Acute Disseminated Paracoccidioidomycosis Septic Shock

September 1988, Vol. 27, No. 7

Cameo

Acute Disseminated Paracoccidioidomycosis Septic Shock RUBEM DAVID AZULAY, M.D., MARIA BEATRIZ VELLOSO, M.D., SUELI YURIKO SUCUIMOTO, M.D., CLEIDE EIKO ISHIDA, M.D., AND ANTONIO CARLOS PEREIRA, JR., M.D.

From the Department of Dermatology, Hospital Universitario C/ement/no Fraga Filho, Universidade Federal do Rio de Janeiro, Rio de Janeiro, Brasil

A

l 5 year-old single girl, a student, mestizo, and Brazilian, came from the State of Maranhao to our hospital due to generalized skin lesions, pains all over the body, and fever since August 1984. The physical examination showed discrete enlargement of the lymph nodes located symmetrically in the cervical, submandibular, axillary, paravertebral, and inquinal regions and hepatomegaly. The skin lesions were vegetating, yaws-like lesions, some ulcerated and others covered by crusts 0.5-2.0 cm in diameter, localized in the face, trunk, and extremities; subcutaneous nodules 0.5-2.0 cm in diameter all over the body; pustules in the face, trunk, and extremities; and ulcerations in the palate, jugal mucosa, and oropharynx. The articulations were voluminous and painful. The patient could hardly move in the bed. The x-rays of the bones showed osteolytic lesions in both clavicles, humerus, radius, ulna, femur, tibia, and fibula. Pathologic fractures in both distal extremities of radius and ulna were observed. The x-rays of the lungs showed an image of residual fibrosis in the superior third of the right lung.

(April 1985). In January 1986, after 1 year of treatment, the immunodifusion was negative. The paracoccidioidin test result was negative. Tests for cellular immunity were negative for vaccinia, trichophytin, and brucell but positive for DNCB and candidin. The hemogram showed hypochromic microcytic anemia. Hepatic function test results were SGOT, 51 U/L; SGPT, 45 U/l; and alkaline phosphatase, 415 U/L. Renal function was normal. Evolution Under treatment with sulfa, the patient's general health became worse. She developed shock, which was considered septic due to P. brasiliensis. Besides the treatment for the shock, amphotericin B was started. In the meantime, the patient presented: 1st- and 2nd-degree atrioventricular block secondary to two cardiorespiratory arrests, pericardial effusion, and pulmonary edema. Cardiac and pulmonary insufficiencies appeared. She was transferred to the unit of intense treatment (UIT) where she was placed on a ventilator and heart monitor. She lost 20 kg of weight. Additional treatment with ketoconazole (400 mg/day) was introduced, and the patient was recovered. The skin lesions healed, she gained weight, and the hepatic function became normal, but the bone lesions persisted. Now she is under treatment with sulfamethoxypyridazine (2 g/day).

Laboratory Findings

Comments

Thirteen blood samples were examined during the patient's hospitalization. All of them were negative for bacteria, but one was positive for Paracoccidioides hrasiliensis. Direct examination and culture from a cutaneous lesion were positive for P. brasiliensis. Histopathology of cutaneous lesions showed tuberculoid granuloma and exsudative polymorphonuclear inflamation and numerous round forms of P. brasiliensis. Serologic test results were positive immunodiffusion (Ouchterlony), counterimmunoelectrophoresis 1:8, macrocomplement fixation 36.0, and immunofluorescence 1:512

Paracoccidioidomycosis was described by Adolpho Lutz^ in 1908 in Brazil. Since then, cases of this disease have been described in all countries of Latin America except Chile. It is also called Brazilian blastomycosis, South American blastomycosis, and Lutz's mycosis. The disease is a systemic mycosis produced by Paracoccidioides brasiliensis, which lives as saprophytes in the soil. It is an air-borne infection in humans. Depending on the immunologic status ofthe patient, one may see (1) subclinical infection, which is revealed by positive paracoccidioidin intradermic test; or (2) symptomatic disease, which may affect practically all organs and tissues.^

Address for correspondence: R. D. Azulay, Av. Atlantica, 3130, Rio de laneiro, 22070, Brasil.

No. 7

PARACOCCIDIOIDOMYCOSIS: SEPTIC SHOCK

This is a peculiar case of paracoccidioidomycosis for the following reasons: 1. The patient had a septicemia due to P. brasiliensis. Very seldom have cases in the literature had positive hemocultures.^ 2. This seems to be the first case of paracoccidioidomycosis that produced septic shock. The positive hemoculture for P. brasiliensis and the negativity for bacteria suggest that etiology for the shock. 3. The bone lesions were numerous and severe, including pathologic fractures. The bone lesions are rather rare (5.9% of 254 patients)" and due to hematogenous dissemination. In addition, the bone lesions did not heal under a long treatment

Azulay et al.

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(2 years), which disagrees with other observations.'' References 1. Lutz A. Uma mycose pseudococcidica localizada na boca e observada no Brasil. Contribuigao aoconhecimento das hyphoblastomycoses americanas. Brasil-med. 1908;22:121124;141-144. 2. Azuiay RD. Contribui^ao ao estudo da Micose de Lutz. Thesis. 1950. 3. Madeira )A, Lacaz CS, Forattini OP. Consideragoes sobre um caso de blastomicose generalizada, com o isolamento do Paracoccidioides brasiliensis a partir do sangue circulante. Hospital. 1947;31:845-954. 4. Silva AF. Contribuifao aos estudos da paracoccidioidomicose no Parana e das lesoes osseas e articulares paracoccidioidicas. Thesis. 1976. 5. Mello Filho A, Sandreschi ET, Rosa IP, et al. Osseous paracoccidioidomycosis: residual lesions after recovery in a patient from 10-16 years old. Castellania. 1975;3:151-154.

Spanish Fly

Spanish fly, or cantharides, is a powder derived from certain crushed beetles which contain less than 0.6% ofthe active ingredient, cantharidin, first isolated by Robiquet in 1810. Cantharidin is the anhydride of a simple aromatic acid, cantharidic acid, which forms biologically active soluble salts with alkali. The acid itself is soluble only in fats, ether, and alcohol. This substance is well known as a vesicant and as a potent irritant to the gastrointestinal and genitourinary mucosa. The lethal dose of cantharidin has been reported to be 10 mg; however, two cases of survival following ingestion of more than 75 mg are on record. The symptoms of cantharidin poisoning are stomatitis with excessive salivation, the appearance of blisters of the oropharynx, nausea, emesis, crampy abdominal pain, bloody diarrhea, and most prominently, increased urinary frequency, urgency, dysuria, and hematuria, since the drug is excreted in the urine. Terminal signs and symptoms are priapism, cardiorespiratory collapse, coma, and death. Cantharidin is also directly toxic to the kidneys, and causes glomerular and tubular necrosis without interstitial involvement. All the beetles which contain cantharidin belong to the order Coleoptera, family Meloidae. It is estimated that there are over 2000 different species in the world which contain this active agent, and over 200 of which exist within the United States. Mylabris cichorii contains the highest percentage of cantharidin; however, the most common source is Cantharis vesicatorea. In the United States, Epicauta vittata can be found in abundant quantities in the summer months, particularly in the western part of the country where they infest tomato plants and are a serious farm pest. Historically, cantharides has been known since Hippocrates, who advocated its use in the treatment of dropsy. To the layman it is known as an aphrodisiac; yet even in the earliest animal experimentations only its toxic manifestations are documented, with priapism as a terminal event. Currently, it is only used in the experimental study of its vesicant action on the skin, as in the treatment of superficial lesions, such as verrucae vulgaris. It should be borne in mind that systemic toxic effects via cutaneous absorption may present a definite hazard in the therapeutic use of cantharides. Recently, Muhsen treated 222 patients with cantharides for bilharzic calcification ofthe bladder mucosa, with some benefit in 1 74 (80%), and he suggests its use in the treatment of interstitial cystitis. In the past, cantharidin was used in veterinary medicine; however, since toxic nephritis and renal failure in subject animals are frequent complications, this application no longer is recommended.—Presto AJ III, Muecke EC. A dose of Spanish fly. JAMA 1970;214;591^592.