Case Report

A Familial Pattern of Multiple Idiopathic Cervical RootResorption in a Father and Son: A 22-Year Follow-Up

Anthony L. Neely* and Sara C. Gordon

Background: The etiology of idiopathic cervicalroot resorption has not been elucidated clearly. How-ever, the process has been linked to trauma, in-tracanal bleaching, and partial-thickness connectivetissue grafts.

Methods: This study describes a familial pattern ofmultiple idiopathic cervical root resorption in a fatherand son.

Results: The father was a healthy 63-year-oldwhite male who presented with the first resorption le-sion in 1983. Twenty-seven additional lesions wereidentified on 16 teeth over 22 years. Five teeth werelost as a result of extensive resorption. The son was ahealthy 43-year-old when a resorption lesion wasidentified in 1993. A lesion identified on another tooth12 years later resulted in extraction.

Conclusions: Close relatives of those affected bymultiple idiopathic cervical root resorption shouldbe examined carefully for cervical resorption. Thisstudy also showed that early treatment can preventor delay the need for extraction. J Periodontol 2007;78:367-371.

KEY WORDS

Cervical; familial; idiopathic; root resorption.

External cervical root resorption is an unusualand vexing problem in dentistry. It is isolatedto one tooth most often but can occur in

multiple sites. Although cervical resorption seems tobe rare, its prevalence is unknown and its etiologyhas not been elucidated clearly. However, the processhas been linked to trauma, intracanal bleaching,1-3

partial-thickness connective tissue graft placement,4

and fresh iliac crest grafts.5-7 Fuss et al.2 associatedexternal cervical root resorption with inflammationcaused by bacteria, but Frank8 demonstrated a lackof inflammation in extensively resorbed areas.

It is quite difficult for clinicians to identify and re-store lesions of cervical root resorption. Generally,lesions are found serendipitously on radiographs orduring clinical examinations when destruction oftooth structure already is advanced. Small lesionsor those on buccal or lingual/palatal surfaces may bedifficult or impossible to discern on radiographs orduring routine clinical examination. Goldberg et al.9

documented this problem in a study of simulated ex-ternal resorption lesions in central and lateral incisorsin human skulls. Small lesions (0.6 mm in diameter)on buccal surfaces could not be detected radiographi-cally, whereas between 74% and 78% of larger lesions(1.8 mm in diameter) could be detected on initial orrepeat examinations.

Sometimes, lesions are too extensive to treat andthe teeth must be extracted. Although lesions oftenprogress and/or recur despite intervention, somecan be halted for long periods of time with treat-ment.10 These lesions present a restorative challengebecause they are located subgingivally and/or inter-proximally, making them difficult to repair. Theycan be difficult to isolate and keep dry in a bloody field.Restorative options are limited because the lesionstypically are on dentin and cementum, surfaces thatare more difficult to bond with restorative materials.However, composite11 andglass ionomer materials10,12

have been used successfully to treat resorption le-sions. Although amalgam alloy also can be used ef-fectively, it presents an esthetic problem in anterior

* Department of Periodontology and Dental Hygiene, School of Dentistry,University of Detroit Mercy, Detroit, MI.

Department of Oral Medicine and Diagnostic Sciences, College ofDentistry, University of Illinois at Chicago, Chicago, IL. doi: 10.1902/jop.2007.060155

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regions because the grayish color can be transmittedthrough the gingiva.

The following cases document a familial pattern formultiple external idiopathic cervical root resorption ina father and son. The resorption lesions occurred over>22 years in the father and 12 years in the son. Al-though a familial pattern has been reported in multipleexternal apical root resorption,13-16 this is believed tobe the first report of a familial pattern for this type ofcervical resorption.

CASE DESCRIPTION AND RESULTS

Case 1The patient was a healthy 63-year-old white male. Hismedical history included vitiligo on the skin, coloncancer treated in 1992 by polypectomy, and a mildheart attack in 1993. He reported being treated forhypothyroidism from age 16 to 21. His wife and fourdaughters reported hypothyroidism treated with hor-mone replacement therapy. His son is presented incase 2.

This patient presented initially in 1983 for treat-ment of a cervical resorptive lesion that extended sub-gingivally on the mesial aspect of the distal root of thelower right third molar. The mesial root had been re-moved many years previously. The patient indicatedthat the reason for the resection had been an earlierepisode of root resorption, but this could not be con-firmed with the previous dentist. This lone molar rootserved as the distal abutment of a four-unit bridge thatextended to the second premolar. A full-thickness ac-cess flap was performed, and the lesion was inspectedand repaired with amalgam.

Because the patient reported a past history of hy-pothyroidism, a complete medical evaluation wasperformed, including an endocrinology evaluation andbone densitometry. Laboratory analysis included ion-ized calcium, urinary calcium, alkaline phosphatase,phosphorus, thyroid-stimulatinghormone,creatinine,serum electrolytes, and a complete blood cell countwith differential. All findings were within normal limits.

Many other resorption lesions were identified overtime. They varied with respect to size and location;nevertheless, some characteristics were similar. Alllesions are summarized in Table 1, and representativelesions are presented below to illustrate the nature ofthe defects and the diagnostic and clinical treatmentdilemma associated with each.

Figure 1 displays two typical resorption lesions onthe maxillary right first and second molars discoveredin 1994 on a routine periodontal maintenance exam-ination. The lesions were asymptomatic, and the pa-tient was unaware of their presence. Aside from theslight bluish color and rolled edematous marginalgingiva, there was no immediate clinical evidenceof underlying pathology (Fig. 1A). However, careful

Table 1.

Resorption Lesions Identified Over 22Years of Observation by Tooth, Surface,and Treatment Provided for Case 1

Tooth

Number

Surface

Involved

Year

Identified Treatment Rendered

6 Buccal 1983 Flap, composite

32 Mesial 1983 Flap, amalgam restoration

2 Buccal 1989 Flap, root canal therapy,amalgam

28 Lingual 1990 Extracted (1990)

32 Mesial* 1990 Extracted (1990)

2 Palatal* 1994 Flap, glass ionomer

3 Palatal 1994 Flap, glass ionomer

13 Buccal 1994 Flap, odontoplasty

14 Mesial 1994 Flap, glass ionomer, extractedfor other reasons (2005)

15 Mesial 1994 Flap, glass ionomer

18 Lingual 1995 Flap, extracted (1995)

19 Buccal 1996 Flap, glass ionomer

13 Buccal 2001 Flap, glass ionomer

19 Lingual 2001 Extracted (2001)

21 Buccal 2001 Referred, treatment unknown

22 Mesial 2001 Referred, treatment unknown

29 Lingual 2001 Extracted (2001)

6 Buccal* 2005 Flap, glass ionomer

6 Mesial* 2005 Flap, glass ionomer

7 Mesial 2005 Flap, glass ionomer

11 Buccal 2005 Flap, glass ionomer

12 Buccal 2005 Flap, glass ionomer

12 Mesial 2005 Flap, odontoplasty

13 Buccal* 2005 Flap, glass ionomer

13 Mesial* 2005 Flap, odontoplasty

13 Palatal* 2005 Flap, odontoplasty

21 Buccal* 2005 Flap, debrided, closed

22 Buccal* 2005 Flap, glass ionomer

Text in bold emphasizes the final fate of the tooth affected.* More than one episode of resorption.

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probing and evaluation with an explorer revealed de-fects below the gingival margin on the roots of bothmolars.Anaccessflapwasraisedtoevaluatethe lesionsand to determine restorability. Despite being large,neither of these lesions extended to the bony crest.The outline of the lesion on the first molar appearedregular and well circumscribed, whereas the secondmolar lesion was jagged and undermined enamel nearthe cemento-enamel junction. After thorough lesiondebridement, osseous recontouring was performed toexpose more tooth structure (Fig. 1C), followed by aglass ionomer restoration and flap closure (Fig. 1D).

Figure 2 shows asymptomatic lesions on the max-illary right canine and lateral incisor detected in 2005with a periodontal probe and explorer. Examinationwith an explorer revealed a large lesion on the facialaspect of the canine and the mesial aspect of the rightmaxillary lateral incisor. Probing depths around thelateral incisor and canine were 3 to 4 mm. A radio-graph of the area (Fig. 2B) showed a diffuse radiolu-cency at the cervical aspect of the canine, and, to alesser extent, on the mesial aspect of the lateral inci-sor. Figure 2C shows the appearance of the soft tissueoverlying the lesion upon initial flap reflection. It dis-plays a lack of overt inflammation. The lesion was ap-parent after complete degranulation of soft tissue(Fig. 2D) and osseous recontouring (Fig. 2E). It wasrepaired with a glass ionomer restoration; the flap

was replaced and sutured to prevent excessive reces-sion in the anterior region (Figs. 2F and 2G). As in allprevious surgical procedures, healing was uneventful.

Figure 3 illustrates the difficulty of identifying le-sions with radiographs alone. Although large lesionscan be seen easily on canine and first premolar teeth(Figs. 3A and 3B), incipient lesions cannot be identi-fied on maxillary first and second premolars (Fig. 3Cand Table 1). The maxillary premolar and mandibularcanine lesions were treated successfully with flap pro-cedures and glass ionomer restorations. The largelesion on the mandibular first premolar was too ex-tensive to be repaired after flap reflection, and willbe removed and replaced by a dental implant. Despitethe extensive nature of the lesion, the patient reportedno symptoms before or after flap surgery.

On two separate occasions, soft tissue immedi-ately overlying resorptive lesions was removed andsubmitted for evaluation by oral and maxillofacial pa-thologists. Histologic evaluation of soft tissue fragments

Figure 1.A) Lingual view of the maxillary right posterior sextant prior to flapreflection. Note that the gingiva appears normal overall with moderateinflammation at the cervical area of the first and second molar areas.B) Palatal view showing the character of the resorption defects on thefirst and second molars after soft tissue debridement. C). This viewshows the shape and extent of the defects after osseous resection(arrows show extent of lesions). A small piece of gutta percha fromprevious root canal therapy can be seen at apical portion of the defectof the second molar ( just below arrow on mesial side of tooth).D) Glass ionomer restorations visible after flap closure.

Figure 2.A) Clinical appearance of the maxillary canine and lateral incisor areain 2005 prior to clinical evaluation with periodontal probe and dentalexplorer. The examination revealed a resorption lesion on the buccalsurface of the canine and mesial surface of the lateral incisor.B) A radiograph of the area revealed evidence of extensive resorptionon the canine and a smaller lesion on the mesial of the lateralincisor. C) View of the soft tissue overlying the resorption lesion afterfull-thickness flap reflection. Note the close proximity of the soft tissuewith minimal overt inflammation. D) Resorption lesion clearly visibleimmediately after soft tissue removal. The lesion was hard and smoothto the explorer tine and did not involve the pulp. The outline of acomposite restoration placed in 1983 to repair a previous episode ofresorption is visible at the coronal aspect the current lesion. E) Canineand premolar area after osseous resection in preparation for arestoration. F) The lesions have been repaired on the canine andlateral incisor with glass ionomer restorations. G). The flap was closedto its original position to minimize gingival recession.

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overlying resorptive lesions on the buccal aspectof the maxillary left first and second premolars re-vealed chronically inflamed epithelial-lined connec-tive tissue. An oral and maxillofacial pathologist haddiagnosed another lesion, submitted years previ-ously, as chronically inflamed granulation tissue.

Case 2The second patient was the 43-year-old son of the pa-tient in case 1. The patient reported being healthy, had

no systemic health concerns, and took no medica-tions. He presented in 2005 with a history of two epi-sodes of external cervical root resorption treated byprevious clinicians.

Figure 4A shows the radiographic appearance ofthe maxillary right first molar in 1993, which seemsto show root resorption. The patients former dentistperformed root canal therapy followed by placementof a crown (Fig. 4B). The tooth has remained asymp-tomatic clinically and radiographically since treat-ment (Fig. 4C).

The maxillary left first molar (Fig. 4D) was ex-tracted after being diagnosed with cervical rootresorption by his dentist in 2005. Subsequent full-mouth clinical examination by the author (ALN) re-vealed no evidence of further root resorption. Becauseof a history of repeated resorption, the patient was ad-vised to undergo complete oral examination every 3months to identify any future lesions in an incipientand treatable stage.

DISCUSSION

This report presents two cases of multiple idiopathicexternal cervical root resorption presenting in a fatherand son over a 22-year period. To the knowledge ofthe authors, this is the first report of a familial patternfor this type of resorption.

The present cases seemed to be true idiopathic re-sorption because none of the usual factors associatedwith root resorption were found. A complete medicalexamination, including evaluation for endocrine dys-function and whole body bone scans of the father, re-vealed no abnormalities. The only unusual finding wasthat the father reported being treated for hypothyroid-ism as a teenager. However, laboratory evaluationsrevealed no current evidence of the condition. Theson was evaluated for endocrine dysfunction as achild and was found to be normal. He reported beingin good physical health at the time of the examination.He did not have dry skin, weight gain, lethargy, tongueenlargement, or other signs of myxedema. A youngerbrother also was tested for hypothyroidism and foundto be normal. His four sisters and mother had hypothy-roidism and were under treatment. No credible linkhas been established between cervical root resorptionand hypothyroidism. Although one coincident findingof hypothyroidism and extracanal invasive root re-sorption has been reported,17 no causal relationshipwas suggested or established.

Acute trauma and excessive orthodontic forcesare factors that also are associated with root resorp-tion.1-3 However, neither patient received orthodontictreatment during the period reported nor displayedevidence of occlusal trauma or parafunctional habits.Hence, it is not likely that either of these conditionscontributed to these findings.

Figure 3.A) Radiograph taken in 2005 of the lower left canine and firstpremolar revealed evidence of resorption lesions. The most extensivelesion was visible on the first premolar. B) This radiograph also showsthe extent of the resorption on the first premolar and absence of alesion on the second premolar. C) Clinically detectable resorptionlesions noted on the buccal and mesial of the maxillary first andbuccal, mesial, and palatal aspect of the second premolar were notvisible on this radiograph. The inability to identify the incipient lesionson radiographs illustrates the importance of thorough periodic clinicalexamination of all roots with an explorer and periodontal probe forsubjects with a history of root resorption.

Figure 4.A) Radiograph taken in 1994 of the upper right first molar of the43-year-old son of case 1 showing a large area of root resorption(asterisks). B) Upper right first molar after root canal therapy. Noclinical photographs available. C) Periapical radiograph of the upperright first molar taken in 2005. No additional changes have beennoted since the root canal was performed and the crown placed.D) Radiograph of the upper left first molar, also taken in 2005, showsa diffuse radiolucency just apical to the crown margin (asterisks).However, extensive root resorption was identified clinically by thepatients general dentist. The tooth was deemed non-restorable andwas extracted. Subsequent clinical examination by the author (ALN)revealed no evidence of resorption lesions.

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The resorption exhibited in case 1 displayed no par-ticular pattern of occurrence; all tooth types were af-fected, although the posterior teeth seemed to beaffected with greater frequency and earlier. However,not all tooth surfaces were affected equally. Of the 28surfaces on 16 teeth affected over 22 years, none oc-curred on a distal surface. This finding is unusual andcontrasts with other investigations that reported in-volvement of all surfaces.10

The most troublesome aspect of case 1 was the in-ability to predict which teeth would be affected andwhen. For instance, the first lesions were identifiedin 1983 but not again until 1989 and 1990. Four yearspassed until the next lesions were noted in 1994,1995, and 1996. No new lesions were detected until2001 and finally 4 years later in 2005. It is possibleand likely that some lesions were present for an unde-termined time before they were detected; the patientmoved in 1996 and was seen only periodically for ex-amination and treatment by the first author. Unfortu-nately, five of the 16 teeth (31.3%) with resorption hadto be extracted because the lesions had progressedtoo far to be repaired. Earlier identification and inter-vention may have delayed or prevented the need forextraction.

A major limitation of the findings in this article is theinherent problem of drawing conclusions from casereports. Because case reports contain only informa-tion gathered from the case(s) that present for evalu-ation, it is not possible to rule out factors other thanthose evaluated. For instance, factors not measuredin the study, such as environmental exposure and/orother endocrine or metabolic conditions, may havecontributed to the observed outcome. Although envi-ronmental exposure(s) seem unlikely because thefather and son had not resided in the same home orstate for >20 years, they cannot be ruled out. Whereasa latent effect of a similar prior environmental or otherexposure(s) may have had an effect on local or sys-temic health, it seems doubtful that the clinical mani-festationsofsuchexposure(s)wouldbeso intermittentand affect such a variable number of teeth.

CONCLUSIONS

These cases demonstrate a familial pattern for exter-nal cervical root resorption. Further study is needed tovalidate this finding and to determine whether there isa genetic predisposition to this condition. There is noevidence that endocrine dysfunction played a role inthese two cases. The results of this study indicatedthat close relatives of those affected by multiple idio-pathic cervical resorption lesions should be examinedcarefully. The results also showed that patients iden-tified with idiopathic cervical resorption must be fol-lowed closely because it can recur, spread rapidly,and affect any area of the mouth.

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Correspondence: Dr. Anthony L. Neely, Department ofPeriodontology and Dental Hygiene, School of Dentistry,University of Detroit Mercy, 8200 W. Outer Dr., Detroit, MI48219. Fax: 313/494-6666; e-mail: neelya@udmercy.edu.

Accepted for publication September 14, 2006.

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