Herpes Simplex Neuralgia

History: A 48-year-old woman was seen because of her concern about a lesion on the left buttock.  She is worried that she had gotten a spider bite six months prior.  It healed but she has had two similar episodes since then.  

The patient is in her usual state of health.  Her only medication is sertraline.  She says she is not under any stress but I know that she has a 20-year-old daughter with psychiatric disease who is a great concern for her.   She has one sexual partner who is asymptomatic.   

O/E:  On examination, the patient has a cluster of resolving vesicular lesions on the left buttock. 

Images: (Photos taken by patient and two weeks after acyclovir)

Further history reveals that she has seen a neurologist for sciatic symptoms down the left leg extending to the foot and has been diagnosed with sciatica, meralgia paresthetica and small fiber neuropathy.  She was prescribed gabapentin for this (which she discontinued).

IMPRESSION:  I believe she has recurrent sacral herpes simplex.  This has also called been called "herpes buttockalis" in Iraq, and "herpes okolealis" in Hawaii. Some of these patients have secodary neurological symptoms.

I discussed this with her and prescribed acyclovir 400 mg t.i.d. which she will take for a month or so and then the dose will be lowered depending on symptoms.

Discussion: This patient interested me, because I saw a similar case in the mid 1980s and researched the topic at that time.  He was in his 60s and had a history of recurrent sciatica and urinary obstruction.  He’d been worked up by neurology, orthopedics and urology.  One day, he came in with sacral HSV and anamnesis revealed that this had been going on a few times a years and seemed to be related to his recurrent HSV. I found the article by a Layzer and Conant¹.  When they wrote it, acyclovir was not on the market, but it was in 1985².  I placed that patient on the then new acyclovir and his chronic neuropathy and urinary symptoms improved.  I have always remembered this man.

 

This recent patient is similar.  I did a Pubmed search on "Sciatica Herpes simplex" and found only 8 hits.  Most old.

I suspect this may be an under-reported entity that is worth discussing.  This woman likely underwent an unnecessary neurological work-up and now is labeled now with "small fiber neuropathy."  It was recently reported in the Online Journal of Community and Patient-Centered Dermatology and Our Dermatology Online.

 

References:

1. R B Layzer, M A Conant. Neuralgia in recurrent herpes simplex. Arch Neurol. 1974 Oct;31(4):233-7.

2. Oral form of acyclovir approved. FDA Drug Bull. 1985 Apr;15(1):3-4.

3. Herpes Okolealis.  OJCPCD August 2015

4.  Sharquie K. et. al. Herpes simplex (Buttockalis) of the buttock is a variant of herpes simplex genitalis Khalifa E. Our Dermatology Online 2020;11(e):e170.1-e170.5.

A Young Girl with Ulcerated Lips

A 14-yr-old girl had severe blisters on the lower lip of 5 days duration.  It was painful and developed into superficial painful ulcerations of the lips.  Then she experienced eye discomfort with eye discharge esp in early morning.  There were no red eyes though.  She did not have any fever or any polyarthralgia.  No genital ulcerations. She is a secondary school student and stays with parents with no unusual habits. There was no family history of similar illness.  There was no recent drug history including OTC products, supplements and traditional chinese medicines. 

Examination was unremarkable except superficial ulcerations on the lower lip and to a certain extent on the upper lip too. The ulcerations was covered with yellowish slough and crusts.  Superficial erosions were noted on the inner buccal mucosa. No genital ulcerations.  No blisters elsewhere. 

Rest of exam unremarkable.

Diagnosis

Aphthous ulcerations - severe

Differentials considered were First episode orolabial HSV infection, drug eruptions, pemphigus vulgaris, erythema multiforme.

Blood counts and biochemistry was done as well as ANA serology.  HSV I and II serology was not done due to financial reasons. If she does not improve I think this patient may require a biopsy.

She was treated with oral prednisolone 20mg bd, topical triamcinolone gel bd and oral azithromycin 250mg daily. Your comments on this patient would be highly appreciated.

References:

1. Mucosal erosions as the presenting symptom in erythema multiforme: a case report. Spencer S, Buhary T, Coulson I, Gayed S. Br J Gen Pract. 2016 Mar;66(644):e222-4.  Free Full Text.

Follow up: Good response to treatment with oral prednisolone and azithromycin. Lesions were drying up and clearing.

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Herpes Zoster in a 10 year-old

The patient is an otherwise healthy ten year-old boy with a two day history of grouped zosteriform vesicles on the left arm, anterior shoulder and upper back.  He has mild discomfort.  No known illnesses and on no medication.  He had two immunizations for varicella at the approopriate ages.

Observation:  All of the grouped vesicles appeared to be of uniform size.

Lab:  A Tzanck smear was positive for multinucleated giant cells.

Diagnosis:  Herpes zoster roughly C3 - 6.

Herpes zoster in children is unusual but not all that rare.  There are a few cases of HZ after vaccination for varicella.  As he felt well, and as the effect of specific antiviral therapy is not striking; after discussion with his mother it was elected to simply follow.  In out opinion, in healthy children and young adults the course of HZ is usually relatively mild and almost never followed by post-herpetic neuralgia.  Immunity seems to wear off over time and it appears that this attenuated vaccine is capable of causing H.Z.  The other possibility is that this is zosteriform simplex.  We did not culture for that. 
While researching this case, we looked up "zosteriform simplex."  An observation (ref 4) indicates that in these patients the vesicles are of uniform size (as we see here).  This would tilt us towards a diagnosis of Zosteriform Herpes Simplex here, and not of vaccine failure.  Should this child get a recurrence, that would clinch the diagnosis.

Reference:
1. Herpes zoster in children.

Peterson N, Goodman S, Peterson M. Cutis. 2016 Aug;98(2):94-5.

Abstract: Herpes zoster (HZ) in immunocompetent children is quite uncommon. Initial exposure to the varicella-zoster virus (VZV) may be from a wild-type or vaccine-related strain. Either strain may cause a latent infection and subsequent eruption of HZ. We present a case of HZ in a 15-month-old boy after receiving the varicella vaccination at 12 months of age. A review of the literature regarding the incidence, clinical characteristics, and diagnosis of HZ in children also is provided.

 

2. Herpes zoster and zosteriform herpes simplex virus infections in immunocompetent adults.

Kalman CM, Laskin OL. Am J Med. 1986 Nov;81(5):775-8.

Abstract: Among 111 immunocompetent patients referred to a general hospital setting with the clinical diagnosis of herpes zoster, viral cultures were obtained from 47 patients. Six of these patients (13 percent) had herpes simplex virus isolated, with four of the six infections involving the facial distribution, and the other two involving the T4 (breast) distribution. Excluding those in whom herpes simplex virus was isolated, the mean age (+/- SD) of the remaining 105 patients was 50 +/- 19 years. Thirty-two percent of the patients were at least 65 years old; however, 39 percent were younger than 40 years of age. Thus, herpes zoster frequently occurs in young, immunocompetent adults. Also, since zosteriform rashes may be caused by herpes simplex virus, viral cultures of lesions are useful to differentiate infections caused by herpes simplex virus from those due to varicella-zoster virus. The need to distinguish between these two viruses may be important with the advent of antiviral drugs and for use of the proper epidemiologic isolation procedures.

3. Varicella Vaccine (Wiki)
Vaccines are less effective among high-risk patients, as well as being more dangerous because they contain attenuated live virus. In a study performed on children with an impaired immune system, 30% had lost the antibody after five years, and 8% had already caught wild chickenpox in that five-year period.

4. Zosteriform herpes simplex and herpes zoster: A clinical clue

Sanath Aithal, Sheela Kuruvila, and Satyaki Ganguly. Indian Dermatol Online J. 2013 Oct-Dec; 4(4): 369.  Free Full Text.

Excerpt: An important clinical observation by many authors that the vesicles of herpes simplex are uniform in size in contrast to the vesicles seen in herpes zoster, which vary in size. In other words, vesicles of herpes simplex are uniform within a cluster.

Sacral Herpes Simplex

The patient is a 77-year-old woman who presents for evaluation of a recurrent localized blistering eruption on the right buttock.  This has happened off and on for 2-3 years.  Before this, she noticed a pain in the right buttock to hip that was attributed to some form of trauma and has had physical therapy for the pain.

EXAMINATION:  The examination shows grouped vesicles on an erythematous base on the right buttock. 

Clinical Picture:

Lab: Tzanck smear was positive for multinucleated giant cells. 

IMPRESSION:  Sacral herpes simplex.  Her buttock and hip pain may be related.  

PLAN:  Acyclovir 400 mg three times a day for seven to ten days.  If her hip pain improves, I would continue the acyclovir for a few months at 400 mg twice to three times a day to see if that impacts the chronic hip pain for which she has had physical therapy without much relief.

Discussion:  Sacral herpes simplex is seen with some regularity, although it has not been well-studied.  In 1974, Lenzer and Conant mentioned sciatica with sacral herpes simplex. I have seen a few memorable cases over the years.  One, in particular was a 70 yo man with sciatica and urinary symptoms that resolved completely when his recurrent sacral HSV was treated with acyclovir and he was maintained on suppressive therapy.

Patient reports:  I completed the full ten day regimen of acyclovir with apparent success - healing of the lesion and elimination  of the ache in my buttock which I had thought was a lingering result of the fall that I had almost two years ago. I am wondering if I should continue with prophylactic use of the acyclovir.

References:

1. Neuralgia in Recurrent Herpes Simplex

Robert B. Layzer, MD; Marcus A. Conant, MD

Arch Neurol. 1974;31(4):233-237.

ABSTRACT: Five patients with recurrent herpes simplex of the skin had unusual neuralgic pains preceding the eruptions by 24 hours or more. Although prodromal neuralgia is an uncommon feature of recurrent herpes, about 15 similar cases have been reported previously. The pain is often diffuse and aching in character and, in contrast with herpes zoster, leaves no sensory or motor deficit. Stereotyped cycles of pain and herpes simplex may occur repeatedly for as long as 20 years. The fact that pain precedes the eruption supports the theory that a persistent latent infection of sensory ganglia is activated during recurrences of herpes simplex.

2. [Recurrent herpes with neuralgia and zones of cutaneous hypoesthesia].

[Article in French]

de la Sayette V, er. Al

Abstract: A 52-year old man presented with recurrent Herpes simplex of the thigh and buttock of 30 years duration. The skin eruption was preceded by pain and sciatica. Surgical excision of the skin area involved modified the site of recurrence. During an attack, the patient developed severe pain and hypoaesthesia in the left half of his chest. The skin lesions were unmodified, and a type 2 Herpes simplex virus was isolated from a vesicle. A clinical examination performed 5 weeks later showed reduced sensitivity to pin prick in the previously painful D5 to D12 territory. Three points are of interest in this case: the site of recurrence moved after surgical excision, pain extended over a wide area and, most of all, persistent hypoaesthesia occurred during a recurrence.

3. Although this review (below) does not mentione HSV neuropathy, I suppose it belongs in this group.

Infectious neuropathies.

Sindic CJ1. Curr Opin Neurol. 2013 Oct;26(5):510-5

Abstract

PURPOSE OF REVIEW: Infectious neuropathies are heterogeneous neuropathies with multiple causes. They still represent an important world health burden and some of them have no current available therapy.

RECENT FINDINGS: Leprosy incidence has decreased by 50% during the last years, but leprosy-related neuropathies still cause severe disability. The pure neuritic leprosy is a diagnostic challenge that may require nerve biopsy or nerve aspiration cytology. The treatment itself may lead to a 'reversal reaction', which further causes injuries to the nerve. HCV-related neuropathies may be related or not to the presence of cryoglobulins. The absence of vasculitis, the most frequent form is a peripheral sensory neuropathy involving small nerve fibers, and more accurately diagnosed by pain-related evoked potentials. HIV-related neuropathy has become the major neurological complication of HIV infection. Both HIV-induced neuropathy and antiretroviral toxic neuropathy are clinically indistinguishable. The existence of an isolated chronic polyneuropathy due to Borrelia burgdorferi remains highly controversial. Lastly, an active infectious ganglioneuritis caused by varicella zoster virus, producing shingles, is the most frequent infectious neuropathy in the world and may cause various neurological complications. Zoster sine herpete remains frequently undiagnosed.

SUMMARY: Recent data have improved our knowledge and diagnostic tools of infectious neuropathies. Treatment of the injured nerves is not yet available, and prevention and rapid diagnosis remain the main priorities for the clinician.