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LETTER TO THE EDITOR
Genitourinary paracoccidioidomycosis complicated with urinary outflow obstruction—a report of two cases and a review of the literature
Paulo Henrique Goulart Fernandes DiasI, Marcelo ZeniI, Guilherme R TaquesII, Frederico R RomeroI, Fernando Henrique Tremel BuenoI, Luiz Carlos Almeida RochaI
I Urology Department, Universidade Federal do Paraná, Curitiba, Paraná, Brazil
II Pathology Department, Universidade Federal do Paraná, Curitiba, Paraná, Brazil
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    "textoCompleto" => "<span class="elsevierStyleSections"><span id="cesec10" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="cestitle10">INTRODUCTION</span><p id="para10" class="elsevierStylePara elsevierViewall">Paracoccidioidomycosis &#40;PCM&#41; is a systemic granulomatous mycosis caused by the fungus <span class="elsevierStyleItalic">Paracoccidioides brasiliensis</span>&#46; Also known as South American blastomycosis and Lutz-Splendore-Almeida disease&#44; PCM is endemic in South and Central America&#44; especially in Brazil&#44; Venezuela&#44; Colombia&#44; Ecuador&#44; and Argentina&#46;<a class="elsevierStyleCrossRef" href="#bib1">1</a> Its chronic form&#44; which accounts for more than 80&#37; of cases&#44; usually affects men between 29 and 60 years old&#8212;predominantly rural workers&#8212;and it is characterized by polymorphic lesions that may affect any organ&#44; in particular the skin&#59; the lymph nodes&#59; the lungs&#59; the oral&#44; nasal&#44; and gastrointestinal mucosa&#59; the adrenals&#59; and the central nervous system&#46;<a class="elsevierStyleCrossRef" href="#bib1">1</a> The genitourinary tract is the least commonly involved system in chronic PCM&#46; In the present paper&#44; we describe two cases of genitourinary PCM located in the penis and the prostate that were treated at our institution&#44; and we review the pertinent literature&#46;</p></span><span id="cesec20" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="cestitle20">CASE REPORT</span><span id="cesec30" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="cestitle30">Case 1</span><p id="para20" class="elsevierStylePara elsevierViewall">A 56-year-old white male presented with a 3-month history of hesitancy&#44; weak urinary stream&#44; intermittency&#44; nocturia&#44; incomplete voiding&#44; terminal dribbling&#44; dysuria&#44; lower abdominal pain&#44; and painful lesions on the penis&#44; along with a lower lip lesion&#46; His history was positive for cigarette smoking&#44; arterial hypertension&#44; and cerebrovascular disease&#46; He had worked in agriculture for 12 years but had been living in urban areas for the past 24 years&#46; On physical examination&#44; the patient had poor hygiene and nutritional conditions&#44; body lesions suggestive of scabies infestation&#44; and a 1&#46;5-cm ulcerated lesion on the lower lip&#46; Pulmonary auscultation revealed a diffuse decrease in breath sounds&#46; A genital exam demonstrated an ulcerated lesion partially exposed on the glans penis caused by poor retractability of the foreskin &#40;<a class="elsevierStyleCrossRefs" href="#fig1-cln_65p1207">Figures 1 and 2</a>&#41;&#59; the scrotum and prostate exams were normal&#46; Laboratory tests revealed normal renal function&#44; a negative HIV ELISA-test&#44; a negative urine culture&#44; a negative PPD&#44; and negative acid-fast bacilli in the sputum&#46; Chest radiography showed diffuse and nodular infiltrates&#46; The patient underwent dorsal incision of the foreskin and Foley catheterization&#44; which immediately drained 1&#44;500 cc of concentrated urine&#46; A penile biopsy was positive for PCM &#40;<a class="elsevierStyleCrossRefs" href="#fig3-cln_65p1207">Figures 3 and 4</a>&#41;&#46; He underwent antibiotic treatment with sulfamethoxazole &#40;800 mg&#41; and trimethoprim &#40;160 mg&#41; twice a day for 6 months&#46; The Foley catheter was removed after 4 weeks of antibiotic treatment&#44; with satisfactory voiding&#46; Unfortunately&#44; the patient was lost to follow-up before the antibiotic treatment was completed&#46;</p><elsevierMultimedia ident="fig1-cln_65p1207"></elsevierMultimedia><elsevierMultimedia ident="fig2-cln_65p1207"></elsevierMultimedia><elsevierMultimedia ident="fig3-cln_65p1207"></elsevierMultimedia><elsevierMultimedia ident="fig4-cln_65p1207"></elsevierMultimedia></span><span id="cesec40" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="cestitle40">Case 2</span><p id="para30" class="elsevierStylePara elsevierViewall">A 59-year-old white male living in a rural area presented with a 3-year history of obstructive and irritative lower urinary tract symptoms&#44; which culminated in acute urinary retention requiring emergency suprapubic cystostomy&#46; His medical history was positive for arterial hypertension and pulmonary PCM treated 2 years earlier with sulfamethoxazole&#47;trimethoprim&#46; On physical examination&#44; the left testis was enlarged and painful&#44; with ipsilateral&#44; painless lymph node enlargement&#46; A prostate exam revealed a grade 1 smooth&#44; elastic&#44; and painless prostate&#46; Laboratory tests showed normal renal function&#44; a PSA concentration of 2&#46;47 ng&#47;mL&#44; proteinuria&#44; leukocyturia&#44; a negative urine culture&#44; a negative PPD&#44; and negative acid-fast bacilli in the sputum&#46; With the suspicion of genitourinary PCM&#44; a US-guided prostate biopsy was performed&#44; which revealed granulomatous chronic prostatitis with fungal structures consistent with PCM &#40;<a class="elsevierStyleCrossRefs" href="#fig5-cln_65p1207">Figures 5 and 6</a>&#41;&#46; He underwent treatment with itraconazole &#40;200 mg&#47;day&#41; for 6 months&#44; followed by transurethral resection of the prostate&#44; which confirmed complete resolution of PCM upon a pathological examination of the specimen&#46; The cystostomy tube was occluded and removed after satisfactory micturition&#46; Improvement of testicular enlargement with itraconazole provided indirect evidence of testis PCM&#44; but the diagnosis was not confirmed pathologically&#46; At 3 years of follow-up&#44; the patient shows no evidence of recurrence&#46;</p><elsevierMultimedia ident="fig5-cln_65p1207"></elsevierMultimedia><elsevierMultimedia ident="fig6-cln_65p1207"></elsevierMultimedia></span></span><span id="cesec50" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="cestitle50">DISCUSSION</span><p id="para40" class="elsevierStylePara elsevierViewall">Genitourinary PCM is rare&#44; with a clinical incidence of 1&#46;6&#37; to 2&#37; among individuals with chronic PCM&#46; When genitourinary PCM is present&#44; the infection is most common in the scrotum&#44;<a class="elsevierStyleCrossRefs" href="#bib2">2-5</a> the epididymis&#44;<a class="elsevierStyleCrossRefs" href="#bib2">2&#44;6</a> the testis&#44;<a class="elsevierStyleCrossRefs" href="#bib2">2&#44;5&#44;7</a> the prostate&#44;<a class="elsevierStyleCrossRefs" href="#bib2">2&#44;6&#44;8&#44;9</a> and the penis&#46;<a class="elsevierStyleCrossRef" href="#bib2">2</a> The exact number of cases&#44; however&#44; cannot be precisely assessed because of underreporting&#44; the reporting of cases in non-indexed local journals&#44; nomenclature problems&#44; and non-routine use of mycological tests in patients with suspected infection&#46;</p><p id="para50" class="elsevierStylePara elsevierViewall">PCM is generally considered to result from the inhalation of spores released into the air&#44; with the lungs being the usual site of primary infection&#46; In the acute&#47;subacute form&#44; superficial and&#47;or visceral lymph node enlargement is the major presentation&#46; In the chronic form&#44; which occurs in 80&#37; to 90&#37; of cases&#44; infection results from reactivation of quiescent lesions&#46; Chronic PCM can manifest after long latency periods&#44; up to 30 years after the individual has left an endemic area&#46;<a class="elsevierStyleCrossRef" href="#bib1">1</a> The lung is the only organ affected in 25&#37; of chronic PCM cases&#44; while the remaining cases are multifocal&#44; with mucocutaneous lesions in the mouth&#44; nose&#44; ears&#44; pubis&#44; and perineal regions&#46; Primary mucocutaneous involvement is uncommon and may be characterized as an inoculation lesion&#46;<a class="elsevierStyleCrossRef" href="#bib4">4</a></p><p id="para60" class="elsevierStylePara elsevierViewall">The age of presentation of genitourinary PCM is often between 30 and 75 years&#44; which is somewhat above the normal range for chronic PCM&#46;<a class="elsevierStyleCrossRef" href="#bib3">3</a> It is almost exclusively a disease of men&#46; Genitourinary PCM is extremely rare in females because feminine hormones presumably protect women of fertile age&#46;<a class="elsevierStyleCrossRefs" href="#bib1">1&#44;10-12</a> Patients almost always present with multifocal disease&#44; with lung infiltrates or other mucocutaneous lesions suggestive of PCM&#46;<a class="elsevierStyleCrossRefs" href="#bib2">2&#44;3</a> Patients usually present with chronic illness&#44; complaining of anorexia&#44; muscle weakness&#44; and weight loss&#46;<a class="elsevierStyleCrossRefs" href="#bib2">2&#44;6&#44;13</a> As with other chronic PCM manifestations&#44; lymphadenopathy is uncommon in genitourinary PCM&#46;</p><p id="para70" class="elsevierStylePara elsevierViewall">Scrotal lesions may present as frequently painful erythematous plaques with well-defined edges and as ulcerated&#44; granulomatous&#44; or verrucous lesions that may be associated with lesions in the inguinal&#44; perineal and perianal regions&#46;<a class="elsevierStyleCrossRefs" href="#bib3">3-5</a> Lesions are usually clean&#44; without secondary infection&#44; and with characteristic fine granulations and hemorrhagic dots&#46;<a class="elsevierStyleCrossRef" href="#bib1">1</a> Scrotal PCM usually results from hematogenic dissemination&#44; as with other forms of chronic PCM&#44; but may occasionally be a consequence of the anal toilet practice of using leaves from contaminated plants&#46;<a class="elsevierStyleCrossRefs" href="#bib1">1&#44;4</a> Diffuse eruptions may simulate syphilis&#44; psoriasis&#44; and lymphomas&#46; If located&#44; they should be differentiated from leishmaniasis&#44; sporotrichosis&#44; tuberculosis&#44; and chromomycosis&#46;<a class="elsevierStyleCrossRef" href="#bib1">1</a></p><p id="para80" class="elsevierStylePara elsevierViewall">Unilateral&#44; hardened&#44; painful enlargement of the testicle and&#47;or epididymis are the main manifestation of PCM epididymo-orchitis&#46;<a class="elsevierStyleCrossRefs" href="#bib2">2&#44;6</a> Epididymitis may coexist with a normal ipsilateral testicle&#46;<a class="elsevierStyleCrossRefs" href="#bib6">6&#44;14</a> A fistulous tract draining purulent secretions rich in parasites may be present&#46;<a class="elsevierStyleCrossRefs" href="#bib1">1&#44;2</a> The differential diagnosis includes specific epididymo-orchitis &#40;e&#46;g&#46;&#44; tuberculosis&#44; brucellosis&#41; and testicular neoplasms&#46;</p><p id="para90" class="elsevierStylePara elsevierViewall">Lesions on the penis may involve the external skin and&#47;or the internal aspect of the foreshaft and the glans penis&#46;<a class="elsevierStyleCrossRef" href="#bib15">15</a> These lesions may be ulcerated&#44; with infiltrated borders and hemorrhagic or seropurulent secretions&#46; They usually do not reproduce the fine microgranulation and hemorrhagic dots observed in mucous membranes&#46; Although penile PCM is usually asymptomatic&#44; the site may be tender&#44; and lower urinary tract symptoms may be present if the infection is located near the external urethral orifice&#46; The differential diagnosis includes penile cancer and ulcerated&#47;papillary sexually transmitted lesions &#40;e&#46;g&#46;&#44; HPV&#44; syphilis&#44; genital herpes&#44; chancroid&#44; lymphogranuloma venereum&#44; and donovanosis&#41;&#46; There is no evidence that penile PCM may be sexually transmitted because the fungus is not in an infectious state &#40;yeast form&#41; inside humans&#46;<a class="elsevierStyleCrossRef" href="#bib1">1</a> The infecting form of the fungus is the mycelium form&#44; found only in nature at a temperature of 25&#176;C&#46;</p><p id="para100" class="elsevierStylePara elsevierViewall">Most lesions in the prostate are asymptomatic and are found during necropsy&#44;<a class="elsevierStyleCrossRef" href="#bib9">9</a> but they may also cause symptoms of lower urinary tract obstruction&#46;<a class="elsevierStyleCrossRefs" href="#bib6">6&#44;8</a> Prostatic lesions may be palpated in a normal or enlarged prostate as indurated lesions during digital rectal examination&#44; mimicking nonspecific chronic prostatitis or prostate cancer&#46;<a class="elsevierStyleCrossRef" href="#bib8">8</a> Associated with prostatic PCM&#44; unilateral kidney exclusion may also occur&#44; presumably as a consequence of hematogenous dissemination of the fungus leading to autonephrectomy&#44; similar to the process observed in kidney tuberculosis&#46;<a class="elsevierStyleCrossRef" href="#bib8">8</a> Prostate PCM has also been described as being involved secondarily via the canalicular path from the epididymis&#46;<a class="elsevierStyleCrossRef" href="#bib16">16</a></p><p id="para110" class="elsevierStylePara elsevierViewall">The diagnosis is made by isolation of the fungus by either histopathological&#44; cytopathological&#44; or direct mycological examination after puncture biopsy or culture&#46; A biopsy with hematoxylin-eosin stain shows a granulomatous pattern&#44; with findings of a double-wall parasite with simple or multiple gemmulation&#46; The granuloma is rich in epithelioid and giant cells&#44; some containing variable amounts of parasites&#46; If few in number&#44; the parasites are better visualized through special stains&#44; such as periodic acid-Schiff or Gomori-Grocott&#46; Serology in PCM has applications in diagnosis and follow-up&#46; PCM treatment includes the use of antifungal drugs&#44; nutritional support&#44; the prevention of opportunist diseases&#44; and treatment of eventual sequelae and complications&#46;<a class="elsevierStyleCrossRef" href="#bib1">1</a></p><p id="para120" class="elsevierStylePara elsevierViewall">Urinary outflow obstruction is an uncommon presentation of paracoccidioidomycosis&#46; This clinical entity can manifest even long after the patient has left an endemic area and should always be born in mind as a differential diagnosis in the presence of multifocal mucocutaneous&#44; pulmonary&#44; and&#47;or genitourinary lesions&#46;</p></span></span>"
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Article information
ISSN: 18075932
Original language: English
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es en pt

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