Int J Oral-Med Sci 8(1):52-59, 2009 Case Report

An Unusual Case of Osteonecrosis and Spontaneous Tooth Exfoliation Following

Trigeminal Herpes Zoster in a HIV Seropositive Patient

Nagaraju Kamarthi,Guru Eriaiah Narasimha,and Ashok Lingappa

Department of Oral Medicine& Radiology Subharti Dental College,Subhartipuram,India Department of Oral Medicine& Radiology Vinayaka Mission’s Sankarachariyar Dental College,Salem,Tamilnadu

State,India Department of Oral Medicine& Radiology Bapuji Dental College,Davangere,India

Correspondence to: Kamarthi Nagaraju Oral lesions associated with human immunodeficiency virus(HIV)are

E-mail: drnagrani1977＠gmail.com often the first overt clinical features of HIV infection.Reports of HIV -associated herpes zoster infection with alveolar bone necrosis and

spontaneous tooth exfoliation are extremely rare,with only four cases

reported in the literature. The exact mechanism of osteonecrosis

induced by herpes zoster infection remains uncertain.An altered oral

environment, decreased host immunity, or immune reconstitution

inflammatory syndrome (IRIS) could further compromise the viable

periodontal status,leading to alveolar bone necrosis and tooth exfolia- tion.We report a case of herpes zoster infection involving two divi- sions of the trigeminal nerve in an HIV-seropositive patient who

further manifested with mandibular and maxillary osteonecrosis and

Keywords: spontaneous mandibular and maxillary tooth exfoliation.The occur-

HIV,herpes zoster,tooth exfoliation, rence of such infection in immunosuppressed individuals,its etiopath- immune reconstitution inflammatory ogenesis, and associated clinical features are briefly reviewed and syndrome (IRIS) discussed.

College and Hospital,Davangere,Karnataka,India, Herpes zoster is an acute viral infection of the with a burning sensation on the right side of his dorsal root ganglion of the spinal cord or extra- mouth of 15 days’duration and pain in the lower medullary cranial nerve ganglia due to reactivation right back tooth region beginning the previous day. of the varicella zoster virus and is characterized by He was apparently asymptomatic 15 days earlier, unilateral vesicular eruption localized to the skin when he developed an itching sensation of the skin and mucous membrane of the dermatome innervated overlying the right side of his face and the right by the affected nerve(1). buccal mucosa, followed by spontaneous develop- Oral manifestations of herpes zoster appear when ment of fever,rash,and vesicles over the right side of the mandibular or maxillary divisions of the the face that eventually ulcerated and became en- trigeminal nerve are affected(2).Herpes zoster infec- crusted.The condition was associated with difficulty tion has been commonly recorded in cases of human chewing and swallowing,a burning sensation of the immunodeficiency virus (HIV) infection; however, eyes,blurring of vision,and spontaneous exfoliation manifestations with extensive alveolar necrosis and of apparently firm teeth #27, #28, and #29 (right spontaneous tooth exfoliation are rare(3). mandible). The patient’s past medical, dental, and family

histories were insignificant. Physical examination

A 43-year-old male patient came to the Depart- revealed vesicles and pustules with crusting on the ment of Oral Medicine and Radiology,Bapuji Dental skin overlying the external ear and upper and lower Int J Oral-Med Sci 8(1):52-59, 2009

Fig.3. Intraoral photograph showing erosions of the right

hemipalate.

Fig.1. Clinical photograph showing hypopigmented, hyper- pigmented,and encrusted lesion areas of the right side of the face(frontal view).

Fig.4. Intraoral photograph showing erosions of the right

buccal mucosa in the retromolar area.

Fig.5. Intraoral photograph showing the alveoli with denud- ed,dry,and yellowish bone devoid of blood clots and missing

Fig.2. Clinical photograph (lateral view). teeth #27,#28,and #29. Int J Oral-Med Sci 8(1):52-59, 2009 lips superimposed over areas of healing with scar ity to heat and cold and the electric pulp vitality test formation.The lesions were confined to the distribu- showed negative responses by all teeth in the first tion of right trigeminal nerve,sparing the eyes and and fourth quadrants; intraoral periapical and pano- an area over the angle of the mandible.There was no ramic images revealed the outlines of the sockets of involvement of the tip of nose,conjunctiva,or other the exfoliated teeth with no evidence of sequestra- regions of the body(Figs.1 and 2). tion(Figs.6 and 7).Based on the history and clinical

Intraoral examination revealed poor oral hygiene features,a diagnosis of osteonecrosis and spontane- and halitosis with erosions over the right retromolar area and right hemipalate that were restricted to the midline (Figs.3 and 4).The alveolar bone missing teeth #27, #28,and #29 was denuded,dry,yellowish, and devoid of blood clots (Fig.5).Tests for sensitiv-

Fig.7. Panoramic images (orthopantomography) showing

mild interdental bone loss and exfoliated teeth #27, #28,and #29.

Fig.8. Clinical photograph at the second visit showing healed

Fig.6. IOPA radiographs showing mild interdental bone loss. cutaneous lesions. Int J Oral-Med Sci 8(1):52-59, 2009

Fig.9. Intraoral photograph at the second visit showing healed oral lesions. Fig.10. Intraoral photographs showing exposed maxillary

alveolar bone. ous tooth exfoliation following herpes zoster infec- tion of the right trigeminal nerve was made.Blood assays (enzyme-linked immunosorbent assay and

Western blot assay)revealed that the patient was seropositive for HIV antibodies. The patient was advised to practice strict oral hygiene measures with

0.02% chlorhexidine mouthwash and was prescribed

800 mg acyclovir taken daily for 1 week and 200 mg tetracycline taken four times daily for 5 days.The patient was further referred to a general physician for the treatment of HIV, where he was counseled and prescribed highly active antiretroviral therapy (HAART).

At the second visit 10 days later,the patient had Fig.11. Intraoral photographs showing exposed mandibular significant relief from the burning sensation and alveolar bone and sockets. tooth pain. There was complete remission of the cutaneous lesions with healing by scarring,hypopig- mentation, and hyperpigmentation (Fig.8)and hea- ling of the oral lesions(Fig.9).However,the teeth in the right maxilla and mandible were becoming pro- gressively mobile with spontaneous painless exfolia- tion of #7, #30,and #31,along with exposure of the alveolar bone and sockets (Figs.10-12). Exposed alveolar bone was removed and sent for histopath- ological investigations and the margins were filed and sutured. Histological sections showed bone se- questra with necrosis and chronic inflammatory cell infiltrate(Fig.13). Fig.12. Intraoral photographs showing exposed alveolar

Within one month,patient returned with a severe bone and sockets. Int J Oral-Med Sci 8(1):52-59, 2009

(18.5% to 22%),followed by the glossopharyngeal

nerve and hypoglossal nerve (5).Trigeminal nerve

involvement is usually unilateral and limited to a

single division.Oral manifestations appear when the

second or third trigeminal division is involved(2).In

our case,both the maxillary and mandibular divi- sions were involved, which was reflected both

intraorally and extraorally. Osseous alterations associated with herpes zoster

infections were first reported by Rose in 1908 (7). Gonnett is credited with being the first to draw

attention to herpes zoster infection-related alveolar

bone necrosis and tooth loss,in 1922(7).In a review Fig.13. Bone sequestra with necrosis and chronic inflamma- tory cell infiltrate (haematoxylin and eosin, magnification of the literature to 2005(8),39 previous reports of 10×). bone necrosis after herpes zoster reactivation with a

fairly common occurrence of spontaneous tooth ex- foliation have been reported and in the past 4 years, four new cases have been added to the literature(8- 11).One of these cases presented a co-infection with

herpes zoster and cytomegalovirus (8). With the

current case,a total of 44 cases of herpes zoster with

osteonecrosis have been reported. Involvement of

both jaws with exfoliation of the teeth in more than

one quadrant,which occurred in the present case,is

Fig.14. Panoramic images (orthopantomography)revealing extremely rare. the healing sockets of exfoliated teeth #7 and #27 through #31. An increased frequency of herpes zoster infection lancinating pain over the right side of his face,espe- has been seen in cases of HIV infection.Reports of a cially along the lower border of the mandible,which similar manifestation in a seropositive individual was suggestive of post-herpetic neuralgia. He was have previously been reported only three times(3,11, prescribed 100 mg carbamazepine (Tegretol) taken 12); our case is the fourth. twice daily and 500μg Diacobal(vitamin B)taken The exact mechanism by which herpes zoster twice daily for one month. Following this regimen, causes bone necrosis is still unclear.Various hypoth- the patient had temporary relief within four weeks. eses concerning the pathogenesis of herpes zoster- Healing of the sockets was remarkable and radio- induced osteonecrosis have been postulated.Hall et graphs showed healing alveolar bone(Fig.14). al.suggested that decreased local host resistance,in

the form of host irradiation obliterative endarteritis, may contribute to subsequent inflammatory and

The incidence of herpes zoster infection in the necrotic alterations due to avascular necrosis (13). general population has been reported to be 5.4% (2, Garty et al.(14)however disagreed,especially in the

4).Herpes zoster infection typically occurs in individ- maxilla due to its rich vascular supply.Decreased uals older than 45 years of age, with the highest host resistance fails to explain the localized effect to incidence among persons 60 to 90 years old (5, 6). the jaws.Cooper et al.(15)postulated that adverse

Herpes zoster usually affects the cranial nerves; the tissue reactions, possibly aided by local chronic trigeminal nerve is the most frequently affected inflammatory changes in the form of advanced per- Int J Oral-Med Sci 8(1):52-59, 2009 iodontal diseases and delayed healing of extraction patient first experienced exfoliation of the man- sockets,resulted in bone necrosis and tooth exfolia- dibular teeth and later, after he was started on tion.Wright et al.(7)postulated that local vasculitis HAART therapy, experienced exfoliation of the caused by a direct extension of the neural inflamma- maxillary teeth and the early occurrence of post- tory process to the adjacent blood vessels leads to herpetic neuralgia. In the present case, we believe infarction of the trigeminal vessels that accompany that a correlation existed between IRIS and herpes the trigeminal nerve supplying the jaws,leading to zoster infection in an HIV-seropositive patient, ischemic necrosis of the periodontal ligament and which led to bone necrosis and tooth exfoliation in alveolar bone, which is well described in cases of more than one quadrant. herpes zoster infection involving the central nervous Knowledge of complications related to this partic- system.Mintz and Anavi postulated that denervation ular viral infection is necessary for comprehensive of the bones resulted in bone necrosis and tooth patient care. Here are the signs, symptoms, and exfoliation (16),which seems unlikely. radiographic and histological features of herpes

Arikawa et al.(17)suggested that osteonecrosis in zoster infection with oral manifestations. herpes zoster infection is probably due to compres- Symptoms of osteonecrosis include fetor oris,deep sion of the alveolar artery due to edema caused by boring pain,exposure of bone,mobility and painless the inflammation of the alveolar nerve in the narrow exfoliation of teeth, and trismus in cases of super- maxillary or mandibular canal,resulting in ischemia imposed secondary infection. Signs include denuda- and subsequent necrosis of the periodontal ligament tion of alveolar bone that does not appear viable, and alveolar bone. Systemic viral infection can recession of underlying soft tissues,mobility of teeth, injure odontoblasts and cause degenerative tissue soft tissue abscess,and persistently draining sinuses changes that result in pulp necrosis(18). due to secondary infection (1,3,7,12-17,21).

The theory put forward by Wright (7),which sug- Radiographic features may vary from clearly vis- gests that vasculitis following viremia from adjacent ible outlines of the sockets of exfoliated teeth to no nerves in the alveolar bone leads to avascular ne- demarcations of bone fragments adjacent to the crosis,appears to be more appropriate.This initial remaining teeth to well demarcated sequestra from infection, however, in the majority of cases is not the surrounding healthy bone (1, 3, 7, 12-17, 21). sufficient to cause bone necrosis and tooth exfolia- Histopathological sections of bony sequestra usually tion, but contributing pre-disposing factors that reveal portions of necrotic bone tissue in the region reduce local and systemic host resistance may cause of the bone marrow space and may show purulent these aggravated responses(7). exudates with bacterial colonies or superficial fungal

Immune reconstitution inflammatory syndrome invasion in cases of secondary infection(1,3,7,12-17, (IRIS)is defined as that circumstance in which pre- 21). existing subclinical or mildly symptomatic infections This condition is managed mainly by a conserva- or inflammatory conditions undergo paradoxical tive approach: good oral hygiene instructions, fre- worsening with a substantial increase in inflamma- quent irrigation of the wound, and antibiotics to tion during the initial months of host immunorecon- control secondary infections and prevent serious stitution(19).This condition is an immunopathogenic complications (13).Sequestrectomy with extraction or hyperinflammatory response to a pathogen that of the involved teeth would be an ideal treatment, was already there as the host immune response is because the prognosis in these patients would be being rapidly reconstituted (19).Ortega et al. (20) remarkable(21). reported a case of mandibular osteonecrosis in a HIV Mintz and Anavi(16,22)have used a closed nasal

-seropositive patient who was on HAART therapy, vestibular double portal drainage system for the signifying the effects of IRIS.In the present case,the drainage and irrigation of the suppurative maxillary Int J Oral-Med Sci 8(1):52-59, 2009 bone.Two vertical incisions were made,one in the submucosally until it was through the nasal ves- unattached mucosa in the canine fossa and the sec- tibular hole.The nonperforated segment of the drain ond at the zygomaticomaxillary buttress.The inci- was inserted first so that it lay extramucosally,while sions were made down to the bone and a subperios- the perforated segment was in the subperiosteal teal tunnel joining them was created with a perios- tunnel against the maxilla (Fig.16).Two catheter teal elevator. The subperiosteal tunneling was adaptors were attached to the lumena of the drain extended anteromedially along the anterior maxillar- tubes. With this drainage system in place, the ne- y wall and the nasal vestibule was penetrated with a crotic maxillary alveolar bone was then irrigated hemostat. A 0.7×20-cm (Fig.15)three-fourths per- with 2 gm cefazolin in 1000 ml of normal saline for 2 forated,flat silicone drain was inserted through the weeks. nostril and brought through the subperiosteal tunnel. An extramucosal U-turn was then made around the distal incision; the drain was brought forward The exact mechanism of osteonecrosis induced by through the anterior incision and then advanced herpes zoster infection remains unclear.It is possible

that poor oral hygiene coupled with decreased host

immunity or IRIS could further compromise an

already fragile periodontal ligament status, thus

leading to alveolar bone necrosis and tooth exfolia- tion. Prompt antibiotic and antiviral medication along

with oral hygiene measures may help to control bone

Fig.15. The nasal vestibular drainage system,consisting of a destruction.More studies are needed to fully under-

0.7×20-cm,three-fourths perforated,flat silicone drain with stand the pathophysiology and complications as- two adaptors.Arrows denote the perforated and nonperforat- ed segments.(Courtesy of: Mintz SM,Anavi Y.J Oral Maxil- sociated with this viral infection for improved pre- lofacial Surg,48: 1344-1345,1990) vention,early detection,and management.

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