|Year : 2015 | Volume
| Issue : 2 | Page : 214-219
|Dental complications of herpes zoster: Two case reports and review of literature
Swati Gupta1, V Sreenivasan2, Prashant B Patil3
1 Department of Oral Medicine and Radiology, Subharti Dental College and Hospital, Subhati University, Meerut, India
2 Department of Oral Medicine and Radiology, Saraswati-Dhanwantari Dental College and Hospital and Post Graduate Research Institute, Dr. Prafulla Patil Educational and Hospital Campus, Pathri Road, National Highway 222, Parbhani(M.S.), India
3 Department of Oral Medicine and Radiology, Navodaya Dental College, Mantralaya Road, Raichur, Karnataka, India
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|Date of Submission||04-Jan-2014|
|Date of Decision||19-Feb-2014|
|Date of Acceptance||10-Apr-2015|
|Date of Web Publication||22-Jun-2015|
| Abstract|| |
Herpes zoster (HZ) (shingles) results due to reactivation of varicella-zoster virus. Unusual dental complications like osteonecrosis, exfoliation of teeth, periodontitis, and calcified and devitalized pulps, periapical lesions, and resorption of roots as well as developmental anomalies such as irregular short roots and missing teeth may arise secondary to involvement of 2 nd or 3 rd division of trigeminal nerve by HZ. Such cases pose both a diagnostic as well as a therapeutic challenge. We report two such rare dental complications of HZ-spontaneous tooth exfoliation and osteonecrosis of the maxilla in a 70-year-old female patient; and multiple periapical pathoses affecting right half of the mandibular teeth in a 45-year-old female patient. Both the patients did not have any associated systemic illness. The aim of this paper was to compare the present cases with all the 46 cases of osteonecrosis and 6 cases of multiple periapical pathoses secondary to trigeminal zoster reported in literature till date The article also throws light that the occurrence of such dental complications of HZ is not entirely dependent on the immune status of the host.
Keywords: Complications, herpes zoster, osteonecrosis, periapical pathoses, spontaneous tooth exfoliation
|How to cite this article:|
Gupta S, Sreenivasan V, Patil PB. Dental complications of herpes zoster: Two case reports and review of literature. Indian J Dent Res 2015;26:214-9
Herpes zoster (HZ) is a well-known viral disease that usually presents as a painful unilateral vesicular rash restricted to the distribution of a sensory nerve.  Trigeminal nerve is the most commonly affected cranial nerve (18.5-22%). , In general, the incidence and burden of HZ complications other than postherpetic neuralgia (PHN) are poorly studied and consequently, reliable epidemiological information is scarce. An observational, retrospective analysis of 1401 HZ cases recorded by dermatologists and general practitioners in Italy showed that the most frequently occurring zoster-related complications, excluding PHN, were ocular complications (5.7%), and facial palsy (0.6%), with the risk increasing with age.  Reports of dental complications are even rarer. These are secondary to HZ of 2 nd and 3 rd division of the trigeminal nerve. These include osteonecrosis, exfoliation of teeth, periodontitis, and calcified and devitalized pulps, periapical lesions and resorption of roots, as well as developmental anomalies such as irregular short roots and missing teeth. 
|How to cite this URL:|
Gupta S, Sreenivasan V, Patil PB. Dental complications of herpes zoster: Two case reports and review of literature. Indian J Dent Res [serial online] 2015 [cited 2018 Nov 16];26:214-9. Available from: http://www.ijdr.in/text.asp?2015/26/2/214/159175
The aim of this paper was to review the literature regarding two such unusual complications secondary to trigeminal zoster - osteonecrosis and multiple periapical pathoses; and to further highlight their rarity with the help of clinical cases of each.
| Case Reports|| |
Case report 1
A 70-year-old female patient reported with burning pain in the right maxillary molar region since 2 months. Her history included a description of unilateral eruptions consistent with HZ of right maxillary division of trigeminal nerve which was accompanied by spontaneous exfoliation of a tooth.
Examination revealed persistent scarring and pigmentation on right side of the face. Right eyelids were swollen associated with some watery discharge from that eye [Figure 1]. There was a deep ulcerative lesion present in the upper right buccal vestibule from 13 to 18 with exposed alveolar bone covered with sloughing. Gingiva in relation to the involved teeth was red, edematous and enlarged. 16 was missing and 17 was grade III mobile [Figure 2] and [Figure 3].
|Figure 1: Profile photograph showing facial scarring and hyperpigmentation post-herpes zoster (case 1)|
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|Figure 2: Intraoral photograph showing deep ulcerative lesion present in upper right buccal vestibule from 13 to 18 (case 1)|
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|Figure 3: Intraoral photograph showing clinical findings in relation to 15, 17, and 18 with absent 16 (case 1)|
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Based on the history and clinical findings, we arrived at a diagnosis of spontaneous tooth exfoliation and alveolar osteonecrosis following HZ infection of the maxillary division of trigeminal nerve. A differential diagnosis of osteomyelitis was also considered.
Radiological examination revealed missing 16, hanging drop appearance of 17, and severe alveolar bone destruction in the 15-18 regions [Figure 4]. Generalized moderate bone loss was also seen. Medical referral and necessary investigations to rule out any underlying systemic condition was performed, which included routine hematological investigations, blood sugar investigation, posteroanterior chest X-ray, and ELISA for HIV. All test results were within normal acceptable limits and ELISA was negative. Patient refused to undergo any further investigation including an ophthalmic examination.
|Figure 4: Panoramic radiograph showing missing 16, hanging drop appearance of 17, and severe alveolar bone destruction in the 15-18 region (case 1)|
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Treatment included extraction of grade III mobile 17 and surgical debridement and removal of the necrotic bone.
Case report 2
A 45-year-old female patient was referred for evaluation of episodes of poorly localized pain and serial involvement of teeth by endodontic pathoses in right lower quadrant over a period of 10 years. There were no apparent cause such as caries, severe wasting disease of teeth (attrition/abrasion/erosion), and periodontal disease. The patient had a history of HZ of the right V3 division of trigeminal nerve before10 years.
On examination, 32 was missing. 31 and 41-46 teeth were previously endodontically treated. A temporary restoration was present in 47 [Figure 5].
|Figure 5: Intraoral photograph of mandibular arch showing the clinical findings in case 2|
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Radiological investigations [Figure 6] revealed adequately obturated root canals of 31, 41-46; and a radiopaque restoration in the crown of 47. Periapical changes were seen from 41 to 48 with blunting of root apex of 41-48.
|Figure 6: Panoramic radiograph showing adequately obturated root canals with periapical changes from 41 to 46 with blunting of root apex of 41-47 (case 2)|
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Routine investigations like complete blood count, erythrocyte sedimentation rate, alkaline phosphatase, serum calcium, serum phosphorus, liver function tests were done. All were within the reference range.
History, clinical and radiographic findings were all suggestive of multiple periapical pathologies and external root resorption limited to one quadrant secondary to HZ of right mandibular division of the trigeminal nerve. All other causes of odontogenic and chronic orofacial pain were considered and ruled out. No specific treatment was instituted because by the time patient was diagnosed to be suffering from this rare complication of HZ, almost all the teeth in that quadrant were already treated endodontically.
| Discussion|| |
Herpes zoster is a sporadic disease. In 1.5-3 of every 1,000 individuals, varicella-zoster virus (VZV) becomes reactivated, causing lesions of localized HZ. The nerves most commonly affected with HZ are C3, T5, L1, L2, and the first division of the trigeminal nerve. The incidence of HZ is more in elderly males and increases with age or degree of immunosuppression. It increases by 15-fold in HIV-infected patients than in uninfected individuals. ,, Triggering factors include stress or local trauma, diminished immune response or in association with malignancies, chronic hepatitis, polymyalgia rheumatica, acute rheumatic fever. , Individuals aged >65 years had almost four times the risk of complications of HZ when compared to those aged <35 years. Both of our patients were above 35 years of age. 
Various dental complications like osteonecrosis, exfoliation of teeth, periodontitis, and calcified and devitalized pulps, periapical lesions, and resorption of roots as well as developmental anomalies such as irregular short roots and missing teeth have been related to HZ.  Both of our patients suffered from HZ of the trigeminal nerve and presented with entirely different dental complications-osteonecrosis; and multiple periapical pathoses and resorption of roots. Neither of them had any associated major systemic illness.
Osseous alterations related to HZ were first reported by Rose in 1908, and in 1922 Gonnet was credited as the first to call attention to the alveolar bone necrosis and tooth loss associated with HZ infection.  So far, 46 cases of osteonecrosis of jaws [Table 1] and [Table 2] ,,,,,,,,,, have been reported without any predilection for maxilla or mandible. Slight male predilection was seen (21M: 15F) with a wide age range of 6-79 years and the mean age of 52 years. Our patient was 70-year-old, but was otherwise healthy as revealed by past history and the other necessary routine investigations. On analysis of the 46 cases, 20 [Table 1] had some form of underlying systemic diseases which included Hodgkins disease (3), reticulum cell sarcoma (1), diffuse histiocytic lymphoma (1), pharyngeal cancer (1), chronic hepatitis and anemia (1), polymyalgia rheumatic (1), acute rheumatic fever (1), leukemia (1), brain tumor diabetes-cytomegalovirus (CMV) infection (1), tuberculosis (1), pneumonia (1), HIV (1). Chemotherapy was associated with 5 such cases and radiotherapy was done in 4 of these, corticosteroids in one of them which could be a predisposing factor by diminishing the local resistance to bone necrosis.  Further analysis revealed that a more severe and fulminant course of this complication (average spontaneous exfoliating teeth [3.75 vs. 2.57]) was seen at a younger age (47 vs. 56) in those with associated systemic illness [Table 1] and [Table 2]. ,,,,,,,,,, We hereby suggest that the severity of this complication can be linked to the underlying systemic illness and not just the age.
|Table 1: Summation of reported cases of trigeminal herpes zoster associated with alveolar osteonecrosis and tooth exfoliation in immunocompetent patients |
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|Table 2: Summation of reported cases of trigeminal herpes zoster associated with alveolar osteonecrosis and tooth exfoliation in those with underlying systemic illness |
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Paradoxically, the number of cases reported was more in immune-competent individuals (26 vs. 20). This probably could either represent a misdiagnosis in immunocompromised individuals or a publication bias towards the publication of articles in immune-competent individuals. However, a genuine predilection for occurrence of this complication in otherwise healthy individuals cannot be ruled out.
An unusual finding in our 70-year-old elderly female patient was the involvement of eyes which can be attributed to the established rami communications between nasal branches of maxillary nerve with external nasal branches of the anterior ethmoidal nerve, branch of nasociliary (ophthalmic nerve). It has also been proposed that occasionally the lacrimal nerve is absent and is replaced by the zygomaticotemporal branch of the maxillary nerve, sometimes conversely. The clinical presentation of our case was very much similar to the case reported by Jain and Rathore. 
Some authors believe that the spontaneous exfoliation of teeth in the area innervated by the affected nerve is an early event occurring during the first 2 weeks of the infection as was seen in our case while others consider this to be a late complication that will occur between the 3 rd and 12 th weeks after onset [Table 1] and [Table 2]. ,,,,,,,,,, Poor oral hygiene and severe generalized periodontal bone loss might have contributed to the severity of the condition in our patient as was also observed in some of the reported cases. , Loss of bone is due to alveolar bone necrosis and/or to necrosis of periodontal ligament. , This necrosis could be a result of ischemia. Wright et al.  postulated that the necrosis could be a result of ischemia related to the infarction of vessels supplying the teeth. Direct invasion of blood vessels by virus spreading from adjacent cranial nerves and segmental granulomatous vasculitis, associated with HZ infection, with multifocal infarcts in the brain, and spinal cord have been reported. Considering the close anatomical relationship between the virus infected fifth cranial nerve branches and blood vessels, this vasculitis component may contribute to the infarction of vessels. 
In the literature review using the search terms - "zoster: Pulp" and " zoster: Periapical", six case reports were found [Table 3] , out of which only 2 showed periapical radiolucencies; multiple devitalization was seen in 2; 2 showed internal root resorption and one was associated with pulpless teeth. 3-8 years elapsed before these complications were reported post-HZ, mostly being reported in females. Wide age range (31-72 years) was also seen. Tooth resorption and periapical lesions as reported by Ramchandani and Mellor  in maxillary 24, 25, 26 was very similar to the present case where all mandibular teeth of 4 th quadrant showed periapical radiolucencies. Our second case also adds to this list of rare dental complication. All her right mandibular teeth were affected with multiple periapical pathoses and root resorption.
|Table 3: Summation of reported cases of pulpal - periapical pathoses as a complication of trigeminal herpes zoster |
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Role of viruses especially herpes virus in causing resorption of roots and such periapical pathoses are still subject to debate. Some implicate viruses to cause an increase in the virulence of bacterial pathogens by enhancing adherence and invasiveness into epithelial cells. Some suggest that the dental pulp could be adversely affected, or there may be injury to odontoblasts and degeneration by HZ. ,, Although some studies have confirmed that there may be an association present between Epstein-Barr virus, CMV and irreversible pulpitis and apical periodontitis, but not enough evidence is found in favor of association of VZV with such endodontic pathoses. ,,
Undoubtedly, the resorption and development of periapical pathoses in the second case is suggestive of role played by zoster virus in its pathogenesis; but whether pulpal involvement or root resorption was responsible for the episodes of poorly localized pain in the involved teeth still remains unclear.
The treatment plan for this complication is empirical and based on anecdotal experiences. It is not clear whether antiseptic irrigants applied through the root canal can reach and affect herpes viruses in the periapical tissue.  Antiviral medications have not been tried in any of the previous reported cases nor was it tried in the present case. A more clear insight into the etiopathogenesis of such complications may help us understand and treat the disease better.
Early diagnosis and prompt treatment of the HZ in the prodromal phase by the use of antiviral agents may aid in reducing the duration and the severity of pain associated, but complications may still occur. Although less reported, these dental complications may either be extremely distressing for the patient who loses teeth spontaneously like our first case or may pose a diagnostic challenge as in our second case.
| Acknowledgments|| |
We extend our sincere thanks to Dr. Nagaraju (Reader), Dr. Shirin Vashisth (Reader), Dr. Sumit Goel (Reader) and Dr. Kanika (Lecturer) for their support.
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Dr. Swati Gupta
Department of Oral Medicine and Radiology, Subharti Dental College and Hospital, Subhati University, Meerut
Source of Support: None, Conflict of Interest: None
[Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6]
[Table 1], [Table 2], [Table 3]
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