perianal abscess and fistula in ano in infancy and childhood: a clinicopathological study

PERIANAL ABSCESS AND FISTULA IN A N 0 IN INFANCY AND CHILDHOOD: A Clinicopathological Study

Ahmed H. Al-Salem, MBBS, FRCSI, FICS, FACS, and Syed Qaisaruddin, M S Qatif, Saudi Arabia

Shahzad S. Qureshi, MBBS, M Med Sci, MRC Path Department of Pathology, Qatif Central Hospital, Qatif, Saudi Arabia

Division of Pediatric Surgery, Department of Surgery, Qatif Central Hospital,

This is a retrospective study of 78 children with perianal abscess and/orfistula in ano presenting during a 6% -year pa'od. Sixty-five were males and I3 females. Their ages at presentation ranged from 22 days to I8 years (median I. 7 year), and the majority of males 7uere below 2 years of age. The 13 females all presented with perianal abscess, the majmity of which grew Staphylococcus aureus (69.2 %). On followup, none of them deuelopedJisttila in ano. Twenty-two of the 65 males (33.8%) jn-esented initially with fistula in ano. The remaining 43 presented with pa 'anal abscess. Four OJ them were found to havefistula in ano at the time of incision and drainage and on followup, and 14 others deuelopedfistula i n ano. Of the 40 cases offistula in ano, all were males; 25 were on the right side and 9 on the left side, 5 had bilateralfistula i n ano, and 1 had two fistulas on the left side at 3 and 5 o'clock positions. Gut-derived organisms were isolated from 88.4 % of the males with perianal abscess. There appears to be a causal relationship between perianal abscess andfist t i la in ano.

Keywords fistula in ano, pediatric, perianal abscess

Perianal abscess (PA) and fistula in ano (FIA) are not uncommon in adults, but their incidence in children is low. They have certain features that appear to distinguish them from those in adults [l]. Controversy still surrounds their etiology. Various pathogenetic factors such as congenital abnormalities of the anal glands [2, 31, abnormal anal crypts of Morgagni [ 11, and ectopic inappropriate epithelium [4, 51 have been cited as important in infants and children.

We present the results of a retrospective review of our cases of perianal abscess and FIA and discuss aspects of etiology.

Received 25 July 1995; accepted 27 December 1995. Address correspondence to Dr. Ahmed H. Al-Salem, P.O. Box 18432, Qatif 3191 1, Saudi Arabia.

Pediatric Pathology & Labmatoly Medicine, 16: 755-764, I996 Copyright 0 1996 Tuylm &Francis 1077-1042/96$12.00 + .OO 755

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756 A. H. AL-SALEM ET AL.

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No of 20. Patients

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37 d

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Age in Years Figure 1. Age at presentation o f male children with perianal abscess and/or fistula in ano

PATIENTS AND METHODS

During the 6X-year period from April 1988 to October 1994,80 children with PA and/or FIA were seen at Qatif Central Hospital, Qatif, Saudi Arabia. The records of these patients were analyzed retrospectively in terms of age at presentation, sex, clinical features, duration of symptoms, associated ill- nesses, operative findings, and outcome. The PA had been treated by incision and drainage and swabs were taken for culture. The FIAs were excised, the tracts laid open, and the specimens sent for histological evaluation.

RESULTS

Eighty children with PA and/or FIA were treated at our hospital during the study period. Two were excluded, both females; one of them developed

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PERIANAL ABSCESS AND FISTULA IN A N 0 757

bilateral FIA and the other developed PA following posterior sagittal anorectoplasty for high anorectal agenesis. Out of 78,65 were males and 13 females. Their ages at presentation ranged from 22 days to 18 years (mean 4.5 years and median 1.7 years). The majority of males, 37 (56.9%) were less than 2 years of age (Figure 1).

Twenty-two of 65 males presented initially with FIA; 17 (77%) of the fistulas were right sided, 4 left sided, and one patient had bilateral FIA (Figure 2). At the time of presentation four had purulent discharge from the fistula opening that grew Staphylococcus aurais in one and Escherichia coli in the other three. Eleven (50%) patients had a history of abscess at the site of FIA prior to their presentation and in five this was recurrent.

The remaining 43 males presented initially with PA (Figure 3). Four were found to have FIA at the time of incision and drainage, 2 on the right and 2 on the left. The bacteriology of the abscess is shown in Table 1. None of them presented with bilateral PA, but on follow-up 14 others developed FIA. Of the 18 who developed FIA, 8 cases were right sided and 5 left sided, 1 had two fistulas on the left side at 3 and 5 o’clock positions, and 4 developed bilateral FIA. We treated a total of 40 children with FIA. All were males. Twenty-five had FIA on the right side and nine on the left; five had bilateral FIA, and one had two fistulas on the left side at 3 and 5 o’clock positions. The remaining 25 patients had PA only.

The 13 females ranged in age from 11 months to 6 years (mean 3.1 years, median 4 years). Nine (69.2%) had right-sided abscess, three had left-sided abscess, and one had bilateral abscesses. The bacteriology of the abscesses is compared with the males in Table 1. The majority grew S. aureus (9 of 13). None of them developed FIA.

None of our patients had predisposing factors such as diabetes melli- tus, Crohn’s disease, leukemia, or neutropenia. All fistulas were found to be low and direct and were treated by excision with laying open of the tract except in four patients in whom the fistulas were discovered intraopera- tively during incision and drainage. These were treated with fistulotomy only. On follow-up ranging from 6 months to 5 years (mean 2.6 years) none of our patients developed recurrence. Two of the patients with FIA were referred to us with recurrent FIA treated at another hospital.

Histological evaluation was available for 27 patients. This revealed chronic nonspecific inflammation in six. Nine of the tracts were lined by granulation tissue. In the remaining 12 patients, the tract was lined by granulation tissue and squamous epithelium in 4; granulation tissue and transitional epithelium in 3; and a mixture of granulation tissue, squamous epithelium, and transitional and glandular epithelium in 5 (Figures 4, 5, and 6).

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Figure 2. Bilateral fistula in a n o in a child

DISCUSSION

Perianal abscess and fistula in ano have been recognized for hundreds of years, but little has been written about them in infants and children and most of the information in the literature is derived from studies of adults. In 1880 Herrmann and Desfosses [6] described small anal glands and suggested that

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Table 1. Causative organisms of perianal abscess ~~~ ~ ~

Males with perianal Females with Males with abscess who perianal abscess perianal abscess subsequently Males with

Causative organisms (13) only (25) developed FIA (18) FIA (4)

E. coli 1 4 7 3 E. coli + S. uureus 1 1 2

1 E. coli t Pseudomonas - 1 E. Cali+ Klebsiellu - 1 E. coli + Proteus - 2

2 1

S. nureus 7 4

- - -

- -

- -

- - E. coli + Streptococcus -

E. coli t propnono- -

S. nureus + group D

fuecalis - - E. coli t anaerobes - - -

bacterium 1 - - - - I

Salmonella - - 2

2 2 I 1 1 1

1

Mixed fecal flora - Coliforrns -

Klebsielln - - Kkbsielkz + Pseudomonas - -

-

- -

- B-hemolytic streptococci - - Hamophilus para- - -

Group C Snlmonelli - - No growth 3 3 3

- inJumntne

- -

- - 1 Proteus mirubilis -

Figure 4. Fistula tract lined by columnar and transitional epithelium. H & E, X100, reproduced at 80%.

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Figure 5. Anal glands surrounded by mixed inflammatory infiltrate and lined by pseudostratified columnar epithelium. H & E, ~ 1 0 0 , reproduced at 80%.

Figure 6. Fistula tract showing areas of transitional epithelium, pseudostratified columnar epithelium, and loss and destruction of epithelial lining. H & E, ~ 4 0 , reproduced at 80%.

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FIAs arise as a result of infection of these anal glands. In 1956 Eisenhammer [7] stated that the PA is the parent of the FIA, which arises from crypto- glandular infection of the anal glands that lie within the intersphincteric space [8,9]. Parks [ 101 suggested that cystic dilations of the anal glands were a necessary precursor to infection, which could be acquired or congenital, and that the individual susceptibility to infection was the result of variation in the number, depth, and shape of these anal glands [ l l ] . Lilius [12], on the other hand, found that anal glands were not present in all individuals. Men have a slightly higher incidence of anal glands than women. Shafik [ 131 suggested that anal intramuscular glands are developmental remnants rather than true glands, with sequestered epithelium from the developing anorectal sinus that perpetuates subsequent infection. Controversy still surrounds the etiology of these conditions, but the general consensus is that FIA is primarily due to infection of the anal glands.

Perianal abscess and FIA in the pediatric age group are infrequent and different from their adult counterpart [ l , 141. Mentzer [15], in a review of 3210 cases of pediatric proctological diseases, showed that PA accounted for 2.5% of the cases. The microbiology of acute PA has been well described in adults. Finegold [ 161 found that more than 75% of microorganisms from PAS in adults were due to gram-negative enteric bacteria and anerobic organisms, and their presence in the pus as opposed to bacteria derived from skin indicates a suspected FIA [17, 181. Seow-Choen et al. [19] found similar organisms in cultures of granulation tissues from FIAs and from acute PAS. Enberget al. [20] found S. aurmsas the most common organism in 29 patients with PA (ages 1 month to 14 years). In their study no sex was noted as 13 of their patients were females, from whom S. aureus is commonly cultured, whereas males commonly grow E. coli and other gastrointestinally derived bacteria. Brook and Martin [21] found anaerobes to be the predominant isolates from children with PA. Our results defined two groups based on their sex. In females the majority (69%) grew S. aurms, whereas in males gut-derived organisms formed 88% of the isolates and E. coli was the most common. Forty-two percent of our male children subsequently developed FIA, which is similar to the findings of Poenaru and Yazbeck [ 141. Others have reported a higher percentage of FIA following PA [22] in children.

The pathogenesis of FIA in children is still controversial. Fitzgerald et al. [3] suggested abnormalities of the anal glands that are present at birth as an important factor in the pathogenesis of FIA in children. This explains the early onset of symptoms and absence of inflammation in many cases as the majority are below 2 years of age [3, 5, 14, 221. Our series confirms the early onset as 70% of our patients were younger than 2 years. I t has been postulated that excessive intrauterine androgenic stimulation leads to the formation of these abnormal glands, explaining the male predominance [3, 231. All our

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patients with FIA were males and none of our female patients with PA developed FIA, similar to previous reports [l, 3, 221. The presence of androgenic hormones has a generalized effect on the anal glands, perhaps explaining the multiple FIAs in some infants and children [3,24]. Six of our patients with FIA had more than one fistula, similar to the findings of Poenaru and Yazbeck [ 141. Others have reported a higher incidence of bilateral and multiple FIAs in infants and children [2, 5, 241.

Shafer et al. [l] suggested abnormal crypts of Morgagni as the cause of PA and FIA in infants and children. They found 3 to 13 abnormal deep crypts that retain bacteria and cause cryptitis leading to PA and fistula formation. Although we found megacrypts contiguous with the fistulas, we tend to agree with Piazza and Radhakrishnan [22] that it is difficult to state that FIAs originate in these crypts. These crypts are also present at other sites and without their incision resulted in no recurrence.

Pople and Ralphs [4] postulated the presence of ectopic columnar and transitional epithelium along the length of the fistula as evidence of congenital origin of FIA presenting in the first few months of life. This was stressed subsequently by Al-Salem et al. [5]. Four of our patients had tracts lined with squamous epithelium; three had transitional epithelium; and five had a mixture of squamous, columnar, transitional, and glandular epithelium. Although these different types of epithelium probably represent inflammation-induced metaplastic changes, the inappropriate types of epithelium may result from entrapment of migrating cells from the urogenital sinus during the development of the perineum [25]. The loss of epithelium from the lining of the fistula in some cases has been attributed to repeated or chronic inflammation and scarring, which is supported by the absence of epithelium in older children with FIA [5].

In summary, FIAs in infants and children have specific features that separate them from their adult counterparts:

1. FIA occurs exclusively in males [ 1, 5, 14 , 221. 2. PA in males less than 2 years of age is associated with FIA in more than

70% of cases, whereas PA in female children does not subsequently lead to FIA [5, 14, 221.

3. The vast majority of FIAs are seen in children less than 2 years of age [l, 3, 5, 14, 221.

4. Bilateral and multiple fistulas are frequent (10-28.6%) [2, 3, 5, 241. 5. The fistulas are usually low, simple, and direct. Complex and high

fistulas are not seen in children [l , 3, 51 except in those with predisposing factors.

6. With proper surgery, fistulotomy or fistulectomy with or without cryptotomy, recurrence is very low [l, 3, 5, 221.

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7. FIA in infancy and childhood is due to congenital abnormalities of the anal glands [2, 31, which may result from excessive intrauterine androgenic stimulation [3, 231. Others have suggested a thickened, markedly irregular dentate line with abnormal crypts capable of trapping bacteria [l], or the presence of ectopic inappropriate epithelium as the result of entrapment of migratory cells from the urogenital sinus during development of the perineum ~ 4 ~ 5 1 .

REFERENCES

1 . Shafer AD, McGlone TI’, Flagan RA. Abnormal crypts o f Morgagni: The cause ofperianal abscess and

2. Dnhaniel J. Anal fistulae in childhood. A n , ] Protocol 1975;26:40-3. 3. Fiugerald &I, Harding B, Ryan W. Fistula-in-ano in childhood: A congenital etio1oby.J Pediatr Surg

1985;20:80-1. 4. Pople I] , Ralphs DNL. An aetioloby for listula in ano. 8 r J Surg 1988;75:9045. 5. AlSalcin AH, LaingW, TalwalkerV. Fistula-in-ano in infancy and chi1dhood.J Pediatr Surg 1994;29:435-8. 6. Herrmann G, Dcsfosses L. Sur la niuqiiense de la region cloacale d u rectum. CR Acad Sci ( I I I )

7. Eisenhammcr S. The internal anal sphincter and anorectal abscess. Surg Gynecol Obstet 1956; 103.50 Id. 8. Lockhart-Mummeiy JP. Discussion on fistula-in-ano. I’roc R Soc Med 1 929;22: 1331-58. 9. Eisenhammer S. A new approach t o the anorcctal fistuloiis abscess based on the high interm;tscular

lesion. Swg Gynecol Obstct 1958; 306:595-9. 10. Parks AG. Pathogenesis and treatment of fistula-in-ano. Br Med J 1961;1:463-9. I I . Hill MR, Sinall CS, Hunt GM, Richards LJ. Development of anal ducts and glands with reference t o

the pathogenesis of anorectal disease. Arch Pathol 1949;47:350-60. 12. Lilius HG. Fistula-in-aim. An investigation of human foetal anal ducts and intramuscular glands and

a clinical study of 150 patients. Acta Chir Scand Suppl 1968;383:1-88. 13. Shafik A. A new concept of the anatomy of the anal sphincter mechanism and the physioloby of

defecation. X. Anorcctal sinus and band: Anatomic nature and surgical significance. Dis Colon Rectum 1980;23: 170-9.

14. Poenani D, Yazheck S. Anal fistula in infants: Etiology, features, management. J Pediatr Surg l993;28: 11945.

15. Menuer CG. Anorectal diseasc. I’ediatr Clin North An l956;3:113. 16. Finegold SM. Anaerobic bacteria in human disease. New York: Academic Press, 1977;386435. 17. Eykyn SJ, Grace RH. The rclevance of microbiology in the management of morcctal sepsis. Ann R

Coll Surg Engl 1986;68:237-9. 18. Grace KH, Harpcr IA, Thompson KG. Anorectal sepsis: Microbiology in relation to fistula-in-ano. Br

J Surg 1982;69:401-3. 19. Seow-Choen F, Hay .A], Heard S, Phillips RKS. Bacteriology of anal fistulae. BrJ Surg 1992;79:27-8. 20. Enberg RN, Cox RH, Burry VF. Perirectal abscess in children. Am J Dis Child 1974;128:360-1. 21. Brook I , Martin WJ. Aerobic and anaerobic bacteriology ofperirectal abscess in children. Pediatrics

1980;66:282-4. 22. Piazza DJ, Kadhakrishnan J. Perianal abscess and fistula-in-ano in children. Dis Colon Rectum

1990;33:101416. 23. Takatsuki S. An etiology of anal fistula in infants. Keio] Med 1976;25: 1 4 . 24. Grant CS, Al-Salem AH, Anim JT, Khwaja MS. Childhood fistula-in-ano: A clinicc~pathological study.

25. Vaniler Putte SCJ. Normal and abnormal development of the anorectum.,] Pedbatr Surg 1986;2 1 :434-40.

listula-in-ano. J I’cdiati- Snrg 1987;22:203-4.

1880;90: 1301-2.

Pediatr Surg Int 1991;6:207-9.

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perianal abscess and fistula in ano in infancy and childhood: a clinicopathological study

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