Nocturnal sedation in a child with facial ulceration

Abstract

Mostly, herpes zoster affects adults and therefore childhood presentation can represent a diagnostic challenge. Childhood herpes zoster, when it occurs, can also be associated with peripheral nerve complications, as illustrated by this case. A 3‐year‐old child who had herpes zoster developed a nasolabial scar resulting in a shallow non‐healing ulcer from being repeatedly picked. Healing was only achieved after nocturnal sedation, with chloral hydrate.

Case

A 3‐year‐old girl developed typical herpes zoster (shingles) of the maxillary division of the left trigeminal nerve and presented 10 days later with striking ulceration and scarring over the left cheek. She had been previously exposed to varicella at the age of 6 months. At the onset of herpes zoster, she received treatment with aciclovir 800 mg five times a day (intravenously for 3 days and as tablets for 2 days). The vesico‐pustular eruption resolved with treatment but left scarring and ulceration of the affected site. She repeatedly picked and excoriated the nasolabial fold and upper cheek, this was particularly marked at night and associated with insomnia.

At presentation, there was a shallow ulcers affecting the left nasolabial fold and the upper cheek, extending over the left ala and onto the lower eyelid, with an area of 10 cm² (Figure 1). There was an adherent yellow serous exudate. Sensation overlying the ulcer and surrounding unaffected skin was normal.

Figure 1.

Extensive disfiguring ulceration and scarring affecting the maxillary division of the left trigeminal nerve.

Various topical measures were tried including the application of polymycin ointment, gelliperm^® dressings and an occlusive face‐mask (as used for burns), without success. Considering the possibility of a postherpetic neuralgia or trigeminal trophic syndrome, oral carbamazepine was given for 2 weeks but was unhelpful. The wound began to show signs of healing after the introduction of chloral hydrate as nocturnal sedation and healed completely within 2 weeks.

Discussion

Chickenpox is a common viral infection of childhood, with most cases occurring before 10 years of age (1). After this initial infection, the virus becomes resident in ganglia along the entire neuraxis. Unlike herpes simplex virus, varicella zoster virus cannot be cultured from human ganglia (2). Recrudescence of latent varicella zoster virus giving rise to shingles affects less than 1 per 1000 person years in children, and up to 12 per 1000 person years in adults aged over 65 years (3). Early onset of shingles, before 60 years of age, is more likely in patients exposed to varicella before 1 year of age (4). Shingles is characterized by sharp pain and tenderness followed by a vesico‐pustular eruption distributed over the affected dermatome, the thorax and face are the most common sites involved. Associated peripheral nerve complications include keratitis due to ophthalmic zoster, osteonecrosis and loss of dentition due to mandibular or maxillary zoster, Ramsay Hunt syndrome, zoster paresis, postherpetic neuralgia and trigeminal trophic syndrome (5, 6).

Trigeminal trophic syndrome was considered in our patient, this is an uncommon disorder in which trophic ulceration follows repetitive subconscious self‐trauma to anaesthetic skin within the trigeminal area. Ulceration typically begins on the ala nasi, may destroy the cartilage, and then spreads towards the cheek. It is caused by neurotophic changes leading to destruction of the small neurones conveying pain and temperature sensation. Many different causes have been identified including shingles, but many reported cases relate to a postoperative complication of gasserian ganglion destruction for postherpetic neuralgia (7). Though the affected skin is anaesthetic (which makes the diagnosis is our case unlikely), patients describe a variety of sensations, which include itching, burning, crawling and tickling; provoking repeated trauma. The most likely cause of the destructive ulceration in our patient is postherpetic neuralgia, and although this is most commonly associated with pain, an itching has also been described (8).

A variety of factors can adversely influence wound healing. A rational approach to wound management included the application of a topical antiseptic to reduce bacterial colonization, an interactive local dressing to promote moist wound healing and a bandage to keep the dressing in place. For facial wounds, in particular postburn injury, and for patients with trigeminal trophic syndrome, specially moulded facial masks can be used to protect the skin (6). Carbamazepine has been often used to treat neuropathic symptoms, it has been successfully used in the management of postherpetic neuralgia and the trigeminal trophic syndrome (5, 9) As involuntary excoriation was considered the main reason for the wound failing to heal in this case, potent nocturnal sedation was introduced; this provided a sufficient period of rest from interference for the wound to start healing.