INTRODUCTION

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Bronchoalveolar lavage (BAL) findings in sarcoidosis include increased numbers of helper T lymphocytes, increased CD4/ CD8 ratios, activated alveolar macrophages, and diverse lymphokines, monokines, and biochemical markers (1). Although important for understanding the pathogenesis of sarcoidosis, these BAL markers are not sufficient for diagnosis. Diagnosis depends on the demonstration of noncaseating granulomas in lung tissue, most often by transbronchial biopsy (1). We present an unusual case of sarcoidosis in which noncaseating granulomas were recovered in BAL fluid.

	CASE REPORT

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A 29-year-old African-American male presented to the Medical College of Georgia (Augusta, GA) with a chief complaint of dyspnea on exertion. Symptoms had progressed over a 6-mo period, to the point that he could no longer walk 20 yd without having to stop because of severe shortness of breath. He also complained of nonproductive cough. Before the onset of illness, he had been in excellent physical condition and was involved in sports. A local physician had treated the patient with multiple antibiotics and short courses of corticosteroids without improvement. He denied fever, chills, night sweats, wheezing, or hemoptysis. There was no change in appetite or weight loss. There was no history of exposure to tuberculosis.

Past medical history was notable for a recent flank laceration, which was healing appropriately. Otherwise there was no history of lung disease or other medical problems. He denied recent travel and was not taking any medications. The patient was a nonsmoker and denied alcohol abuse or use of illicit drugs. He had worked for the past 2 yr at a local foundry, where he was exposed to silica and did not wear a mask. Family history was negative for lung disease.

Physical examination revealed a large, healthy-appearing black male with resting tachypnea. Pulse was 110 beats/min, blood pressure was 140/70, respiratory rate was 26 bpm, and temperature was 37° C. Weight was 242 lb. The skin was normal. There was no lymphadenopathy. Chest examination revealed fine basilar crackles bilaterally. The heart was normal except for tachycardia. An abdominal examination revealed no hepatosplenomegaly. Extremities were normal and there was no clubbing.

Basic laboratory studies, including a complete blood count (CBC), electrolytes, and liver function studies, were normal. Angiotensin I-converting enzyme (ACE) was normal at 34 U/L (range, 8-52 U/L) and ionized calcium was within normal limits. A test for human immunodeficiency virus (HIV) was negative.

Chest X-ray showed diffuse, bilateral reticulonodular infiltrates (Figure 1). Pulmonary function tests demonstrated a moderate restrictive defect: FVC, 1.34 L (26% predicted), FEV₁, 1.14 L (27% predicted), and FEV₁/FVC, 85%. Lung volumes showed a total lung capacity (TLC) of 3.39 L (52% predicted), and the diffusing capacity (DL_CO) was 11.57 ml/ min/mm Hg (30% predicted). Arterial blood gas on room air was pH 7.38, Pa_CO2 was 38.7 mm Hg, and Pa_O2 was 56 mm Hg.

View larger version (127K): [in this window] [in a new window]   Figure 1.   Chest X-ray showing diffuse bilateral reticulonodular infiltrates.

Fiberoptic bronchoscopy revealed red edematous airways with a "cobblestone" appearance suggestive of endobronchial sarcoidosis. BAL was performed in the right middle lobe, using five 20-ml aliquots of saline. BAL fluid was collected in a trap, which was removed from the bronchoscopy suction apparatus before lung biopsy. After BAL, four transbronchial biopsies were performed in the right middle lobe under fluoroscopy. Cultures and stains of BAL fluid were negative for infectious organisms. The BAL fluid was centrifuged, and smears of the sediment were fixed in 95% ethanol and stained with Papanicolaou stain. The smears contained widely scattered tight aggregates of epithelioid cells and scattered Langhans' multinucleated giant cells (Figure 2). Otherwise, the BAL fluid contained numerous alveolar macrophages and a moderate number of lymphocytes. A cell differential showed 57% macrophages, 34% lymphocytes, 7% neutrophils, and 2% eosinophils. Transbronchial biopsies showed extensive noncaseating granulomas that were negative for mycobacterial and fungal organisms by special stains. The patient was diagnosed with sarcoidosis and was started on prednisone (60 mg daily) with gradual improvement in clinical symptoms and spirometry.

View larger version (125K): [in this window] [in a new window]   Figure 2.   Bronchoalveolar lavage (BAL) fluid stained with papanicolaou stain (high power) showing a tight aggregate of epithelioid cells (arrow) and a Langhans' giant cell (arrowhead ).

	DISCUSSION

TOP ABSTRACT INTRODUCTION CASE REPORT DISCUSSION REFERENCES

BAL is thought to mirror parenchymal inflammation in the interstitial lung diseases (4, 6). In sarcoidosis, BAL recovers activated lymphocytes and alveolar macrophages, which are the precursors of granuloma formation (2). Nevertheless, we know of no prior reports of intact granulomas in the BAL fluid of patients with sarcoidosis. This is surprising in view of the high prevalence of sarcoidosis, its diagnosis by bronchoscopy, and the large number of research studies using BAL. In the present case, we were surprised that epithelioid cells and Langhans' giant cells were easily discernible in the BAL fluid by the cytopathologist (J.H.C.) before histologic examination of the transbronchial biopsies.

The BAL granulomas in this patient could reflect the severity of his disease process. He had resting tachypnea and impressive radiographic and physiologic abnormalities. It is reasonable to assume that his lungs contained a large burden of granulomas, of which a few found their way into the BAL fluid. Alternatively, granulomas might have originated from the bronchial mucosa, since the airway examination was consistent with endobronchial sarcoidosis. Although the bronchial mucosa was not biopsied, a few granulomas could have been mechanically disrupted from the airway wall by the lavage process or by scope trauma. Nevertheless, neither severe alveolar inflammation nor endobronchial involvement is unusual in sarcoidosis, and it remains surprising that intact granulomas have not been previously reported.

It is clear that the epithelioid and Langhans' giant cells were well formed and close in appearance to their counterparts in lung tissue. They did not resemble the previously described phenomenon of one or more lymphocytes adhering to alveolar macrophages in the BAL fluid of patients with sarcoidosis (6, 7). Although not previously reported in sarcoidosis, BAL granulomas have been reported in two cases of pulmonary tuberculosis (8).