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KATHMANDU UNIVERSITY MEDICAL JOURNAL Necrotizing in Varicella Zoster Infection Lamichhane S, Humagain M, Subba M, Neupane M, Dawadi A

Department of , ABSTRACT Dhulikhel Hospital, Kathmandu University Hospital, Necrotizing are acute periodontal conditions which can Kathmandu University School of Medical Sciences, present with simple to more complex life threatening condition where Dhulikhel, Kavre, Nepal. it can perforate the skin of cheek. They are found in patients with severe illness, stress and reduced immunity. The prevalence of this whole group of disease is usually very low and is stated frequently as the first manifestation in HIV infection. Corresponding Author Herpes zoster, a common dermatological condition is a secondary infection due to Simant Lamichhane re-activation of Varicella zoster virus in a person previously acquiring a chicken pox Department of Periodontology, in his/her lifetime. There are limited scientific literatures citing the occurrence of necrotizing stomatitis in varicella zoster infection. Thus, here we present a detail Dhulikhel Hospital, Kathmandu University Hospital, about a case and management of 46 years old female who had a hospital stay of Kathmandu University School of Medical Sciences, over 5 days for treatment of Herpes zoster infection of mandibular nerve presented Dhulikhel, Kavre, Nepal. with necrotizing stomatitis within oral cavity. E-mail: [email protected] KEY WORDS Citation HIV, Necrotizing stomatitis, Varicella zoster infection

Lamichhane S, Humagain M, Subba M, Neupane M, Dawadi A. Necrotizing Stomatitis in Varicella Zoster Infection. Kathmandu Univ Med J. 2020;70(2):210-3.

INTRODUCTION Necrotizing stomatitis is a severe inflammatory condition skin infection which occurs in younger age group isa of the and the oral cavity in which soft tissue primary disease caused by VZV. Herpes zoster (HZ), or necrosis extends beyond the gingiva and bone denudation , is the painful eruption of a rash, usually unilateral. may occur through the alveolar mucosa, with larger areas Post herpetic neuralgia with tingling sensation is a common of osteitis and formation of bone sequestrum.1 It is one feature in HZ. HZ is caused by reactivation of the varicella of the variant of the broad category of disease called as zoster virus (VZV) which remained latent in the dorsal Necrotizing periodontal disease (NPD).2 It typically occurs ganglion cells of sensory nerves. HZ is a common disease in severely immune-compromised patients. In addition with a lifetime risk of 10-30% which increases to 50% to immunosuppression, , psychological among individuals ≥ 85 years.4 stress, insufficient sleep, inadequate , pre- existing gingivitis or periodontitis, tobacco consumption, alcoholism, age, seasonal variations, etc. are the suggested CASE REPORT etiology and risk factors for the development of3 NPDs. A 46 year old female patient reported to the Department Prevalence of necrotizing stomatitis are not estimated and of Periodontology, Kathmandu University School of are only limited to case reports whereas its predecessor Medical Sciences, Dhulikhel with a chief complaint of Necrotizing gingivitis prevalence in the general population gingival swelling in lower left back region of mouth since is estimated to be around 0.51 to 3.3% which is also not a 1 week. Medical history revealed that she had a hospital worldwide prevalence.3 stay for 5 days for management of Herpes zoster infection Chicken pox and Herpes zoster (HZ), both are caused by of mandibular nerve with dissemination. Laboratory Varicella zoster virus (VZV). Chicken pox, a very common investigations suggested that she was HIV seronegative.

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Her personal history revealed that she was a chronic current smoker (23.25 pack-years) and had a poor socio- economic background. Extra-oral examination revealed the presence of severely hyperpigmented patches which were present unilaterally on left side of the face. (fig. 1) On intraoral examination, her oral hygiene was poor, severely inflamed, edematous gingiva with spontaneous bleeding on slight provocation was noted along with exposure of alveolar bone with respect to 33 and 34. (fig. 2) The periodontal diagnosis was made as necrotizing stomatitis (Hornig and Cohen Stage Erythematous gingival margins 6) whose prevalence is estimated to be only 1% of total NPDs.5 Figure 3. 3% Hydrogen Figure 4. 2nd visit Intra-oral peroxide prepared from 6% . 2nd visit Patient conditions were evaluated. The gingival margins were erythematous without any superficial necrotic areas. The overt was dramatically reduced but exposing more amount of alveolar bone in the affected

area. (fig. 4) The area was irrigated using 3% H2O2. Patient was asked to follow the same instructions given previously Necrosis of interdental papilla and was recalled after 5 days.

Figure 2. Intra-oral photograph rd (First visit) 3 visit Figure 1. Extra-oral photograph Patient was re-evaluated and digital intra-oral periapical The case was managed using the standard treatment radiograph (IOPAR) was taken. (fig. 5) The IOPAR clearly protocol for necrotizing periodontal disease as follows: revealed the extensive destruction of bone identified as dead necrotic bone (sequestrum) which was more 1st visit radiopaque than surrounding bone involving till the apex The goals of the initial therapy followed at the first visit of number 35 and periodontal widening was noted only involves the treatment of involved area to reduce around 36. (fig. 6) Considering the rapid nature of the the bacterial load and removal of necrotic tissue so that disease, extraction of the affected tooth 34 and 35 along repair and regeneration of normal tissue barriers can be with sequestrectomy was planned. reestablished. The area was anesthetized by use of topical anesthetic (2% Lignocaine gel). Small cotton pellets were moistened in 3% hydrogen peroxide solution which delivers nascent oxygen to the area where anaerobic bacteria are believed to be predominant. Cotton pellets were intended for single use and any sweeping motion which could possibly transfer infection to adjacent non affected sites 6 was prevented. Increased loss of attachment Bony sequestrum Supragingival ultrasonic scaling was performed. Patient Figure 5. 3rd visit Intra-oral Figure 6. IOPAR X-ray was counseled about potential risk of future and she was asked to use only water and ultrasoft The area was anesthetized by use of mental nerve only for toothbrushing. Due to known facts of unavailability block technique with 2% Lignocaine hydrochloride and of 3% hydrogen peroxide oral mouthrinse in the country, 1:2,00,0000 epinephrine. Extraction of tooth 34 and 35

3% concentration was prepared by adding 45 ml 6% H2O2 was performed along with removal of sequestrum and was and 55 ml of distilled water in our department. (fig. 3) sent for histopathological examination. (fig. 7) The area 500 mg and 400 mg three times was sutured for healing with primary intention and recalled daily was prescribed for 1 week. Patient was instructed to after 10 days for suture removal. (fig. 8) take Ibuprofen and paracetamol combination analgesics whenever required and was asked to visit after 2 days in Follow up visits the department. Suture removal was done but the wound closure was not achieved primarily. The large defect was present

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a

Figure 7. Extraction of tooth Figure 8. Suturing at the along with sequestrectomy surgical day on the buccal aspect of the extracted site. (fig. 9) So, for further anticipation of restorative therapy on mind to gain keratinized tissue secondary suturing was performed and bleeding was intentionally induced. (fig.10) Histopathological slides confirmed the specimen was of dead bone where there was empty bony lacuna. In a viable bone, the osteocytes are present within the bony lacuna.7 (fig. 11)

b

Figure 13 a,b. 3D CBCT imaging at 6 months showing resolution of bony defects.

DISCUSSION

Figure 9. Extraction of tooth Figure 10. Extraction of tooth NPDs are more common in HIV seropositive patients and can along with sequestrectomy along with sequestrectomy be the 1st manifestations of HIV infection or an indication of immune deterioration. In HIV-positive patients, decreased peripheral CD4 lymphocytes are correlated with NPDs. The HIV patients with NPDs were 20.8 times more likely to have CD4+ cell counts below 200 cells/mm3 than patients without NPDs which suggests severe immune deficiency in HIV-NPDs patients.8 As with other periodontal diseases, the etiology for NPDs appears to be polymicrobial. The bacterial etiology of NPD (fusobacterium) was first Figure 12. Healing after 1 month demonstrated by Plaut in 1894 and Vincent in 1896 using electron microscopy. Since then, many bacteria and viruses have been isolated from NPDs lesion. Culture studies Empty lacunae of bone devoid of osteocytes identified P. intermedia, Treponema, Selenomonas and Figure 11. Histopathological Fusobacterium species and are considered as “constant slide showing dead necrotic flora” in NPD . The microbiota associated with NPD bone. in HIV is like that of periodontitis in non HIV patients, with some specific features, such as presence and invasion of Healing was better and greatly improved at 1 month Candida albicans, herpes viruses or superinfecting bacterial period. (fig. 12) Patient was kept on regular follow ups. species.3 Thus, in the recent past the role of Herpes virus Considering the possibility of implant therapy, 3D CBCT has been advocated for aggressive forms of periodontal (cone beam computed tomography) scans were performed disease. There are total of eight human herpes virus (HHV) after 6 month of tooth extraction. CBCT scan showed good of which varicella zoster is HHV-3. HHV-2 ( bone fill all around the defect area. However slight buccal 2) and HHV-3 (VZV) are isolated from severe periodontal collapse was seen at the crestal area. (fig. 13 a and b) Patient disease and the periodontal herpes virus total copy counts still complained of tingling sensation in the auricular region can exceed the total bacterial cell counts.9 The risk behind so was sent for evaluation to dermatology department and the presence of HHV in periodontal sites are : Studies was kept on close observation for rehabilitation with oral have found that the sites where HHV are isolated has implants.

Page 212 Case Note VOL. 18 | NO. 2 | ISSUE 70 | APRIL-JUNE 2020 more amount of periodontal destruction, can persist in prompt action to limit the periodontal destruction and slight periodontal sites for longer duration, can replicate in oral/ variation in management protocol can give us a chance to gingival epithelial cells and herpes-bacteria co-interaction offer patient a near-natural prosthetic rehabilitation. can pose an ongoing risk for disease progression.9,10 So, necrotizing stomatitis is a rare clinical entity which is seen ACKNOWLEDGEMENT once in a long while. Its close association with HIV patients is sometimes breached and seen in non-HIV patients as We would like to pay our sincere gratitude towards our well. The viral etiology of periodontal disease has time patient for her kind co-operation and Dr. Ritesh Sri, Assistant and again led the periodontal scientific committee towards professor in department of Oral pathology, KUSMS for finding the exact link of pathogenesis and formulation of the histopathological examination and providing us the management protocol for virus associated periodontal photographs of histopathological slides. disease. The aggressive nature of this disease demands

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