Bronchioloalveolar Carcinoma Accompanied by Severe Bronchorrhea

resistant organism (ie, MDR-TB) apparently led to nosocomial infection with two other drug-resistant organisms (vancomycin-resistant enterococcus and multidrug-resistant Acinetobacter). As one commentator has stated, "multiple resistance . .. is the therapeutic anathema of the present decade." 10 ACKNOWLEDGMENTS: We thank Paul Bellman, MD, and Jose Giron, MD, for providing information on the source-patient.

noembryonic stains, which were localized in the apical region of tumor cells. (CHEST 1996; 110:281-82) Key words: bronchioloalveolar carcinoma (BAC); bronchorrhea; carcinoembryonic antigen (CEA) gastrointestinal cancer-associated antigen (CA19-9) Abbreviations: BAC=bronchioloalveolar cell carcinoma; CEA= carcinoembryonic antigen

REFERENCES

1 Beck-Sague C, Dooley SW, Hutton MD, eta!. Hospital outbreak of multidrug-resistant Mycobacterium tuberculosis infections: factors in transmission to staff and HIV-infected patients. JAMA 1992; 268:1280-86 2 Pierce JR, Sims SL, Holman GH. Transmission of tuberculosis to hospital workers by a patient with AIDS. Chest 1992; 101:581-82 3 Centers for Disease Control and Prevention. Nosocomial transmission of multidrug-resistant tuberculosis among HIV-infected persons-Florida and New York. MMWR 1991; 40:585-91 4 American Thoracic Society. Treatment of tuberculosis and tuberculosis infection in adults and children. Am J Respir Grit Care Med 1994; 149:1359-74 5 Centers for Disease Control and Prevention. Management of persons exposed to multidrug resistant tuberculosis. MMWR 1992; 41(RR-ll):61-71 6 Hom DL, Hewlett D , Alfalla C, et a!. limited tolerance of ofloxacin and pyrazinamide prophylaxis against tuberculosis [letter). N Eng! J Med 1994; 330:1241 7 Brausch LM, Bass JB. The treatment of tuberculosis. Med Clin North Am 1993; 77:1277-88 8 O'Brien RJ, Lyle MA, Snider DE. llifabutin (ansamycin LM 427): a new rifamycin-$ derivative for the treatment of mycobacterial diseases. Rev Infect Dis 1987; 9:519-30 9 Centers for Disease Control and Prevention. Severe isoniazidassociated hepatitis-New York, 1991-1993. MMWR 1993; 42:545-47 10 Levy SB. Confronting multidrug resistance: arole for each of us. JAMA 1993; 269:1840-42

Bronchioloalveolar Carcinoma Accompanied by Severe Bronchorrhea* Noriko Hidaka, MD; and Koichi Nagao, MD

We report an unusual case of bronchioloalveolar carcinoma characterized by production of a large quantity of sputum accompanied by drastic electrolyte and fluid loss. The sputum contained a high level of gastrointestinal cancer-associated antigen (CA19-9) and carcinoembryonic antigen (CEA). An immunohistochemical study of tumor cells showed the specific distribution of gastrointestinal cancer-associated antigen and carci*From the Department of Internal Medicine (Dr. Hidaka) and the Department of Surgical Pathology (Dr. Nagao), Teikyo University Scliool of Medicine, Ichihara, Japan. Manuscript received September ""9, 1995; revision accepted December 6. Reprint requests: Dr. Hidaka, Third Department of Internal Medicine, Teik_lfO University School of Medicine, 3426-3 Anesaki, Ichihara, ChifJa 299-01, Japan

A

striking feature ofbronchioloalveolar carcinoma (BAC) is the production of large quantities of sputum. Recently, biochemical constituents of the sputum have been studied; 1-3 however, few reports are available concerning the levels of tumor markers. We studied the carcinoembryonic antigen (CEA) and the gastrointestinal cancer-associated antigen (CAl9-9) levels in the sputum produced by a patient with BAC suffering from severe bronchorrhea. The sputum contained significantly higher levels of these tumor markers than the serum.

CASE REPORT

A 49-year-old woman developed a cough accompanied by clear watery sputum of 300 to 400 mUd. A chest radiograph showed diffuse lesions in both lungs. At bronchoscopy, a large amount of watery fluid was observed in the bronchus of the lower lobe of the left lung. The diagnosis of BAC was made based on the histologic examination of specimens obtained through transbronchial lung biopsy. All sputum expectorated was collected and measured. The analysis of the sputum revealed that CEA and CA19-9 were present at high levels (CEA, 612.7 nglmL; CA19-9, 33,057 U/mL). Despite these findings, serum levels of these tumor markers were not significantly elevated (CEA, 0.5 nglmL [normal range, <5 nglmL); CA19-9, 91.5 unit/mL [normal range, <35 unitlmL)). Measurement of electrolytes in the sputum were as follows: sodium, 134 mEq!L; chloride, 116 mEq/L; potassium, 7.4 mEq!L. Corticosteroids, atropine sulfate, and cytotoxic drugs failed to reduce the sputum volume. Several months later, the amount of sputum increased to the maximum of9 L daily. The patient always had to bend forward to expectorate the sputum. Approximately 10 L fluids were administered daily to restore serum electrolyte levels and water balance. Soon after, her condition deteriorated and she died of severe respiratory failure. The autopsy revealed that the primary site was the lower lobe of the left lung: Both lungs revealed an extensive presence of tumor cells. Microscopic examination showed that the tumor cells were tall and columnar containing periodic acid-Schiff-positive and Alcian blue-negative substances. Immunohistochemical evaluation showed that tumor cells were strongly stained by CEA and CA19-9 antibodies (Fig 1).

DISCUSSION

Production of large amounts of sputum is one of the unique features of BAC.l This patient produced an extremely large volume of sputum, much greater than in other cases reported. 1-3 Because the sputum of the patient contained almost the same concentration of electrolytes as the serum, loss of an CHEST /110/1/ JULY, 1996

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2 Lopez-Vidriero MT, Reid L. Bronchial mucus in health and disease. Br Med Bull 1978; 34:63-4 3 Homma H, Kira S, Takahashi Y, et al. A case of alveolar cell carcinoma accompanied by fluid and electrolyte depletion through production of voluminous amounts oflung liquid. Am Rev Respir Dis 1975; 3:857-62 4 Atkinson BF, Ernst CS, Herlyn M, et al. Gastrointestinal cancer-associated antigen in immunoperoxidase assay. Cancer Res 1982; 42:4820-23 5 Grigioni WF, Biagini G, Garbisa S, et al. Immunohistochemical study of basement membrane antigens in bronchioloalveolar carcinoma. Am J Pathol1987; 128:217-24 6 Bando M, Shimizu E , Mukai J, et al. Two cases of a bronchioloalveolar cell carcinoma of the lung with special reference to the significance ofCA19-9. Japan J Cancer Clin 1991; 37:1197-1202

Primary Systemic Amyloidosis Complicated by Massive Thrombosis* Frank J. Cools, MD; Marc M. Kockx, MD, PhD; Guy E. Boeckxstaens, MD, PhD; Paul Van den Heuvel, MD; and Jean-Jacques Cuykens, MD

FIGURE 1. Immunoperoxidase staining with anti-CEA (top, A) and anti-CA19-9 (bottom, B) monoclonal antibody. The secretory surface of the tumor cells lining the alveolar spaces was strongly stained (originals x50).

enormous amount of water and electrolytes through the lungs occurred. In general, it is speculated that active secretion of tumor cells of BAC growing along the alveolar walls produces the sputum. 3 CA 19-9 antigen has been detected in tumor cells ofBAC as well as in normal bronchial glands. 4 One of the characteristic findings of the present case was that the levels of tumor markers in the sputum were signiflcantly higher than those in the serum. The CA19-9 and CEA immunohistochemical staining of tumor cells revealed that the secretory surface stained stronger than the cytoplasm. These findings suggest that the tumor cells produced these tumor markers and secreted them predominantly into the alveolar spaces. BAC cells are known to grow along the alveolar lining without disrupting the underlying basement membrane. 5 This could partially explain the higher levels of tumor markers in the sputum as opposed to the levels in the serum. Based on this study and a review of the literature,6 BAC cells may in certain cases have a strong tendency to produce CA19-9 and CEA and to secrete them into the sputum. Therefore, the examination of the level of CA 19-9 and CEA in sputum can be useful in suggesting a diagnosis of BAC; however, further investigations are necessary before the clinical implications of these findings become clear. REFERENCES

1 Spiro SG, Lopez-Vidriero MT, Charman J, et al. Bronchorrhea in a case of alveolar cell carcinoma. J Clin Path 1975; 28:60-5

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We present a case of primary systemic amyloidosis complicated by multiple thrombotic events and initially presenting with a massive thrombosis of the inferior vena cava. Widespread infiltration of the vascular tree by amyloid was found at the time of autopsy. In addition, we report successful treatment of the massive inferior vena cava thrombosis with systemic thrombolysis. (CHEST 1996; 110:282-84) Key words: amyloidosis; inferior vena cava; thrombolysis; thrombosis Abbreviations: APTT =activated partial thromboplastin time

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rimary systemic amyloidosis, complicated by multiple thrombotic events, was first evidenced in a patient who had massive thrombosis of the inferior vena cava. Systemic thrombolysis was successfully used to treat the massive thrombosis of the vena cava; however, the patient died of a small-bowel infarction. CASE REPORT

A 64-year-old white man with a medical history of a normal coronary angiogram 2 years earlier and no other medical problems was admitted to the hospital because of transient episodes of swelling *From the Department of Internal Medicine (Drs. Cools, Boeckxstaens, and Cuxkens), Pathologx (Dr. Kockx), and Cardiology (Dr. Van den Heuvel), Middelheim Hospital, Antwerp, Belgium. Manuscript received May 11, 1995; revision accepted December 2E. Reprint requests: Dr. Cuykens, AZ Middelheim (5D), Lindendreef, B-2020 Antwerp, Belgium Selected Reports