with right upper-quadrant pain and jaundice. A high index of suspicion leads to early diagnosis and treatment. In children, acute acalculous cholecystitis most often results from antecedent bacteremia and localization of bacteria in the gallbladder. Although the majority of cases have been managed with surgical intervention, medical treatment alone can be successful. Our case is an example of the successful treatment of such a patient with antibiotics and no surgical intervention.

Accepted June 17, 2002.

References

(1.) Hawkins PE, Graham FB, Holliday P. Gallbladder disease in children. Am J Surg 1966;111:741-744.

(2.) Glenn F, Becker CG. Acute acalculous cholecystitis: An increasing entity. Ann Surg 1982;195:131-136.

(3.) Sievert W, Vakil NB. Emergencies of the biliary tract. Gastroenterol Clin North Am 1988;l7:245-264.

(4.) Tsakayannis DE, Kozakewich HP, Lillehei CW. Acalculous cholecystitis in children. J Pediatr Surg 1996;31:127-131.

(5.) Shapiro MJ, Luchtefeld WB, Kurzweil S, Kaminski DL, Durham RM, Mazuski JE. Acute acalculous cholecystitis in the critically ill. Am Surg 1994;60:335-339.

(6.) Frazee RC, Nagorney DM, Mucha P Jr. Acute acalculous cholecystitis. Mayo Clin Proc 1989:64:163-167.

(7.) Temberg JL, Keating JP. Acute acalculous cholecystitis: Complication of other illnesses in childhood. Arch Surg 1975;110:543-547.

(8.) Vilaichone RK, Mahachai V, Wilde H. Acute acalculous cholecystitis in leptospirosis. J Clin Gastroenterol 1999;29:280-283.

(9.) Thambidoral CR, Shyamala J, Sarala R, Vatsala RB, Tamizhisai S. Acute acalculous cholecystitis associated with enteric fever in children. Pediatr Infect Dis J l995;14:812-813.

(10.) Winkler AP, Gleich S. Acute acalculous cholecystitis caused by Salmonella typhi in an 11-year-old. Pediatr Infect Dis J 1988;7:125-128.

(11.) James RA, Morris G, Winter R, Keston-Jones M. Acalculous cholecystitis due to Salmonella virchow, Br J Clin Pract 1990:44:767-768.

(12.) Thomas WE, Thornton JR, Thompson MH. Staphylococcal acalculous cholecystitis. Br J Surg 198l;68:136.

(13.) Hiatt JR, Kobayashi MR, Doty JE, Ramming KP. Acalculous candida cholecystitis: A complication of critical surgical illness. Am Surg 1991;57:825-829.

(14.) Kuzu MA, Ozturk Y, Ozbek H, Soran A. Acalculous cholecystitis: Ascariasis as an unusual cause. J Gastroenterol 1996;31:747-749.

(15.) Sanchez R, Portilla J, Boix v, Memo E, Murcia JM. Acalculous cholecystitis associated with Plasmodium falciparum malaria. Clin Infect Dis 2000;31:622-623.

(16.) Fernandes ET, Hollabaugh RS, Boulden TF, Angel C. Gangrenous acalculous cholecystitis in a premature infant. J Pediatr Surg 1989;24:608-609.

(17.) Nash JA, Cohen SA. Gallbladder and biliary tract disease in AIDS. Gastroenterol Clin North Am 1997;26:323-335.

(18.) Mirvis SE, Vainright JR, Nelson AW, Johnston GS, Shorr R, Rodriguez A, et al. The diagnosis of acute acalculous cholecystitis: A comparison of sonography, scintigraphy, and CT. AJR Am J Roentgenol 1986;147:1171-1175.

(19.) Gutman H, Kott I, Haddad M, Reiss R. Changing trends in surgery for benign gallbladder disease. Am J Gastroenterol 1988;83:545-548.

RELATED ARTICLE: Key Points

* Acute acalculous cholecystitis, an inflammation of the gallbladder in the absence of gallstones, usually occurs in critically ill patients.

* Acute acalculous cholecystitis is rare in the pediatric age group.

* In children, acute acalculous cholecystitis most often is a result of antecedent bacteremia and seeding of the gallbladder.

* Although the majority of cases previously were managed with surgical intervention, medical management alone can be curative, as it was in our patient.

* A high index of suspicion is helpful in making an early diagnosis and initiating proper management.

Case Report

A previously healthy 12-year-old white boy was admitted to our institution with a 4-day history of back pain. Two days before presentation, jaundice, right-sided abdominal pain, fever with chills, and a diffuse, nonpruritic, maculopapular rash had developed. A physical examination disclosed that he was in marked discomfort because of back and abdominal pain. His body temperature was 37.9[degrees]C (100.2[degrees]F), his respiratory rate was 16 breaths/min, and his heart rate was 94 beats/min. The patient had icteric sclerae and a diffuse erythematous maculopapular rash that spared the palms and soles. He also had periumbilical and right upper quadrant tenderness, mild hepatomegaly, tendemess in the left sacroiliac area, and limitation of movement of the left hip joint. Laboratory studies showed a white blood cell count of 7,400/[mm.sup.3] (1% myelocytes, 2% metamyelocytes, 20% band forms, 60% neutrophils, 6% lymphocytes, 4% monocytes, and 7% eosinophils) and a platelet count of 162,000/[mm.sup.3]. The erythro cyte sedimentation rate was 50 mm/h. The total serum bilirubin value was 10.5 mg/dl with a direct fraction of 6.8 mg/dl. The results of liver function tests, including aspartate aminotransaminase and alanine aminotransaminase, lactate dehydrogenase, creatme kinase, alkaline phosphatase, prothrombin time, and partial thromboplastin time, were normal. Blood urea nitrogen, creatinine, amylase, and lipase values also were normal. Urine and serum drug screens were negative.

Blood was drawn for culture, and treatment was started with intravenous ampicillin/sulbactam, cefepime, and metronidazole to provide antimicrobial coverage for possible causative organisms of acute cholecystitis. X-rays of the abdomen and the left lower extremity showed no abnormalities. Ultrasonography of the abdomen revealed a small amount of ascites and gallbladder sludge. Computed tomography (CT) of the abdomen revealed mild splenomegaly. Serologic tests for hepatitis B, hepatitis C, Epstein-Barr virus, human immuno-deficiency virus, Leptospira species, Mycoplasma species, parvovirus B19, Rickettsia rickettsii, Bartonella henselae, and cytomegalovirus were negative. Hepatobiliary scintigraphy revealing nonvisualization of the gallbladder 4 hours after injection was suggestive of acute acalculous cholecystitis. The blood culture obtained at admission grew Staphylococcus aureus (S. aureus) susceptible to oxacillin, ampicillian/sulbactam, ceftriaxone, cephalothin, clindamycin, rifampin, and vancomycin. Withi n 2 days of antibiotic therapy, the patient's abdominal pain improved and his jaundice subsided, but his left-sided lower-back and hip pain persisted. Magnetic resonance imaging of the pelvis showed changes consistent with left sacroiliitis, osteomyelitis of the left iliac wing, and an extraperitoneal inflammatory mass near the sacrosciatic foramen and along the pelvic sidewall. A percutaneous CT-guided biopsy of the heterogeneous mass was performed. Tissue Gram stains and cultures were negative. The patient completed a 3-week course of treatment with intravenous ampicillin/ sulbactam, ceftriaxone, and oral metronidazole. This treatment was followed by 3 weeks of oral cephalexin to complete the treatment of osteomyelitis. He had a full recovery. At the end of therapy, magnetic resonance imaging of the pelvis showed resolution of the extraperitoneal inflammatory mass, and the patient's erythrocyte sedimentation rate was 17 mm/h.

From the Department of Pediatrics, Wayne State University School of Medicine, and the Division of Infectious Diseases, Children's Hospital of Michigan, Detroit, MI.

Reprint requests to Basim I. Asmar, MD, Division of Infectious Diseases, Children's Hospital of Michigan, 3901 Beaubien Blvd., Detroit, MI 48201. Email: [e-mail]

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