Scleroderma PDF

Case Report/Clinical Techniques
Root Resorption Associated with Mandibular Bone

Erosion in a Patient with Scleroderma
Maria Antonia Zancanaro de Figueiredo, BDS, PhD*
Jose Antonio Poli de Figueiredo, BDS, MSc, PhD and
Stephen Porter, MD, PhD, FDS RCS, FDS RCSE*
Abstract
A rare feature of mandibular bone erosion and external
apical resorption of a mandibular left third molar (tooth
#17) in a patient with scleroderma is described. Scleroderma is characterized by vascular and fibrous changes
of the mucocutaneous surfaces and viscera caused by
immunologically mediated connective tissue disorders.
A dental panoramic tomogram of the patient revealed
notable erosion of the left ramus, the inferior border of
the mandible, and the left coronoid process. Resorption
of the distal root of the tooth #17 was notable, coincident with the mandibular erosive process, and such
association has not yet been reported. The report details the possible cause of this unusual presentation of
tooth root resorption. The increased physical pressure
of the tight facial mucocutaneous tissues from the
scleroderma is likely to have participated in the resorptive process. (J Endod 2008;34:102103)
Key Words
Mandibular erosion, resorption, root, scleroderma
cleroderma is a group of immunologically mediated connective tissue disorders,

characterized by vascular and fibrous changes of the mucocutaneous surfaces and
viscera. It can give rise to a wide range of clinical features but in general can be classified
as localized or systemic (1, 2). Although rare, scleroderma has a worldwide distribution, usually manifesting clinically in early middle life, and it is 3 to 4 times more
common in women than men (1). The prevalence of scleroderma may be increasing as
a consequence of improved diagnostic methods and/or improvements in management
and, hence, patient survival (3). A wide pattern of abnormalities of soft and hard tissues
of the face and mouth can arise. Typical features include orofacial telangiectasia, tightening of the skin of the face gives rise to a loss of wrinkling, nasal beaking, and
microstomia. Salivary gland dysfunction manifesting as secondary Sjgrens syndrome
is common, and together with difficulties in maintaining good oral hygiene (as a consequence of microstomia and reduced manual dexterity), there may be an increased
risk of caries (4, 5).
Loss of facial hard tissue, presumably because of pressure atrophy, is a rare
feature. There is only one report of root resorption associated with scleroderma (6),
but to date there have been no reported instances of root resorption associated with
mandibular bone erosion in patients with scleroderma. The present report details the
features of a patient with resorption of the mandibular ramus and angle and external
resorption of the distal root of an adjacent permanent molar.
Case Report
From the *Oral Medicine Unit and Endodontology Unit,
UCL Eastman Dental Institute, London, United Kingdom; and
Oral Medicine Unit, Pontifical Catholic University of Rio

Grande do Sul, Sao Lucas Hospital, Porto Alegre, Rio Grande
do Sul, Brazil.
Address requests for reprints to Dr Jose Antonio Poli de
Figueiredo, Unit of Endodontology, UCL Eastman Dental Institute, 256 Grays Inn Road, London, UK WC1X 8LD. E-mail
address: j.figueiredo@eastman.ucl.ac.uk.
0099-2399/$0 - see front matter
Copyright 2008 by the American Association of
Endodontists.
doi:10.1016/j.joen.2007.10.021
A 43-year-old female patient was referred to the Oral Medicine unit of UCL Eastman
Dental Institute. Her principal complaint was difficulty eating as a consequence of loss
of some of her lower teeth. Clinical examination revealed facial telangiectasia and
microstomia, with an interincisal opening of approximately 2 cm. Intraorally, there was
no xerostomia, but there were a number of telangiectasia present on the hard palate and
the left and right anterior lateral borders of the tongue. There was no obvious sign of
active caries. Although there was generalized chronic marginal periodontitis, there was
no mobility of the present teeth.
A dental panoramic tomogram (Fig. 1) revealed notable erosion of the left ramus,
the inferior border of the mandible, and the left coronoid process. The roots of tooth
#30 were still in situ, and there was radiologic evidence of caries at the crown of tooth
#31. There was radiologic evidence of generalized chronic periodontitis. Of note, there
was mild apical resorption of the distal root of tooth #17 (Fig. 2). This resorption was
coincident with the mandibular erosive process. The distal root seemed blunted, and
the periodontal ligament displayed normal radiographic features. The tooth #17 responded positively to a sensitivity test with tetrafluorethane (Endo Ice, Whaledent,
Mahwah, NJ), which was similar to the other teeth present in the left quadrant. The tooth
root resorption was a radiographic finding. No specific treatment was suggested other
than follow-up radiographic assessment in 2 years.
Discussion
Bony resorption is an uncommon but recognized complication of longstanding
scleroderma. This resorption, presumably caused by pressure atrophy by the tight
mucocutaneous tissues, affects the inferior body of the mandible (giving rise to a
pregonial notch), angle, coronoid process of the mandible condylar process, and
102
de Figueiredo et al.
JOE Volume 34, Number 1, January 2008
Case Report/Clinical Techniques

occasionally the zygoma (2). Widening of the radiographic image of the
periodontal ligament may also arise. The only recorded finding of root
resorption tries to explain the process as a consequence of regressive
changes of pulpal tissue within root canals secondary to the systemic
sclerosis (6). However, in the present case, tooth sensitivity test was
performed, and the pulp reaction to the cold test was similar to the
unaffected teeth.
The present patient had root resorption of the apical third of the
distal root of a mandibular third molar adjacent to a site of gross
external bony resorption. The presence of the mandibular right
third molar enabled the comparison with an unaffected distal root.
It is likely that the affected root had greater resorption than that
detected radiologically (7).
The hard tissues (dentine, cementum, and enamel) of permanent
teeth do not normally undergo resorption (8), probably because of the
anti-invasion factor, a potent collagenase inhibitor present in cartilage,
blood vessel walls, and teeth (9). Resorption of permanent teeth is
usually the result of trauma or chronic inflammation of the pulp and/or
periodontal tissues. External resorption can also be induced by increased physical pressure in the periodontal ligament associated with
orthodontic tooth movement, tumors, or tooth eruption (10, 11). Resorption on the external root surface usually accompanies simultaneous
reactions within the alveolar bone; indeed, the process of tooth resorption is considered to be similar to that of bone resorption (9).
The etiology of different types of root resorption requires 2 phases:
injury to the protective tissues (mechanical or chemical) and stimulation by infection or pressure (11). Teeth with apical periodontitis are
likely to undergo inflammatory root resorption. It has been hypothesized that the oncotic pressure in periapical lesions caused by the presence of large protein molecules provides a common pressure pathway
for the different types of root resorption (12). However, the treatment is
related to the stimulating factors (13, 14).
The resorption of the present patient is unlikely to have been the
consequence of local occlusal forces. The present external resorption
would potentially be the consequence of the erosive process affecting
the adjacent mandible. This may have occurred as a result of the tight
facial mucocutaneous tissues from the scleroderma, initiating cell signaling to inhibit the anti-invasion factor in the cementum or periodontal
membrane cells (9) surrounding the periapex of this distal root. Additionally, there is deviation in the calcium and phosphorus contents and
Figure 1. A dental panoramic tomogram showing the affected left mandible with
erosion and resorption of the distal root of lower left third molar.
JOE Volume 34, Number 1, January 2008
Figure 2. A closer radiographic view of the area comprising the lower left third
molar and the eroded mandible.
an absence of magnesium in the dentine and decrease in the calciumphosphorus ratio in patients with scleroderma (15). This would allow
the root resorption to occur together with the bone resorptive process,
thus maintaining a minimal amount of trabecular and compact bone in
the area surrounding the distal root of the lower third molar. If the root
were maintained at its normal size and mandibular erosion continued,
the root apex would have become exposed, weakening the mandible.
The root resorption could be perceived to be a useful defense mechanism to maintain same normal bone structure and, thus, lessen the risk
of pathological fracture.
References
1. Ferri C, Valentini G, Cozzi F, et al. Systemic sclerosis: demographic, clinical and
serological features and survival in 1012 Italian patients. Medicine 2002;81:139 53.
2. Chaffee NR. CREST syndrome: clinical manifestations and dental management. J
Prosthodont 1998;7:155 60.
3. Mayes MD. Scleroderma epidemiology. Rheum Dis Clin North Am 2003;29:239 54.
4. Porter SR, Scully C. Oral manifestations of connective tissue diseases. Dent Update
2007 (in press).
5. Poole JL, Brewer C, Rossie K, Good CC, Conte C, Steen V. Factors related to oral
hygiene in persons with scleroderma. Int J Dent Hyg 2005;3:137.
6. Rout PG, Hamburger J, Potts AJC. Orofacial radiographic manifestations of systemic
sclerosis. Dentomaxillofac Radiol 1996;25:193 6.
7. Laux M, Abbott PV, Pajarola G, Nair PNR. Apical inflammatory root resorption: a
correlative radiographic and histological assessment. Int Endod J 2000;33:48393.
8. Pindborg JJ. Pathology of the dental hard tissues, 1st ed. Copenhagen: Munksgaard;
1970.
9. Lindskog S, Hammarstrm L. Evidence in favor of anti-invasion factor in cementum
or periodontal membrane of human teeth. Scand J Dent Res 1980;88:1613.
10. Gunraj MN. Dental root resorption. Oral Surg Oral Med Oral Pathol Oral Radiol
Endod 1999;88:64753.
11. Fuss Z, Tsesis I, Lin S. Root resorption: diagnosis, classification and treatment
choices based on stimulation factors. Dent Traumatol 2003;19:175 82.
12. Vier FV, Figueiredo JAP. Prevalence of different periapical lesions associated with
human teeth and their correlation with the presence and extension of apical external
root resorption. Int Endod J 2002;35:710 9.
13. Wood P, Rees JS. An unusual case of furcation external resorption. Int Endod J
2000;33:530 3.
14. Fayad S, Steffensen B. Root resorptions in a patient with hemifacial atrophy. J Endod
1994;20:299 303.
15. Knychalska-Karwan Z, Pawlicki R, Karwan T. Morphology and microanalysis of teeth
in scleroderma. Folia Histochem Cytobiol 1986;24:257 62.
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Case Report/Clinical Techniques

Root Resorption Associated with Mandibular Bone

cleroderma is a group of immunologically mediated connective tissue disorders,

Oral Medicine Unit, Pontifical Catholic University of Rio

JOE Volume 34, Number 1, January 2008

Case Report/Clinical Techniques

JOE Volume 34, Number 1, January 2008

Root Resorption and Mandibular Bone Erosion with Scleroderma

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