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Eosinophilic Ulcer (EU) is a rare self-limiting chronic benign lesion of the oral mucosa with pathogenesis still unclear, however it may resemble malignancies, traumatic ulcerations and some infections such as deep fungal infections, tuberculosis and primary syphilis. This is a case report of a patient with EU in the lateral border of the tongue with no history of associated trauma and refractory to treatment with drugs. The ulcer rapidly healed after an incisional biopsy and the definite diagnosis was achieved only combining histologic findings and the clinical follow-up.
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Eosinophilic ulcer of oral mucosa: a case report
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Eosinophilic ulcer of oral mucosa: a case report
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                                                                                                         Case report


    Eosinophilic ulcer of oral mucosa: a case report

    Marcelo Carlos Bortoluzzi, MD1                            Introduction
    Fabrício Passador-Santos, MD2




                                                                                                   li
    Diogo L. Capella, MS3                                     Eosinophilic ulcer (EU) is a rare self-limiting chronic be-
    Gabriel Manfro, MD4                                       nign lesion of the oral mucosa. Clinically, the ulceration




                                                                                                na
    Rudy José Nodari Jr, MD5                                  has been most frequently found in tongue and it is char-
    Andréia Antoniuk Presta, MD6                              acterized by the presence of mildly indurated borders
                                                              which may resemble malignancies, traumatic ulcerations
                                                              and some infections such as deep fungal infections, tu-




                                                                                             io
    1
      Stomatology and Maxillofacial Surgery Professor,        berculosis and primary syphilis (1-8). EU also has been
    School of Dentistry, Health Bioscience Postgraduate       referred as traumatic ulcerative granuloma with stromal
    Program, Tissue Aspects for Health Prognosis and Inter-   eosinophilia, traumatic eosinophilic granuloma, trau-




                                                                         az
    vention Laboratory (LAPROG), Santa Terezinha Univer-      matic granuloma and ulcerative eosinophilic granuloma.
    sity Hospital (HUST), Oeste de Santa Catarina             In infants, usually on the ventral surface of the anterior
    University (UNOESC)                                       tongue secondary to trauma from newly erupted primary
    2
      Department of Pathology, São Leopoldo Mandic            teeth, EU is referred as Riga-Fede disease (1-8).




                                                                       rn
    Institute and Research Centre                             The pathogenesis of the EU is still unclear, however the
    3
      School of Dentistry, Oeste de Santa Catarina            lesion is thought to be reactive since trauma has been
    University (UNOESC)                                       implicated as initiating factor, nevertheless trauma can-
    4
      Head and Neck Surgery Service,                          not be demonstrated in many cases. Histopathological




                                                              te
    Santa Terezinha University Hospital (UNOESC)              findings consist of eosinophil-rich mixed infiltrates in-
    5
      Health Bioscience Postgraduate Program, Tissue As-      volving the superficial mucosa and the deeper muscle
    pects for Health Prognosis and Intervention Laboratory    layer, accompanied by a population of large mononucle-
    (LAPROG), Oeste de Santa Catarina University
                                                      In      ar cells that may correspond either to histiocytic cells,
    (UNOESC)                                                  myofobroblastic cells or activated lymphoid cells. Immu-
    6
      Health Bioscience Postgraduate Program,                 nohistochemical studies showed the expression of CD3,
    Oeste de Santa Catarina University (UNOESC)               CD4, CD8, CD20, CD43, cytotoxic granules (TIA-1) and
                                                              for the large cells expression of CD30, vimentin and
                                                  ni

                                                              CD68. Lymphocytes may also express the proliferation
    Corresponding author:                                     marker Ki-67 in small and large cells. The markers ALK
    Marcelo Carlos Bortoluzzi, MD                             (anaplastic lymphoma kinase), CD5, Beta-F1 and CD56
                                           io


    Universidade do Oeste de Santa Catarina (UNOESC)          were all described as negative. The differential histo-
    Faculdade de Odontologia                                  pathologic diagnosis may include Langerhans cell his-
    Av. Getúlio Vargas, 2125, Bairro Flor da Serra,           tiocytosis, Kimura disease, angiolymphoid hyperplasia
                                 iz



    Joaçaba, Santa Catarina, Brazil CEP (ZIP) 89600-000       with eosinophilia, atypical histiocytic granuloma, CD30+
    Phones: + 55 49 3551 2047 or + 55 49 3551 2112            lymphoproliferative disorder of the oral mucosa, lympho-
    E-mail: mbortoluzzi@gmail.com                             ma, allergic reactions and parasitic diseases. Definite
                 Ed




         or marcelo.bortoluzzi@unoesc.edu.br                  diagnosis can be achieved only by combining histologic
                                                              findings with clinical follow-up (1-10).

    Summary
                                                              Case report
    Eosinophilic Ulcer (EU) is a rare self-limiting chronic
 IC




    benign lesion of the oral mucosa with pathogenesis        A 33 year-old male patient searched treatment due a
    still unclear, however it may resemble malignancies,      solitary and painful ulcerative lesion in his right lateral
    traumatic ulcerations and some infections such as         border of the tongue with two months of evolution and
    deep fungal infections, tuberculosis and primary          with no history of associated trauma (Fig. 1). Examina-
C




    syphilis. This is a case report of a patient with EU      tion revealed a painful ulcer with relative firmness but not
    in the lateral border of the tongue with no history of    indurated, containing borders bringing two aspects, ery-
    associated trauma and refractory to treatment with        thematous or nodular, with a central ulcer reaching 4-5
    drugs. The ulcer rapidly healed after an incisional       cm in diameter that had a white-yellow fibrinous base.
©




    biopsy and the definite diagnosis was achieved            There were no significant regional lymphadenopathies.
    only combining histologic findings and the clinical       The patient was non smoker and had a past of alcoholism
    follow-up.                                                (has stopped for more than a year) with no actual alcohol
                                                              abuse. The patient reported a previous clinical treatment
    Key words: eosinophilic ulcer, oral mucosa, ulcer-        with his physician with no improvement or healing of the
    ative eosinophilic granuloma.                             lesion. He was admitted to Santa Terezinha University


    Annali di Stomatologia 2012; III (1): 11-13                                                                        11
    M. Bortoluzzi et al.



                                                                         nohistochemistry for CD68 antibody revealed a diffuse
                                                                         pattern of stain mostly in large cells localized from the
                                                                         surface of the ulcerated area until the skeletal muscle.
                                                                         Positive stain for CD34 antibody was observed only in
                                                                         blood vessels present within the lesion. Periodic acid-




                                                                                                                 li
                                                                         Schiff (PAS) and Ziehl-Nielssen stains were negative for
                                                                         microorganisms. At this point the histopathological diag-




                                                                                                              na
                                                                         nosis was inconclusive, however it was possible to reach
                                                                         the diagnosis of EU by combining histologic findings with
                                                                         the clinical follow-up.
                                                                         Forty-five days later the patient went through a cor-
                                                                         rective surgery due to the permanence of the exofitic




                                                                                                           io
                                                                         growths in the tongue and the histopathological analysis
                                                                         of this removed tissue brought none additional or inter-
                                                                         esting information. After an additional period of 30 days




                                                                                    az
                                                                         the tongue showed a complete healing (Fig. 4).
    Fig. 1. Initial clinical aspect of the eosinophilic ulcer.
                                                                         Discussion




                                                                                  rn
                                                                         Eosinophilic ulcer of the oral mucosa may be best re-
                                                                         garded as a reactive pattern of unclear etiology (5) and
                                                                         is characterized as rare benign entity which occurs as




                                                                         te
                                                                 In
                                                      ni
                                           io


    Fig. 2. Clinical aspect of the lesion following the incisional bi-
    opsy.
                                 iz



    Hospital due to his clinical debilitating condition and due
    the complaint of severe pain with significant impairment
    of food intake. He was medicated empirically with intra-
                 Ed




    venous Cephazolin 500 mg (three times a day), topical
                                                                         Fig. 3. Histopathological aspect of the tongue lesion. Notice that
    Tetracycline (three times a day, mouthwash), topical cor-
                                                                         the infiltrate extends from the ulcer base deep into subjacent
    ticoid (Dexamethazone, three times a day, mouthwash),
                                                                         skeletal muscle (H&E; 100x).
    non steroidal antiinflammatory and opioid analgesics for
    five days with no signs of healing. Preparing for a bi-
    opsy, blood tests were taken with none alterations found
 IC




    in complete blood count, hemosedimentation velocity,
    HIV test, rheumatoid factor, antinuclear antibody and
    fluorescent treponemal antibody-absorption test (FTA-
    ABS) though, C-reactive protein showed to be very high
C




    (51.61 mg/L while the normal parameter is below than
    6.5 mg/L). Chest radiographs where normal. An incision-
    al biopsy was performed and the patient was delivered
    from the hospital. Ten days postbiopsy the ulcer was
©




    markedly diminished in size (Fig. 2).
    The histopathological findings showed an ulcerated area
    covered with fibrinoid material. A dense polymorphous
    inflammatory infiltrate was seen and with numerous eo-
    sinophils (Fig. 3). The infiltrate extends from the ulcer
    base deep into subjacent skeletal muscle. There were
    no mitotic figures or nuclear atypia in the infiltrate. Immu-        Fig. 4. Final aspect of the tongue.


    12                                                                                            Annali di Stomatologia 2012; III (1): 11-13
    Eosinophilic ulcer of oral mucosa: a case report



    an ulcerated and sometimes with nodular-ulcerated le-          diagnosis and monitoring these patients are important to
    sion which is most frequently located on the tongue. The       avoid possible complications or overtreatment due to the
    entity brings several clinical differential diagnoses which    difficulties that arise in the histopathological exam, since
    include mainly malignancies, infectious diseases, auto-        there is no specific hallmark of the disease and, there-
    immune diseases or others (2,3,5-8,10).                        fore, the histopathological diagnosis of EU is by exclu-
    In our case the immunohistochemical study showed               sion (5). Monitoring is also important because low-grade




                                                                                                           li
    positivity for CD68 similarly to what was found by oth-        lymphoma in routine microscopic exam, for example,




                                                                                                        na
    ers (1,4,7), and this is a routine macrophage marker,          may only be recognized retrospectively, when lesions
    which may present immunohistochemical expression for           recur several times, spread to other areas, or develop
    monocytes, macrophages, neutrophils, basophils, large          more pronounced malignant features microscopically
    lymphocytes, myeloid precursors, osteoclasts, mast cell,       (4).
    synovial cells, Langerhans’ cell histiocytosis, interdigi-




                                                                                                     io
    tating reticulum cell sarcomas and melanomas. CD68
    has been described as having diagnostic utility for true       References
    histiocytic lymphomas and granulocytic sarcoma (11). In




                                                                              az
    the present case, the CD34 immunohistochemical reac-            1. Regezi JA, Zarbo RJ, Daniels TE, Greenspan JS. Oral
    tion was expressed in normal endothelial cells. The main           traumatic granuloma. Characterization of the cellular infil-
    diagnostic utility for CD34 is the identification of endo-         trate. Oral Surg Oral Med Oral Pathol. 1993;75(6):723-727.
    thelial differentiation. It is sensitive regardless of tumor    2. Lourenço SV, Silva MA, Nico MM. An ulcer on the lip. Clin




                                                                            rn
    grade, recognizing greater than 85% of angiosarcomas               Exp Dermatol. 2005;30(2):199-200.
    and Kaposi’s sarcomas (11).                                     3. Segura S, Romero D, Mascaró JM Jr, Colomo L, Ferrando
    In our case a rapid improvement of the ulcer occurred af-          J, Estrach T. Eosinophilic ulcer of the oral mucosa: another
    ter the incisional biopsy similarly to what was described




                                                                   te
                                                                       histological simulator of CD30+ lymphoproliferative disor-
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    havior is unknown. Accordingly to Eleni et al. (8) this         4. Hirshberg A, Amariglio N, Akrish S, Yahalom R, Rosen-
    behavior may be indicative that the healing response is            baum H, Okon E, et al. Traumatic ulcerative granuloma
    reactivated to the surgical intervention.
                                                           In          with stromal eosinophilia: a reactive lesion of the oral mu-
    Several therapeutic options for EU of the oral mucosa in           cosa. Am J Clin Pathol. 2006;126(4):522-529.
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                                                                       a distinct entity or a non-specific reactive pattern? Oral Dis.
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                                                  ni
                                                                       2008;14(4):287-295.
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                                                                    6. Kumar SK, Dhyllon A, Sedghizadeh PP. Indurated tongue
    otics as well as topical corticosteroids showed no results
                                                                       lesion. J Am Dent Assoc. 2008;139(2):159-161.
    with none signs of healing of the ulcer. Intralesional and
                                           io


                                                                    7. Boffano P, Gallesio C, Campisi P, Roccia F. Traumatic ul-
    systemic corticosteroids where not an option since deep
                                                                       cerative granuloma with stromal eosinophilia of the retro-
    fungal infection where considered as clinical hypothesis.
                                                                       molar region. J Craniofac Surg. 2009;20(6):2150-2152.
    Accordingly to el-Mofty et al. (9) in a review of 38 cases,
                                 iz



                                                                    8. Eleni G, Panagiotis S, Andreas K, Georgia A. Traumatic ul-
    suggest that cell-mediated immunity might play an im-
                                                                       cerative granuloma with stromal eosinophilia: a lesion with
    portant role in the pathogenesis of EU, since that topical,
    as well as systemic prednisone, is effective in the treat-         alarming histopathologic presentation and benign clinical
                 Ed




    ment of this disorder. None response with that medicine            course. Am J Dermatopathol. 2011;33(2):192-194.
    was obtained in this case as described above and this           9. el-Mofty SK, Swanson PE, Wick MR, Miller AS. Eosino-
    is in accordance with Segura and Pujol (5) which stated            philic ulcer of the oral mucosa. Report of 38 new cases
    that the use of topical steroids does not have conclusive          with immunohistochemical observations. Oral Surg Oral
    evidence of efficacy in treating EU.                               Med Oral Pathol. 1993;75(6):716-722.
    Surgical excision is the most commonly cited treatment         10. Ada S, Seckin D, Tarhan E, Buyuklu F, Cakmak O, Arikan
 IC




    procedure among the different therapies used and this              U. Eosinophilic ulcer of the tongue. Australas J Dermatol.
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C
©




    Annali di Stomatologia 2012; III (1): 11-13                                                                                    13
