F1cu1u: 1. Tense swelling of the left lower limb, with trophic ulcerations and local globular swellings.
right costodiaphragmatic sinus and opacification of the right horizontal fissure. A specimen obtained by means of thoracentesis was a moderately lactescent liquid; the supernatant did not clarify when centrifuged. Laboratory examination revealed the following values: lymphocytes, 100 percent; cholesterol, 93 mgld; triglycerides, 412 mg/di. After evacuation, the liquid reappeared. Clinical examination did not show changes in the nervous system. The patient had normal height and secondary sexual characteristics, as well as positive X sex chromatin. Like her brother, the patient has no other congenital anomalies (eg, craniofacial or visceral abnormalities or yellow nails), although they were assiduously sought. Family anamnesis showed that the parents (aged 43 and 41) are not kindred, are in good health, have no lymphedema and no pulmonary complaints in their antecedents, and have no other children. No cases similar to those of the patients described were reported in the family among more distant relatives, nor were there any other congenital anomalies or genetic diseases. Paternity tests and confidential anamnesis allowed rejection of the hypothesis that the father was not in fact the biologic father of the patients. The peculiarity of these cases resides in the unilaterality of the lymphedema, the peripubertal spontaneous occurrence of the condition, the association with chylothorax, and the familial character of the disease, which , however, did not manifest itself in the parents, a fact that contravenes the dominant transmission observed in other studies.' .. Two hypotheses are possible: (l) The parents may have crude, oligosymptomatic forms of the disease or may have undiscovered congenital anomalies not associated with lymphedema. (2) The transmission of the disease is autosomal recessive, which could be a novelty in the literature. Traian Mihaescu, M.D., Liliana ~s, M.D., and \eronica Zbranca, M.D., Clinic of fulmonary Diseases, lasi, Romania
The patient's 22-year-old sister had lymphedema localized in the left lower extremity; the condition had appeared shortly after the onset of menarche at age 14 (Fig 2). Since August 1989, she had experienced episodes of fever, shivers, and myalgia. A chest x-ray examination showed a homogeneous intense opacity occupying the
REFERENCES 1 Goodman RM. Familial lymphedema of the Meige type. Am J Med 1962;32:6.'51-56 2 Der Kaloustian VN, Kurban AK. Genetic diseases of the skin. Berlin: Springer-Verlag, 1979; 247-49 3 Esterly JR. Congenital hereditary lympbedema. J Med Genet 1965; 2:93-8 4 Lewis JN, Wald ER. Lymphedema praecox. J Pediatr 1984; 104:
641-48
Myxedematous
Pleural Effusion
To the Editor:
F1GURF. 2. Circumferential edema of the left shank and foot, which was irreducible by compression or elevation.
We would like to comment on myxedematous pleural effusion because our recent experience' was somewhat different from that described by Gottehrer et al• in the November 1990 issue of Chest. They stated that "patients with hypothyroid pleural effusions are asymptomatic" and that the "small, noninflammatory effusions" are "of minor clinical importance." On the contrary, however, our patient had bilateral, massive, refractory pleural effusions and several episodes of apnea in spite of various kinds of treatment. Definitive diagnosis was delayed until six months after admission. All of his clinical problems, such as the refractory pleural effusion, respiratory embarrassment, and even the hearing impairment, disappeared after thyroid hormone replacement. In addition, the predominant cells in the pleural fluid were neutrophils in the beginning but subsequently became lymphocytes (Table I). Furthermore, the positive periodic acid-Schiff stain of the pleural effusion cytologic specimen and the high protein content of the pleural fluid in this patient support the thesis of extravasation of the CHEST I 101 I 1 I JANUARY. 1992
291
Table 1- Pleural Fluid Characteristics of the lbtient
Date
Appear.mc.-e
Protein, gtdl
Dec24 Mar30 Jun 24
Serosanguineous Yellow, clear Yellow, clear
5.4 4.0 4.0
Cell Count*
LymphocyteNeutrophil Ratio
700 300 200
30:70 99:1 80:20
*Values are numbers of cells per microliter. hygroscopic mucopolysaccharides into the body cavities as a mechanism of myxedematous pleural effusion. 3 Myxedematous pleural effusion is rare and is not yet fully understood. We hope that our experience will be of some help in understanding what myxedematous pleural effusion is.
BAL aspirated 8uid in the same receptacle. Thus, we obtain fewer diagnoses with BAL exclusively than when we performed bronchial wash without aspirating 8uid after BAL. In our experience, the diagnostic yield of bronchoscopy with BAL for M tuberculosis infection is significantly greater than that fur bronchial wash (odds ratio=6.67; con6dence interval, 1.253 to 35.45). Therefure, BAL, 8uid aspiration after BAL, and bronchial wash should be performed and a postbronchoscopy sputum specimen should be obtained when bronchoscopy is performed in patients suspected of having pulmonary tuberculosis, especially if previous sputum smears were negative. Recently, the American Thoracic Society' reported the utility of BAL for diagnosis of pulmonary tuberculosis.
Javier de Gracia,
M.D., Ph.D.; Victor Curull, M.D.; Rafael Vidal, M.D., Ph.D.; and Ferran Morell, M.D., Ph.D., Pneumology Service, Hospital General UnloersUario \fJll d'Hebron, Barcelona, Spain
Chih-Yu Hsu, B.M., and Shinn-Twng Gong, M.D., Cathay General Hospital, Taipei, Taiwan, Republic of China Reprint requests: Dr. Hsu, Cathay General Hospital, No. 280, Sec. 4, Jen-Ai Road, Taipei 106, Taiwan, Republic of China. REFERENCES l Hsu CY, Gong ST. Pleural effusion as an initial clinical presentation of myxedema: report of a case. J Formosan Med Assoc 1990; 89:470-74 2 Gottehrer A, Roa J, Stanford GG, Chernow B, Salm SA. Hypothyroidism and pleural effusions. Chest 1990; 98:1130-132 3 Sachdev Y, Hall R. Effusions into body cavities in hypothyroidism. Lancet 1975; 1:564-66
Bronchoscopy with Bronchoalveolar Lavage In the Diagnosis of Pulmonary Tuberculosis To the Editor: In the January 1991 issue of Chest, Baughman et al• reported the utility of bronchoscopy with bronchoalveolar lavage (BAL) in the diagnosis of Mycobacterium tuberculosis infections, and they concluded that BAL is useful for this indication. However, the M tuberculosis infection was diagnosed with only BAL in one of 29 cases, when BAL and bronchial wash specimens were obtained together. In a prospective study previously reported by our group," we performed fiberoptic bronchoscopy with BAL and bronchial wash and obtained postbronchoscopy sputum specimens in 20 of 222 patients suspected of having pulmonary tuberculosis (all patients had three consecutive early morning sputum specimens or gastric aspiration smears that were negative for acid-fast bacilli, and seven also had negative LOwenstein cultures). The BAL 8uid specimens provided the highest yield for diagnosis of pulmonary tuberculosis (15117 (88 percent]); in seven of the cases, the BAL 8uid specimen was the only positive source. Bronchial wash was positive in only nine (53 percent) patients. Bronchial washes were performed immediately after inspection of the tracheobronchial tree, and the specimens obtained were collected for study before BAL 8uids were obtained. The differences between the results of our study and those of the study by Baughman et al could be due to different methodology. In the latter study the bronchial wash included the aspirated 8uid obtained after BAL; the diagnostic value of that 8uid should be attributed to BAL, not to bronchial wash. Because of economic considerations, we now collect bronchial wash specimens and post-
292
REFERENCES
l Baughman RP, Dohn MN, Loudon RG, Frame PT. Bronchoscopy with bronchoalveolar lavage in tuberculosis and fungal infections. Chest 1991; 99:92-7 2 de Gracia J, Curull V, Vidal R, Morell F. Diagnostic value of bronchoalveolar lavage in suspected pulmonary tuberculosis. Chest 1988; 93:329-32 3 American Thoracic Society. Clinical role ofbronchoalveolar lavage in adults with pulmonary disease. Am Rev Respir Dis 1990; 140:481-86
Usefulness of Bronchoscopy Diagnosis of Tuberculosis
In the
1b the Editor: The study by Baughman et al, 1 which appeared in the January 1991 issue of Chest, exaggerated the importance of bronchoscopy (or at least minimized the elJectiveness of sputum examination) in the diagnosis of tuberculosis. Of 99 patients with tuberculosis, only 50 underwent bronchoscopy. Of the 50 patients who underwent bronchoscopy, 20 did so only as part of the study, after the diagnosis had been made. Of the 30 patients who required bronchoscopy, nine had prebronchoscopy cultures which were later found to be positive. Three with negative bronchoscopy specimens had positive postbronchoscopy sputum cultures. Four patients had positive urine cultures; it is not said whether those overlap with the above groups. Thus, only 14 to 18 of the 99 patients (14 to 18 percent) required bronchoscopy for diagnosis. This is a signi6cant percentage, but not what the authors imply. Forty-nine patients did not undergo bronchoscopy. Of those 49, 33 had positive sputum specimens; 16 patients had extrapulmonary tuberculosis only. The correct sensitivity for sputum examination is 71 percent (57/80), obtained by adding those 33 patients to the authors' 24147 (51 percent). Sixty percent of the patients who underwent bronchoscopy did so because the sputum examination was negative. The authors, by calculating the sensitivity of sputum examination on the basis of the bronchoscopy group alone, have eliminated those patients in whom the sputum was positive. Flexible bronchoscopy is helpful in the diagnosis of tuberculosis. However, the data, ifnot the authors' conclusions, demonstrate that examination of sputum is effective. The main problem is not the insensitivity of the sputum examination, but the failure to consider the diagnosis in a particular case. u In only 47 of the 50 bronchoscopy patients was prebronchoscopy sputum obtained. Could it be that 10 Communlcalions to the Editor
right costodiaphragmatic sinus and opacification of the right horizontal fissure. A specimen obtained by means of thoracentesis was a moderately lactescent liquid; the supernatant did not clarify when centrifuged. Laboratory examination revealed the following values: lymphocytes, 100 percent; cholesterol, 93 mgld; triglycerides, 412 mg/di. After evacuation, the liquid reappeared. Clinical examination did not show changes in the nervous system. The patient had normal height and secondary sexual characteristics, as well as positive X sex chromatin. Like her brother, the patient has no other congenital anomalies (eg, craniofacial or visceral abnormalities or yellow nails), although they were assiduously sought. Family anamnesis showed that the parents (aged 43 and 41) are not kindred, are in good health, have no lymphedema and no pulmonary complaints in their antecedents, and have no other children. No cases similar to those of the patients described were reported in the family among more distant relatives, nor were there any other congenital anomalies or genetic diseases. Paternity tests and confidential anamnesis allowed rejection of the hypothesis that the father was not in fact the biologic father of the patients. The peculiarity of these cases resides in the unilaterality of the lymphedema, the peripubertal spontaneous occurrence of the condition, the association with chylothorax, and the familial character of the disease, which , however, did not manifest itself in the parents, a fact that contravenes the dominant transmission observed in other studies.' .. Two hypotheses are possible: (l) The parents may have crude, oligosymptomatic forms of the disease or may have undiscovered congenital anomalies not associated with lymphedema. (2) The transmission of the disease is autosomal recessive, which could be a novelty in the literature. Traian Mihaescu, M.D., Liliana ~s, M.D., and \eronica Zbranca, M.D., Clinic of fulmonary Diseases, lasi, Romania
The patient's 22-year-old sister had lymphedema localized in the left lower extremity; the condition had appeared shortly after the onset of menarche at age 14 (Fig 2). Since August 1989, she had experienced episodes of fever, shivers, and myalgia. A chest x-ray examination showed a homogeneous intense opacity occupying the
REFERENCES 1 Goodman RM. Familial lymphedema of the Meige type. Am J Med 1962;32:6.'51-56 2 Der Kaloustian VN, Kurban AK. Genetic diseases of the skin. Berlin: Springer-Verlag, 1979; 247-49 3 Esterly JR. Congenital hereditary lympbedema. J Med Genet 1965; 2:93-8 4 Lewis JN, Wald ER. Lymphedema praecox. J Pediatr 1984; 104:
641-48
Myxedematous
Pleural Effusion
To the Editor:
F1GURF. 2. Circumferential edema of the left shank and foot, which was irreducible by compression or elevation.
We would like to comment on myxedematous pleural effusion because our recent experience' was somewhat different from that described by Gottehrer et al• in the November 1990 issue of Chest. They stated that "patients with hypothyroid pleural effusions are asymptomatic" and that the "small, noninflammatory effusions" are "of minor clinical importance." On the contrary, however, our patient had bilateral, massive, refractory pleural effusions and several episodes of apnea in spite of various kinds of treatment. Definitive diagnosis was delayed until six months after admission. All of his clinical problems, such as the refractory pleural effusion, respiratory embarrassment, and even the hearing impairment, disappeared after thyroid hormone replacement. In addition, the predominant cells in the pleural fluid were neutrophils in the beginning but subsequently became lymphocytes (Table I). Furthermore, the positive periodic acid-Schiff stain of the pleural effusion cytologic specimen and the high protein content of the pleural fluid in this patient support the thesis of extravasation of the CHEST I 101 I 1 I JANUARY. 1992
291
Table 1- Pleural Fluid Characteristics of the lbtient
Date
Appear.mc.-e
Protein, gtdl
Dec24 Mar30 Jun 24
Serosanguineous Yellow, clear Yellow, clear
5.4 4.0 4.0
Cell Count*
LymphocyteNeutrophil Ratio
700 300 200
30:70 99:1 80:20
*Values are numbers of cells per microliter. hygroscopic mucopolysaccharides into the body cavities as a mechanism of myxedematous pleural effusion. 3 Myxedematous pleural effusion is rare and is not yet fully understood. We hope that our experience will be of some help in understanding what myxedematous pleural effusion is.
BAL aspirated 8uid in the same receptacle. Thus, we obtain fewer diagnoses with BAL exclusively than when we performed bronchial wash without aspirating 8uid after BAL. In our experience, the diagnostic yield of bronchoscopy with BAL for M tuberculosis infection is significantly greater than that fur bronchial wash (odds ratio=6.67; con6dence interval, 1.253 to 35.45). Therefure, BAL, 8uid aspiration after BAL, and bronchial wash should be performed and a postbronchoscopy sputum specimen should be obtained when bronchoscopy is performed in patients suspected of having pulmonary tuberculosis, especially if previous sputum smears were negative. Recently, the American Thoracic Society' reported the utility of BAL for diagnosis of pulmonary tuberculosis.
Javier de Gracia,
M.D., Ph.D.; Victor Curull, M.D.; Rafael Vidal, M.D., Ph.D.; and Ferran Morell, M.D., Ph.D., Pneumology Service, Hospital General UnloersUario \fJll d'Hebron, Barcelona, Spain
Chih-Yu Hsu, B.M., and Shinn-Twng Gong, M.D., Cathay General Hospital, Taipei, Taiwan, Republic of China Reprint requests: Dr. Hsu, Cathay General Hospital, No. 280, Sec. 4, Jen-Ai Road, Taipei 106, Taiwan, Republic of China. REFERENCES l Hsu CY, Gong ST. Pleural effusion as an initial clinical presentation of myxedema: report of a case. J Formosan Med Assoc 1990; 89:470-74 2 Gottehrer A, Roa J, Stanford GG, Chernow B, Salm SA. Hypothyroidism and pleural effusions. Chest 1990; 98:1130-132 3 Sachdev Y, Hall R. Effusions into body cavities in hypothyroidism. Lancet 1975; 1:564-66
Bronchoscopy with Bronchoalveolar Lavage In the Diagnosis of Pulmonary Tuberculosis To the Editor: In the January 1991 issue of Chest, Baughman et al• reported the utility of bronchoscopy with bronchoalveolar lavage (BAL) in the diagnosis of Mycobacterium tuberculosis infections, and they concluded that BAL is useful for this indication. However, the M tuberculosis infection was diagnosed with only BAL in one of 29 cases, when BAL and bronchial wash specimens were obtained together. In a prospective study previously reported by our group," we performed fiberoptic bronchoscopy with BAL and bronchial wash and obtained postbronchoscopy sputum specimens in 20 of 222 patients suspected of having pulmonary tuberculosis (all patients had three consecutive early morning sputum specimens or gastric aspiration smears that were negative for acid-fast bacilli, and seven also had negative LOwenstein cultures). The BAL 8uid specimens provided the highest yield for diagnosis of pulmonary tuberculosis (15117 (88 percent]); in seven of the cases, the BAL 8uid specimen was the only positive source. Bronchial wash was positive in only nine (53 percent) patients. Bronchial washes were performed immediately after inspection of the tracheobronchial tree, and the specimens obtained were collected for study before BAL 8uids were obtained. The differences between the results of our study and those of the study by Baughman et al could be due to different methodology. In the latter study the bronchial wash included the aspirated 8uid obtained after BAL; the diagnostic value of that 8uid should be attributed to BAL, not to bronchial wash. Because of economic considerations, we now collect bronchial wash specimens and post-
292
REFERENCES
l Baughman RP, Dohn MN, Loudon RG, Frame PT. Bronchoscopy with bronchoalveolar lavage in tuberculosis and fungal infections. Chest 1991; 99:92-7 2 de Gracia J, Curull V, Vidal R, Morell F. Diagnostic value of bronchoalveolar lavage in suspected pulmonary tuberculosis. Chest 1988; 93:329-32 3 American Thoracic Society. Clinical role ofbronchoalveolar lavage in adults with pulmonary disease. Am Rev Respir Dis 1990; 140:481-86
Usefulness of Bronchoscopy Diagnosis of Tuberculosis
In the
1b the Editor: The study by Baughman et al, 1 which appeared in the January 1991 issue of Chest, exaggerated the importance of bronchoscopy (or at least minimized the elJectiveness of sputum examination) in the diagnosis of tuberculosis. Of 99 patients with tuberculosis, only 50 underwent bronchoscopy. Of the 50 patients who underwent bronchoscopy, 20 did so only as part of the study, after the diagnosis had been made. Of the 30 patients who required bronchoscopy, nine had prebronchoscopy cultures which were later found to be positive. Three with negative bronchoscopy specimens had positive postbronchoscopy sputum cultures. Four patients had positive urine cultures; it is not said whether those overlap with the above groups. Thus, only 14 to 18 of the 99 patients (14 to 18 percent) required bronchoscopy for diagnosis. This is a signi6cant percentage, but not what the authors imply. Forty-nine patients did not undergo bronchoscopy. Of those 49, 33 had positive sputum specimens; 16 patients had extrapulmonary tuberculosis only. The correct sensitivity for sputum examination is 71 percent (57/80), obtained by adding those 33 patients to the authors' 24147 (51 percent). Sixty percent of the patients who underwent bronchoscopy did so because the sputum examination was negative. The authors, by calculating the sensitivity of sputum examination on the basis of the bronchoscopy group alone, have eliminated those patients in whom the sputum was positive. Flexible bronchoscopy is helpful in the diagnosis of tuberculosis. However, the data, ifnot the authors' conclusions, demonstrate that examination of sputum is effective. The main problem is not the insensitivity of the sputum examination, but the failure to consider the diagnosis in a particular case. u In only 47 of the 50 bronchoscopy patients was prebronchoscopy sputum obtained. Could it be that 10 Communlcalions to the Editor
