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Dentomaxillofacial Radiology (2005) 34, 240–246

q 2005 The British Institute of Radiology


http://dmfr.birjournals.org

CASE REPORT
Multiple idiopathic external apical root resorption: report
of four cases
SS Cholia1,2, PHR Wilson*,1 and J Makdissi2
1
Unit of Restorative Dentistry, Guy’s Hospital, London SE1 9RT, UK; 2Department 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 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

Deciduous teeth are exfoliated as a result of root resorption. Using this classification we will describe four cases of
This is a physiological process, thought to arise from the multiple idiopathic external apical root resorption
pressure of erupting teeth.1,2 Physiological root resorption is (MIEARR) and consider the aetiology, presentation and
a cyclic process involving spurts of resorptive activity the available treatment options.
followed by periods of attempted repair. This results in
variable deciduous tooth mobility before ultimate exfolia-
tion. In contrast, root resorption in permanent teeth is
pathological.3 Case reports
The process of root resorption involves a complex
interaction of inflammatory cells, resorbing cells, hard Case 1
tissue, cytokines and enzymes such as collagenase, matrix A 28-year-old Caucasian male was referred to the Unit of
metalloproteinase and cysteine proteinase.4 The periodontal Restorative Dentistry regarding shortening of all his molar
ligament is a specialized connective tissue which acts as a and premolar roots. The patient did not have any specific
barrier between the alveolar bone and cementum.5 Loca- concerns with regard to his teeth, and his general dental
lized damage or loss of periodontal ligament renders the practitioner discovered the resorption incidentally on
denuded cementum surface chemotactic to clastic cells such routine intraoral radiographs. The past medical history
as osteoclasts, macrophages and monocytes.2,4,6 – 8 This can revealed he had sustained a mid-facial fracture in a road
result in root resorption. In cases where multiple teeth are traffic accident 2 years previously. His family history was
involved, Löe and Waerhaug9 have suggested that the dental unremarkable.
tissues become part of the osseous system and thus subject On examination the patient had a minimally restored
to remodelling. dentition and relatively poor oral hygiene, with both
Several different types of pathological root resorption supragingival and subgingival calculus evident. He also
are recognized (Table 1)3 and it can be difficult for the displayed a lateral and anterior open bite, which was a
clinician to differentiate between them. Resorption can be result of the facial fracture. The patient indicated that this
broadly classified as either internal or external, and usually occlusal relationship was uncomfortable and he was aware
involves one tooth.3 External root resorption can further be of grinding his contacting teeth.
defined according to the site affected as cervical, apical or A panoramic radiograph revealed moderate to severe
intraradicular. apical root resorption affecting all maxillary and mandib-
ular posterior teeth. The severity of the resorption
increased in the more posterior teeth (Figures 1 and 2).
*Correspondence to: Paul HR Wilson, Unit of Restorative Dentistry, Floor 26,
In some teeth this had progressed almost to the radicular
Guy’s Hospital, London Bridge SE1 9RT, UK;
E-mail: Paul.Wilson@gstt.sthames.nhs.uk furcation, yet there was no evidence of alveolar bone loss
Received 1 November 2004; accepted 8 February 2005 or periradicular periodontitis. Haematological and
Multiple idiopathic external apical root resorption
SS Cholia et al 241

Table 1 Classification and aetiological factors in pathological root permanent canines (13, 23). On examination 28, 38, 36, 46
resorption3 and 48 were missing. His incisal relationship was Class III.
Site Type Aetiology There was generalized bleeding on probing with evidence
Internal Trauma of supragingival and subgingival calculus present. Perio-
Infection dontal probing depths were generally 3 mm, with the
External Surface Trauma exception of 16, 26 and 37, which displayed probing depths
Inflammatory Trauma of greater than 6 mm.
Infection
Replacement (Ankylosis) Avulsion and re-implantation A panoramic radiograph (Figure 3) revealed a mini-
Luxation mally restored permanent dentition and evidence of
Transplantation generalized root resorption. The upper first molar teeth
Pressure Orthodontic tooth movement (16, 26) appeared to be the most severely affected, with 16
Excessive occlusal forces
Impacted teeth showing complete loss of the distobuccal root. There was
Supernumerary teeth also evidence of apical root resorption of the lower right
Tumours second molar (47) and blunting of all the upper premolar
Cysts roots. Haematological and biochemical screening was
Related to systemic conditions Hyperparathyroidism
Paget’s disease within the normal range.
Papillon-Lefèvre syndrome From the clinical and radiographic features a diagnosis
Bone dysplasia of MIEARR was made. The patient was placed on long
Renal disease term review, during which the upper right first molar (16)
Hepatic disease
Invasive (Cervical) Trauma
exfoliated spontaneously (Figures 4 and 5). The resulting
Orthodontic tooth movement edentulous space was left unrestored.
Periodontal treatment
Intracoronal tooth bleaching
Unknown Case 3
Idiopathic Unknown A 38-year-old Caucasian male was referred to the Unit of
Restorative Dentistry by the South Thames Cleft Lip and
biochemical screening was within the normal range. Palate Service at Guy’s Hospital. The patient complained
A diagnosis of MIEARR was made radiographically. of a loose upper fixed bridge. He had had a bilateral cleft
In light of the patient’s discomfort with his traumati- lip and palate repair in the past and recently a rhinoplasty.
cally induced malocclusion and tooth grinding habit, Otherwise, his medical history was unremarkable. The past
reversible occlusal therapy was indicated. An upper all- dental history revealed irregular dental attendance. His
acrylic resin bite-raising appliance was constructed family history was also unremarkable.
(Michigan splint)10 to distribute the occlusal loads On extraoral examination the patient had obvious
uniformly. The patient was then placed on regular review. scarring from the lip repair. Intraorally, there was a
heavily restored dentition with complex maxillary fixed
Case 2 partial dentures (bridges). The bridge restoring the upper
A 37-year-old Arabic male was referred to the Unit of right quadrant had debonded on the upper right central
Restorative Dentistry by his dentist regarding root resorp- incisor abutment. Caries could be detected at the retainer
tion affecting all molar teeth, but most severely the upper margins. The patient also had a fractured amalgam
first molars (16, 26). The patient complained of increased restoration on the lower right first molar (46).
mobility of 16 and occasional gingival bleeding when Radiographic examination revealed carious breakdown
brushing. There was no history of trauma and his past of the aforementioned mesial abutment and marked
medical and family histories were unremarkable. The past generalized radicular resorption associated with all upper
dental history revealed congenitally missing maxillary and 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

Dentomaxillofacial Radiology
Multiple idiopathic external apical root resorption
242 SS Cholia et al

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

in the maxilla compared with the mandible. Serum as a pier abutment. The patient was kept under regular
biochemistry was within the normal range. review in order to monitor the MIEARR.
The clinical and radiographic features suggested
diagnoses of a failing maxillary dental prosthesis second- Case 4
ary to a carious abutment and MIEARR. The upper right A 39-year-old Caucasian female was referred to the Unit of
central incisor root was extracted and the maxillary bridges Restorative Dentistry by her dentist regarding the lower left
were replaced by a combination of single unit crowns on third molar tooth (38), which was becoming increasingly
the premolar teeth and a fixed bridge between the upper
canines. The upper left central incisor (21) was employed

Figure 4 Photograph of exfoliated 16 (a view from below) of Case 2 Figure 5 Photograph of exfoliated 16 (lateral view)

Dentomaxillofacial Radiology
Multiple idiopathic external apical root resorption
SS Cholia et al 243

Figure 6 Panoramic radiograph of Case 3 showing heavily restored dentition with generalized root resorption

loose. There was no pain or sepsis associated with this tooth. with generalized rapidly progressive cervical root resorp-
The past medical history was uneventful and the patient had tion. The authors believed that the cervical damage was
traumatically lost the upper left central incisor (21) several associated with a functional hepatic disturbance. Their
years earlier. There was no family history of root resorption. treatment involved dietary intervention so that liver
The panoramic radiograph revealed the following function tests returned to a normal range. This simple
features (Figure 7): intervention appeared to be effective in halting the process
in this case.
† Moderate to severe radicular resorption associated with Since Muller and Rony,11 it has been postulated that
the maxillary and mandibular third molar teeth (18, 28, external root resorption has several other causes (Table 1).
38, 48); It appears to be a relatively common incidental radio-
† Early resorption of the distobuccal roots of the upper graphic finding in isolated teeth, but uncommon in a
right and left second molar teeth (17, 27) and distal generalized form.3 Local causes are thought to be the most
roots of the lower right and left second molar teeth (37, frequent, caused by excessive pressure and inflammation.
47); Mechanisms include large orthodontic forces, occlusal
† Localized radiolucency of the bone associated with the trauma, impacted teeth, re-implanted teeth, periradicular
third molar teeth; and infection or tooth bleaching.3 Many systemic abnormalities
† A symmetrical root resorption pattern, with progression have been implicated, which include hormonal disturb-
anteroposteriorly. ances,12 hypophosphatasia, 13 hyperparathyroidism, 14
Paget’s disease,15 Papillon-Lefèvre syndrome,16 renal
Electric pulp testing indicated that all teeth, except the disease,17 hepatic disease18 and bone dysplasia.19 It should
upper right central incisor (11), were vital. No haemato- be noted that arrested root development in radiotherapy,20
logical or biochemical investigations were undertaken. dental dyplasia,21 hypothyroidism22 and Stevens-Johnson
From the radiographic appearance a diagnosis of syndrome23 can have a similar radiographic presentation to
MIEARR was made. All third molars were extracted and external root resorption. Stafne and Slocumb24 in a study of
the patient was placed on regular review. 179 root resorption cases failed to find any definite
associations with systemic disease. Newman13 reported a
similar finding in a study of 47 individuals with idiopathic
Discussion root resorption. In contrast, Gunraj8 has suggested that
changes in the host cellular immune system may be
Muller and Rony11 first documented idiopathic external root implicated. The cases presented in this article did not have
resorption in 1930 in a case report of a 36-year-old woman obvious local or systemic causative factors for their root

Figure 7 Panoramic radiograph of Case 4 showing variable resorption of molar teeth

Dentomaxillofacial Radiology
Multiple idiopathic external apical root resorption
244 SS Cholia et al

resorption patterns. Case 1 suffered a facial fracture but the logical process where increased tooth mobility
presentation of resorption was symmetrical and involved reported;
both upper and lower jaws. Case 3 had sustained surgical † Commonly found as an incidental finding on radio-
trauma as a result of cleft lip and palate correction, but graphs; and
once again the presentation of tooth resorption was † Intramaxillary and intermaxillary symmetrical pattern
symmetrical in both jaws. Cases 2 and 4 had no obvious of root resorption.
aetiological factors.
In an attempt to explain the cause of idiopathic external With no absolute aetiological factors identified, treatment
root resorption, Pinska and Jarzynka25 first suggested of MIEARR depends largely on the presenting symptoms
genetic susceptibility in their report of a family with and the extent and the severity of root resorption. The usual
generalized root resorption. Newman13 then followed this treatment is the extraction of teeth of poor prognosis and
with a study of 37 families. Newman contacted first-degree long-term monitoring of the remaining dentition using
relatives of affected probands and assessed external root serial radiographs, periodontal measures, sensibility tests or
resorption from periapical radiographs. A tentative genetic patient symptoms. This was the option chosen for the
association was found. Six families displayed an autosomal majority of our cases. Edentulous saddles may be restored
dominant inheritance pattern, three families an autosomal using adhesive or conventional fixed bridges, removable
recessive pattern, while three individuals displayed a partial dentures or osseointegrated implants. Abutment
spontaneous phenotype. Unfortunately, the small sample teeth must be carefully assessed for root resorption. The
size meant that these results were not statistically success of long-term osseointegration in sites where root
significant. The most compelling evidence for a genetic resorption has been active is unknown.35 In severe cases the
association with MIEARR came from Saravia et al.26 The only option available may be extraction of all teeth and
authors described 14-year-old monozygotic twins, who construction of a complete denture.
presented with identical clinical and radiographic patterns If adverse occlusal loading or occlusal trauma is
of MIEARR. Despite this evidence, in all our cases the suspected then adjustment of occlusal interferences or
family histories were inconclusive. provision of an occlusal appliance to remove the influence
External root resorption which develops in the absence of such interferences may be indicated. This was employed
of a plausible cause is termed idiopathic (Table 1). By for Case 1. If irreversible occlusal therapy is to be
definition, idiopathic external root resorption is a diagnosis undertaken, then practice of the occlusal adjustment on
of exclusion. From the number of reported cases in the accurately articulated study models is required pre-
dental literature, multiple idiopathic external cervical root operatively. A more invasive approach involves endodontic
resorption (MIECRR)27 appears to be more common than treatment of the affected teeth. This has been well
MIEARR. MIECRR is associated with younger females documented for inflammatory root resorption, where
and is unrelated to any significant medical condition or calcium hydroxide is the current intraradicular medicament
dental abnormality.27 Radiographically, the process of choice.36 However, a common finding in MIEARR is that
appears to initiate at the cemento – enamel junction and teeth remain vital even after extensive root resorption. It has
continues until the lesion coalesces. Occasionally it been suggested that Ledermix (Triamcinolone acetonide
spontaneously arrests. The number of teeth affected is and Demeclocycline calcium; Lederle Laboratories, UK)
thought to range from 5 to 24 in a single dentition, with no inhibits the proliferation of dentinoclasts37 and it may prove
site or side predilection. effective when mixed with calcium hydroxide. An exper-
A recent literature search for MIEARR identified ten imental approach may be calcitonin38 as an intracanal
published case reports describing 11 patients,14,18,26,28 – 34 medicament. Calcitonin inhibits osteoclast motility and
to which we have added the four cases presented in this retraction and could be potentially useful in modifying the
article (Table 2; n ¼ 15). These 15 cases indicate that resorptive process. Postlethwaite and Hamilton29 planned to
MIEARR affects a wide age range of patients, from 14 extirpate the pulps and apply intraradicular calcium
years to 39 years old. In contrast to MIECRR, males hydroxide to half of the affected teeth in their case of
appear to be more frequently affected by MIEARR than MIEARR. Unfortunately they have not reported the
females, with a male:female ratio of 11:4. In addition, outcome. Rivera and Walton31 stated that MIEARR does
MIEARR appears to have a predilection for premolar and not seem to be mediated by or have its source from the dental
molar regions (Table 2). In contrast, there was no site pulp. Therefore, in the absence of pulpal symptoms,
specific relationship reported for MIECRR.27 Other endodontic therapy cannot be indicated for MIEARR. In
common features of the MIEARR cases appear to be: the future bioactive molecules capable of modifying the
process of root resorption may become available which
† Normal clinical appearance of teeth and periodontal target the periradicular resorptive process.39
tissues;
† Root resorption associated with vital teeth and
endodontically treated teeth; Conclusions
† Lack of periodontal and periradicular inflammation;
† Alveolar bone levels within normal limits; From the published literature and these four new cases,
† Absence of local aetiological factors; MIEARR affects a wide age range of individuals, with
† Patients asymptomatic until very late in the patho- males affected more frequently than females. There

Dentomaxillofacial Radiology
Multiple idiopathic external apical root resorption
SS Cholia et al 245

Table 2 Reported cases of multiple idiopathic external apical root resorption


Reference Year Region Sex Age (years) Teeth affected
28
Soni and La Velle 1970 Apical M 34 14
24, 25
35, 36, 37
45, 46, 48
Cowie and Wright33 1981 Apical M 27 14, 15, 16, 17
24, 25, 26, 27
37
Belanger and Coke14 1985 Apical M 14 All permanent teeth
Brooks34 1986 Apical M 17 16, 35, 36, 46
Pankhurst et al18 1988 Apical M 30 15, 16, 17
25, 26, 28
37, 38
46, 47, 48
Saravia and Meyer26 1989 Apical 2 £ F (Twins) 14 14, 15, 16, 17
24, 25, 26, 27
34, 35, 36, 37
44, 45, 46, 47
Posthewaite and Hamilton29 1989 Apical M 14 11, 12, 13, 14, 15
21, 22, 23, 24, 25
31, 32, 33, 34, 35
41, 42, 43, 44, 45
Yusof and Ghazali30 1989 Apical M 35 14, 15
21, 24, 25, 26
34, 35
41, 44, 45
Rivera and Walton31 1994 Apical M 24 All permanent teeth
Di Domizio et al32 2000 Apical F 26 All permanent teeth
Cholia et al (this study) 2004 Apical M 27 14, 15, 16, 17
24, 25, 26, 27
34, 35, 36, 37
38, 44, 45, 47, 48
M 38 14, 15, 16, 17
24, 25, 26, 27
44, 45, 48
M 37 14, 15, 16, 17
18, 24, 25, 26
27, 28, 34, 35
36, 37, 38, 44
45, 46, 47, 48
F 39 14, 15, 16, 17
18, 24, 26, 27
28, 35, 36, 37
46, 47

appears to be a predilection for premolar and molar teeth, However, endodontic therapy is not indicated at the present
in a symmetrical pattern of expression. The current time. The aetiology of MIEARR remains unknown, but it is
mainstay of management for affected individuals remains hoped that the discovery of the molecular and cellular
long-term monitoring, but occlusal therapy and restorative mechanism of root resorption will yield new methods of
dental treatment have a role in appropriate circumstances. treatment.

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