CASE REPORT Multiple idiopathic external apical root resorption: report of four cases SS Cholia 1,2 , PHR Wilson* ,1 and J Makdissi 2 1 Unit of Restorative Dentistry, Guy’s Hospital, London SE1 9RT, UK; 2 Department of Dental Radiology, Guy’s Hospital, London SE1 9RT, UKMultiple idiopathic external root resorption is an unusual condition that may present in a cervical or an apical form. In this article, we review the published literature relating to multiple idiopathic external apical root resorption and present four clinical cases. We consider the aetiology of this condition and discuss the various treatment options. Dentomaxillofacial Radiology (2005) 34, 240–246. doi: 10.1259/dmfr/74146718 Keywords: tooth root resorption, dental pathology, idiopathic condition Introduction Dec iduous teet h are exf oli ated as a res ult of roo t resorption. This is a physiological process, thought to arise from the pre ssure of erupti ng tee th. 1,2 Physio logica l root resorp tion is a cyc lic process involv ing spur ts of resorpt ive act ivi ty fol lowe d by per iods of att empted repair . Thi s res ult s in variable deciduous tooth mobility before ultimate exfolia- tion. In contra st, roo t reso rpt ion in per manent tee th is pathological. 3 The process of root res orp tion invol ves a comple x interaction of inflammatory cel ls, res orbing cel ls, har d tissue, cytokines and enzymes such as collagenase, matrix metall oprote inase and cystei ne protei nase. 4 The period ontal ligament is a specialized connective tissue which acts as a barrier between the alveolar bone and cementum. 5 Loca- lized damage or loss of periodontal ligament renders the denuded cement um sur fac e che mot actic to cla sticcells such as osteoclasts, macrophages and monocytes. 2,4,6– 8 This can result in root resorption. In cases where multiple teeth are invol ved, Lo ¨e andWaerha ug 9 have suggest ed tha t the dental tissues become part of the osseous system and thus subject to remodelling. Several different types of pathological root resorption are recognized (Table 1) 3 and it can be difficult for the clinic ian to dif ferent iate between them. Resorpt ion can be broadly classified as either internal or external, and usually involves one tooth. 3 External root resorption can further be defined according to the site affected as cervical, apical or intraradicular. Using this classification we will describe four cases ofmultiple idiopathic external apical root resorption (MIEARR) and consider the aetio logy, presentati on and the available treatment options. Case reports Case 1 A 28-year-old Caucasian male was referred to the Unit ofRestorative Dentistry regarding shortening of all his molar and premolar roots. The patient did not have any specific concerns with regard to his teeth, and his general dental pract itioner discovered the resorption incidental ly on routin e int raoral radiographs. The past med ica l his tor y revealed he had sustained a mid-facial fracture in a road traffic accident 2 years previously. His family history was unremarkable. On examination the patient had a minimally restored dentit ion and rel ativ ely poor ora l hygiene, wit h bot h supragi ngival and subging ival calculus evident. He also displayed a lateral and anterior open bite, which was a result of the facial fracture. The patient indicated that this occlusal relationship was uncomfortable and he was aware of grinding his contacting teeth. A panoramic radiograph revealed moderate to severe apical root resorption affecting all maxillary and mandib- ular pos ter ior teeth. The severity of the resorpti on increased in the more posterior teeth (Figures 1 and 2). In some teeth this had progressed almost to the radicular furcation, yet there was no evidence of alveolar bone loss or periradicular peri odontiti s. Haematological and *Corr esponde nce to: Paul HR Wilso n, Unit of Resto rativ e Denti stry, Floor 26, Guy’s Hospital, London Bridge SE1 9RT, UK; E-mail: [email protected]Recei ved 1 Novemb er 2004; accept ed 8 Febru ary 2005 Dentomaxillofacial Radiology (2005) 34, Dentomaxillofacial Radiology (2005) 34, 240–246 q 2005 The British Institute of Radiology http:/ /dmfr.birjournal s.org
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8/14/2019 Dentomaxillofacial Radiology (2005) 34, 240–246 q 2005 the British Institute
1Unit of Restorative Dentistry, Guy’s Hospital, London SE1 9RT, UK; 2 Department of Dental Radiology, Guy’s Hospital, London SE1 9RT, UK
Multiple idiopathic external root resorption is an unusual condition that may present in a cervical oran apical form. In this article, we review the published literature relating to multiple idiopathicexternal apical root resorption and present four clinical cases. We consider the aetiology of this
condition and discuss the various treatment options. Dentomaxillofacial Radiology (2005) 34, 240–246. doi: 10.1259/dmfr/74146718
Deciduous teeth are exfoliated as a result of root resorption.This is a physiological process, thought to arise from thepressure of erupting teeth.1,2 Physiological root resorption isa cyclic process involving spurts of resorptive activityfollowed by periods of attempted repair. This results invariable deciduous tooth mobility before ultimate exfolia-tion. In contrast, root resorption in permanent teeth is
pathological.3
The process of root resorption involves a complexinteraction of inflammatory cells, resorbing cells, hardtissue, cytokines and enzymes such as collagenase, matrixmetalloproteinase and cysteine proteinase.4 The periodontalligament is a specialized connective tissue which acts as abarrier between the alveolar bone and cementum.5 Loca-lized damage or loss of periodontal ligament renders thedenuded cementum surface chemotactic to clasticcells suchas osteoclasts, macrophages and monocytes.2,4,6– 8 This canresult in root resorption. In cases where multiple teeth areinvolved, Loe andWaerhaug9 have suggested that the dentaltissues become part of the osseous system and thus subjectto remodelling.
Several different types of pathological root resorptionare recognized (Table 1)3 and it can be difficult for theclinician to differentiate between them. Resorption can bebroadly classified as either internal or external, and usuallyinvolves one tooth.3 External root resorption can further bedefined according to the site affected as cervical, apical orintraradicular.
Using this classification we will describe four cases of multiple idiopathic external apical root resorption(MIEARR) and consider the aetiology, presentation andthe available treatment options.
Case reports
Case 1A 28-year-old Caucasian male was referred to the Unit of Restorative Dentistry regarding shortening of all his molarand premolar roots. The patient did not have any specificconcerns with regard to his teeth, and his general dentalpractitioner discovered the resorption incidentally onroutine intraoral radiographs. The past medical historyrevealed he had sustained a mid-facial fracture in a roadtraffic accident 2 years previously. His family history wasunremarkable.
On examination the patient had a minimally restoreddentition and relatively poor oral hygiene, with both
supragingival and subgingival calculus evident. He alsodisplayed a lateral and anterior open bite, which was aresult of the facial fracture. The patient indicated that thisocclusal relationship was uncomfortable and he was awareof grinding his contacting teeth.
A panoramic radiograph revealed moderate to severeapical root resorption affecting all maxillary and mandib-ular posterior teeth. The severity of the resorptionincreased in the more posterior teeth (Figures 1 and 2).In some teeth this had progressed almost to the radicularfurcation, yet there was no evidence of alveolar bone lossor periradicular periodontitis. Haematological and
*Correspondence to: Paul HR Wilson, Unit of Restorative Dentistry, Floor 26,
biochemical screening was within the normal range.A diagnosis of MIEARR was made radiographically.
In light of the patient’s discomfort with his traumati-cally induced malocclusion and tooth grinding habit,reversible occlusal therapy was indicated. An upper all-acrylic resin bite-raising appliance was constructed(Michigan splint)10 to distribute the occlusal loadsuniformly. The patient was then placed on regular review.
Case 2
A 37-year-old Arabic male was referred to the Unit of Restorative Dentistry by his dentist regarding root resorp-tion affecting all molar teeth, but most severely the upperfirst molars (16, 26). The patient complained of increasedmobility of 16 and occasional gingival bleeding whenbrushing. There was no history of trauma and his pastmedical and family histories were unremarkable. The pastdental history revealed congenitally missing maxillary
permanent canines (13, 23). On examination 28, 38, 36, 46and 48 were missing. His incisal relationship was Class III.There was generalized bleeding on probing with evidenceof supragingival and subgingival calculus present. Perio-dontal probing depths were generally 3 mm, with theexception of 16, 26 and 37, which displayed probing depths
of greater than 6 mm.A panoramic radiograph (Figure 3) revealed a mini-
mally restored permanent dentition and evidence of generalized root resorption. The upper first molar teeth(16, 26) appeared to be the most severely affected, with 16showing complete loss of the distobuccal root. There wasalso evidence of apical root resorption of the lower rightsecond molar (47) and blunting of all the upper premolarroots. Haematological and biochemical screening waswithin the normal range.
From the clinical and radiographic features a diagnosisof MIEARR was made. The patient was placed on longterm review, during which the upper right first molar (16)exfoliated spontaneously (Figures 4 and 5). The resulting
edentulous space was left unrestored.
Case 3
A 38-year-old Caucasian male was referred to the Unit of Restorative Dentistry by the South Thames Cleft Lip andPalate Service at Guy’s Hospital. The patient complainedof a loose upper fixed bridge. He had had a bilateral cleftlip and palate repair in the past and recently a rhinoplasty.Otherwise, his medical history was unremarkable. The pastdental history revealed irregular dental attendance. Hisfamily history was also unremarkable.
On extraoral examination the patient had obviousscarring from the lip repair. Intraorally, there was a
heavily restored dentition with complex maxillary fixedpartial dentures (bridges). The bridge restoring the upperright quadrant had debonded on the upper right centralincisor abutment. Caries could be detected at the retainermargins. The patient also had a fractured amalgamrestoration on the lower right first molar (46).
Radiographic examination revealed carious breakdownof the aforementioned mesial abutment and markedgeneralized radicular resorption associated with all upperand lower teeth (Figure 6). Resorption was more advanced
Figure 1 Panoramic radiograph of Case 1 showing severe root resorption of the permanent molars in all four quadrants and moderate root resorption of premolars
Table 1 Classification and aetiological factors in pathological rootresorption3
Site Type Aetiology
Internal TraumaInfection
External Surface Trauma
Inflammatory TraumaInfection
Replacement (Ankylosis) Avulsion and re-implantationLuxationTransplantation
diagnoses of a failing maxillary dental prosthesis second-
ary to a carious abutment and MIEARR. The upper right
central incisor root was extracted and the maxillary bridges
were replaced by a combination of single unit crowns on
the premolar teeth and a fixed bridge between the upper
canines. The upper left central incisor (21) was employed
as a pier abutment. The patient was kept under regularreview in order to monitor the MIEARR.
Case 4A 39-year-old Caucasian female was referred to the Unit of Restorative Dentistry by her dentist regarding the lower leftthird molar tooth (38), which was becoming increasingly
Figure 2 Full-mouth periapical radiographs of Case 1
Figure 3 Panoramic radiograph of Case 2 showing extensive resorption of the roots of the left and right first molars of Case 2
Figure 4 Photograph of exfoliated 16 (a view from below) of Case 2 Figure 5 Photograph of exfoliated 16 (lateral view)
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loose. There was no pain or sepsis associated with this tooth.The past medical history was uneventful and the patient hadtraumatically lost the upper left central incisor (21) severalyears earlier. There was no family history of root resorption.
The panoramic radiograph revealed the following
features (Figure 7):
† Moderate to severe radicular resorption associated withthe maxillary and mandibular third molar teeth (18, 28,38, 48);
† Early resorption of the distobuccal roots of the upperright and left second molar teeth (17, 27) and distalroots of the lower right and left second molar teeth (37,47);
† Localized radiolucency of the bone associated with thethird molar teeth; and
† A symmetrical root resorption pattern, with progressionanteroposteriorly.
Electric pulp testing indicated that all teeth, except theupper right central incisor (11), were vital. No haemato-logical or biochemical investigations were undertaken.
From the radiographic appearance a diagnosis of MIEARR was made. All third molars were extracted andthe patient was placed on regular review.
Discussion
Muller and Rony11 first documented idiopathic external rootresorption in 1930 in a case report of a 36-year-old woman
with generalized rapidly progressive cervical root resorp-tion. The authors believed that the cervical damage wasassociated with a functional hepatic disturbance. Theirtreatment involved dietary intervention so that liverfunction tests returned to a normal range. This simple
intervention appeared to be effective in halting the processin this case.Since Muller and Rony,11 it has been postulated that
external root resorption has several other causes (Table 1).It appears to be a relatively common incidental radio-graphic finding in isolated teeth, but uncommon in ageneralized form.3 Local causes are thought to be the mostfrequent, caused by excessive pressure and inflammation.Mechanisms include large orthodontic forces, occlusaltrauma, impacted teeth, re-implanted teeth, periradicularinfection or tooth bleaching.3 Many systemic abnormalitieshave been implicated, which include hormonal disturb-ances,12 hypophosphatasia,13 hyperparathyroidism,14
It shouldbe noted that arrested root development in radiotherapy,20
dental dyplasia,21 hypothyroidism22 and Stevens-Johnsonsyndrome23 can have a similar radiographic presentation toexternal root resorption. Stafne and Slocumb24 in a study of 179 root resorption cases failed to find any definiteassociations with systemic disease. Newman13 reported asimilar finding in a study of 47 individuals with idiopathicroot resorption. In contrast, Gunraj8 has suggested thatchanges in the host cellular immune system may beimplicated. The cases presented in this article did not haveobvious local or systemic causative factors for their root
Figure 6 Panoramic radiograph of Case 3 showing heavily restored dentition with generalized root resorption
Figure 7 Panoramic radiograph of Case 4 showing variable resorption of molar teeth
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resorption patterns. Case 1 suffered a facial fracture but thepresentation of resorption was symmetrical and involvedboth upper and lower jaws. Case 3 had sustained surgicaltrauma as a result of cleft lip and palate correction, butonce again the presentation of tooth resorption wassymmetrical in both jaws. Cases 2 and 4 had no obvious
aetiological factors.In an attempt to explain the cause of idiopathic external
root resorption, Pinska and Jarzynka25 first suggestedgenetic susceptibility in their report of a family withgeneralized root resorption. Newman13 then followed thiswith a study of 37 families. Newman contacted first-degreerelatives of affected probands and assessed external rootresorption from periapical radiographs. A tentative geneticassociation was found. Six families displayed an autosomaldominant inheritance pattern, three families an autosomalrecessive pattern, while three individuals displayed aspontaneous phenotype. Unfortunately, the small samplesize meant that these results were not statisticallysignificant. The most compelling evidence for a genetic
association with MIEARR came from Saravia et al.26 Theauthors described 14-year-old monozygotic twins, whopresented with identical clinical and radiographic patternsof MIEARR. Despite this evidence, in all our cases thefamily histories were inconclusive.
External root resorption which develops in the absenceof a plausible cause is termed idiopathic (Table 1). Bydefinition, idiopathic external root resorption is a diagnosisof exclusion. From the number of reported cases in thedental literature, multiple idiopathic external cervical rootresorption (MIECRR)27 appears to be more common thanMIEARR. MIECRR is associated with younger femalesand is unrelated to any significant medical condition ordental abnormality.27 Radiographically, the process
appears to initiate at the cemento– enamel junction andcontinues until the lesion coalesces. Occasionally itspontaneously arrests. The number of teeth affected isthought to range from 5 to 24 in a single dentition, with nosite or side predilection.
A recent literature search for MIEARR identified tenpublished case reports describing 11 patients,14,18,26,28–34
to which we have added the four cases presented in thisarticle (Table 2; n ¼ 15). These 15 cases indicate thatMIEARR affects a wide age range of patients, from 14years to 39 years old. In contrast to MIECRR, malesappear to be more frequently affected by MIEARR thanfemales, with a male:female ratio of 11:4. In addition,MIEARR appears to have a predilection for premolar and
molar regions (Table 2). In contrast, there was no sitespecific relationship reported for MIECRR.27 Othercommon features of the MIEARR cases appear to be:
† Normal clinical appearance of teeth and periodontaltissues;
† Root resorption associated with vital teeth andendodontically treated teeth;
† Lack of periodontal and periradicular inflammation;† Alveolar bone levels within normal limits;† Absence of local aetiological factors;† Patients asymptomatic until very late in the patho-
logical process where increased tooth mobilityreported;
† Commonly found as an incidental finding on radio-graphs; and
† Intramaxillary and intermaxillary symmetrical patternof root resorption.
With no absolute aetiological factors identified, treatmentof MIEARR depends largely on the presenting symptomsand the extent and the severity of root resorption. The usualtreatment is the extraction of teeth of poor prognosis andlong-term monitoring of the remaining dentition usingserial radiographs, periodontal measures, sensibility tests orpatient symptoms. This was the option chosen for themajority of our cases. Edentulous saddles may be restoredusing adhesive or conventional fixed bridges, removablepartial dentures or osseointegrated implants. Abutmentteeth must be carefully assessed for root resorption. Thesuccess of long-term osseointegration in sites where rootresorption has been active is unknown.35 In severe cases the
only option available may be extraction of all teeth andconstruction of a complete denture.
If adverse occlusal loading or occlusal trauma issuspected then adjustment of occlusal interferences orprovision of an occlusal appliance to remove the influenceof such interferences may be indicated. This was employedfor Case 1. If irreversible occlusal therapy is to beundertaken, then practice of the occlusal adjustment onaccurately articulated study models is required pre-operatively. A more invasive approach involves endodontictreatment of the affected teeth. This has been welldocumented for inflammatory root resorption, wherecalcium hydroxide is the current intraradicular medicamentof choice.36 However, a common finding in MIEARR is that
teeth remain vital even after extensive root resorption. It hasbeen suggested that Ledermix (Triamcinolone acetonideand Demeclocycline calcium; Lederle Laboratories, UK)inhibits the proliferation of dentinoclasts37 and it may proveeffective when mixed with calcium hydroxide. An exper-imental approach may be calcitonin38 as an intracanalmedicament. Calcitonin inhibits osteoclast motility andretraction and could be potentially useful in modifying theresorptive process. Postlethwaite and Hamilton29 planned toextirpate the pulps and apply intraradicular calciumhydroxide to half of the affected teeth in their case of MIEARR. Unfortunately they have not reported theoutcome. Rivera and Walton31 stated that MIEARR doesnot seem tobe mediatedby orhaveits sourcefromthe dental
pulp. Therefore, in the absence of pulpal symptoms,endodontic therapy cannot be indicated for MIEARR. Inthe future bioactive molecules capable of modifying theprocess of root resorption may become available whichtarget the periradicular resorptive process.39
Conclusions
From the published literature and these four new cases,MIEARR affects a wide age range of individuals, withmales affected more frequently than females. There
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appears to be a predilection for premolar and molar teeth,in a symmetrical pattern of expression. The currentmainstay of management for affected individuals remainslong-term monitoring, but occlusal therapy and restorativedental treatment have a role in appropriate circumstances.
However, endodontic therapy is not indicated at the presenttime. The aetiology of MIEARR remains unknown, but it ishoped that the discovery of the molecular and cellularmechanism of root resorption will yield new methods of treatment.
References
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3. Bakland LK. Root resorption. Dent Clin North Am 1992; 36:491–507.
4. Ne RF, Witherspoon DE, Gutmann JL. Tooth resorption. Quintes-sence Int 1999; 30: 9–25.
5. Brezniak N, Wasserstein A. Orthodontically induced inflammatoryroot resorption. Part I: The basic science aspects. Angle Orthod 2002;72: 175–179.
6. Sasaki T, Shimizu T, Watanabe C, Hiyoshi Y. Cellular roles inphysiological root resorption of deciduous teeth in the cat. J Dent Res1990; 69: 67–74.
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Rivera and Walton31 1994 Apical M 24 All permanent teethDi Domizio et al32 2000 Apical F 26 All permanent teethCholia et al (this study) 2004 Apical M 27 14, 15, 16, 17
Dent 1986; 34: 385–386.35. Marx RE, Garg AK. Bone structure, metabolism, and physiology: its
impact on dental implantology. Implant Dent 1998; 7: 267–276.36. Trope M. Clinical management of the avulsed tooth. Dent Clin North
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