Granuloma inguinal (Donovanosis) - Universidad Peruana ...

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Delgado WA, Gonzalo E, Meneses LV, Lama JR.inferior. El paciente nunca consultóacerca de las lesiones ubicadas en lasregiones perianal y gingival. Elcompromiso submandibular se presentócomo masas granulomatosas, simétricas,indoloras, clínicamente eran muysemejantes a las lesiones de donovanosisque se desarrollan en el perineo (20,21).Las lesiones de la cavidad oralestaban confinadas a toda la encíavestibular y lingual inferior y semanifestaron como ulceracionesmicrogranulomatosas combinadas conáreas de apariencia esponjosa. Lasencías sangraban fácilmente durante lamasticación y a la menor manipulación.De acuerdo a la presentación clínica, seconsideraron los siguientes diagnósticodiferenciales : paracoccidiodomicosis,histoplasmosis y tuberculosis oralprimariaEl diagnóstico definitivo se estableciópor estudio histopatológico de biopsiastomadas de la lesión gingival. Lassecciones de H&E mostraban una marcadareacción histiocítica granulomatosacon numerosos neutrófilos y escasonúmero de células plasmáticas ylinfocitos. Los cuerpos de Donovanfueron identificados dentro de loshistiocitos usando la coloración deWarthin-Starry. La apariencia esponjosade la encía, correspondía histológicamentea la acumulación superficial degrandes histiocitos que producía atrofiadel epitelio.Cuando se utiliza la coloración deWarthin-Starry es muy importanterecordar que la Klebsiellarhinoscleromatis también se tiñe conesta técnica. La forma y tamaño de estosdos microorganismos son similarescuando se usa esta coloración (22).Recientemente, ha sido propuesta lareclasificación del C.granulomatiscomo Klesiella granulomatis debidoal hecho que el microorganismo muestraun alto grado de identificación conespecies de Klebsiella que sonpatógenos para los humanos (23).La diferenciación entre donovanosisy rinoescleroma se establece considerandoque rinoescleroma afecta la narizy también el tracto respiratorio superiore inferior, siendo el compromiso oral muyraro y cuando esto ocurre las lesionesestán localizadas en el paladar blandoy duro, encías y labio superior. Por otrolado, histológicamente se observanagregados compactos de histiocitos decitoplasma espumoso de aparienciaxantomatosa, conteniendo algunos deellos varios núcleos (células deMikulicz), además existe un densoinfiltrado de células plasmáticas ynumerosos cuerpos de Russell(22,24).Los neutrófilos no son tannumerosos como en donovanosis.Un aspecto notorio de este caso esla ausencia de lesiones en el resto de lacavidad oral, a pesar del extensocompromiso de la encía inferior y laprolongada duración de la infección.También es importante recalcar que nohubo compromiso sistémico, aúncuando, las lesiones de donovanosispersistieron por varios meses. Eltratamiento con doxiciclina fue efectivo,dejando solamente secuelas cicatrizalesen la piel pero no en la encía.Puesto que el paciente tenía tambiéncompromiso de la región anal, es difícilprobar que las lesiones gingivales osubmaxilares representaban manifestacionesprimarias o secundarias. Elcomportamiento sexual del paciente másbien sugiere que las tres áreasanatómicas afectadas representabanmúltiples sitios primarios de infecciónpor C.granulomatis. Sin embargo, nopuede descartarse completamente laposibilidad que las lesiones submaxilaresy orales hayan sido el resultadode autoinoculación.El presente reporte es un excelenteejemplo de manifestación extragenital dedonovanosis. Debido a que la cavidadoral representa el sitio extragenital máscomunmente afectado y siendo laslesiones típicamente crónicas yasintomáticas, en el diagnóstico diferencialde lesiones granulomatosas de lamucosa oral, es fundamental considerardonovanosis. Esto es parti-cularmenteimportante cuando en la historia delpaciente no se obtienen datos epidemiológicosrelacionados con infeccionesgranulomatosas endémicas y el pacientees sexualmente activo. La biopsia y elfrotis teñidos con coloracionesespecíficas son fundamentales paraestablecer el diagnóstico correcto.Granuloma inguinale (Donovanosis) ofthe gingiva and submandibular regionsDelgado WA, Gotuzzo E, MenesesLV, Lama JR. Granuloma inguinale(Donovanosis) of the submandibularregion and gingiva. Rev EstomatolHerediana 2005; 15(1):Donovanosis is a sexually transmittedinfection produce by the gram negativebacterium Calymmatobacteriumgranulomatis. The disease was firstdescribed by Mc Leod in India underthe name of “serpiginous ulcers” (1).Donovanosis is better known as granulomainguinale, since this infection predominantlyaffects the anogenital areas.In 1905, Donovan (2) described thepresence of what are now called“Donovan bodies” in the exudate froman oral granulomatous lesion of a patientwho also had a genital lesion.The primary lesion starts as a smallpapule or indurated nodule, usually occurringafter an incubation period of oneto four weeks; however, longer periodsof incubation have been reported (3).Lesions are characteristically painless.In men the anatomical sites more commonlyinvolved are the prepuce, coronalsulcus and penile shaft. Rectal lesionshave been reported in homosexualmen (4). In women, labial, vaginal andcervical lesions may occur (4).Dissemination of donovanosisthrough hematogenous spread, self- inoculationor both, can result in bone,visceral and skin lesions (3,5). There areseveral reports of oral lesions of granulomainguinale found in both men andwomen. Primary or secondary lesionsaffecting the oral mucosa have beenreported (6-12).A case of donovanosis involvingthe mandibular gingiva, submandibularregion and perianal skin in a 19-yearold homosexual man is described.Case reportA 19 year old man with bilateralsubmandibular granulomatous masses63
Granuloma inguinal ...was admitted to the Tropical MedicineInstitute Alexander von Humboldt of thePeruvian University Cayetano Heredia.Eighteen months before he had developedsubcutaneous nodules in both submandibularregions which had been increasingslowly in size and had becomeulcerated. Later the nodules developedchronic exuberant granulomatousmasses. At this time, the lesions weredrainage and dicloxacillin was prescribedfor treatment. Biopsy of the lesions werenot performed. He continued receivingmany short courses of different types ofantibiotics without benefit.Upon examination the patient appearedseverely ill and extremelydebilitated.He had chronic diarrhea andmarked anasarca. Abdominal examinationrevealed ascitis. The liver, spleenand lymph nodes could not be palpated.The scrotum presented marked edema.The patient stated he was a crossdressinghomosexual sex worker sincethe age of 14. He has had multiple sexpartners and rarely used condoms. Healways lived in Lima and denied travelto the peruvian jungle or highlands.There was no history of tuberculosis.Skin findingsAt the anal region a painless ulceratedmass of irregular form, approximately3 cm. in diameter was found.Symmetrical large granulomatous exuberantmasses were present in the submandibularregion bilaterally. The surfacewere ulcerated with serosanguinousdischarge. The borders of themasses were sharply demarcated fromthe adjacent skin. The lesions were friable(Fig.1, 2). No cervical lymphadenopathywas detected.Oral findingsBoth vestibular and lingual aspectsof the mandibular gingiva appearedspongy and hyperplastic. In some areas,the texture of the surface wasmicrogranulomatous showing areas ofulceration and bleeding (Fig. 3). Themucosa adjacent to the root remnant ofthe first right lower molar bled easily(Fig. 4). The gingival lesions were painlessand the patient was not aware ofthem. The rest of the oral mucosa includingthe tongue and tonsils werenormal. Apart from the molar remmantthe rest of the teeth were in good condition.A panoramic radiograph did notshow bone alterations.Laboratory studiesThe following abnormalities werefound :Hematocrite 19% (N: 45%)Erythrocyte sedimentation rate58% mm/h (N: 1 -10mm/h)Neutrophils 83% (55-70%)Lymphocytes 12% (25-33%)Total serum proteins4.83 g/dl (N: 6.1-7.9 g/dl)Serum albumin1.5 g/dl (N: 3.5-4.8 g/dl)Serum globulin3.33 g/dl (N: 2.6-3.1 g/dl)Serum vitamin B12180.0 pg/ml (N: 200-1000 pg/dl)Serum folic acid2 ng/ml (N: 3.0-25.0 ng/dl)The results of two HIV ELISA tests,VDRL and HBsAg were negative. Gramstain of the smears of the skin lesionsshowed isolated gram (+) coccus andacid-fast stain was negative.Gingival biopsyThe hematoxilin and esosin (H&E)sections obtained from two vestibulargingival biopsies showed accumulationsof large histiocytes with pale cytoplasmlocated immediately below the epitheliumwhich occupied the entire lamina propria(Fig. 5). Together with this cells manyneutrophils and few plasma cells and lymphocyteswere present (Fig. 6). Ulceratedareas were observed. Inside the cytoplasmof the histiocytes slightly basophilicsmall bodies could be detected athigher power. Sections stained with PASand acid fast stains were negative, butthe Warthin-Starry stain disclosed manyblack rod-shaped bodies inside the histiocytes(Fig.7). This finding together withthe history of the patient established thediagnosis of donovanosis.SmearsOnce the histopathologic diagnosiswas established, smears from thesubmandibular lesions were takenwhich were stained with Warthin-Starrystain. This study was also positive forDonovan bodies. (Fig.8).Other biopsiesSpecimens obtained from the skinof the submandibular and anal regionsdemonstrated similar histopathologicfeatures to the gingival tissues. TheWarthin-Starry stain was also positivefor Donovan bodies.TreatmentThe patient was hydrated and providedan adequate diet. He was treatedwith supplements of folic acid and vitaminB12. Doxycycline 100mg twice a daywas administered for eighty days. Theskin lesions disappeared slowly leavingfibrous scars in the perianal and submandibularregions (Fig. 9). The oral lesionshealed without scarring (Fig. 10).DiscussionDonovanosis is a mildly contagious,chronic, slowly progressive infectionproduced by the microorganism Calymmatobacteriumgranulomatis. Lesionsare usually localized to anorectal andinguinal skin. It also affects the genitaliaand oral mucosa. Occasionally, otherparts of the body can be involved. Clinically,it is characterized by the developmentof a velvety or beefy, red, granulomatouspainless ulceration that expandsslowly. Definitive diagnosis isestablished by detection of Donovanbodies in smears and biopsies takenfrom affected areas. Donovan bodies aredisclosed inside the cytoplasm of histiocytesusing special stains. Thesmears can be stained with Giemsa,Leishman, Wright or Warthin-Starrytechniques. In tissue sections, Giemsastain and particularly silver stains suchas Warthin-Starry demostrate the microorganism.Warthin-Starry stainclearly shows Donovan bodies as darkrod-shaped bodies measuring 0.5 - 0.7um x 1-1.5 um (3).Granuloma inguinale is seldomsystemic.Only 25% or less of cases affectthe inguinal region. Therefore, theterm “granuloma donovani” was pro64Rev Estomatol Herediana 2005; 15 (1)
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