Periodontics

Generalized aggressive periodontitis in a prepubertaipatient: A case report

Ay?en Bodur, DDS, PhDVHaluk Bodur, DDS, PhD /̂Belgin Bal, DDS, PhDVKöksalBalo|,DDS, PhD'

A 10-year-old boy presented wilh generalized gingival inflammation, extensive aiveoiar bone loss, andtooth mobility. Ciinjcal and radiographie examination suppiemenled by microbioiogic investigation led to adiagnosis ol classically termed prepubertai periodontitis (now i<nown as generalized aggressive periodan-titis). Other than severe periodontitis, the child was systemicaily healthy. Neither unusual infections norabnormalities in neutrophil functions were detected. Microbiologie examinations by culture revealed thepresence of the periodontal pathogen Actlncbacillus acfmomyceiemcomiians. Treatment consisted ofextraction of mobile teeth, supragingivai and subgingival debridement, subgingival curettage, and rootplaning combined with a 1 -week prescription of a combination of metronidazoie and amoxiciliin. Scanningelectron microscopy of extracted teeth revealed iiypoplastic and aplastic cementum at the periodontallyexposed and intact surfaces. Ciinicai and microbiologie toilow-up was continued over a 1 -year period. Noperiodontal lesions have been detected, and A actinomycetemcomitans could not be isoiated from thesubgingivai areas of the remaining teeth at the end of the first year Since A actinomycetemcomitans wasthe main pathogen present in the subgingivai microflora of the patient, it might play a key role in the etiol-ogy of prepubertai periodontitis. (Ouintessence Int 2001-,32:303-308)

Key words: Actinobaciiius actinomycQtemcomitans, generalized aggressive periodontitis, periodontal therapy

Periodontal diseases that cause rapid and severeperiodontal destructions in children prior to

puberty arc rather rare.'"^ Most of the reported casesin dental literature have been associated with diseasessuch as Papillon-Lefèvre syndrome (PLS),'̂ '' hypophos-phatasia,*' neutropenia,*-^ leukemia,'"'" histíocytosisX,"̂ " and leukocyte adhesion deficiency (LAD).'"''̂

In 1983, periodontitis in young children wasdescribed as a distinct clinical entity and termed pre-pubertai periodontitis by Page and coworkers/^recently reclassified as a form of generalized aggres-sive periodontitis," In this article, the classic terminol-ogy will be used. Although this entity is rare and iittleis known about its etiology and pathogenesis, it is ahighly destructive form of early-onset periodontitis.

Prepubertai periodontitis generally occurs in local-ized or generalized forms, and both the primary andpermanent dentition may be involved.' Generalizedprepubertai periodontitis (GPP) is characterized byacute inflammation, proliferation, and cleft formation

of the gingiva around all teeth with rapid alveolar bonedestrucfion, and seems to be resistant to conventionalperiodontal treatment. Also, the generalized form maybe associated with recurrent and sometimes life-threat-ening infections.'^ The localized form of prepubertaiperiodontifis involves only a few teeth and is charac-terized by minor inflammafion and slower bone loss.

However, some of the patients described in the lit-erature are otherwise healthy prepubertai children.''^Pathogenic bacteria-especially Actinobacillus actino-mycetemcomitans-have been considered to be possi-ble etiologic factors. Cementum defects and/or func-tional defects in host defense systems have beensuggested to be predisposing factors in the develop-ment of periodontitis in children,^-"'•'°

The aim of this report is to present a 1-year follow-up of a systemicaily healthy prepubertai periodontitispatient with clinical, radiologie, and laboratoryfindings and the patient's response to periodontaltreatment.

'Deparlment oí Perlodontology, School of Dentistry, Univetsity of Gazi,Ankara, Turkey.

^Department of Pedodoniics, School o( Dentistry, University of GazJ,Ankara, Turkey.

Reprint requests: Dr Ayçen Bodur, Gazi Üriveisitesi DifhekimligiFahiiltesi, Periodontoloji Anabilim Dalr, Emek Mali. Biçkek Cad. 82. Sok..06510 Ankara, Turkey. E-mail; aysenbodur@hotmaiLcom

CASE REPORT

A 10-year-old hoy who had extensive periodontal honeloss and tooth mobility was referred to our periodon-tology clinic by his dentist. He had not received anyperiodontal therapy before. According to information

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Fig 1a Ciinicai appearance of the patient's maxiilary permanentteeth before periodontai treatment.

Fig 1b Ciinicai appearance ol the palient's mandibuiar perma-nent teeth before periodontal treatment.

provided by his family, the patient's primary teeth haderupted normally, buf they had migrated and started toexfoliate at the age of 4. Although his permanent teethhad hegun fo erupt normally, gingival inflammation,mobility of the teeth, and halitosis were noted in ashort time.

The patient's past medical history was not remark-able, and he weighed 26 kg and was 144 cm in height.Consanguinity existed between his parents, who werefourth cousins; however, his 1 brother, 3 sisters, andparents had no known periodonfal or systemic disease.

The ciinical oral examination revealed a permanentdentition with 4 maxillary incisors, 4 mandibularincisors, 1 parfially erupted maxillary left secondmolar, and 1 mandibular right canine. Tlie permanentfirst molars had been lost prior fo fhis examination,inflammation and proliferation of fhe gingiva wifh atendency fo bleeding were noted, especially aroundthe maxiilary and mandibular incisors. There were 6to 10-mm probing depths around fhe maxillary andmandibuiar central incisors, and fhe incisors wereloose and extremely mobile. There was no calculuspresent, but high amounts of bacterial plaque wereseen (Figs la and lb). The submandibular lymphnodes were normal af fhe time of fhe examination.

A radiographie examinafion consisfing of pano-ramic and periapicai films revealed fhaf severe boneloss had occurred around fhe maxillary and mandibu-lar leff cenfral incisors, the maxillary righf cenfralincisor, and the maxillary and mandibular laterals.Remaining permanenf feefh were in fhe process oferupfion, and fhe only unaffected partially eruptedtooth appears to be the mandibular righf canine.There was some degree of bone loss around the maxil-lary second molars {Figs 2 and 3).

Because of the early exfoliation of the primary teethand fhe pronounced bone loss of the permanent denfi-fion, the patient was referred for a complete medicalevaluation fo rule ouf any underlying systemic disease.

The results of fhe medical evaluation, including thelaboratory values, were within normal limits for fhepatient's age. A chemotaxis assay was performedaccording to the method described by Boyden. '̂ Achemotacfic index was calculated; oriented migrafion{toward chemotactic factor) divided by spontaneousmigration {toward control medium). Flow cytometrywas used to perform CD18 analysis, and the result ispresented as a percentage of the positive cells. Theneuf rophil chemotaxis was found to be normal, and fheCD18 value was within normal limits. The lahoratoryfindings are depicted in Tahle 1. The skull and skeletalradiographie surveys revealed no abnormalities.

For further evaluation, subgingival plaque was sam-pled from fhe deepest pockets using paper points.Microbiologie cuituring of the plaque was performedon selective media for A actinomycetemcomitans fol-lowed by a catalase-acfivity fesf.̂ ^ The culturesrevealed the presence of A actinomycetemcomitans onaii the sites sampled.

According to the clinical and radiologie featuresand the hacterial profile, the diagnosis of prepubertalperiodonfifis was made.

The treatment plan consisted of extractions of themaxillary left central incisor, mandibular left and righfcentral incisors, oral hygiene instructions, supragingi-val and subgingival debridement, subgingival curet-fage, and root planing.

The exfracfed feeth were examined by a scanningelectron microscope {SEM, JOEL-JSM 6400). Theroots of the teeth exhibited hypoplastic and aplasfic

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Fig 2a (left) Radiograph of the patient'smaxillary left central and lateral incisors.

Fig 2b (right) Radiograph of ihe patient'smandibular right central and lateral incisors

Fig 3 Panoramic radiograpti.

cementum at the periodontally exposed and intact sur-faces (Fig 4).

The patient was instructed in how to improve oralhygiene and prescribed a 0.2% chlorhexidine mouth-ritise to use twice a day, followed by systemic adminis-tration of an antibiotic regimen (250 mg metronida-zole plus 375 mg amoxicillin) 3 times a day,̂ ^ inconjunction with supragingival and subgingivaldebridement for a period of 7 days. Further, Fie wastreated on 4 occasions by curettage under local anes-ihesia. Figure 5 demonstrates healing 2 weeks aftercompletion of the initial phase of periodontal treat-ment. Normal overbite and overjet relationships wereobtained by orthodontic therapy tbat was performedby a removable appliance 4 weeks after the comple-tion of the initial phase of periodontal treatment.Figure 6 demonstrates the clinical condition of thepatient 1 month after the completion of orthodontic

TABLE 1

HGBWBCPLTHCTRBCMCV

Laboratory results

Patient

12.1 g/dL6500/mm^358 X 10̂ /mmä35.3 (%]4.44 (JL79.4 m̂ = fL

Blood glucose 93 % mglevel

ALPIgGIgMIgAC3C4CD18

237 U/L1505 mg/dL74 mg/dL273 mg/dL119 mg/dL26 mg/dL85.6 (%)

Chemotactic index 94.8/21.2 = 4.47

1Normal range

11.5-15.5 g/dL4500-13,500/mm3150^00 X 1O'/mm335-45 (%)4-5 2 |JL

77-95 m^ = fL70-110mg(%)

130-560 U/L842-1943 mg/dL54-392 mg/dL62-390 mg/dL50-120 mg/dL20-50 mg/dL> 85 {%)104/20.6 = 5.07 (control)

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Fig 4 The root surface of the extracted niaxliiary left centralincisor (scanning eiectron micrograph; otiginai magnificationX12)

Fig 5 Clinicai appearance afte- completion of the initial phaseof *periodontai treatment i

TABLE 2 Mean clinical patient results before (BPT)and after (APT) periodontal treatment

Gingivai index(Loe and Siiness]

Tooth No, BPT

11 2121 2.631 2,332 2,141 2,342 2,6

Ext = exlracted.

APT

0Ext

Ext

1

Ext

1.6

Plaque index(SI 1 ness and Loe)

BPT APT

2,6 0,63 Ext

2.6 Ext2,3 0.62,8 Ext2.3 0,5

Probing deptn(mm)

BPT APT

7.1 2.338,8 Exl10,8 Exl6.6 1,6610.1 Ext4,6 2,16

Fig 6 Clinical appearance after correction of crossbite. There ismild gingival inflammation around tiie mandibular inoisors.

therapy. Since then, he has been monitored and hashad oral hygiene regimen at 1-month intervals for ayear in order to avoid inflammation due to poor oralhygiene. The clinical findings of the teeth before andafter periodontai treatment are depicted in Tahle 2,

Actinobacillus actinomycetemcomitans was notdetected from the sampled sites of the present denti-tion at the 12-month evaluation, and the diseaseappeared to be arrested.

DISCUSSION

Over the last 40 years, several severe cases of perio-dontitis, generally associated with certain systemic dis-eases, have been reported in children,''-" Any associa-tion between the susceptible systemic disorders andthe case presented here has been discarded because of

the results from the biochemical, physical, and clinicalexaminations,

Prepubertal periodontitis is one of the destructiveforms of early-onset periodontitis. Although the onsetof the disease is during or immediately after eruptionof the primary teeth, in some cases, it may not be diag-nosed until 7 to 9 years of age,'"

In this case, 3 of the main characteristics of prepu-bertal periodontitis described in the literature'̂ '̂ ''•'̂were observed during the examinatioti: mobility andloss of teeth, presence of putative periodontai patho-gens, and lack of cementum.

The patient presented in this study is a 10-year-oldhoy who had lost all his primary teeth and permanentfirst molars before the first examination. He alsoexhibited some features of severe destruction in theperiodontai apparatus of his remaining teeth asdescribed hy Page et al,'^ Although the onset of the

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Bodur et ai •

disease in our patient could not be established and itwas not possible to determine wbetber tbe periodonti-tis had afieeted the primary dentition, the dental bis-tory tbat was reported by tbe patient's family led us toassume that disease bad begun at least 3 to 4 yearsbefore bis presentation to our clinic. Tberefore, on thebasis of these findings, be was diagnosed as havingprepubertal periodontitis because tbe onset of perio-dontitis occurred before tbe circumpubertal period.

Despite having severe periodontitis, tbe cbild wasapparently systemically healthy and showed neitberunusual infections nor abnormalities in neutrophilfunctions. Our findings were similar with the GPPcases tbat were reported by Lopez.̂ Linden et aF^andYosbida-Minami et al" also have demonstrated thatprepubertal periodontitis can occur in tbe absence ofdetectable neutropbil defects. In contrast to thesereports, some otber investigators noted a decrease inneutrophil cbemotaxis.̂ ^^* According to Page et al,^''itwas suggested tbat tbe generalized form of prepubertalperiodontitis is part of tbe LAD syndrome. On tbemolecular level, the severe form of LAD is associatedwitb an undetectable expression of CD18, and tbemoderate form is associated witb an expression rang-ing between 3°lo and 30% CD18,̂ ^ In a case report onfamilies, Sbapira et al'" pointed out tbat the higb lev-els of CD18 proved tbe impossibility of tbe history ofLAD in one of their presented cases. In our case, lym-phocytes from tbe patient were found to express bigblevels of CD18, ruling out the possibility of LAD. Tbemonocyte functions were not tested because thepatient did not have an increased susceptibility toinfections.

Page and Baab^* pointed out that in some prepuber-tal periodontitis patients, the major determinant of thedisease might be the abnormal deposition of rootcementum. They postulated tbat abnormal cementumor lack of cementum would result in abnormal attach-ment and make tbe affected teeth bigbly susceptible toinvasion by periodontal patbogens. In tbeir bistologi-cal study, Lopez et aP' stated tbat a bigb percentage ofrésorption, unrelated to inflammation, was found inthe cementum of all tbe teetb that were extracted dueto prepubertal periodontitis. In the present case, weexamined the extracted teetb by SEM, and tbe cemen-tum was found to be bypoplastic and aplastic in someunexposed areas.

Althougb A actinomycelemcomitans bas been sus-pected to be the main patbogen in tbe etiology oflocalized juvenile periodontitis (LJP),̂ ^ it bas also beendetected from tbe subgingival plaque samples of manyprepubescent patients witb severe periodontitis.^'^'^ Inour case, A actinomycetemcomilans was detectedfrom tbe subgingival areas of all the affected teetb.Actinobaciilus actinomycetemcomitans is suggested to

be more easily transmitted to bumans witb nonmatureor nonstable oral subgingival microfloras. Sus-ceptibility can occur during the eruption and sheddingof teetb and would explain why the first molars andincisors are the first teetb affected witb LJP and GPP,̂

In our case, we extracted the teetb tbat were mostaffected from periodontal disease, because tbe extrac-tion migbt belp remove tbe habitats and potentialreservoirs of periodontal patbogens. A combination ofa systemic antibiotic regimen witb subgingivalmecbanical debridement and a continuous systematicmeebanieal plaque control program bas been sbownto be a valuable therapeutic approach in early-onsetperiodontitis patients,̂ ^ In order to control plaque, tbepatient was instructed at the first week of treatment touse 0.2% chlorbexidine moutbrinse.

Tbe results of the present case report indicate tbattbere is no evidence for defects in tbe immunologiefunetions of tbe patient witb prepubertal periodontitis,tbat the underlying cause of prepubertal periodontitismigbt not always be related to LAD, and that A actin-omycetemcomitans is suspected to have played a keyrole in the etiology of this case. The disease appearedto have been arrested after tbe establisbment of tbeperiodontal treatment. Also, tbe suspected pathogencould not be isolated from tbe subgingival areas of theremaining teeth at the end of tbe first year of monitor-ing. Clinical and microbiologie follow-up is planned.

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