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BMJ Case Reports 2013; doi:10.1136/bcr-2012-008487
  • CASE REPORT

Eosinophilic ulcer of the tongue in an 80-year-old Iranian woman after a psychologically stressful event

  1. Hossein Navabii4
  1. 1Department of Radiation Oncology, Shahid Sadoghi University of Medical Sciences, Yazd, Iran
  2. 2Department of Pathology, Shohaday-e-Kargar Hospital, Yazd, Iran
  3. 3Department of Pathology, Shohaday-e- Karegar Hospital, Yazd, Iran
  4. 4Shahid Sadoghi Hospital, Yazd, Iran
  1. Correspondence to Dr Ali Akhavan, ali52akhavan{at}yahoo.com

Summary

Eosinophilic ulcer of the oral mucosa is a benign self-limiting, chronic lesion more frequently seen in the oral cavity or tongue that can mimic squamous cell carcinoma. The pathogenesis of this problem is unknown. In this paper, we present a case of an 80-year-old woman with eosinophilic ulcer of the tongue after a psychologically stressful event.

Background

Eosinophilic ulcer of the oral mucosa (EUOM) is an uncommon self-limiting oral condition most commonly presenting as an ulcer or sometimes as a raised and indurated submucosal mass.1–10 It has been referred to as various entities in medical literature such as: ulcerated granuloma eosinophilicum diutinum, traumatic granuloma of the tongue, eosinophilic granuloma of the tongue, eosinophilic ulcer of the tongue, traumatic eosinophilic granuloma, atypical histiocytic granuloma (AHG) and ulcerative eosinophilic granuloma of the tongue.1–3 6–10 It also occurs in infants under 2 years of age at the anterior labial mucosa or at the ventral surface of the tongue, which is considered as a result of friction between the tongue or lip and teeth and is termed Riga-Fede disease.1–3 6–10 Although some specialists propose that the trauma caused by accidental bites or by repeated thrusting against sharp, misplaced or fractured teeth has a major role in this disease, however; its exact pathogenesis is obscure.2–10 Stress-induced psychoneuroimmunological factors may also have a role in pathogenesis.5 In this paper, we present a case with this disease occurring subsequent to a psychologically stressful event.

Case presentation

The patient was an 80-year-old woman referred to a private ENT office with a painful white lesion in the lateral aspect of her tongue. The lesion suddenly presented since 2 weeks previously, after her husband's death, and grew rapidly. She recalled no traumatic event. The patient complained of odinophagia; however, she could not explain the dysphagia. She was not a smoker and did not drink alcohol. The patient had taken atenolol for hypertension. On physical examination, she was found to be in good general condition. Head and neck examination revealed a 3 cm tender and indurated ulcer with inflammated borders in the lateral aspect of her tongue that was covered with a fibronoid membrane. There was no lymphadenopathy. Since the lesion mimicked a squamous cell carcinoma (SCC) of the tongue, an excisional biopsy was performed of the specimen and sent to the pathologist.

Investigations

Pathological examination revealed polymorphic inflammatory cells including neutrophils, macrophages and numerous eosinophil infiltration that extended to the submucosa and muscles (figure 1).

Figure 1

Polymorphic inflammatory cells especially eosionophils infiltration that is extending to the submocusa and muscles.

Differential diagnosis

Owing to the rapid growth, ulcerative nature and indurated borders of the usually single lesion, this condition can mimic SCC.1 ,2 ,6 In our patient, the clinical feature was so similar to SCC that the ENT specialist talked to the patient and her family about the necessity of hemiglosectomy and ipsilateral lymph node dissection after confirming the diagnosis by pathology. Other tumoral lesions such as lymphoma, lymphangioma, minor salivary gland tumours and metastatic tumours may be included in the differential diagnosis.1 Some infectious diseases such as primary syphilis, oral tuberculosis and fungal infections are found in the clinical differential diagnosis list.2 Sarcoidosis, discoid lupus erythematosus, Wegener's granolumatosis and eosinophilic granoluma are also included in the list of differential diagnosis.2 Microscopic examination, however; easily diagnosis EUOM from other clinical conditions, such as that of our patient.

The disease is microscopically similar to AHG, angiolymphoid hyperplasia with eosinophilia and Kimura disease.2

Treatment

Cholorhexidyn mouth wash, penicillin V tablets 500 mg four times a day and ibuprofen tablets 400 mg three times a day were prescribed.

Outcome and follow-up

Two weeks later, the lesion recovered completely. Now, 14 months later, the patient is well and her disease has not recurred.

Discussion

The aetiology and pathophysiology of EUOM remain poorly understood. Some authors propose a relation with trauma. The evidences that reinforced this theory include: the case of some patients remembering accidental biting of their tongue before the disease; the disease having two peaks of age incidence, the first one in children at the primary teeth eruption, and the second in old age on the occasion of missing and more commonly malposed teeth, dental appliances and dentures.2 The disease is more common in the ventral and lateral aspects of the tongue that are most exposed to trauma. In experimental studies, repeated injuries to the tongue in rats demonstrate similar lesions.4 Others have suggested that trauma is only a contributing factor in the development of EUOM and could facilitate the entry of unidentified aetiological factors such as microorganisms, toxins or foreign proteins into the underlying tissue to cause an inflammatory response.1 ,2 ,4 On the other hand, trauma is identified in less than 50% of cases.1 Histologically, EUOM shows a diffuse polymorphic inflammatory infiltrate, rich in eosinophils, involving the superficial mucosa and a deeper muscle layer. A typically large mononuclear cell scattered within the inflammatory infiltrate has been described and its origins have been a matter of debate in some patients;2 ,4 ,8 however, they were not seen in our patient. According to a suggestion about the immunohistochemical characteristics of these cells by Regezi et al, they are the combinations of macrophages and the oral counterparts to dermal dendrocytes. They also found an abundance of smaller CD3-positive T-lymphocytes.11 El-Mofty et al12 suggested that they may be myofibroblasts. Ficarra et al13 and Segura et al4 described the proliferation of CD30 atypical large mononuclear cells then as leading to the conclusion that EUOM could be included within the spectrum of CD30 lymphoproliferative disorders. Alobeid et al14 described three patients with oral mucosal lesions and EUOM features in one of whom the oral lesion was followed by skin nodules. All oral lesions contained atypical large lymphoid cells, which expressed T-cell markers and CD30. PCR analysis showed a monoclonal rearrangement of the TCRγ chain gene in all lesions and, in one patient, the same rearrangement in the oral and cutaneous specimens. These studies led to the conclusion that EUOM could be included within the spectrum of CD30 lymphoproliferative disorders. On the other hand, Elzay15 reported the increased numbers of mast cells in this disease and suggested that the interactions between mast cells and eosinophils might play a pathogenetic role. Atopic dermatitis (AD) is a chronic and relapsing inflammatory skin disease characterised by eczematous skin lesions, xerosis, lichenification and severe pruritus.16 This disease has some similarities to EUOM. Both of them have an obscure aetiology and similar inflammatory cells such as eosinophils and mast cells. The clinical occurrence of AD is often associated with psychological stress. In response to stress, the upregulation of neuropeptide mediators in the brain, endocrine organs and peripheral nervous system directly affects immune and resident cells in the.16 The substance P (SP) becomes elevated in the brain in response to many different types of psychological stressors. Lymphocytes, leucocytes, macrophages and mast cells have receptors for SP, and these cells can be stimulated by SP and through this route. Our patient's case was similar to that reported by Ribeiro et al.5 She had experienced a psychologically stressful event before her problem occurred. Stressors may play a role in developing EUOM.17

Learning points

  • Eosinophilic ulcer of the oral mucosa (EUOM) can mimic squamous cell carcinoma and must be considered in differential diagnosis.

  • The aetiology of EUOM is unknown. Psychological stressors may have a role.

  • EOUM is usually self-limited and recovers after 2–4 weeks.

Footnotes

  • Competing interests None.

  • Patient consent Obtained.

  • Provenance and peer review Not commissioned; externally peer reviewed.

References

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