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BRIEF COMMUNICATION
Year : 2020  |  Volume : 8  |  Issue : 2  |  Page : 65-67

Herpes zoster ophthalmicus-related oculomotor palsy in a pediatric patient


1 Department of Pediatric Ophthalmology and Strabismus, Narayana Nethralaya-3, Bengaluru, Karnataka, India
2 Department of Cataract and Neuro-Ophthalmology, Narayana Nethralaya-3, Bengaluru, Karnataka, India
3 Department of Retina, Narayana Nethralaya-3, Bengaluru, Karnataka, India
4 Department of Cornea and Refractive Surgery, Narayana Nethralaya-3, Bengaluru, Karnataka, India

Date of Submission19-Mar-2019
Date of Decision04-Apr-2019
Date of Acceptance16-Sep-2019
Date of Web Publication2-Jul-2020

Correspondence Address:
Vimal Krishna Rajput
Department of Pediatric Ophthalmology and Strabismus, Narayana Nethralaya-3, 37 Castle Street, Ashok Nagar, Bengaluru - 560 025, Karnataka
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jcor.jcor_20_19

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  Abstract 


Herpes zoster ophthalmicus (HZO) is a rare form of shingles that typically presents with prodromal symptoms followed by rash along the V1 or V2 dermatomes. Associated ophthalmoplegia is even rarer. About 99% of adults with a history of chickenpox and up to 90% of those without have serology positive for varicella-zoster. Age and immune incompetence are the main risk factors. In the current report, we describe an immunocompetent 16-year-old girl who presented with pupil invoinvolving complete third nerve palsy associated with HZO. After an extensive literature search, we could not find any similar case reported before.

Keywords: Herpes zoster ophthalmicus, oculomotor nerve palsy, pediatric patient


How to cite this article:
Rajput VK, Aroor CD, Kumar AK, Bhandary P. Herpes zoster ophthalmicus-related oculomotor palsy in a pediatric patient. J Clin Ophthalmol Res 2020;8:65-7

How to cite this URL:
Rajput VK, Aroor CD, Kumar AK, Bhandary P. Herpes zoster ophthalmicus-related oculomotor palsy in a pediatric patient. J Clin Ophthalmol Res [serial online] 2020 [cited 2020 Aug 5];8:65-7. Available from: http://www.jcor.in/text.asp?2020/8/2/65/288842




  Introduction Top


Herpes zoster ophthalmicus (HZO) is a rare form of shingles that typically presents with prodromal symptoms followed by rash along the V1 or V2 dermatomes. Associated ophthalmoplegia is even rarer. About 99% of adults with a history of chicken pox and up to 90% of those without, have serology positive for varicella zoster. Age and immune incompetence are the main risk factors. In the current report, we describe an immunocompetent 16 year old girl who presented with pupil involving complete third nerve palsy associated with HZO. After an extensive literature search, we could not find any similar case reported before.


  Case Report Top


A 16-year-old girl presented to our neuro-ophthalmology clinic with drooping of the left upper eyelid and blurring of vision in the left eye for a week.

She was treated elsewhere for conjunctivitis 2 weeks earlier. However, when she developed a rash on the left side of her face 2 days later, she was diagnosed with herpes zoster ophthalmicus (HZO) and started on oral and topical acyclovir (Acivir 400 mg tablets, Cipla Ltd., three times a day and Acivir eye ointment 3% w/w, Cipla Ltd., five times a day). She was currently using acyclovir ointment five times a day and fluorometholone (0.1% w/v, Allergan India Pvt. Ltd.) and moxifloxacin (0.5% w/v, Cipla Ltd.) eye drops four times a day.

On examination, a rash in healing stage was seen on the left side of her face in the distribution of ophthalmic division of trigeminal nerve (V1), reaching up to the tip of her nose. Her right eye was essentially normal. The left eye showed complete ptosis [Figure 1]. On lifting the eyelid manually, visual acuity was recorded to be 6/12 (Snellen) which improved to 6/6 with pinhole. Color vision was normal. The left eye was in exotropic position. The conjunctiva was congested and had papillary reaction (secondary to use of topical acyclovir). Corneal sensations were absent, and cornea showed punctate epithelial erosions (PEEs). Pupil was 8 mm dilated [Figure 2], fixed, not reacting to light (direct, consensual, or near-reflex) with no relative afferent papillary defect. Extraocular examination [Figure 3] revealed limitation of elevation (−5), depression (−4), and adduction (−4); abduction and intorsion were present. Facial sensations were normal and equal on both sides. Fundus examination did not reveal any abnormality.
Figure 1: The profile picture of the patient showing the skin lesions of herpes zoster on the left side of the face in the dermatomal distribution of the trigeminal nerve and complete left eye ptosis. Note that Hutchinson sign is present

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Figure 2: The anterior segment photograph of the left eye demonstrates mid-dilated pupil

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Figure 3: External photographs of extraocular movements. Note the limitation of adduction, elevation, and depression in the left eye

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A provisional diagnosis of the pupil involving complete third nerve palsy secondary to herpes zoster (HZ) was made. Magnetic resonance angiography was advised to rule out compressive lesions. Complete blood count, sugar profile, and tests for human immunodeficiency virus (HIV), hepatitis C virus, and hepatitis B surface antigen were advised and were reported to be normal. In view of its typical clinical picture and financial constraints, polymerase chain reaction test or serological tests were not advised. Neuroimaging did not reveal any compressive lesion but showed signs of inflammation around left oculomotor nerve. In consultation with the pediatric neurologist, option of oral steroids under cover of antiviral was advised (prednisolone tablet 1 mg/kg/day, tapering by 10 mg/week, along with acyclovir tablet 400 mg three times a day).

The patient was lost to follow-up for 8 months. On review, we noted that she had taken oral prednisolone 1 mg/kg/day (Wysolone, Pfizer Ltd., 40 mg tapered over 4 weeks) under cover of oral acyclovir (Acivir 400 mg tablets, Cipla Ltd., three times a day). Now, the ptosis had partially improved (MRD1 of 2 mm). Cornea still showed diffuse PEEs with epithelial haze. The pupil was mid-dilated (4 mm). The eyes were central on Hirschberg. Extraocular movements were full, except elevation that showed −2 limitation. Cover test was orthophoric in primary and reading positions. Worth 4 dot test did not reveal diplopia or suppression of the left eye. A short course of low potent topical steroids and copious lubrication was advised for corneal involvement.


  Discussion Top


HZ is a common disease affecting elderly patients due to reduction in the level of T-cell immunity to varicella-zoster virus, following initial infection and senescence.[1] HZ occurs by reactivation of latent virus in the ganglions following primary infection with varicella-zoster and occurs mostly in thoracic and cranial sensory ganglia.[2],[3] In addition to sensory neurons, HZ may also affect motor neurons in rare cases.[4] When the ophthalmic division of the trigeminal nerve is affected, the condition is referred to as HZO which occurs in 10%–25% of HZ, with 50% of these cases infecting the eye.[2],[3]

Although not uncommon in adults, HZ is rarely found in children, having an incidence of 42:100,000 person-years.[5] HZO may cause extraocular muscle palsies of the third, fourth, and sixth cranial nerves in 11%–29% of patients.[2] The third nerve is the most common site among them and the fourth the least.[2],[3],[4] Complete ophthalmoplegia is a very rare complication of HZO; only few cases have been reported in the literature.[6],[7]

The extraocular muscle palsies usually appear 2–4 weeks after the rash but sometimes occur simultaneously with the rash or more than 4 weeks later.[4] Involvement of the tip of the nose (Hutchinson sign) is a strong predictor of ocular involvement. Our patient developed palsy 2 weeks after the rash that involved the tip of her nose.

The girl, in this case, did not have an underlying systemic disease, had not been vaccinated against chickenpox, and did not have a history of a priori chickenpox infection. Yet, she presented with an HZO, suggesting that sometimes she may have contracted a (mild or subclinical form of) varicella-zoster infection. Similar cases of HZO in immunocompetent children without a history of varicella vaccination have also been reported in the literature.[8],[9] Likewise, studies of zoster in pediatric patients demonstrate that childhood zoster is not always a harbinger of an underlying immunodeficiency, HIV infection, or malignancy.[5] HZO-related ophthalmoplegia in otherwise healthy pediatric patients has been only been reported as single isolated case of the sixth nerve palsy in contralateral eye [9] and trochlear nerve palsy.[10]

The prognosis of ophthalmoplegia is generally favorable. The duration of diplopia associated with ocular motor palsy was reported to be 2–23 months. However, 87.5% of diplopia cases recovered within 1 year.[2] Our patient though lost to routine follow-up was orthophoric in primary position when reviewed after 8 months. Ptosis, however, was only partially improved.


  Conclusion Top


The case described here may be the first to report a complete oculomotor nerve ophthalmoplegia with pupil involvement secondary to HZO in a pediatric age group. It has a favorable outcome, and antiviral agents and corticosteroids may be used to hasten the recovery process.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Cohen JI. Clinical practice: Herpes zoster. N Engl J Med 2013;369:255-63.  Back to cited text no. 1
    
2.
Shin HM, Lew H, Yun YS. A case of complete ophthalmoplegia in herpes zoster ophthalmicus. Korean J Ophthalmol 2005;19:302-4.  Back to cited text no. 2
    
3.
Delengocky T, Bui CM. Complete ophthalmoplegia with pupillary involvement as an initial clinical presentation of herpes zoster ophthalmicus. J Am Osteopath Assoc 2008;108:615-21.  Back to cited text no. 3
    
4.
Shin MK, Choi CP, Lee MH. A case of herpes zoster with abducens palsy. J Korean Med Sci 2007;22:905-7.  Back to cited text no. 4
    
5.
Soeteman M, Willems RP, Busari JO. Herpes zoster ophthalmicus in an otherwise healthy 2-year-old child. BMJ Case Rep 2012;2012. pii: bcr2012007015.  Back to cited text no. 5
    
6.
Binder NR, Holland GN, Hosea S, Silverberg ML. Herpes zoster ophthalmicus in an otherwise-healthy child. J AAPOS 2005;9:597-8.  Back to cited text no. 6
    
7.
Im M, Kim BJ, Seo YJ, Park JK, Lee JH. Complete ophthalmoplegia after herpes zoster. Clin Exp Dermatol 2007;32:162-4.  Back to cited text no. 7
    
8.
Sanjay S, Chan EW, Gopal L, Hegde SR, Chang BC. Complete unilateral ophthalmoplegia in herpes zoster ophthalmicus. J Neuroophthalmol 2009;29:325-37.  Back to cited text no. 8
    
9.
Liao W, Chu G, Hutnik CM. Herpes zoster ophthalmicus and sixth nerve palsy in a pediatric patient. Can J Ophthalmol 2007;42:152-3.  Back to cited text no. 9
    
10.
Ryu WY, Kim NY, Kwon YH, Ahn HB. Herpes zoster ophthalmicus with isolated trochlear nerve palsy in an otherwise healthy 13.year.old girl. J AAPOS 2014;18:193.5.  Back to cited text no. 10
    


    Figures

  [Figure 1], [Figure 2], [Figure 3]



 

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