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Mandibular osteonecrosis and dental exfoliation after trigeminal zoster in an HIV-infected patient: case report and review of the literature

Cloarec, Nicolasa; Zaegel-Faucher, Oliviaa; Bregigeon, Sylviea; Cano, Carla E.a; Chossegros, Cyrilb,d; Wajszczak, Benoitb; Poizot-Martin, Isabellea,c,d

doi: 10.1097/QAD.0000000000000122

aAPHM Sainte-Marguerite, Service d’immuno-hématologie clinique

bAPHM Timone, Service de stomatologie et chirurgie maxillo-faciale

cInserm, U912 (SESSTIM)

dAix-Marseille Univ, Marseille, France.

Correspondence to Nicolas Cloarec, MD, Service d’immuno-hématologie Clinique, CHU Sainte Marguerite, 270, Boulevard Sainte Marguerite, BP29, 13274 Marseille Cedex 9, France. Tel: +33 (0)4 91 74 56 96; fax: +33 (0)4 91 74 50 69; e-mail:

Received 4 September, 2013

Revised 18 October, 2013

Accepted 18 October, 2013

Herpes zoster is a classical concern among HIV-infected patients; its incidence can reach up to five cases per 100 persons per year [1].

Trigeminal nerve is a frequent localization (about 20% of cases) and its classical complications are postherpetic neuralgia, facial palsy, loss of earring and encephalitis.

Less well known are the maxillofacial complications including periodontitis, tooth exfoliation and osteonecrosis of jaw (ONJ) [2].

We report such a case of mandibular osteonecrosis with teeth exfoliation and make a review of the literature.

A 50-year-old HIV-infected male patient was hospitalized for an extensive facial zoster associated with a perforated otitis and peripheral facial palsy (Ramsay–Hunt syndrome).

He was diagnosed for HIV in 1989 and treated from 1994. He was staged C3 in the CDC classification. His CD4+ T cell count was 314 cells/μl (20%) and viral load was less than 50 copies/ml. Ongoing combined antiretroviral therapy (cART) was etravirine, darunavir boosted with ritonavir and tenofovir/emtricitabine.

Other medical records were an opioid substitution by oral buprenorphine and allergy to penicillin and cotrimoxazole. Clinically, the patient was apyretic, the skin rash was localized in the region linked to the right mandibular branch of the trigeminal nerve, otoscopy showed purulent flow and oral examination showed precarious bucco-dental status but no acute abscess or periodontitis.

The diagnosis of zoster was confirmed by genomic amplification of varicella zoster virus on peripheral blood samples and on cutaneous swab.

The patient received 7 days of intravenous aciclovir [10 mg/kg three times daily (t.i.d.)] relayed with oral valaciclovir (1000 mg t.i.d.) for an overall 15-day treatment.

He also received an antibiotic treatment by clindamycin.

Amitryptilin (20 mg t.i.d.) was prescribed in prevention of postzoster neuralgia and had to be majored up to 60 mg t.i.d. because of important dysesthesia.

Complete resolution of the skin lesions was obtained within 3 weeks.

Five weeks after the onset of the zoster, the patient experienced spontaneous, multiple (five) and nonalgic tooth exfoliation in the eruption territory.

Clinical examination revealed an ulcero-necrotic gingivitis in the right mandible territory. It was associated with bone exposure with no bleeding at contact. Teeth 43 and 44 were mobile and were finally spontaneously exfoliated a few days later.

Tomographic examination showed a well limited osteonecrosis next to the exfoliated teeth (Fig. 1).

Fig. 1

Fig. 1

A conservative treatment was decided and anti-VZV therapy by valacyclovir (1000 mg t.i.d.) was restarted for 15 more days.

A review of the literature allowed us to identify 45 cases in addition to the present case. The first case was reported in 1955 [3] and our case was, up to now, the third case associated with a Ramsay–Hunt syndrome.

Thirty-three cases occurred along the HIV/AIDS era, of which 14 (42%) were documented to be HIV-positive, nine were HIV-negative and, for 10 patients, the status was unknown.

The delay of appearance of ONJ after the zoster event was only reported in 14 cases, with a mean of 5 weeks (0.5–24).

Among the 25 cases seronegative or of unknown status, nine presented a severe immunocompromising condition but, for seven of them, there was no evidence of an underlying morbidity.

We missed information with respect to the determination of the specific roles of immune deficiency (HIV staging, cART) or the effect of zoster treatment on the onset of ONJ.

The mechanism of osteonecrosis during Herpes zoster infection is still debated and seems to be multifactorial.

Ischemic injury of alveolar bone could be due to a local vasculitis [4], vasoconstriction through the sympathetic innervations [5], mechanic compression of the alveolar artery by the swelled alveolar nerve [6] or a hypercoagulable state.

Nervous mechanisms could also be implicated, as there is an important role of innervations on regulation of bone mass [7].

Infectious process could play a role, either by direct odontoblast targeting by VZV or by superinfection with other agents (mostly by actinomyces species), and has been reported in other causes of ONJ [8].

With regard to prior oral condition, osteonecrosis of the jaw is a rare but disabling complication of trigeminal zoster. It is still poorly known and mostly reported in odontologic journals.

Its incidence seems to be higher in HIV-infected patients, but more data are needed to reach any conclusion on the precise roles of immune status and antiretroviral regimen.

Its pathogenia is still unclear but could include vascular, nervous and infectious factors.

Being aware of this possible complication could lead the clinician to extend the length of VZV treatment when facing a trigeminal zoster in a patient with risk factors or underlying comorbidity.

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This work was not supported by any grants from any institution.

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Conflicts of interest

There are no conflicts of interest.

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