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Journal of Case Reports
Corneal Involvement in Herpes Zoster Maxillaris: A Rare Occurrence
Shovna Dash, Gayatree Mohanty
Department of Ophthalmology, Kalinga Institute of Medical Sciences & Pradyumna Bal Memorial Hospital, Bhubaneswar, Odisha, India.
Corresponding Author:
Dr. Gayatree Mohanty 
Email: mgayatree@gmail.com
Received: 03-MAY-2020 Accepted: 20-JUL-2020 Published Online: 20-AUG-2020
DOI: http://dx.doi.org/10.17659/01.2020.0045
Abstract
Background: Herpes zoster ophthalmicus occurs due to reactivation  of  latent  varicella-zoster  virus  in  the  trigeminal  ganglion,  commonly  involving  the  ophthalmic  division.  To  the  best  of  our  knowledge,  ocular  involvement  in  zoster  cases  affecting  the  maxillary  division  have  been  rarely  reported. Case Report: We  present  two  young  females  with  herpes  zoster  ophthalmicus  involving  the  maxillary  division  with  corneal  involvement. Conclusion: All  cases  of  zoster  infection  should  be  referred  to  an  ophthalmologist  for  early  intervention  and  prevent  sequelae  like  corneal  opacity.  Maxillary  zoster  may  sometimes  precede  ophthalmic  zoster  and  zoster  ophthalmicus  may  occur  without  skin  rashes,  involving  only  the  cornea  or  iris.
Keywords : Cornea, Corneal Opacity, Herpes Zoster Ophthalmicus, Ophthalmologists, Varicella Zoster Virus Infection.
Introduction

Herpes  zoster  ophthalmicus  is  a  neurocutaneous  disorder  caused  by  the  Human  Herpes  Virus-3,  the  same  virus  that  causes  varicella.  Approximately  13%  of  the  patients  present  with  infections  involving  any  of  the  three  branches  of  the  trigeminal  nerve.  However  involvement  of  the  maxillary  division is  quite  rare,  approximately  seen  in  1.7%  of  the  cases.  We  come  across  innumerable  cases  of  herpes  zoster  infection  in  our  clinic  and   are  well-versed  with  it.  It’s  easy  to  spot  prototype  cases  of  herpes  zoster  ophthalmicus  but  it  is  rare  to  encounter  cases  with  only  the  maxillary  division  being  involved  and  detect  ocular  involvement.  Here  is  a  brief  report  of  two  young  females  with  similar  findings.
Our   hospital  based  observational  study  has  been  approved  by  the  Institutional  Ethics  Committee  according  to  the  tenets  of  the  declaration  of  Helsinki. Written  informed  consent  was  taken  from  the  patient  for  collection  of  his/her  details  and  images.

Case Report

Two  young  females   presented  with  complaints  of  decreased  vision  in  left  eye  with  vesicular  eruptions  over  their  left  cheek,  side  of  nose  and  upper  lip.  Both  patients  had  multiple  eruptions  in  various  stages  on  the  left  side  of  face,  over  the  malar  eminence, side  of  nose,  tip of nose, upper  lip  and  near  the  angle  of  mouth. There  was  mild  lower  lid  edema  and  ciliary  congestion  in  the  left  eye  in  both  the  cases. A corneal sensation of the left eye was diminished.  Fluorescein staining was negative in both cases.  Ocular movements and intraocular pressure   were normal.
Our first case had  a  diffuse  patch  of  anterior  stromal  infiltration  in  the  lower  one-third  of  the  cornea  without  epithelial  involvement  and   best corrected  visual  acuity  6/6(p)   and   6/18 [Fig.1]. In second case, a  limbal  phlyctenular  lesion  was  associated  with  conjunctival  congestion  at  nine  o’clock  position  and  best  corrected  visual  acuity  6/6  and   6/12(p) [Fig.2].




A clinical diagnosis of maxillary zoster  was made based on the typical, sharp limitation of vesicular eruptions within a specific area of neural  distribution. There was no oral mucosal ulceration. Otorhinolaryngology and dental examination supported the diagnosis of maxillary zoster.  Routine investigations were done. Blood sugar was normal.  HIV and syphilis were ruled out by serological tests. Oral  acyclovir  800 mg  5 times  a  day for 7 days was prescribed, supplemented  by  NSAIDs. Antibiotic drops were prescribed along  with  antibiotic  ointment  for  the  lesions.

Discussion

Herpes  zoster  ophthalmicus  occurs  due  to  reactivation  of  latent  varicella-zoster  virus (VZV)  in  the  trigeminal  ganglion.  The   reactivated virus has a predilection for  the  ophthalmic  division  of  the  trigeminal  nerve [1]. The  ophthalmic  division  of  the  trigeminal  nerve  is  20  times  more  commonly  involved  than  the  maxillary  or  mandibular  nerves.  This  case  report  is  regarding  herpes  zoster  infection  of  the  maxillary  nerve,  which  is  quite  rare,  approximately  seen  in  1.7%  of  the  cases [2,3].
The  ophthalmic  division  of  the  trigeminal  nerve  divides  into  three  main  branches:  the  frontal   nerve,  the  lacrimal  nerve  and  the  nasociliary  nerve.  The  nasociliary  branch  innervates  the  ciliary  body,  iris,  cornea  and  conjunctiva.  Its  terminal  branch  is  the  anterior  ethmoidal nerve,  which  innervates  the  sides  of  the  tip  of  the  nose (alae nasae)  via  the  external  nasal  nerve.  Hutchinson  observed  that  ocular  involvement  is  more  commonly  seen  if  the  herpes zoster opthalmicus  rash  involves  the  alae  nasae [4]. Up to 85% of such cases may develop  ocular  involvement [5]. If  Hutchinson’s  sign  is  absent  ocular  involvement  is  less  likely,  however  can  still  occur  and  is  likely  to  be  present  if  the  patient  reports  reduced  vision,  eye  pain  or  photophobia.  Additionally, reduced corneal sensation is a useful sign of ocular involvement.  Thus,  urgent  consultation  with  an  ophthalmologist  is  indispensable  in  all  cases  of  herpes  zoster  with  Hutchinson  sign.  Early treatment can prevent ophthalmologic complications and post-herpetic neuralgia [6].
In  the  present  cases,  the  corneal  involvement  can  be  attributed  to transverion of  zoster  virus    by   direct  neural  connection  and/or  by  direct  spread  through  the  tissues  to  other  nerves.  There  are  established  rami  communications  between  nasal  branches  of  maxillary  nerve  with  external  nasal  branches  of  the  anterior  ethmoidal  nerve  (branch  of  nasociliary  nerve).  These  neuronal  connections  may  explain  the  involvement  of  cornea  with  isolated  maxillary  zoster.   Occasionally  lacrimal  nerve  is  absent  and  is  replaced  by  zygomatico-temporal  branch  of  maxillary  nerve,  sometimes  conversely [7].

Conclusion 

In  our  cases  there  could  be  a  possibility  of  malinnervation  of  cornea  with  a  branch  of  the  maxillary  nerve.  Maxillary  zoster  may  sometimes  precede  ophthalmic  zoster  and  zoster  ophthalmicus  may  occur  without  skin  rashes,  involving  only  the  cornea  or  iris.  All cases  of  zoster  infestation  should  be  referred   to  an  ophthalmologist  to  prevent  sequelae   like  corneal  opacity.

Contributors: SD: manuscript writing and the conception of the work; GM was responsible for revising it critically. SD will act as a study guarantor. All authors approved the final version of this manuscript and are responsible for all aspects of this study.
Funding: None; Competing interests: None stated.

References
  1. Ostler HB, Thygeson P. The ocular manifestations of herpes zoster, varicella, infectious mononucleosis and cytomegalovirus disease.  Surv Ophthalmol. 1976;21:148.
  2. Patil S, Srinivas K, Reddy SBH, Gupta M.  Prodromal herpes zoster mimicking odontalgia- A diagnostic challenge.  Ethiop J Health Sci. 2013;23:73-77.  
  3. Tandon MP, Verma SK.  Herpes zoster maxillaries:  A case report.  Indian J Ophthalmol. 1987;35:160-161. 
  4. Hutchinson J.  A  clinical  report  on  herpes  zoster  frontalis  ophthalmicus  (shingles  affecting the  forehead  and  nose).  R Lond Ophthalmic Hosp Rep. 1865;5:191-215.
  5. Zaal MJ, Völker-Dieben HJ, D’Amaro J. Prognostic value of Hutchinson’s sign in acute herpes zoster ophthalmicus. Graefes Arch Clin Exp Ophthalmol. 2003;241:187-191.
  6. Matsuura H, Senoo A.  Herpes zoster and Hutchinson’s sign. QJM: An International Journal of Medicine. 2018;111:483.
  7. Jain S, Rathore MK.  Maxillary zoster with corneal involvement.  Indian J Ophthalmol. 2004;52:323.
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Dash S, Mohanty GCorneal Involvement in Herpes Zoster Maxillaris: A Rare Occurrence.JCR 2020;10:170-172
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Dash S, Mohanty GCorneal Involvement in Herpes Zoster Maxillaris: A Rare Occurrence.JCR [serial online] 2020[cited 2020 Nov 29];10:170-172. Available from: http://www.casereports.in/articles/10/3/Corneal-Involvement-in-Herpes-Zoster-Maxillaris.html
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