0 Pp the g Ss ae volume 77. : : Némero 1 Janeiro - Fevereiro 2002Pemande, Mocs, Knacfes& Fernones 6s Caso Clinico / Case Report Botriomicose cutanea: relato de caso* Cutaneous botryomycosis: a case report” ‘Nurimar C Femandes” Juan Piro Maceira* Trocy Guedes Knacks * [ilo Femandes * Resumo: £ descrito wm caso de botviomicose cutinea em um paciente adulro do sexo masculino; O dlingnético fat haseado nos aspects clinics, hacterologico « histopatol6gico. O pe esquerdo apre “entava aumento de volume ¢ mltipls fistulas que drenavam pus e sangue sem grins. Do material de fessezio parcial da lesio, foram isolados em cultura Pseudomonas aeruginosa, Proteus mirabilis © Escberchia coli; 40 exame histopatolgco, fo encontrado grinulo contendo cocos Gram-positivo, O piciente fol tratado com eiprolaxacina por via intravenosa durante dois meses; apés um segundo des bridamento cirigico, 2 eiprofloxacina foi administra por via oral durante nove meses com recupe- ‘cio complet. Palavras-chave: Infegoes por Pseudomonas, infeexbes por Proteus; Infeccbes por Escherichia coli Summary: A case of cutaneous botryomycoss was diagnosed in a white, adult male witout any underlying disease. Toe diagnosis was based on clinical, bacteriological and bistoogte grounds ‘Selig end multiple fistulae on the lf foot draining pus and Blood without granules were assessed by partial resvection. Pseudomonas aeruginosa, Proteus mirabilis and Escherichia coli were isolated from the culture ard bistopathologic examination disclosed granules containing Gram-positive cocet ‘Te patient uss kept on intravenous ciprofloxacin for 2 months, a second surgical debridement was performed and ciprofloxacin was administered orally for 9 months ith full recovery ‘Rey words. Pseudosionas infections; Proteus infections; Escherichia coli nections INTRODUGAO INTRODUCTION ‘A Botromicose ¢ uma infoogio bacteria granvlo- rmatosa da pele e viscera, caracterizada microscopicamen te pela presenga de grinuios lembrando aqueles do miceto imac actinomicose, As lexdes cutineas so ernicas © supu: rativase semelbantes acistos, abscess, fstles, néduos, Placas ou lesdes wleeradas. Sto sofitrias ow miltiplas © Tocalizam-se principalmente aas areas expostas (cabeya. brapos, peras) ou genitais “=” A patogénese permancee obscura; a doenga. parece esr celacionada, de_aiguma "i gina gs ee tc aes le An nas Derma lode nc. 771 5-7. jon fe. 2002 Botyomyeosit is a granulomatous hacteriol infection ofthe skin and viscera, which is characterized ricraseopicaly by the presence of granules that resen- Ble those of mycetoma and actinomycosis. The skin lesions are chronic and suppurative and may resemble cysts, abscesses, fistulae, nodules. plagues or wleerated Tesions. They ore solitary or multiple ond mainly on exposed areas (head, arms, legs) or genitals! The pathogenesis remains obseure: it seems thatthe disense ata os Se ee ae SO scacucse inp orn cee rae Dagan hie cect Paco85 Pamand, Moccia Kactfine & Ferandes forma, 20 equilbrio entre © nimero de organismos e as detesas do hospedeico “*” Sao descritos defitos neutof 0s na forma visceral, e especulam-se anormalidades imu- nolégicas * A aparéncia geral de cisto sebéceo infestado Sugere que algum corpo estranho (osso de peixe,palha de vassoura) tena desempenhado um papel na formasio ou Perpetuagio da lesio. Podem estar envolvides também musculos, poncuroses, tend6es © ostos ‘A causa mais fequente & 0 Staphylococcus aureus, ‘mbora tenhum sido desritos tamisém Pseudomonas spe cies, Escherichia col, Proteus vulgaris como agentes eto cos dessa infeegdo.'*" Rotriomicose é daenga muito rera em humanos, pois menos de 100 casos foram deseritos, dos «qusis a botriomicose eutinea & responsive por 75%" Os fatores predisponentes para batriomicase visceral incluem fbrosecstca." diabetes mellitus," trapia cam esters" ¢ infecgdo pelo HIV" A hotiomicose cutnea & deserita raramente na literatura estrangeira, havendo também poucos relatos no Brasil ***? Wroclawski ‘© cols” descreveram 0 primeio caso brasileiro de botriont- ose cutines (Tabela 1). O tratamento depende do organis: ‘mo causal e sua sensibildade aos anibiécicos." (Os autores descrevem um caso de botriomicose euté- ‘nea em um paciente sem nenhuma doenea subjacente que se recuperou completamente apis desbridamenta cirigica © ‘erapiaantimicrobiana de longa duracao, O principal objet ‘may by related in some way 10 a balance between num: bers of organisms and host defenses.” Neutrophilie defects are described inthe visceral form and immunolo: ‘gic anomalies are speculated" The gross appearance of an infected sebaceous eyst suggests that a foreign body Gish bone, broom straw) has played a rae initiating or perpetuating the lesion. Muscles, aponeurosis, tendons ‘and bones can be involved too, ‘Staphylococcus aureus & the moat frequent cause but Pseudomonas species, Escherichia coli, Proteus vulgaris have also been reported asthe etiologic agent of this infec- tion" Botryomycasis is a very rare disease in humans Since less than a hundred cases have been reported: ofthese, cutaneous botrronneosi accounts for 7326 ofthe cases." Predisposing factors for visceral borryomycosis Include cystic fibrosis diabetes mellitus", steroid the- apy "and HIV infection.“ Cutaneous booryomy- cnsis 1s rarely reported in the foreign literature!” and there are alo few reports in Brazil" Wroclawsk etal’ repor ted the frst Brazilian case of extansous botryomycasis (lable 1). The weament 1s dependent upon the particular ‘causative organism and ts sensitivity to antibiotis We describe a case of cutaneous borryomycosis i 4 patient without any known underlying illness who fully recovered after surgical debridement and long-term anti: crobial therapy. The main purpose of this paper is 10 ‘Tabela 1 — Botiromicose cutinea: casas brasilecos, Table 1: Cutaneous botryomycosis: brazilian cases efteg tumor xan (5 das fas ‘Saladin Cefutia (20 dias do), Getic (5 das de SUKI TMP SRM (Occ moni) ‘Tehomobiy, Mtge Sola do péeaquedo Staph aureus Give 1083 (5) tumor veruseso—SMX/TMP (-) lesolevericous amor. Sari daioage Toweney, = M14 Student 9 couro cabeuis, trax, Staph aureus Improvement 1966) al, pem, bapa ne Peni (18 5) aca, tho- Surgical dinage aillae, lear Braga, renee : 12 facies, tice Inprosement 19870), 5 An box Dermatol, Ri de Sone, 771) 65-70, fn 2002 Fie TARELIONATO. DE NOTASFernanda, Mostra, Kole & Fermandes aumento gsr ‘o peesquerdo| vo deste tmbulho € enftizar aque nessa condieao a interns540 pode ser necessiria por longo periodo a fim de se ebter um ‘resulta sais, RELATO DE CASO Um homem branco, ‘com 37 anos de idade, motorists, da regio urbane do Rio de Janciro, foi admitido no Hospital Universitario CClementino Frage Filho (UFRJ) em janeiro de 1999, Nove ‘meses antes, ele havianotado aumento de volume progres- sivo do hilux enquerdo,sendo tratado com drogas atin. gicas sem resposta, Negava febre, perda de peso, dor ou trauma local. Ao exame clinico observou-se um impres- sionante aumento de volume do pé esquerdo, com milti= plas fstlas drenando pus amarelo e sangue, mas sem gri- hiulos (Figura 1). O paciente foi submetido a ressecyi0 parcial da massa, Estudos micol6gicos nfo revelaram fun- 0s, ¢ 0 exame bacerioldgico mostrou cocos Gram-posi fives € bastonetes Gram-negativos. Nas culturas cres- cceram Prewdomonas aeruginosa, Proteus mirabilis ¢ Escherichia coli. Cortes histlégieos corados pela H&E, Grocott - Gomori e Gram - Brown - Brean mostraram um proceso inflamatirio necrdtico granulomataso, agudo € frénico, no qual se observou um grimulo com cocos Gram-positivos em seu interior (figuras 2 € 3). Estudos Iaboratoriais na momento da admissio revelacam hemos- @ Page 1 impress oft fot emphasize that this condition ‘may require a inpatont rea ‘ment for @ long period to ‘achieve a good outcome CASE REPORT 4 37-year-old white male, driver from the urban area of Rio de Janeiro was admitted to Hospital Universitirio Clementino Fraga Filho (UFRJ) n January 1999, Nine month« before he had begun experiencing pro- agressive swelling on his left hallux, which was treated With antifengal drugs withowt response. He denied fever, weight loss, pain or focal trauma At clinical examina- tion, an impressive enlargement of the left foot was obser- ved with mullpl fistulae draining yellow pus and blood, but no gramules (Figure 1). The patient was submited fo 4@ partial resection of the mass. Mycologic studies did not reveal any fingus and the bacteriologic examination Showed Gram:positive cocci and Gram-negative rods Cultures yielded Pseudomonas aeruginosa, Proteus mi bilis and Escherichia coli growths. The histological sec- tion stained by H&E, Grocott ~ Gomori methenamine - silver nitrate and Gram ~ Brown - Brena techniques sho ‘wed an acute and chronic inflammatory, necrotizing and {granulomatous process, where one granule was seen with Gram-positive cocct inside it (Figures 2 and 3) Laboratory studies ot the time of admission revealed a igure 2 Grlnulo boiromicco (H&E AON) 0 grimulo € pequene a pigment / gure 2 Botryonpeotie granule i "G8 400) the granule foal and nonpgmentad An bras Dermatol, Ri Jamia, 7(16370, fon fe 200. Tigi 5 Gclnulo hosiomiciace (1 & E1003) deposigo de ‘Subninctsconsoflica na perl Go grinulo gure 3 Botromyectieranale (1 & 1001: daposition of Sonnopbc abst on te porpbery of eranuleSF, Maite eaciae& Ferner core ap sedimentagdo de 65mm, exame HIV negativo, radiogeafias normais dos 108805 do pé. Administrou-se cipro foxacina (400mg a cada 12 horas) por via intravenosa de acordo com (© antibiograma, durante 64 dias Realizou-se um nova desbridamen: to cirirgico,e 0 paciente fi mant «lo com eiprofloxaciaa oral (S0.ms cada 12 horas) durante aove meses. Observou-se cura clinica (Figura 4), e 4 hemossedimentagio fetormou ao normal, euidado Focal incliu limpezs vigorosa com solupio de povidine e creme de slfaizina de prata ras éceasuleeradas. NZo foram observados efeitos coat tis durante 0 segeimenta, DIScUSsAO, A patogénese da botriomicose ainda nao fo estabe- lecids. Os chaos imunol6gicos no aso brasileiro cama PPD (Onn), esteproquinase-esteptodanase (Onn) io- micina (Omm), vaccinia (2mm), aumento de 1gG e gama alobulina sugerem uma possvelalteragio nas defesos do hospedeiro No paciente em questio observou-sealterag da emossedimentago: s outros achados laboratriis encontravam-se dentro dos limites norma envolvimento sstémice, ‘Ao exame do pus, gros amareloesbranquigadas da tamanho de eabeeas de alfinete, podem ser vsios. Os auto- Fes ndo observaram essas estnturas, como 0 fizeram joutrs.**" Caso no ocoraeliminaglo dos aos, eles serdo vistos nos corteshistolgicos (HE). Os aris so besof Tico ecircundados por material eosinoflico ratiado (fend meno Splendore-Hoeppli ©. diagnéstic.clinico diferencia. de oriomicose faz-se principalmente com micetoma (actitomicetoma) © sctinomicose. O micetoma é ums infecyio que se disserina Jentamente em tecidos contigues,inclind asso. Os pis sto © principal focal em que dterminados fungos (eumicetora) ‘0 hactrias aerdbicas superiores (atinomicetoma) si inc- clades por pequenos traumas. Ocorre drenagem contend ros caracteristics por meio de fistulas que se estendem Daca a pele!" Os agentes etiolégicas do micetoma mais ‘comumente observades no Brasil sii 08 fungos Prev ddllescheria boyd, Cephalosporium (Aeremonium) 5p. Maret grisea e 0 actinomiceta Nocardia brasiliensis! © ‘exame microsodpico do pus, exsudato ou material de bib © 0 havia | Gee afer 11 ‘months of therapy sedimentation rate of 65 mm, a negative HIV test and normal radiographs of the foot bones, Intravenous ciprofloxacin (400 img every 12 hours) according to antibiogram, was administered for 64 days. A new surgical débri dement was performed and the patient was then kept on oral cipraflosacin (500 mg every 12 hours) for 9 months. The cure was seen clinically (Figure 4 and the sedimentation rate returned f0 normal. The local care included vigorous cleanliness with povidone solution and 2% sl: ver sulfadiazine cream on the ulcerated areas. No side effects were observed during the follow-up Discussion The pathogenesis of botrvomvcosis is not yet ‘established. The inmunological findings in one Bracilion case such as: PPD (0 orm), streptokinase-sieptodornase (0mm), oidiomvein 10 nm), vaccinia (2 mn), increased IgG and gama globulin suggest a possible change in the host's defenses" In our patient. an elevation in the eryth- rocyte sedimentation rate, other laboratory findings within normal limits and no systemic anolvement were observed, On gross examination ofthe pus, pinkeodt-size whitish yellow granules can be evident. We didnot obser ve these structures unlike other authors.” If there is no discharge of granules, che histological sections (#6) will disclose them. The granules are hasophilic and surrow ded by radiated eosinophilic material (Splendore-Hoeppli phenomenon) Differential clinical diagnosis of botrvomycosis is nan with myeesoma (actinomyesioma) and aetinom costs. Myeetoma 1s an infection that slowly spreads to contiguous tissue, including bone. The foot is the main site where,certain fungi (eumycetoma) or higher aerobic bacteria factinomyceioma) have been inoculated by Iminor trauma. There is drainage containing the charac teristic granules through sinus tracts extending to the shin." The etiologic agents of mycetomna mast common!y seen in Brasil ane the fungi Pseudallescheria. boydi Cephalosporium (Acremoniuiny sp, Maducella grisea and ‘the actinomycete Nocardia brasiliensis” Microscopic examination of pus. exsudete or biopsy material revealsForan Macs Kroc Farandes sia evela grdnulos, que so os principals indicadores diag- ‘ésticos, A observagdo do tamanho, forma, core consistén- ia dos grinulos pode levar freqlentemente 0 investizador ‘experimentada a idenificar a espicie de fungo. Para exame dirt, os grinuls podem ser montados ero ua lamin com uma gota de KOH a 10% © esmagados com laminvls. Podem-se observar grénulos cumicsticos contendo hifas de dois a Gym de largura que geralmente apresentam células grandes, edemaciadas, globosas (com até 15jm ou mais) na ‘margem.” Os grinulesactinomieéticos possuem laments ‘com didmetro de mei a (sam, bem como elements coc6i- des ebacilares; filaments finos eramficados Gram-posti- vos, em fundo de material Gram-negativo ‘Actinomicose € uma infecslo crdnica supueaiva causada por um dos varios actinomicetos anaerdbicos ou microaeriilos (fetomyces isracite Actinomyces bovis) nenhium dos quais causa actinomicetoma." A actinomicose aparece na regido cervicofacial, peito ou abdémen com seis de deenagem das visceras. Na bidpsia, ha prnals em focos piogénieos ciundados par fibroseeinflamagto crS- nies, A colorago pelo Grain dos gros esmagades distingue prontamente as granulos de betriomicose daqueles da acti- homicose, O Gram de teide (Gram Weigert ou Brown © ‘Brenn} é eficaz para revelar as ramificagtes delicadas das hitas de actinomicetos¢ cocos ou bacilos de bottiomicose. [No paciente em andlise nfo havia drenagem de ed tls, mas as cultures da massa revelaram creseimento de Pseudomonas aeruginosa, Proteus mirabilis Escherichia coli ‘© Gram dos cortes histolépieosrevelou orgunismos Gram-positivos dentro do gr. ‘Staphylococcus aureus & 0 patégeno bacteriano habitual, mas a8 bastécias Grotn-negativas também podem ser responsiveis*"'* ou pode ser identifcado mais de um ‘agente caustl,” Os achados bacterioligicos. isto, covos Gram-positives/erinulos Gram-positivos © bastonetes Gramn-nepatives/Prendlomonas, Proteus, Escherichia estto em acorde, ‘Aa redor do grimula se apresentava uma camade de neutrfils, linfcitos, eosindfiles, eélulas plasuéticas Aibroblastos, além de histiécitose cul pigentes de corpo estranho. O depésito de substincia eosinofiliea na perifevia os grinulos botriomicéticas, em forma de coroa ou de fstruturas radiadas, represents o fendmeno Splendore- Hoeppi°*" "5! Essa estratura eosinoflca a0 redor dos srinulos & PAS-positiva, No pacionte aqui tatado, essa ‘leporigan de material eosinolica 40 redor dos microorga nisms foi observada pela coloragdo H&E, ‘0s grinulos nem sempre S30 visiveis em todos os comes mictoscépicos; no caso em questo foram necessi- ries virios cortes de tecido mentados em parafina pact ‘bservar apenas um gringlo, Deve ser enfatizado que « amstia foi excisadacirurgicamente Nocardia brasiliensis pode causar uma info sub- ‘nen ap trauma, doen gue lembra muito a esporotico- An rae Dermal Rio de Sei, P7857 jan se 2002. the presence of the granules which are the primary dio nostic indicators. Observation of the size, shape, color ‘and consistency of the granules may often lead the expe Fenced investigator 10 the identification of the fungal species. For dvect examination, granules can be mou ted in a drop of 10% KOH on a'slide and crushed under 4 coverslip. Eumycotic granules can be seen with 2 - 10 {6 um wide hyphae thot often have large. globose swollen cells (up 10 13 yim or more) at the margin” Aetinomycotic gramules have filaments with a diameter 0f0.5 0 1 jm as well as caccoid to bacillary elements Gram-positive, narrow branching filaments in a back ground of Gram-negative cementing material Actinomycoris ks @ chronic ‘uppurative infection caused hy one of several anaerobic or microaerophilc act- Inomycetes (Actinomyees israeli and Actinomyces bovis) none of sshich couses actinomycetoma.” Aetinomycosis appears in the cervicofacial area, chest ot abdomen ard raining sinuses ariginate inthe viscera, On biopsy. there fare granules in pyogenic foct surrounded by fibrosis ard chronic inflammation. Gram stain ofa crushed granule re: thy distinguishes the granules of btryaomycosis rom those fof actinomycete, A tissue Gram siain (Gram Weigert or Brown and Brenn) is effective for revealing the branching delicate hyphae of the actinomyeete and the cocci or baci 1 of banyomycass ‘In our patient there was no discharge of gromules, but cultures of the mass yelded Pseudomonas aeruginosa Proteus mirabilis and Escherichia coi “A Gram stain ofthe histological section disclosed Gram postive organisms inside the gramme. Staphylococcus aureus isthe usual bacterial patho gen but Gram-negative bacteria also ea be responsible "® ar more than one causative agent can be identified" Our bacteriological findings, that is, Gram-positive cacci/Gram-pasitive granules and Gram-negative ‘rads/Pseudomonas, Proteus, Escherichia are in accordance. In tissue sections immediately surrounding the gr ule was a layer of neutrophils. domphocytes, eosinophils plasma cel, fibroblasts as well as histioestes and foreign body giont cells, The depasit of easinophilic substance on the periphery of botryompcotie granules with a crow shape or ratioted siructures represent the Splendore- Hoepols phenomenon. °°" This eosinophilic shell “round the granule PAS positive. In our patient this depo- sition of eosinophilic material aroud the mcraorganiss ‘was observed in Hf & stn, The gramules are not always visible in every micros copie section: in our case many sections though the paral fin-mounted nssue were necessary to see anty one granule In gection, ft must be emphstze shar the sample was @sur~ ically excised mass. ‘Nocardia brasiliensis may couse @ Subcutaneous Infection following trauma, a disease closely. rexembling sporotrichosis. The distinction between myceroma undRemand, Mace, Krackis & Forandes Se. distngio entre mieetome e outasinfeopbes causadas pelos mesmos agentes repousa sobre a formario de grinulos {ea demonstragdo de foco subcutineo deinfecgo primera” Selecionou-se ciprofloxacina de acordo com 0 anibiograma por ser eficaz também contea bactérias Gram-positivas, inclindo Staphylococcus aureus. Foi dada preferéncia ini- cialmente& vi parenteral devido & magnitude das lesses, [Neste caso 2 drenagem cirirgica ea ansbioticotera- pia de acordo com 0 antibiograma resltaram em recup (80 completa. Os autores consideram que o tratamento de longa duragéo (IL meses) e a boa tolerncia e adesto do paciente foram devisivos para o sucesso dos resultados. [REFERENCIAS / REFERENCES LBonifz A, Carasco E. Botriomicose. Int J Derm 1996; 35(0)381-8 2Cont-Diaz 1A, Almeida EA, Roadan M, Algorta G, Otero M. Cutaneous Doaryomyeosi. Repiet of the two fist Uriguayan ‘ass. 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