Traumatic ulcers with a hidden cause: A case report

Case report

A 19-year-old male was referred in urgently by their general dental practitioner to the Oral Medicine department. The patient was initially seen in September 2018 and complained of a 2-month history of worsening oral ulceration. He reported no other mucocutaneous lesions and that this was his first episode of oral ulceration. The patient was fit and well; he took no regular medications and had no allergies. He was a non-smoker and drank 20 units of alcohol weekly. Intra-oral clinical examination revealed four irregular ulcers in the oral cavity ranging from 2 cm x 5 mm up to 2 cm x 3 cm (Figure 1). The smallest ulcer was in the lower left quadrant, and the largest in the upper right quadrant.

Extensive haematoserological investigations were undertaken via the patient's general medical practitioner while the oral ulceration was present. Table 1 shows the investigations carried out by the patient's general medical practitioner. All investigations came back unremarkable. The patient had an incisional biopsy of the ulcer present in the right buccal mucosa carried out by the Oral Surgery department on the day of presentation. The initial diagnoses were that of possible traumatic ulceration or pemphigus vulgaris; however, the latter was considered to be highly unlikely.

Results from the biopsy revealed a part-ulcerated mucosa with an area of epithelial loss covered by a thin fibrinopurulent exudate. The epithelium at the ulcer edge was hyperplastic with thick surface parakeratosis. There were no features of any background lichenoid inflammatory lesion, nor any intra-or subepithelial separation to suggest vesiculobullous disease. No granulomatous inflammation was seen. There was also no evidence of dysplasia or neoplasia (Figure 2). The sample also displayed a negative direct immunofluorescence result and was screened for IgM, IgG, IgA, C3 and fibrinogen.

The only feature identified that might explain the aetiology was that the ulcer edge was hyperplastic with thick surface parakeratosis. This feature is typically encountered with traumatic ulceration. No other features were identified that might explain the aetiology.

Following investigations, the diagnosis was probable traumatic ulceration. This diagnosis immediately raised concerns over self-inflicted trauma. The patient repeatedly denied any history of trauma (thermal, chemical or mechanical) and any recreational drug use. The oral ulceration was treated with a 2-week course of 30-mg prednisolone. The patient was also advised to use Difflam mouth rinse and Corsodyl mouthwash. The patient was reviewed 8 days after the prednisolone commenced. There were no new lesions and the oral ulcerations had begun to granulate. He was advised to complete the 2-week course of prednisolone and to also use Flixonase TDS mouthwash until the ulcers had completely healed. The patient had no further episodes of oral ulceration.

Discussion

Oral ulceration is a common manifestation in both primary and secondary care. Therefore, it is important that practitioners are able to recognize the features of the presenting oral ulceration in order to establish the potential cause of ulceration. This will allow the patient to be treated appropriately and improve patient outcomes.

Most commonly, oral ulceration is caused by trauma.¹ Traumatic ulceration can be attributed to physical, thermal or chemical injury. Physical injury can include local aetiologies such as sharp restorations, broken teeth or a prosthodontic appliance. Ulceration caused by chemical injury is less common than that of physical injury. Chemical injury may be caused by medications (such as aspirin), recreational drugs or materials used during dental procedures.^7,8 Dental treatment itself may also be the cause of the ulceration.⁹

Traumatic ulceration is not suspected to have a gender predominance or age predilection. These ulcers are most commonly encountered on the tongue, lips or buccal mucosa.¹⁰ They often have an easily identifiable aetiology and present as single ulcers. Clinically, they present with a slightly raised or reddish border and a yellow necrotic pseudomembrane that can readily be wiped off. Provided that the injury is removed, these ulcers normally heal within 10 days.¹¹ For this reason, they are generally classified as being ‘acute’ due to their abrupt onset and short duration.¹² In some cases, the ulcer may be due to chronic trauma, and this will produce an ulcer with a keratotic margin.¹³ In cases where traumatic ulcers are asymptomatic, removal of the causative factor is satisfactory. If any clinical suspicion is present, a biopsy may be indicated. Biopsies should also be undertaken if the ulcer persists and shows no signs of healing up to 14 days after the causative factor has been removed.¹⁴ Although in this case prednisolone was prescribed, it should be noted that this is not normally a first line of treatment for traumatic ulceration. For symptomatic patients with traumatic ulceration, treatment may include topical analgesics, mouthwashes such as Difflam or Corsodyl, or topical corticosteroids. Prednisolone or other systemic corticosteroids may be indicated in severe cases of ulceration or for the treatment of pemphigus vulgaris.¹⁵

If patients present with atypical features, such as in this case, factitious injury should be considered as a possible cause. This is a behavioural disturbance that leads to deliberate self-harm. Factitious oral injuries have been reported as being rare in the maxillofacial region and have mostly been reported in paediatric patients or patients with mental health disorders.¹⁶ The literature also reports some cases whereby oral ulceration has been caused by deliberately swallowing caustic agents.¹⁷ In comparison to accidental or iatrogenic injuries, which are often more acute and self-limiting in nature, factitious injuries have a tendency to be more chronic in nature.¹⁸ Typical features also include: clinical features inconsistent with the history, usually in an otherwise healthy mouth; and in areas accessible to the patient. Suspected factitious injuries should be investigated thoroughly; however, denial by the patient is often ecountered.¹⁹ Clinicians should consider a referral for psychiatric evaluation should they deem this necessary, because in a small number of cases, factitious injury may be the initial presentation of some serious underlying psychological problems.²⁰

Oro-genital stimulation has become increasingly more popular and it has been reported that 90% of individuals under the age of 25 years of age practise oro-gential sex. Although trauma to the oral mucosa resulting from oro-genital sexual practice may have a low prevalence, it is important that it is still considered as a possible aetiology with atypical traumatic ulceration. Among oro-genital stimulation, fellatio is the most traumatic action, which may cause oral ulceration or laceration to the buccal mucosa and cheeks. These symptoms generally tend to be asymptomatic, and heal within 7–10 days.¹⁹

The literature also reports cases of oral ulceration with atypical features in recreational drug users. Most commonly reported are cases of ulceration caused by cocaine and ecstasy, often with ulceration presenting in the buccal sulcus or on the gingivae, which is the site of drug application.^6,21,22 Cannabis, cocaine and ecstasy are the most commonly reported drugs used by young individuals, and illicit drug use has been reported to be increasing year after year in the UK. In this case, the patient denied any recreational drug use; however, it is important that recreational drug use as a cause of atypical oral ulceration in young adults is considered by clinicians.²³

Conclusion

An accurate and detailed history is often critical to diagnose and identify the aetiology of traumatic ulceration. This case reveals the complexity and challenges of such diagnostic issues, in particular the investigation, initial and ongoing management of an apparent hidden cause or oral ulceration when the histopathology favours a traumatic cause.