Abstract: In addition to the familiar acuteinflammation of the appendix, chronicinflammation has long been a controversialdisease entity. However, some recent reportsannounced that chronic recurrent appendicitisis by no means an established disease of theappendix. Therefore, we reviewed ourappendectomies in children to determine theunderestimated cases of chronic recurrentappendicitis and discuss this disease entity inchildren.

The records of children who had undergoneappendectomy upon initial clinical diagnosis ofappendicitis were reviewed for a period of 42months. Incidental appendectomies wereexcluded. The diagnoses of acute appendicitis,negative explorations and chronic recurrentappendicitis were made on clinical andhistopathological grounds.

Of 72 children who underwent appendectomy,three cases showed chronic inflammation of theappendix, five cases showed normal appendixand the remaining were of acute inflammationhistopathologically. Three girls of eight, 10 and12 years old also had clinical presentation ofchronic recurrent appendicitis with previoussimilar attacks of right lower abdominal painand vomiting and showed relief of thesesymptoms in the postoperative follow-up.

For children with previous similar attacks ofright lower abdominal pain and vomitingchronic recurrent appendicitis should beconsidered in differential diagnosis after athorough clinical examination.

Key Words: Appendicitis, ChronicRecurrent; Abdomen, Acute; Children

Introduction

Although acute appendicitis is the most commondisease of the appendix, chronic recurrent appendicitis isa previously condemned but existing disease of theappendix (1-9). Many reviews and case reports of chronicrecurrent appendicitis in children and adults have alsofeatured in non-English literature (10-12). Because ofthe general belief and attitude that seeks to avoidunnecessary operations shonly abdominal pain exist,especially when the signs and symptoms are atypical,many children with chronic recurrent appendicitis suffer asimilar attack of pain, vomiting and other symptoms.

The present study aims to present our experiencewith chronic recurrent appendicitis in children and discussthis entity in the light of the literature.

Materials and Methods

The clinical and histopathological records of childrenwho underwent appendectomy following suspicion ofappendicitis at the Department of Pediatric Surgery

between 15 June 1998 and 15 December 2001 werereviewed. Incidental appendectomies were not taken intoaccount. Cases of acute and chronic inflammationtogether with negative explorations were recorded.Diagnosis of chronic recurrent appendicitis was made onthe basis of a history of similar, recurrent attacks of rightlower abdominal quadrant pain leading to appendectomyand histopathological diagnosis of chronic inflammationof the appendix and relief of signs and symptoms afterappendectomy. Chronic inflammation was diagnosedwhen lymphocytes and eosinophils were present withinthe appendiceal wall with associated fibrosis in thehistopathological examination with hematoxylin and eosin(1,2,13-15).

Results

A total of 72 children underwent appendectomy from15 June 1998 to 15 December 2001. There were 43boys and 29 girls. The male to female ratio was 1,5. Themedian and mean standard deviation of the ages of allpatients were 7 and 7,2 3,8 years respectively.

Turk J Med Sci32 (2002) 351-354' TBTAK

351

Received: March 06, 2002

Departments of 1Pediatric Surgery,

2Pathology Faculty of Medicine, Fatih

University, Ankara - Turkey

Fatih ANDIRAN1

Sabriye DAYI1

Muzaffer AYDERE2

Hseyin STN2

Chronic Recurrent Appendicitis in Children:An Insidious and Neglected Cause of SurgicalAbdomen

Histopathologically, five cases of normal appendixes andthree cases of chronic appendiceal inflammation asdescribed above were found, while the remaining 64cases were of acute inflammation. Table 1 shows thedescriptive analysis and the diagnoses of the patients andTable 2 summarizes the properties of three cases ofchronic recurrent appendicitis. The histopathologicalexaminations of 12-, 8- and 10- year-old girlsconfirmatory for chronic recurrent appendicitis are alsoshown in Figures 1-3 respectively.

Discussion

The existence of chronic recurrent appendicitis haslong been controversial and has recently beenreconsidered and re-addressed (1-8). While acuteappendicitis presents with a more typical sequence ofsigns and symptoms, the presentation of chronicrecurrent appendicitis is more subtle. After earlyattempts to describe chronic and recurrent appendicitis inthe last decades of the 19th century, the term of chronicrecurrent appendicitis was then condemned for sometime (1,2,16,17). However, recent reports from largeseries of patients have re-established the term andconcept of chronic recurrent appendicitis (1-4,7).

The diagnostic criteria for chronic recurrentappendicitis are a history of similar, recurrent attacks ofright lower quadrant pain leading to appendectomy, ahistopathological diagnosis of chronic inflammation of theappendix and relief of symptoms after appendectomy(1,2,13-15). If acute inflammation is presented withsimilar criteria, it is termed recurrent appendicitis only.Chronic recurrent appendicitis comprises about 1,5 to10% of all appendix inflammations (1-4). However, theprecise disease mechanism of chronic recurrent

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Table 1. The descriptive analysis and histopathological results ofpatients who underwent appendectomy.

BOYS GIRLS TOTAL

No. of Cases 43 29 72Age range 2-12 4-15 2-15Mean age SD 7.03.5 8.83.6 7.23.8Median age 7 8 7Chronic infl. - 3 3Normal 1 4 5Acute infl. 42 22 64

(Ages were given in years; No, = number, SD = standard deviation; infl.= inflammation)

Table 2. The properties of three cases of chronic recurrent appendicitis.

PATIENT 1 PATIENT 2 PATIENT 3

Age (years) 12 8 10Sex girl girl girlClinical presentation and RLQ pain, bilious RLQ pain RLQ pain, biliousduration of symptom vomiting for 3 days for 2 days vomiting for one weekPrevious similar attack(s) twice within last year once six months prior once three months priorPhysical examination RLQ tenderness RLQ tenderness RLQ tendernessFever absent absent absentWhite blood cell count/mL 7900 8800 11,200Hemoglobin value (g/dL) 15 12.8 13.4Urinalysis normal normal normalFibrinogen (mg/dL) 227 - 258C-reactive protein (mg/L) < 3.6 < 3.6 < 3.6Plain abdominal X-ray nonspecific nonspecific local ileus in RLQAbdominopelvic USG and color right adnexial cyst of normal normalDoppler USG for ovaries Morgagni of 3 cm diamLaporotomy findings no free fluid, ovaries normal, no free fluid, ovaries normal, no free fluid, ovaries normal,

Morgagni cyst excised, appendix thickened with appendix tense and thickened withappendix grayish and thickened local adhesion increased vascularity in the wall

Histopathological examination Lypmphocyte Lypmphocyte Lypmphocyteof the appendix infiltration and fibrosis infiltration and fibrosis infiltration and fibrosisPostoperative complaints complete relief complete relief complete relief

(RLQ = right lower quadrant, USG = ultrasonography, diam = diameter)

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appendicitis is obscure. Besides the various inflammatoryreactions, changes in neuroendocrine cells and formationof traumatic neuromas were held responsible for thedisease (18,19).

In children, a recent clinicopathological study showedchronic active inflammation with increased numbers ofimmunocompetent cells, mostly T lymphocytes,subsequent scarring and an increase in the number ofneural cells (1). Appendixes in chronic appendicitis casesharbor a chronic inflammatory reaction of an unknownetiology mediated by T lymphocytes and increasedlymphoid tissue with concomitant hyperplasia of germinalcenters indicating a simultaneous stimulation of B cellmediated immune response (1). Also, appendices frompatients with prolonged clinical symptoms defined aschronic appendicitis were shown to have vascular celladhesion molecule-1 (VCAM-1) expression (20).Panneuronal marker protein gene product 9.5 (PGP9.5)was found to be increased in chronic appendicitis as aneuronal factor in the pathophysiology of the disease andpain symptoms (21).

When reviewing our patients who had undergoneappendectomy, the rates of chronic recurrent appendicitisand negative explorations were 4.1% and 6.9%respectively, which are comparable to those in theliterature (1-4). However, we have not encounteredrecurrent acute appendicitis clinically, as we believe thatsimilar attacks of symptoms and signs in acuteappendicitis cases may have been neglected or omitted inthe past history of the records. In the differentialdiagnosis of appendicitis, acute or chronic, barium enemaexamination and computerized tomography might havebeen helpful in our cases (2,7,9). On the other hand, bothtechniques have their own handicaps (7,8).

In this study, all patients with chronic recurrentappendicitis were female, although in the literature thereis no such sex predilection. While the reason for thisfemale sex predilection in our series may depend on thesize of the patient population, it may also result from thedevelopmental nature of adolescent and pre-adolescentgirls, which could be investigated in prospective studies.Meanwhile, attention should be paid to pathologiesrelated to adnexa when the underlying etiology ofabdominal pain is explored because of the nature of theproblems of this sex, especially at preadolescent andadolescent ages. However, this must not mislead or maskthe differential diagnosis of chronic recurrent diagnosis.

Figure 1. Prominent fibrosis and fatty infiltration in the wall of theappendix of Case 1 (hematoxylin-eosin, x 50).

Figure 2. Photomicrograph of the appendix of Case 2 showing thedispersed submucosal lymph follicles, fibrosis in themuscular layer and fatty infiltration (hematoxylin-eosin, x50).

Figure 3. Eroded mucosal surface, dispersed submucosal lymphfollicles, fibrosis in the muscular layer and fatty infiltrationof the appendiceal wall of Case 3 (hematoxylin-eosin, x50).

We should also emphasize that the reluctance tooperate on chronic recurrent appendicitis because of theviews that disfavor this term may needlessly delay thesurgical treatment and prevent relief from a surgicallycurable problem in children with chronic recurrentappendicitis. Therefore, upon suspicion of chronicrecurrent appendicitis in a patient with right lowerquadrant pain and tenderness with previous similarattacks, the treatment of choice must be surgicalexploration and appendectomy.

In conclusion, chronic recurrent appendicitis should beconsidered in differential diagnosis in the evaluation of achild with abdominal pain. A history of prior similarepisodes of pain, especially in the right lower quadrant

with a tenderness on palpation where other diseases areexcluded, should favor the possibility of chronic recurrentappendicitis and prompt surgical exploration.Furthermore, special care must be given in cases of thefemale sex in the differential diagnosis of acute abdominalpain.

Corresponding author:Fatih ANDIRANFatih University, Faculty of Medicine,Department of Pediatric Surgery,Ciftlik Cad. No: 57, Emek 06510 Ankara / TURKEYe-mail: fandiran@ttnet.net.tr

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