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Revista Española de Cirugía Oral y Maxilofacial

versión impresa ISSN 1130-0558

Rev Esp Cirug Oral y Maxilofac vol.31 no.4  jul./ago. 2009

 

CASO CLÍNICO

 

Eosinophilic ulcer of oral mucosa

Úlcera eosinófila de la mucosa oral

 

 

A.C. Bencini1. C.A. Bencini2, V. Strada3, M. Florencia Soldavini4, G.M. Bruno4, M.F. Cordeu4, M.A. Cotignola4

1 Cirujano Bucomaxilofacial. Presidente Electo de la Sociedad Argentina de Cirugía y Traumatología Bucomaxilofacial. Encargado del área de Cirugía Bucomaxilofacial en el H.I.E.A. y C. San Juan de Dios de la Plata, Argentina. Miembro de la SACyTBMF; ALACIBU; SECOM; IAOMS.
2 Cirujano Bucomaxilofacial. Prof. Titular de Cirugía B Facultad de Odontología de la Universidad Nacional de La Plata, Argentina. Prof. Consulto en Cirugía BMF del Servicio de Odontología y Cirugía Bucomaxilofacial del H.I.E.A. y C. San Juan de Dios de la Plata, Argentina. Miembro de la SACyTBMF; ALACIBU; SECOM; IAOMS.
3 Médica patóloga – H.I.E.A. y C. San Juan de Dios de la Plata, Argentina.
4 Miembros del Servicio de Odontología y Cirugía Bucomaxilofacial del H.I.E.A. y C. San Juan de Dios de la Plata, Argentina.

Correspondence

 

 


ABSTRACT

Eosinophilic Ulcer of the Oral Mucosa, an entity, poorly deciphers in world-wide literature. It is defined as a self-limited, benign injury that although it can appear in different areas of the buccal cavity it presents a noticeable predilection via the ventral mucosa of the tongue. Clinically, one looks like an ulcer with hard and risen edges. The histo-pathological findings are typical and consist of a rich infiltrated mix of eosinophils, as well as a population of large mononuclear cells. Recent reports based on immunohistochemical studies allow us to confirm the presence of large atypical lymphocytes CD30+ and therefore include this lesion injury in the spectrum of lympho proliferative disorder simulators. Despite this the etiopathogenic mechanism remains unknown and local trauma still plays an unexplained roll; although the majority of publications have explained the phenomena as a reactive mechanism. The importance of this injury is established by its differential diagnostic because of its clinical similarity to Spinocellular Carcinoma, Histoplasmosis, syphilitic chancre, Ulcer Tuberculosis, Epidermoid Carcinoma and others. In our work the literature is reviewed and clinical characteristics, histo-pathologies and alternative therapies are discussed. We use the case of a young patient who has a biopsy in an effort to diagnose with certainty has a relapse of the lesion which directs the treatment towards combined surgery and local intra lesion cortico therapy which led to successful remission.

Key words: Ulcer; Eosinophils; Lympho proliferative disorders; Differential diagnostic.


RESUMEN

La Úlcera Eosinófila de la Mucosa Oral, es una entidad poco frecuente, pobremente descrita en la literatura mundial. Se define como una lesión benigna autolimitada que si bien puede presentarse en distintas áreas de la cavidad bucal, presenta una marcada predilección por la mucosa ventral de la lengua. Clínicamente, se presenta como una lesión ulcerada de bordes indurados y sobreelevados. Los hallazgos histopatológicos son característicos y consisten en un infiltrado mixto rico en eosinófilos, acompañado de una población de grandes células mononucleadas. Recientes artículos basados en estudios inmunohistoquimicos, permiten afirmar la presencia de grandes linfocitos atípicos CD30+ y por lo tanto, incluir esta lesión en el espectro de las entidades simuladoras de desordenes linfoproliferativos.
A pesar de esto, el mecanismo etiopatogenico permanece oscuro y el trauma local juega un rol todavía no dilucidado; aunque se halla presente en la mayoría de las publicaciones, explicando el fenómeno como un mecanismo reactivo. La importancia de esta lesión, radica en su diagnostico diferencial por su semejanza clínica al carcinoma espinocelular, histoplasmosis, chancro sifilítico, Úlcera tuberculosa, carcinoma epidermoide y otras.
En nuestro trabajo se revisa la literatura y se discuten la características clínicas, histopatológicas y alternativas terapéuticas, a partir del artículo de un caso clínico en una paciente joven, que luego de la biopsia escisión como método para el diagnostico de certeza, se produce una recidiva de la lesión; lo que orientó el tratamiento hacia la cirugía combinada con corticoterapia local intralesional, logrando su remisión.

Palabras clave: Terceros molares; Colgajo lineal; Colgajo triangular; Edema; Trismus; Dolor.


 

Introduction

The word ulcer is used to define "the loss of chronic evolution substance that affects the dermis and doesn't scar" (Grinspan, D.; 1975).

Eosinophilic Ulcer of the Oral Mucosa is an infrequent, benign entity characterized by one or many ulcers whose borders are elevated and hardened without eosinophil in the blood. It is not described very much in the literature and over time it has receive various denominations like xantoendotheliome, juvenile xantoma, xanthogranulome, granulome eosinophylic diutium of the tongue and finally eosinophilic ulcer of the oral mucosa. In the 2002 edition of the Argentinean Dermatologists Society magazine the eosinophilic ulcer of the oral mucosa was included in the Fitzpatrick classification (Table I), of eosinophil skin illnesses. It was included in group 1 where eosinophils form part of a histological diagnostic pattern, subgroup 1b which is characterized by eosinophile tissue.

It could appear in any etary group, with sex distinction and usually appearing on the mucous membrane of the tongue.

In 1964 Baskar and Lilly,2,3 established that etiopathology is traumatic and the majority of authors agree, or at least they agree that the trauma would be a determining factor in tissue damage. In fact, this entity was first described in 1881 by an Italian doctor named Antonio Riga.3 Histological cases were published in 1890 by F. Fede hence the illness is named Riga-Fede. This pathology is the expression of Eosinophil Ulcer of the Oral Mucosa in children, caused by the trauma of suction over the inferior incisors and lactation.

Others propose a more complex immunity mechanism measured by T lymphocytes.

Recently, thanks to immunohistochemistry techniques it has been possible to show that, at least in some cases, there are proliferations of lymphocytes T CD30+, which can be clonal or not; including EUOM in the spectrum of lymph proliferative disorders.6 But certainly the etiology is still unknown.

Within the pathologies that present an ulcer like elemental lesion of the buccal cavity, a differential diagnostic can be carried out for espinocellular carcinoma, syphilis, traumatic ulcer, tuberculosis, histhoplamosis and others.8,9,11

 

Materials and Methods

In August of 2005 a female patient, age 13, 53 kilos of average physique, from Madariaga (located on the interior of the province of Buenas Aires) went to Servios de Odontlogia y Cirguia Bucomaxilofacial del Hospital Interzonal Especializad en Agudos y Cronicos San Juan de Dios de la Plata, Buenos Aires with an ulcerated lesion on her tongue that the patient had detected 4 months prior. The patient did not have a family history of any illnesses. 4 months prior she noticed a lesion on her tongue (without noticing anything else on her body) for which other doctors prescribed antiseptic mouthwash, antiviral medicine (cream and pill form), oral corticoids, and atimycotics, none of which resulted in complete remission.

Upon clinical inspection an ulcer measuring 1.5 x 2 cm was observed on the ventral face of the tongue. The ulcer had over elevated borders, erythematous, was not hemorrhagic and was whitish in color (Fig. 1). Upon palpation its borders were hard but the centre of the lesion was not. The ulcer seems to be mobile and not deeply adhered. The patient doesn't report spontaneous pain even during the palpation. Neither local nor regional adenopathies were detected. We did not observe dental irregularities or sharp borders of anteroinferior teeth, with class 1 occlusion of Angle.

The results of the laboratory studies showed no abnormalities except on erytho sedimentation of 46 mm in the first hour (Westergreen Method).

 

Treatment

The patient was given amoxicillin and 2 g of clavulanic acid one hour before surgery and was operated on under local anesthetic (carticaine 4%). A biopsy was performed encompassing the lesion (Figs. 2 and 3). The sample was sent to be anatomic pathologically studied, asking for a mycological and viral study in order to rule out mycotic or viral etiologies. The results of these tests were negative. The anatomic pathological diagnosis is Eosinophil Tongue Ulcer.

 

Progress

Four months after surgery the lesion relapsed. The relapse lesion is much smaller than the first one (Fig. 4), so another scission biopsy is performed but this time involves a deeper area in order to eliminate the rhabdomyositis produced by the eosinophils (Med Cutan Iber Lat Am 2003; 31:213-4). This act confirms the Eosinophil Ulcer diagnosis. 21 days after the 2 nd intervention another relapse is found so intra lesion cortico therapy is carried out, rather sub mucosa, with betamethasone acetate 3 mg/ml Disodic Betamethosone phosphate 3.9 mg/ml of the dosis of 1 ml plus l ml of carticain 4% per cm 2 including the lesion every 7 days (Fig. 6); excellent therapeutic results were found.

The lesion reduced in size by 75% by the second week with complete remission of the lesion after 45 days (Fig. 8).

 

Discussion

There is a lot of controversy among pathologists, dermatologists, clinicians and surgeons when establishing an etiology and treatment for Eosinophil Ulcer of the oral mucosa.

In the majority of cases the lesion consists of an infrequent inflammatory benign process that is characterized by an ulceration with clear borders which is sometimes non painful, located on the ventral face of the tongue.

Many others give this entity a genuinely traumatic etiology and assure that in the case of adults, it is the illness or ulcer that in children is called Riga- Fede. (Gonzalez, Ja; et al. 2003)(Elzay, R. 1983).14,15

There are multiple causes of ulcers in the oral mucosa: Chemical, Physical and thermal trauma; infectious agents (bacteria, virus, fungus, parasites, etc.); Allergic reaction; Malignant and Benign Neoplasias; Systematic Illnesses; Psychosomatic Disorders (relapsing apathas, major apathas, etc); lympho proliferative disorders (eosinophil ulcers, etc.). In the majority of studies the etiological agent suggested is trauma. In an experiment with rats Bhaskar and Lilly created eosinophil ulcers using tweezers to create trauma in the tongue.2

Other authors (including us) assure that this lesion is the result of a complex and intricate phenomena related to lympho proliferative disorders (expressing lymphocytes CD-4; CD-8; CD-3; CD-1a and occasionally CD-30+ (Ki-1) in 70% of cases). Thanks to the current techniques in use available to study immuno histo-chemistry the true etiopathology is being discovered.6,12

The oral mucosa eosinophil ulcer has been mainly described on the tongue in infants, adults and the elderly. In a series of 70 cases of patients aged 1-82 years old, Elzay found 37 cases on the tongue.15

The role that eosinophils play in this type of lesion is not well established. It is however, well known that in the healing models of animal wounds these cells normally produce TGF-a and TGF-ß1 that are important in the healing process. In the case of oral mucous eosinophil ulcer in humans synthesis of these factors it is not significant what would partially explain delayed healing of these ulcers. In addition to this it is important to highlight that the deposit of fibrin and blood clots in blood vessels interferes more in the scarring process.

In some cases healing takes place spontaneously between 1 day and 1 year. Recurrence is usually more frequent and has been described in isolated cases.10,12,13,15 Mezei and col. found relapses in 15% of cases.11

The case that we present illustrates the clinical and evolutionary characteristics as well as treatment of ulcerative lesion, apparently lympho proliferative, in which in a short time becomes large in size and doesn't respond to antibiotic treatment, antivirals, etc. Its histo-pathological diagnosis corresponds to Eosinophil Ulcer of the Oral mucosa.

Many therapeutic alternatives have been proposed, for example antibiotics, cryosurgery, corticoids, surgery combined with intra lesional cortico therapy (Ficarra, G; et al. 1997)9 and the simple clinical control of waiting for spontaneous remission. All of the above described have been presented in the literature with successful outcomes.

In our case we chose combined surgery and intra lesional cortico therapy which gave us a positive result.

 

Conclusion

It is very important to get an accurate diagnosis of this pathology. Mainly because of its clinical similarity to other entities that respond to treatment and evolution very differently.

Due to this lesions characteristics and scarce presence, when it comes to treatment there is not one specific protocol. We think it is convenient to evaluate each patient keeping in mind: the size of the lesion, its location, time of evolution, age, habits of patient, relapses, etc; knowing that in the majority of the publications it ends up having etiology of trauma where it would be enough to eliminate the cause and wait for spontaneous remission. In the case of our patient the lesion is lympho proliferative, because of this we chose combined surgery and intra lesional cortico therapy and the results were positive.

 

 

Correspondence:
Prof. Dr. Adrián Carlos Bencini
Diagonal 74 nº 2571 entre 20 y 21
CP1900 La Plata. Buenos Aires. Argentina
Tel/fax: 54 221 451 18 59
e-mail: adrianbencini@speedy.com.ar

Recibido: 04.06.2007
Aceptado: 15.10.2008

 

 

References

1. Publicación de la Sociedad Argentina de Dermatología "Enfermedades eosinofílicas cutáneas". Dermatología Argentina. 2002;4:82-196.        [ Links ]

2. Bhaskar, S; Lilly, G. Traumatic granuloma of de thongue (human and experimental). Oral Surg, Oral Med, Oral Patol. 1964;18:206-18.        [ Links ]

3. Antonio Guzmán, Gloria Mendoza "Dientes natales y Enfermedad de Riga-Fede". Dermatol pediatr lat 2005;3:152-7.        [ Links ]

4. Liliana Vizcaya Barraza, J.M. Nuñez Martinez, J.M. Branzo Fernandez, Reynaldo Falcon Escobedo. "Úlcera eosinofica de lengua". ADM 2003;13:36-9.        [ Links ]

5. Wilson A. Delgado, Italo Funes. "Úlcera eosinofílica de lengua". Rev. Estomatol Herediatra 2003;13:36-9.        [ Links ]

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7. García M, Pagerols X, Curco N, Tarroch X, Vives P. "Eosinophilic ulcer of the oral mucosa: 11 cases". Ann Dermatol Venereol. 2002;129:871-3.        [ Links ]

8. Melgarejo Moreno PJ, Hellin Meseguer D, Ruiz Macia JA. "Eosinophilic ulcers of the tongue as an adverse reaction to carbamazepine" An Otorrinolaringol Ibero Am. 1998;25:167-71.        [ Links ]

9. Ficarra G, Prignano F, Romagnoli P. "Traumatic eosinophilic granuloma of the oral mucosa: a CD30+(Ki-1) lymphoproliferative disorder?" Oral Oncol. 1997;33: 375-9.        [ Links ]

10. Velez A, Alamillos FJ, Dean A, Rodas J, Acosta A. "Eosinophilic ulcer of the oral mucosa: report of a recurrent case on the tongue" Clin Exp Dermatol. 1997; 22:154-6.        [ Links ]

11. Mezei MM, Tron VA, Stewart WD, Rivers JK. "Eosinophilic ulcer of the oral mucosa" J Am Acad Dermatol. 1995;33:734-40.        [ Links ]

12. Mofty SK, Swanson PE, Wick MR, Miller AS. "Eosinophilic ulcer of the oral mucosa. Report of 38 new cases with immunohistochemical observations" Oral Surg Oral Med Oral Pathol. 1993;75:716-22.        [ Links ]

13. Doyle JL, Geary W, Baden E "Eosinophilic ulcer" J Oral Maxillofac Surg. 1989;47: 349-52.        [ Links ]

14. Gonzales JA, Rivas MA, Medina MA, Ocaña, J. Úlcera eosinófila de la Mucosa Oral. Med cutan Iber Lat Am. 2003:31:213-4.        [ Links ]

15. Elzay R. Traumatic ulcerative granuloma with estromal eosinophilia (Riga-Fededisease and traumatic eosinophilic granuloma). Oral Surg 1983;55:497-506.        [ Links ]

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