Malacoplakia is a rare granulomatous disease well described in the urinary tract if it were not that which rarely involves the lung We report for the first time.

Malacoplakia is a rare granulomatous disease well described in the urinary tract if it were not that which rarely involves the lung We report for the first time, to our knowledge, tracheal localization of this unusual disorder. The larynx and probably kidneys were also involved. Differential diagnosis, physiopathology, and treatments are discussed.

Malacoplakia is a rare chronic granulomatous inflammation that consists of tumor-like accumulation of macrophages with characteristic features--the Von Hansemann solitary abode; squalids associated with large intracellular and extracellular calcified structures--the pathognomonic Michaelis-Gutmann (MG) bodies.[1,2]

First described in the subdued urinary tract, malacoplakia has been reported in a wide variety of organs and can offer almost anywhere.[2] In 75 percent of cases, malacoplakia is associated with Escherichia coli infection, however other infection with bacteria, including Mycobacteria and fungi, have been reported.[1,3] An underlying debilitating state and immunosuppression are as a common thing [i]or[/i] matter noted and can play a character in the pathogenesis of this disease.[1]

Pulmonary involvement is not common[4] and sole eight cases were previously described.[5-11] We report a unique case of laryngotracheal malacoplakia, associated with E coli pneumonia. To our knowledge, this is the first reported case of tracheal localization of this unusual disorder.

CASE REPORT

A 32-year-old white woman, a heavy smoker (40 cigarettes a day for 10 years), with no relevant medical history, was admitted to the hospital in December 1991 for treatment of a chronic pulmonary infection and hemoptysis. Since March 1991 she had been treated for returning episodes of fever, cough, and festering expectoration with various antibiotics, yet the symptoms reappeared after discontinuing therapy. In October 1991 she bring outed hemoptysis and complained of hoarseness. A chest radiograph was finally performed in December 1991 and showed right lower lobe pneumonia and a lingular infiltrate. At this time, findings from the physical examination were unremarkable do not include for fever (38 [degrees] C) Laboratory studies revealed the following: normal kindred gases; WBC, 6 x [10sup9]/L (071 neutrophils, 011 lymphocytes); CD4 lymphocyte account 0.28 x [10.sup.9]/L; erythrocyte sedimentation rate, 85 mm/h; normal comes of hepatic and renal function tests; normal accrues of urinanalysis; negative human immunodeficiency virus (HIV) serologic test; and normal immunoglobulin flats Direct laryngoscopic examination was performed and showed a tumor-like formation upon the anterior portion of the left arytenoid cartilage and upon the posterior portion of the false vocal cord. fulvid raised nodules were also seen just above the anterior portion of the right vocal cord. Bronchoscopy revealed multiple, fulvid smooth, irregular, small plaques (3 to 4 mm) upon the posterolateral face of the trachea without apparent involvement of the bronchial tree (Fig 1)

Pathologic examination of mucosa from laryngeal and tracheal biopsy specimens showed in the lamina propria commingling sheets of large histiocytes with abundant, finely granular, and vacuolated cytoplasm (Von Hansemann cells) There were also intracellular and extracellular, globular target-like, and calcified structures that correspond to the pathognomonic MG bodies (Fig 2) These findings were consistent with a diagnosis of malacoplakia. Special stains for acid-fast organisms, bacteria, and fungi were negative if it were not that electron microscopy revealed intracytoplasmic bacteria (Fig 3)

agricultures of protected specimen brush in the lower right lobe grew E coli (3 x [10sup4] ml) sensitive to several antibiotics. civilizations of tracheal biopsy samples were positive for the same E coli. refinement of bronchial aspirate grew Mycobacterium avium-intracellulare MAIC).

Urinary refinement was negative but abdominal echography and comput tomographic scan showed abnormal kidneys with a pseudotumoral aspect. The right psoas muscle was also invaded by the agency of a tumor mass close to the kidney. A biopsy specimen of these lesions could not be obtained because the patient refused further investigations. An antibiotic treatment based forward sensitivity patterns of E coli was begun with ofloxacin, 400 mg/d Unfortunately, the patient was unavailable for follow-up 1 month after hospital discharge, and therefore long-term evolution could not be evaluated.

DISCUSSION

To our knowledge, this is the first report of tracheal localization of malacoplakia. Indeed, this disease is known to affect principally the bladder and the reasonable urinary tract where 60 percent of cases present itself but many other organs can be involved.[1,2] Pulmonary malacoplakia has already been reported nevertheless it remains rare.[4] single eight cases were previously described.[5-11] Endobronchial malacoplakia has been reported as an occlusive mass,[6,8] nevertheless tracheal involvement has never been described before (to our knowledge). In public patient, tracheal lesions resemble those reported in other false organs like bladder or colon[1] No other macroscopic lesions could be erect on careful bronchoscopic examination of the bronchial tree However, distal airways involvement is possible and could encourage obstructive pneumonia. A laryngeal lesion was also erect Laryngeal malacoplakia was previously described in merely one patient, a heavy smoker without other predisposing factors. This patient did not have extralaryngeal localization and excision of these lesions l to definitive cure[12] Initially the gros appearance of the laryngotracheal lesions l us to a diagnosis of papillomatosis which can present itself in the same location and can have similar clinical presentation, still microscopic features are quite different.[13]

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