MULTIPLE IDIOPATHIC CERVICAL ROOT

RESORPTION: A CASE REPORT

J. Wu1, L. Y. Lin, J. Yang, X. F. Chen, J. Y. Ge, J. R. Wu
& W. B. Sun

INTERNATIONAL ENDODONTIC
JOURNAL
• Introduction
• Root resorption is defined as progressive or transitory loss of dentine and cementum,
• which is a normal physiological process resulting in exfoliation of deciduous teeth. However,
• root resorption in permanent teeth is largely pathological and results in tooth loss.
• The classification of root resorption is difficult and complex, and various categories
• with subdivisions have been suggested based on the site and type of resorptive process,
• its clinical and histological appearance or the cause (Andreasen 1985, Tronstad
• 1988, Bakland 1992, Heithersay 2007, Darcey & Qualtrough 2013). The classification
• system proposed by Andreasen is most widely acknowledged (Darcey & Qualtrough
• 2013), according to which root resorption can be simply classified as internal and external.
• The latter is divided into three subcategories: surface resorption, inflammatory
• resorption and replacement resorption. Recent reviews have proposed a new category
• called ‘cervical root resorption’, which is different from other types of external root
• resorption (Makkes & Thoden van Velzen 1975, Heithersay 2004, Darcey & Qualtrough
• 2013).
• Cervical root resorption also referred to as invasive cervical resorption is a relatively
• uncommon, insidious and often aggressive form of external tooth resorption that occurs at
• the cement–enamel junction (CEJ), below the epithelial attachment (Tronstad 1988,
Heithersay
• 2004). Well-recognized causes of root resorption include orthodontic therapy, eruption
• of the underlying permanent dentition, benign and malignant neoplasms, chronic
• periodontal or periapical infections, Paget’s disease of the bone and trauma to the jaws.
• When none of these causes are present, root resorption at the CEJ is termed ‘idiopathic
• cervical root resorption’ (Fish 1941, Stafne & Slocumb 1944, Sullivan & Jolly 1957, Hopkins
• & Adams 1979, George & Miller 1986). Idiopathic cervical root resorption may affect multiple
• teeth (a minimum of three) in a condition known as multiple idiopathic cervical root
• resorption (MICRR) (Liang et al. 2003), which is also called ‘multiple idiopathic root
resorption’
• (Kerr et al. 1970) and ‘multiple idiopathic external resorption’ (Moody et al. 1990).
• Multiple idiopathic cervical root resorption can progress rapidly over a short time,
• which leads to a large irreversible loss of tooth structure. MICRR was first identified by
• Mueller & Rony (1930) and is rarely described in the literature. Table 1 shows reported
• cases of MICRR based on the references of Liang et al. (2003) and Iwamatsu-Kobayashi
• et al. (2005); two cases, one in which orthodontic treatment was given and one in
• which MICRR occurred in deciduous teeth, were excluded. Only approximately 30
• clearly identified cases have been reported (Neely & Gordon 2007, Liu 2008, von Arx
• et al. 2009, Arora et al. 2012, Roy et al. 2012, Haeberle 2013, Jiang et al. 2014).
Fourteen
• cases were reported between 1930 and 2000, and 16 cases after 2000. The
• mechanisms involved in MICRR are poorly understood, and this condition has been
• poorly managed.
• This article presents a rare severe case of MICRR in a healthy young adult female
• involving 29 teeth and leading to the loss of 23 teeth over a period of only 3 years.
• Case report
• Patient
• A healthy 27-year-old Chinese female was first referred to the
Department of Periodontology,
• Institute and Hospital of Stomatology Nanjing University
Medical School in February
• 2012. At the initial examination, she complained of severe
tenderness and
• increased mobility of her maxillary teeth; previous
orthopantomograms (OPGs) were
• available from November 2010 onwards.
• Medical and family/social history
• The patient was healthy with no record of systemic disease; her previous medical history
• revealed no significant causative factors. There was no history of similar problems
• in any of her maternal or paternal ancestors. The family had owned a cat for 5 years.
• Dental history and clinical assessment
• Overview
• In November 2010, the patient had consulted another hospital complaining of tenderness
• in the left mandibular teeth. An OPG taken at the time indicated extensive cervical
• radiolucent areas in teeth 32–35 (according to the FDI tooth numbering system), which
• were extracted. In May 2011 and September 2011, teeth 31, 41 and 36 were extracted
• for the same reason, and tooth 11 had fractured during chewing.
• No history of orthodontic therapy, bleaching, bruxism or periodontal treatment was
• reported. Tooth 11 had been fractured when she had a fall in 1993, after which she
• underwent intracoronal restoration in 2000.
• Clinical
• Clinical assessment
• Oral hygiene was poor, and generalized mild chronic gingivitis and gingival hyperplasia
• were noted. Teeth 11, 17, 18 and 36–41 had been extracted. Teeth 12, 14–16, 22, 24 and
• 25 were grossly mobile (Grade 2) and tender, and teeth 37, 46 and 47 had resin composite
• restorations. No other abnormalities were observed on visual or tactile examination.
• The patient underwent a complete haematological investigation including tests for
• cortisol, C-reactive protein, immunoglobulin A, immunoglobulin G, complement C3,
• complement C4, immunoglobulin M, adrenocorticotropic hormone, insulin, and antinuclear
• antibody levels, and relevant ionic (calcium and phosphorous), enzymatic (alkaline
• phosphatase) and endocrinal investigations (T3, T4 and parathyroid hormone). The test
• results were within normal limits.
• Between November 2010 and December 2013, 23 teeth were extracted or fractured
• because of root resorption. When the teeth were extracted, soft tissues including gingival
• tissue, granulation tissue at the CEJ and overlying resorptive lesions were removed
• and submitted for evaluation by oral and maxillofacial pathologists. The extracted crown
• was processed for scanning electron microscope (SEM) observation, and granulation
• tissue overlying the resorptive lesions was processed for transmission electron microscope
• (TEM) evaluation. Oral photographs (Figs 1–4), and OPG and cone beam computed
• tomography (CBCT) scans were obtained.
• Radiographic assessment (including CBCT)
• Orthopantomograms showed the extent and progression of resorptive lesions and tooth
• loss (Fig. 5). The OPG taken in May 2011 showed circumscribed radiolucent lesions in
• the cervical region of tooth 31; the mesial surfaces of teeth 12, 14, 18, 36 and 41; and
• the distal surfaces of tooth 17. Teeth 32–35 had been extracted. The OPG taken in February
• 2012 revealed circumferential cervical lesions in teeth 12–16, 21–25 and 42–45
• and the mesial surfaces of teeth 26, 37 and 46. Teeth 17, 18, 31, 36 and 41 had been
• extracted. The OPG taken in June 2012 illustrated that the resorptive lesions of teeth
• 43–46 had enlarged significantly; furthermore, teeth 15, 16, 24, 25 and 42 had been
• extracted. The OPG taken in November 2012 revealed that the distal surface of tooth
• 37 was involved and that teeth 12 and 43–45 had been extracted. The OPG taken in
• June 2013 (not shown) illustrated that 48 teeth were involved and that teeth 14, 22 and
• 46 had been extracted.
• A comparison of the OPGs taken over time showed a variable pattern of resorptive
• lesion progression. The resorptive lesion originated at the mesial or distal CEJ, usually
• progressed to involve the entire cervical region, and then extended not only into the
• crown but also into the root. The defects enlarged and encroached on the pulpal tissue;
• the height of the alveolar tissue was unchanged without attachment loss; most of the
• resorptive lesions increased in size rapidly (multiple teeth), whilst the size of others
• increased slowly (teeth 13, 23, 26 and 37).
• Cone beam computed tomography demonstrated that the lesions were generally larger
• than was evident from periapical films or OPGs. Buccal and lingual/palatal lesionswere
also more readily visible on CBCT images (Fig. 6). The extent of lesion progression
• was demonstrated by axial slice images of the cervical region. The lesion in tooth
• 21 began at the mesial or distal CEJ and progressed to involve the entire cervical
• region.
• Histological features
• The appearance of gingival tissue was consistent with chronic nonspecific gingivitis:
• hyperplastic gingival epithelium with mild chronic inflammatory cell infiltration was
• observed. The granulation tissue at the CEJ illustrated a hyperplastic mucosal epithelium
• with increased infiltration of capillary and mononuclear cells, mainly lymphocytes
• and plasma cells (Fig. 7). The resorptive lesion was filled with highly vascularized
• fibrous connective tissue consisting of numerous vacuolar structures, lymphocytes and
• mononucleated and multinucleated cells (Fig. 8).
• SEM and TEM findings
• Scanning electron microscope imaging (Fig. 9) showed that the inner surface of the
• crown had extensive areas with lacunar resorption; the borders of these worm-eaten
• lacunae were distinct and irregular. TEM imaging (Fig. 10) showed that the resorptive
• lesions had abundant lysosomes throughout the cytoplasm and heavy deposits of reaction
• products in variously sized lysosomes