ABSTRACT ÖZET

Varicella-Zoster is a disease which causes varicella

(chickenpox) and herpes zoster (HZ). The prodro-

mal syndromes of HZ are burning, tingling, itching

occuring in the skin over the nerve distribution. Oral

vesicles usually appear after the skin manifestations.

The most common complication of HZ involving the

trigeminal nerve is a prolonged postherpetic neu-

ralgia. Rarely reported complications following oral

involvement with herpes zoster are devitalized teeth,

internal resorption and spontaneous exfoliation of the

teeth with osteomyelitis of the alveolar bone. This is

a case report of a patient with HZ infection of trige-

minal nerve involvement that resulted in mandibular

alveolar bone necrosis. Careful dental and radiological

examination is necessary to prevent unnecessary and

delayed treatment for the patient.

Herpes zoster, varisella ya da herpes zoster (HZ) has-

talığına yol açmaktadır. Hastalığın prodramal semp-

tomları deride oluşan ağrı ve kaşınmadır. Hastalığın

ağız içi belirtileri deri lezyonlarından sonra ortaya

çıkar. HZ’nin en sık rastlanılan komplikasyonu uzun

süre devam eden postherpetik nevraljidir. HZ’de daha

nadir görülen belirtiler dişlerin devitalize olması, inter-

nal rezorpsiyon ve dişlerin osteomyelitine bağlı dişle-

rin eksfoliyasyonudur. Bu olgu sunumunda trigeminal

sinir tutulumu olan hastanın HZ enfeksiyonunu taki-

ben oluşan mandibular alveol kemiğinin nekrozunun

sunulmaktadır. Hastalara gereksiz ve/veya gecikmiş

tedavi uygulamalarının engellenmesi için dikkatli ve

detaylı klinik ve radyografik değerlendirmelere ihtiyaç

duyulmaktadır.

Hacettepe Diş Hekimliği Fakültesi DergisiCilt: 33, Sayı: 1, Sayfa: 31-35, 2009

Mandibular Osteomyelitis FollowingTrigeminal Herpes Zoster Infection

Herpes Zoster Sonrası Gelişen Mandibular Osteomyelit Enfeksiyonu

*Dr.Erinç ÖnEM, *Dr.Esin AlpÖZ, *prof.Dr. Servet Kandemİr, **Dr.Cemal AkAy* Ege University Faculty of Dentistry Department of Oral Diagnosis and Radiology**Ege University Faculty of Dentistry Department of Oral and Maxillofacial Surgery

KEYWORDSHerpes zoster, osteomyelitis, postherpetic neuralgia

ANAHTAR KELİMELERHerpes zoster, osteomyelit, postherpetic nevralji

OLGU RAPORU (Case Report)

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INTRODUCTION

Varicella-Zoster is a disease which causes var-icella (chickenpox) and herpes zoster (HZ). HZ occurs after reactivation of latent Varicella-zoster virüs (VZV) in sensory ganglia, which lies dor-mant in the dorsal root ganglia of the spinal cord or the extramedullary cranial nerve ganglia of pa-tient with diminished immunity to the virus1-3.

The virus spreads along the nerves of the asso-ciated dermatome causing vesicular eruptions. HZ of the trigeminal nerve is associated with painful vesicles of the skin and oral mucosa of the af-fected branch of the nerve. Usually, oral vesicles appear after skin manifestations. Oral vesicles rupture and coalesce presenting as large mucosal erosions. Prodromal pain may ocur in the distri-bution of the trigeminal nerve some days before vesicular eruptions and this pain may mimick toothache or pulpitis4.

Patient with HZ infections usually progress through three stages: (a) prodromal stage (b) ac-tive stage (also called acute stage) and (c) chronic stage5,6.

The prodromal syndrome stage presents it-self as sensation described as burning, tingling, itching, boring, prickly or knife-like occuring in the skin over the nerve distribution. This usually proceedes the rash of the active stage by a few hours to several days5,6. Infections occur unilat-eraly almost exclusively in middle aged and elderly patient1. This infection characterized by the ap-pearance of vesicles that occur on the skin along the pathway of an involved sensory nerve usually are associated with severe pain7,8. When branches of the trigeminal nerve are involved, lesions may appear on the face, in the mouth, in the eye, or on the tongue8. Prodromal pain that occurs in the distribution of the trigeminal nerve several days before the vesicular eruptions may simulate pulpitis. Three or four days later, papules devel-op, which rapidly become vesicles7. Oral vesicles usually appear after the skin manifestations. The most common complication of HZ involving the trigeminal nerve is a prolonged postherpetic

neuralgia7,9. Rarely reported complications follow-ing oral involvement with herpes zoster are devi-talized teeth, internal resorption and spontaneous exfoliation of the teeth with osteomyelitis of the alveolar bone1,3,7.

Gonnet was the first in 1922 to describe al-veolar bone necrosis and tooth loss in associa-tionwith Herpes Zoster (HZ) infection.10 Osteo-necrosis following HZ infection often presents as painless exfoliation of teeth in the involved area. This occurs after the acute phase of the infection has subsided. The pathogenesis of the osteonecrosis is unclear although an alteration of the vascular supply to the affected bone has been postulated10,11.

Few reports on HZ infection and oral com-plications including osteonecrosis and tooth ex-foliation have been published4,12. This is a case report of a patient with HZ infection of trigemi-nal nerve involvement that resulted in mandibular alveolar bone necrosis.

The objectives of this paper are (i) to present a brief review of Herpes zoster (ii) to highlight the role of the dentist in diagnosis and management of HZ of the trigeminal nerve.

CASE REPORT

A 76-year old man previously diagnosed with Herpes zoster, was admitted to the Dental Clinic of Ege University (Figure 1). The clinical exami-nation revealed generalized hyperesthesia over the vesico-bullous lesions on the left trigeminal nerve. The alveolar process became exposed in the premolar area of left mandibulary bone (Fig-ure 2). The panoramic and periapical radiograph showed mandibular alveolar bone necrosis (Fig-ure 3).

The patient was placed on antibiotic therapy (Penicilin G) for 21 days. Following a week af-ter the antibiotic therapy, the sequestrum was removed and the left second incisor teeth was extracted (Figure 4). The histological diagnosis following the biopsy was reported as osteomyeli-tis (Figure 5).

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Follow-up controls and radiographs were tak-en 2 weeks later, showing complete resolution of the lesions (Figure 6,7). However, the patient still has persistent pain resembling postherpetic neuralgia.

DISCUSSION

Trigeminal nerve involvement in HZ is usually unilateral and limited to a single division, more often the ophtalmic division. Oral manifestations appear when the second (maxillary) or third (mandibular) trigeminal divisions are affected. Frequently the intraoral lesions are associated with cutaneous lesions affecting the correspond-ing area innervated by the affected sensory nerve as present in our case.

FIGURE 1

Photograph showing the preoperative clinical appearance of the patient with expansile lesion involving the maxilla.

FIGURE 2

Intraoral photograph showing the preoperative clinical appearance of the patient.

FIGURE 3

Preoperative panoramic and periapical radiograph demonstrating the lesion.

Fig 3 inset: Intra-operative photograph of the patient.

FIGURE 4

Macroscopic view of the sequestrum.

FIGURE 5

Microscopic view showing histological appearance of the lesion.

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The most debilitating complication of HZ is pain associated with acute neuritis and post-her-petic neuralgia7. In our case the patient’s main complaint was the post-herpetic neuralgia. On rare occasions, involvement of bone following trigeminal HZ has been reported. Review of lit-erature demonstrate that the common feature for all the cases is the unilateral involvement of bony structures confined to regions innervated by the affected nerve7.

Spontaneous exfoliation of teeth in the area innervated by the affected nerve has been re-ported. Some authors believe that this is an

early event occurring during the first 2 weeks of the infection while others consider this as a late complication that will occur between the third to twelfth week after the onset1,3,4. Loss of teeth is due to alveolar bone necrosis and to necrosis of the periodontal ligament. After tooth extrac-tion, healing of the periodontal tissues is usually slower than normal, and frequently fragments of necrotic bone remaining after the extraction need to be removed to preserve the height of the alveolar process3,4,7.

The HZ infection can occasionally cause os-teonecrosis, either by affecting the innervation of the periosteum or by a direct vasculotropic effect of the virus, both of which lead to altera-tion of the blood flow to the affected area. The role of vascular alteration in the development of osteonecrosis is further suppported by the fact that osteonecrosis usually occurs in patients with compromised vascularity because of aging, irra-diation, or chronic inflammation3,7,8. One of the earliest signs of osteonecrosis of the jaw bones associated with HZ infection is spontaneous ex-foliation of teeth prior to overt signs of osteone-crosis10. Further, it seems reasonable to assume that preexisting pulpal or periodontal inflamma-tory conditions have the potential to contribute to a greater probability of tooth exfoliation and bone necrosis3. This infection characterized by the appearance of vesicles that occur on the skin and mucous membrane along the pathway of an involved sensory nerve is usually associated with severe pain. Rarely reported complications fol-lowing oral involvement with herpes zoster are devitalized teeth, internal resorption and sponta-neous exfoliation of the teeth with osteomyelitis of the alveolar bone1-5,8,9. Routinely patients with HZ are seen by their physicians for treatment. However, the dentist is often involved in the initial diagnosis of this disease and therefore must be fa-miliar with its differential diagnosis. The diagno-sis of HZ is clear when the prodromal symptoms are present and the dermatomal vesicular rash is present. Careful history and dental examination usually rule out other pathology.

FIGURE 6

Intra-oral photograph of the patient after the operation.

FIGURE 7

Follow-up panoramic radiograph taken 2 weeks after the operation.

Fig 7 inset: Follow-up periapical radiograph taken 2 weeks after the operation.

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CONClUSION

This is a case report of a patient with HZ infec-tion of trigeminal nerve involvement that resulted in mandibular alveolar bone necrosis4,5,7,9,12. Her-pes zoster of the trigeminal nerve is a disease that falls within the diagnostic purview of all dentists and dental specialists. Thorough knowledge of this disease will prevent unnecessary and delayed treatment for the patient.10-13

REFERENCES

1. Meer S, Coleman H, Altini M, Alexander T. Mandibular osteomyelits and tooth exfoliation following zoster-CMV co-infection. Oral Surg Oral Med Oral Pathol 2006; 101: 70-75.

2. Folusa J, Vincent I, Kolude B. Herpes Zoster Infection of the Maxilla: Case Report. J Oral Maxillofac Surg 1999; 57: 1249-1251.

3. Mintz SM, Anavi Y. Maxillary osteomyelitis and spontaneous tooth exfoliaton after herpes zoster. Oral Surg Oral Med Oral Pathol 1992; 73:664-666.

4. Siwamogstham P, Kuansuwan C, Reichart PA. Herpes Zoster in HIV infection with osteonecrosis of the jaw and tooth exfoliation. Oral Diseases 2006; 12:500-505.

5. Strommen GL, Pucina F, Tight RR, Beck CL. Human infection with H. Zoster: etiology, pathophysiology, diagnosis, clinical course and treatment. Pharmacotheraphy 1988; 8:52-68

6. Carmichael JK. Treatment of H. Zoster and postherpetic neuralgia. American Family Practice 1991; 44:203-210.

7. Muto T, Tsuchiya H, Sato K, Kanazawa M. Tooth exfoliation and necrosis of the mandible-A rare complication following trigeminal herpes zoster: Report of case. J Oral Maxillofac Surg 1990; 48:1000-1003.

8. Mendieta C, Miranda J, Brunet LI, Gargallo J, Berini L. Alveolar bone necrosis and tooth evaluation foolowing herpes zoster ınfection: A review of the literature and case reports. J Periodontol 2005; 76:148-153.

9. Barrett A, Katelaris CH, Morris JGL, Schifter M. Zoster sine herpete of the trigeminal nerve. Oral Surg Oral Med Oral Pathol 1993;75:173-175.

10. Mostofi R, Robinson MH, Freije S. Spontaneous tooth exfoliaton and herpes zoster infection of the fifth cranial nerve.J Oral Maxillofac Surg 1987;45:264-266.

11. Millar EP, Troulis MJ. Herpes zoster of the trigeminal nerve: The dentists’ role in diagnosis and treatment. Canadian Dental Journal 1994; 60:450-453.

12. Owotade FJ, Ugboko VI, Kolude B. Herpes Zoster Infection of the Maxilla: Case Report. J Oral Maxillofac Surg 1999;57:1249-1251

13. Tidwell E, Hutson B, Burkhart N, Gutmann JL, Ellis CD. Herpes zoster of the trigeminal nerve third branch: a case report and review of the literature. International Endodontic Journal 1999; 32:61-66.

CORRESPONDING ADRESS

Dr. Esin ALPÖZEge University Faculty of Dentistry Department of Oral Diagnosis and Radiology, Bornova, İzmir 35100, Turkey

Telephone no: +90 232 3881081 Fax No: +90 232 3880325 e-mail address: esinalpz@yahoo.com

Geliş Tarihi : 11.11.2008 Received Date : 11 November 2008 Kabul Tarihi : 06.04.2009 Accepted Date : 06 April 2009