Cryptogenic Hepatic Abscess in Two Uncompromised Children

By Elizabeth Harrington and Michael A. Bleicher

New York and Elmhurst, New York

O A pre-mortem diagnosis of cryptogenic liver abscess in children is rare, but this diagnosis must be considered in the evaluation for pyrexia of unknown origin, Two previously healthy children were suspected of harboring liver abscesses. Radio- isotopic, sonographic, and angiographic evidence supported the clinical diagnosis. Operative drainage was performed in each case. No source for either abscess was found and no cause established. Anaer- obic bacteria, microaerophilic streptococcus and Fusobacterium necrophorum, each in pure culture, were retrieved on culture of the pus from each child's abscess. Experience gleaned from these two cases emphasizes the possibility of an hepatic abscess existing in the uncompromised child with fever of unknown origin. A cryptogenic hepatic abscess may occur in a child with only mild gastroin- testinal complaints and in a child with sickle cell disease. Recovery is attributed to suspicion of diag- nosis, prompt investigation, operative drainage, effective culture technique with isolation of organ- ism and appropriate antibiotic coverage.

INDEX WORDS: Hepatic abscess.

A N otherwise healthy child who presents with fever of unknown origin poses a diagnostic

challenge. Whether or not abdominal complaints accompany the fever, a diagnosis of hepatic abscess should be considered. Recently, on our pediatric surgical service, two cases of non- amebic pyogenic hepatic abscess in uncomprom- ised children emphasized the degree of suspicion necessary for evaluation, confirmation, and treatment of cryptogenic liver abscess.

CASE REPORTS

Case 1

A 17-yr-old, white male complained of fever and chills, of 1 mo duration, associated with epigastric and right shoulder pain on deep inspiration. He had been passing 6-8 loose

From Division o f Pediatric Surgery, Department o f Surgery New York and Elmhurst, New York The Mount Sinai Medical Center, The Mount Sinai Hospital, N.Y.C., N.Y., and City Hospital Center at Elmhurst, N.Y.

Address reprint requests to Michael A. Bleicher, M.D., Chief, Division o f Pediatric Surgery, Department o f Surgery, The Mount Sinai Hospital, lOOth Street and Fifth Avenue, New York, N.Y. 10029.

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stools per day unaccompanied by blood or mucus. Over the preceding 3 mo he had lost 20 lb.

Pertinent physical findings included fever of 101~ and a tender, enlarged liver, spanning 12 cm, palpable 4 cm below the right costal margin.

Significant laboratory data included: erythrocyte sedi- mentation rate: 134 mm/hr hematocrit: 34%, total WBC count: 18,800/cu mm with 68% segmented polymorphonu- clear cells, 9% bands, 21% lymphocytes and 2% monocytes. Serum chemistries were normal, as were liver function tests. Stool cultures and preparations for ova and parasites were negative on several occasions, hemagglutinin titres for amoe- bae were negative, blood cultures did not grow any organ- isms.

Radiologic studies of the chest and abdomen, intravenous pyelography, and contrast study of the colon were entirely normal. Sonography demonstrated a very large right hepatic defect, well-delineated from normal liver, anteriorly and inferiorly. Findings were consistent wth a diagnosis of hepatic abscess. Radionuclide scan of the liver with 99roTE sulfur colloid demonstrated left and right lower lobe enlarge- ment with protrusion towards the right costal margin. The parenchymal pattern was quite abnormal with a large defect in the right lobe visualized best on posterior projection. The findings were consistent wth right hepatic abscess. Selective hepatic angiography demonstrated splayed vasculature about a large right hepatic mass, also consistent with the presence of a right hepatic abscess.

Despite the extensive investigations performed, no etiology for the hepatic abscess was ever delineated. However, armed with supportive evidence of the clinically suspected hepatic abscess, laparotomy with drainage of the right lobe liver abscess was undertaken. Microaerophilic streptococci were cultured from the abscess and the patient was treated with intravenous aqueous penicillin for 21 days thereafter. Although a right pleural effusion developed early in the post- operative course, this was successfully drained by closed tube thoracostomy. The patient fully recovered and was discharged from the hospital. He is well 9 mo later.

Case 2

A 5-yr-old, black female with sickle cell disease was hospitalized with fever spikes of 2 wk duration with no additional complaints. Months before, the child had enjoyed good health after treatment with pencillin for an upper respiratory infection, months before.

Pertinent physical findings included: 102~ fever, scleral icterus, enlarged liver spanning 10 cm, and palpable 3 cm below the right costal margin.

Significant laboratory data included: hemoglobin: 6.2 g/100 ml, total WBC count: 14,800/cu mm with left shift. Sickle cell preparation was positive and hemoglobin electro- phoresis revealed an S-S pattern. Chemistries were normal, save for total serum bilirubin of 4.0 mg/100 ml with a direct fraction of 2.2 mg/100 ml. Alkaline phosphatase was normal

660 Journal of Pediatric Surgery, Vol. 15, No. 5 (October), 1980

HEPATIC ABSCESS 661

for age; SGOT and SGPT were mildly elevated. Negative serial cultures of all orifices were obtained.

Chest x-ray and abdominal films were normal. Radionu- clide scan of the liver revealed a filling defect in the postero- superior aspect of liver; the remainder of liver was uniformly normal and the spleen small. Selective hepatic arteriogram demonstrated an avascular filling defect in the posterosupe- rior aspect of liver with splaying of vessels.

At operation, the right lobe hepatic abscess was success- fully drained through a thoracoabdominal approach. Upon exploration of the peritoneal cavity, no source for the abscess was found. Abscess culture was reported as Fusobacterium necrophorum.

Preoperative intravenous penicillin and chloramphenicol therapy diminished the fever spikes. Drainage of the abscess was accompanied by defervescence. Penicillin and chloram- phenicol were continued for 3 wk after surgery and the patient's temperature remained normal. Drug fever super- vened, but upon cessation of antibiotic therapy, the patient's temperature returned to normal.

She was discharged from hospital four weeks after surgery in good general health with a well-healed wound. Follow-up revealed an afebrile, healthy child, engaging in normal activity.

DISCUSSION

N o n - a m e b i c pyogenic hepat ic abscess in chil- dren is an uncommon problem. When present it genera l ly occurs in a compromised host, a child with mal ignancy , 1 sept icemia , 1 or chronic granu- lomatous disease. 2 Moreover , the diagnosis often is not discovered until autopsy. 3

Four possible mechanisms or routes of access to the liver by pyogenic organisms exist. 4 They include: t r auma , the hepa t ic ar tery , the por ta l vein or umbi l ica l vein, and invasion from contig- uous s t ructures , such as the bile duct. W h e n no route of infection has been indentif ied, the etiol- ogy is considered cryptogenic .

O u r two cases i l lus t ra te the nonspecific na tu re of symptoms and signs. These include fever, le thargy, malaise , vomit ing, weight loss, abdomi- nal pain, abdomina l dis tent ion, d ia r rhea , hepa- tomegaly , splenomegaly , j aundice , and anemia .

Bacter ia commonly found in hepat ic abscess include Escherichia coli, Streptococcus, and Staphylococcus. However , anaerobic bac te r ia p robab ly exist as the p redominan t o rganism more often than apprec ia ted . 5'6

Cul tures from the hepat ic abscesses in our chi ldren yielded Microaerophilic streptococci and Fusobacterium necrophorum. Microaero- phil ic s treptococci a re g r am posit ive obl igate anaerobes , resident among the normal bac te r ia l flora of the upper respi ra tory , gas t rointes t inal , and urogeni ta l t racts . Fusobacterium necropho- rum are anaerobic g r a m negative baci l l i of the fami ly Bacteroidaceae. These organisms nor- mal ly reside in the upper resp i ra tory and occa- sionally, in the gas t ro in tes t ina l t ract .

In nei ther pat ient was a route for infection identif ied and the et iology of the hepa t ic abscess therefore remains cryptogenic. However , we may postu la te tha t in te rcur ren t gas t ro in tes t ina l infection with inflamed bowel mucosa in the first child permi t ted access of bac ter ia to the portal system.

The second child with sickle cell d isease may have had an increased suscept ibi l i ty to infection. Common to chi ldren with sickle cell d isease are mul t ip le small thromboses , progressing to organ infarct ions, caused by an increased viscosity of hemoglobin S. 7 Should cel lular in tegr i ty be d is turbed by this mechanism, infect ion with abscess format ion may supervene upon any infarct ion. 8 Another possibi l i ty for infect ion in the pa t ien t with sickle cell disease is the func- t ional ly asplenic status, which promotes fai lure in c lea rance of bac te r ia from the c i rcula t ion or causes specific an t ibody deficiency.

SUMMARY

Cryptogenic hepat ic abscess in chi ldren is rare. In the two chi ldren presented, the diagnosis was considered dur ing evaluat ion for fever of unknown origin. W o r k - u p included rout ine blood chemistr ies , liver function tests, rout ine radio- logic studies, sonography, rad ionucl ide scan of liver, and angiography.

Diagnosis was confirmed at l apa ro tomy and rare anaerobic bac te r ia were cul tured f rom each case. Each child recovered af ter surgery and af ter a course of ant ib iot ics admin i s t e red for 3 wk.

REFERENCES

1. Kaplan SL, Feigin RD: Pyogenic liver abscess in disease: Correlation betwen pathogenesis and clinical find- normal children with fever of unknown origin. Pediatrics ings. Pediatrics 48:730-739, 1971 58:614-616, 1976 3. Dehner LP, Kissane JM: Pyogenic hepatic abscess in

2. Johnston RB, Baehner RL: Chronic granulomatous infancy and childhood. J Pediatr 74:763-773, 1969

662 HARRINGTON AND BLEICHER

4. Stokes JF: Cryptogenic liver abscess. Lancet 1:355- 358, 1960

5. Gorbach SL, Bartlett JG: Anaerobic infections. N Engl J Med 290:1177-1184, 1974

6. Patterson DK, Ozeran RS, Glantz GJ et al: Pyogenic liver abscess due to microaerophilic streptococci. Ann Surg 165:362-376, 1967

7. Shulman ST Beem MO: A unique presentation of sickle cell disease: Pyogenic hepatic abscess. Pediatrics 47:1019-1022, 1971

8. Brittain HP, de la Torre A, Willey EN: A case of sickle cell disease with an abscess arising in an infarct of the liver. Ann Intern Med 65:560-563, 1966