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 Table of Contents  
CASE REPORT
Year : 2022  |  Volume : 24  |  Issue : 3  |  Page : 143-145

Herpes Zoster Duplex Symmetricus along with Ramsay Hunt Syndrome in an Acute Abdomen


1 Department of Dermatology, CH (SC), Pune, Maharashtra, India
2 Department of Dermatology, AFMC, Pune, Maharashtra, India

Date of Submission13-Oct-2020
Date of Decision02-Dec-2020
Date of Acceptance12-Dec-2020
Date of Web Publication21-Jan-2022

Correspondence Address:
Surg Lt Cdr (Dr) Siddharth Bhatt
Department of Dermatology, AFMC, Pune, Maharashtra
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jmms.jmms_152_20

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  Abstract 


Herpes zoster (HZ) duplex symmetricus is an extremely rare variant of multidermatomal HZ where there is the involvement of bilaterally symmetrical dermatomes. This generally occurs in immuno-compromised individuals and is unusual in immunocompetent host. When occurring over the face, it can be associated with Ramsay Hunt syndrome. Bilateral symmetrical lesions present a unique diagnostic dilemma owing to morphological similarities with Eczema Herpeticum and rarely enteroviral infections. In such cases, correct interpretation of serological tests helps in the diagnosis of the condition. Here, we present the case of HZ duplex symmetricus occurring in an immune-competent individual over bilateral V1 3 and C4 dermatome (left) along with facial palsy.

Keywords: Herpes zoster duplex symmetricus, multidermatomal herpes zoster, Ramsay Hunt syndrome, varicella zoster virus


How to cite this article:
Vashisht D, Pathania V, Bhatt S, Shah N. Herpes Zoster Duplex Symmetricus along with Ramsay Hunt Syndrome in an Acute Abdomen. J Mar Med Soc 2022;24, Suppl S1:143-5

How to cite this URL:
Vashisht D, Pathania V, Bhatt S, Shah N. Herpes Zoster Duplex Symmetricus along with Ramsay Hunt Syndrome in an Acute Abdomen. J Mar Med Soc [serial online] 2022 [cited 2022 Aug 9];24, Suppl S1:143-5. Available from: https://www.marinemedicalsociety.in/text.asp?2022/24/3/143/336185




  Introduction Top


Varicella zoster virus (VZV), a double-stranded DNA virus, causes chicken pox in childhood; thereafter stays dormant in the dorsal root ganglia/cranial nerve ganglia for remaining life. Reactivation occurs due to transient or persistent immunosuppression, wherein it travels along the axon and causes localized vesicular rash in a dermatomal distribution, called herpes zoster (HZ) or Shingles.

HZ is a self-limiting, localized infection in immunocompetent. Disseminated rash is generally seen with immunosuppression.[1]

Few lesions (<20) can occur in adjacent dermatomes, but extensive involvement is unusual in immunocompetent. Even rarer is the involvement of bilateral dermatomes which has a reported incidence of 0.1%.[1] Even among bilateral involvement, occurrence of lesions over symmetrical dermatome called HZ duplex symmetricus is an absolute rarity.[2] Bilateral HZ on the face poses diagnostic dilemma due to morphological similarities with eczema herpeticum and rarely enteroviral infections.[3]

Here, we present the case of HZ duplex symmetricus over the face with Ramsay Hunt syndrome in an immunocompetent individual with acute abdomen.


  Case Report Top


A 60-year-old male, known case of bilateral sensorineural hearing loss was admitted as a case of sub-acute intestinal obstruction and acute appendicitis. On the 2nd day of admission, he developed redness, burning, and deep throbbing pain over the lower lip and left cheek followed by the appearance of multiple grouped fluid-filled lesions over both sides of the face and nape of the neck (left). He also complained of ulceration inside the mouth associated with burning sensation and difficulty in eating.

Examination revealed multiple, grouped vesicles over erythematous base, few erosions and hemorrhagic crusts, over bilateral preauricular region, cheeks, bridge of the nose, forehead (right), eyelid (left), ala of the nose (left), chin, and nape of the neck (left) [Figure 1]. Oral mucosa showed multiple erosions and shallow ulcers on the hard palate on either sides [Figure 2]. Motor examination revealed weakness over frontalis, orbicularis oris, and buccinator on the left side. The weber test was centralized.
Figure 1: Involvement of bilateral cheeks, bridge of nose, forehead (right), eyelid (left), ala of nose (left) , chin and nape of the neck (left) with multiple grouped vesicle over erythematous base and few crusted erosions with hemorrhagic crusts

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Figure 2: Involvement of the hard palate on the both sides with multiple crusted erosions. Weakness on the left side appreciable

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Tzanck smear showed acantholytic cells and multinucleate giant cells [Figure 3]. Serology for VZV IgG showed titers of 1349 mIU/L on day 09 after infection which increased to 1518 mIU/L on day 25 after infection. VZV IgM titers were normal. Herpes simplex virus (HSV) serology was normal. Enzyme-linked immunosorbent assay human immunodeficiency virus-1 and 2 and COVID-19 reverse transcription polymerase chain reaction was negative. Contrast-enhanced computed tomography of the abdomen and pelvis revealed acute appendicitis and subacute intestinal obstruction. Malignancy workup was normal. Peripheral blood smear showed neutrophilia, shift to left, no atypical cells were noted. The patient was diagnosed as a case of HZ duplex symmetricus (B/L V1, V2, V3 and unilateral C4 dermatome [Left]) with Ramsay Hunt syndrome.
Figure 3: Tzanck smear from the vesicle showing acantholytic cells and multinucleate giant cells

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He was treated with injection Acyclovir 10 mg/kg 08 hourly for 7 days and later shifted to oral acyclovir 800 mg five times a day for further 7 days. Tablet prednisolone 60 mg OD for 10 days for facial nerve palsy in consultation with Otorhinolaryngologist was given. Patient responded favorably, cutaneous lesions started crusting by the 3rd day, and the facial weakness resolved completely at the end of therapy [Figure 4].
Figure 4: Resolution of lesions and facial weakness

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  Discussion Top


Multidermatomal HZ is the marker of immunosuppression and is rarely seen in immunocompetent individuals.[1] There are only four case reports of HZ duplex symmetricus of the face but none with Ramsay Hunt syndrome.[1]

Widespread and distinct structure involvement may result due to numerous anatomical interconnections between the spinal nerve, i.e., VII, VIII, IX, X, XI, XII, and upper cervical nerves referred to as spinal accessory nerve plexus, although this can have individual variations.[4]

Eczema herpeticum caused by HSV can have similar vesicular eruptions over the face, however with a preexisting dermatoses or atopic dermatitis. Interestingly, HSV is also associated with Bell's palsy.

Tzanck smear is an easy, bed side method of demonstrating acantholysis and multinucleate giant cells in the smear from the blister fluid. However, as similar cytology can be seen with HSV, differentiation cannot be based on it solely.

VZV IgG, IgM, and IgA antibodies appear 1 week after the appearance of rash and show highest titers at 1 month. Thereafter, IgM and IgA antibodies decrease rapidly within few months.[5] VZV specific IgG levels have a functional half-life of 50 years due to long-lived plasma cells.[6] Although the presence of IgM antibody along with a characteristic rash is diagnostic for reactivation, it has low sensitivity, cross-reacts with other herpes viruses, and has a high false-negative rate with high IgG titer.[5] VZV IgG titers at 3 weeks post-HZ are higher in participants with severe HZ, due to a higher antigen load, whereas VZV IgA and IgM titers are rarely increased after HZ.[6]

IgG avidity testing developed is an effective way to distinguish between primary infection and reactivation. IgG of individuals infected in the past binds with higher affinity with the antigen as compared to primary infection.[7]

Polymerase chain reaction is the most sensitive test for VZV detection, but its utility is limited due to high cost. Other methods employed in the research settings are viral culture and direct fluorescent antibody testing.[7]

HSV IgG become detectable 3–4 weeks after infection and then persist lifelong, whereas HSV IgM become detectable at 9–10 days and remain detectable up to a maximum of 6 weeks.[8] HSV IgM response also weakens with recurrent infections.[9]

VZV serology in our patient was done on day 09 and 25. Rising titer of IgG and normal titer of IgM were found, which conformed with the expected trend. HSV serology on day 9 showed normal IgM and IgG titers.

Ramsay Hunt syndrome is characterized by vesicles over ear canal, anterior two thirds of the tongue and hard palate, ipsilateral facial palsy, hearing loss, tinnitus, and vertigo due to involvement of vestibulocochlear nerve.[10] Facial palsy in Ramsay Hunt syndrome is most severe 1 week after occurrence and thereafter recovers gradually. Treatment started beyond 72 h can have permanent sequela;[10] hence, early diagnosis and prompt management is vital to prevent deformity.

This case is being reported for extremely rare presentation of varicella zoster, i.e., HZ duplex symmetricus along with Ramsay Hunt syndrome and highlights the correct interpretation of VZV serology.

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.
Dai S, Huang X, Chen Y, Wang M, Zheng H, Zeng K, et al. Bilateral asymmetrical herpes-zoster with Ramsay hunt syndrome in an immunocompetent adult. Virol J 2020;17:123.  Back to cited text no. 1
    
2.
Gambhir ML, Aggarwal Y, Pandey K, Malhotra SK. herpes zoster duplex symmetricus in an immunocompetent 70-year female. Our Dermatol Online 2014;5:306-7.  Back to cited text no. 2
    
3.
Mathes EF, Oza V, Frieden IJ, Cordoro KM, Yagi S, Howard R, et al. “Eczema coxsackium” and unusual cutaneous findings in an enterovirus outbreak. Pediatrics 2013;132:e149-57.  Back to cited text no. 3
    
4.
Hashemilar M, Ghabili K, Shoja MM, Savadi-Oskouei D, Keyvani H. Varicella-zoster virus reactivation from multiple ganglia: A case report. J Med Case Rep 2009;3:9134.  Back to cited text no. 4
    
5.
Min SW, Kim YS, Nahm FS, Yoo da H, Choi E, Lee PB, et al. The positive duration of varicella zoster immunoglobulin M antibody test in herpes zoster. Medicine (Baltimore) 2016;95:e4616.  Back to cited text no. 5
    
6.
Laing KJ, Ouwendijk WJD, Koelle DM, Verjans GMGM. Immunobiology of varicella-zoster virus infection. J Infect Dis 2018;218:S68-S74.  Back to cited text no. 6
    
7.
CDC. Cdc.gov. 2020. Chickenpox Interpreting Laboratory Tests Varicella [cited 2020 Jan 20]. Available from: https://www.cdc.gov/chickenpox/lab testing/ lab tests.html. [Last cited on 2020 Jan 20].  Back to cited text no. 7
    
8.
Page J, Taylor J, Tideman RL, Seifert C, Marks C, Cunningham A, et al. Is HSV serology useful for the management of first episode genital herpes? Sex Transm Infect 2003;79:276-9.  Back to cited text no. 8
    
9.
Kühn JE, Dunkler G, Munk K, Braun RW. Analysis of the IgM and IgG antibody response against herpes simplex virus type 1 (HSV-1) structural and nonstructural proteins. J Med Virol 1987;23:135-50.  Back to cited text no. 9
    
10.
Crouch AE, Andaloro C. Ramsay Hunt Syndrome. StatPearls Publishing; 2020 [cited 2020 Jan 20]. Available from: https://www.ncbi.nlm.nih.gov/books/ NBK557409/. [Last cited on 2020 Jan 20].  Back to cited text no. 10
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4]



 

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