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Endgames Spot Diagnosis

Intranasal pain in a patient with Behçet’s disease

BMJ 2020; 368 doi: https://doi.org/10.1136/bmj.m525 (Published 11 March 2020) Cite this as: BMJ 2020;368:m525
  1. Koichi Tsunoda, PhD, otolaryngologist,
  2. Rinako Endo, otolaryngologist
  1. National Hospital Organization Tokyo Medical Center, Tokyo, Japan
  1. Correspondence to K Tsunoda tsunodakoichi{at}kankakuki.go.jp

A 52 year old woman with Behçet’s disease presented with two to three days of severe pain in her left nostril. She had Behçet’s associated bowel, oral, and cerebral vasculitis and had been taking prednisolone for 20 years.

She reported occasional bouts of aphthous stomatitis (a known complication of Behçet’s disease) inside her mouth and on her lips, face, and hands, and said that topical corticosteroid was effective in these instances.

She also stated that blisters that were different from the aphthous ulcers sometimes appeared on her face, lips, and inside her mouth when the dose of steroid increased; these responded well to topical aciclovir so were assumed to be herpes simplex virus (HSV).

Six months earlier she had experienced a previous episode of similar nostril pain that didn’t respond to topical corticosteroid ointment, and she noticed an improvement when she tried vidarabine ointment that she already had at home.

On inspection, there was nothing visible externally on or around her nose. Examination of her ear, face, tongue, oral cavity, pharynx, larynx, head and neck area was also normal. However, examination with a nasal speculum showed blisters inside her left nostril.

Nasal endoscopy was performed to observe the blisters in more detail. There was no evidence of aphthous stomatitis but there were blisters (fig 1) directly behind the nasal hair inside her left nasal alar.

Fig 1
Fig 1

Endoscopic image showing blisters directly behind the nasal hair inside her left nasal alar

HSV IgM was negative but HSV IgG was positive.

What is the most likely diagnosis?

Answer

Intranasal herpes simplex in someone who is immunocompromised owing to long term corticosteroid therapy.

Intranasal HSV infection is not commonly reported1; however, this is the most likely diagnosis based on the patient’s medical history of glucocorticoid therapy and presumed HSV.

HSV IgM was negative but HSV IgG was positive, which, in keeping with the presumed HSV history, suggested that this is not a first HSV infection.

Current and recent doses of glucocorticoid have the greatest impact on infection risk, but the cumulative impact of doses taken in the past two to three years also affects the risk.2

Other dermatological manifestations of Behçet’s include erythema nodosum and aphthous stomatitis. Erythema nodosum was not considered because of the mucocutaneous location and the absence of erythema; however, aphthous stomatitis was a possible differential.

The following features can help to distinguish Behçet’s aphthous ulcers from HSV blisters:

  • Aphthous ulcers do not blister

  • In Behçet’s, several aphthous ulcers usually occur simultaneously, most commonly in the oral cavity.

It would be reasonable to treat this case as HSV because there is blistering and because previous blisters responded to aciclovir.

If blisters do not respond to HSV medication it would be feasible to consider biopsy to rule out squamous cell carcinoma.

Learning point

  • Consider intranasal examination when patients present with nasal pain.

  • Consider HSV infection in immunocompromised patients.

Patient outcome

We advised the woman to stop taking the oral steroid immediately and start taking oral famciclovir for HSV. Seven days later the pain and blisters disappeared. The woman started taking prednisolone 2 mg/day for the maintenance of her Behçet’s disease.

One year later she has not had any HSV or Behçet’s symptoms.

Footnotes

References

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