J Bronchol Intervent Pulmonol
Volume 21, Number 1, January 2014
histoplasmosis being the most common etiology in the United States and tuberculosis in the rest of the world. However, primary endobronchial lesions, such as calcified endobronchial infection and aspirated foreign body, are responsible for broncholithiasis in a small portion of patients, as illustrated in this case.2 The chemical composition of a broncholith is similar to that of bone, consisting of 85% to 90% of calcium phosphate and 10% to 15% of calcium carbonate.3 The most common presenting symptom of broncholithiasis is nonproductive cough although fever, hemoptysis, localized wheezing, chest pain, and lithoptysis (stone expectoration) can also be present. Its complications, including recurrent pneumonia, massive hemoptysis, and fistula formation between the bronchi and adjacent mediastinal structures, are infrequent but had also been reported.4 Lithoptysis, the most common presenting symptoms in the prebronchoscopy era, was considered as pathognomonic sign of broncholithiasis but it is nowadays rare.1 Plain chest x-ray often fails to show calcification of broncholith though sign of obstruction, such as atelectasis and bronchiectasis, can be prominent. Computed tomography of chest usually provides more useful information. Presence of endobronchial or peribronchial calcified nodules with associated findings of bronchial obstruction is very suggestive for the diagnosis.2 Bronchoscopy is diagnostic when broncholiths are revealed although sometimes it is difficult to distinguish broncholith from tumor base on the gross appearance. r
2014 Lippincott Williams & Wilkins
Letters to the Editor
FIGURE 1. A, Chest x-ray revealed right upper lobe atelectasis. B, Chest computed tomography showed a small soft tissue with peripheral calcification in the right upper lobe bronchus (arrow). C, Bronchoscopy revealed an easily movable yellow mass in the right upper lobe bronchus. D, Microscopic examination of the mass demonstrated calcified degenerated vegetable matter surrounded by leukocyte and gram-positive filamentous bacteria.
Therapeutic options include observation, bronchoscopic removal, and surgery as spontaneous broncholith expectoration may occur. However, bronchoscopic treatment or surgery should be exploited in symptomatic patient to avoid its potentially life-threatening complications. Bronchoscopic treatment can be considered in an uncomplicated case with easily movable broncholith, as in this case, otherwise surgical resection including segmentectomy, lobectomy, or pneumectomy, depending upon the size and location of broncholith is preferred.2,4 Patompong Ungprasert, Narat Srivali, Michael A. Bauer, Lee C. Edmonds,
MD MD MD MD
Department of Internal Medicine Bassett Medical Center Columbia University College of Physicians and Surgeons Cooperstown, NY
REFERENCES 1. Anwer M, Venkatram S. Broncholithiasis: “incidental finding during bronchoscopy”—case report and review of the literature. J Bronchol Intervent Pulmonol. 2011;18:181–183. 2. Seo JB, Song KS, Lee JS, et al. Broncholithiasis: review of the causes with radiologic-pathologic correlation. Radiographics. 2002;22:S199–S213. 3. Dixon GF, Donnerberg RL, Schonfeld SA, et al. Advances in the diagnosis and treatment of broncholithiasis. Am Rev Respir Dis. 1984;129:1028–1030. 4. De S, De S. Broncholithiasis. Lung India. 2008;25:152–154.
Video-assisted Thoracoscopic Decortication of Trapped Lung in a Patient With Yellow Nail Syndrome Disclosure: There is no conflict of interest or other disclosures.
www.bronchology.com |
103
Letters to the Editor
To the Editor: A 26-year-old Yemenite male presented with complaints of chronic shortness of breath, dyspnea, pleural effusions, and recurrent respiratory infections. At age 5, he developed lymphedema and was diagnosed with yellow nail syndrome (YNS). He underwent pericardial window at age 10 for pericardial effusion and myringotomy at age 19 to drain an effusion from the middle ear. He has several cousins with chronic lymphedema. In the 2 years before the current admission, he had undergone rightsided thoracentesis twice for recurrent pleural effusions. Pleural fluid analysis on both occasions revealed a serosanguinous, lymphocyte-predominant exudate. Cultures for bacteria, mycobacteria, and fungi were negative. Cytologic examination was negative for malignant cells. On the current admission, CT scan (Fig. 1) showed a large, loculated right and a moderate left pleural effusion. The patient underwent VATS and the right parietal and visceral pleura were found to be fibrotic with areas of calcification. A large amount of debris and approximately 100 mL of fluid were removed. The parietal pleura, which was 3 to 4 mm thick in some areas, was removed entirely down to the chest wall. The lung remained trapped by a dense visceral pleural peel. This was excised permitting almost complete reexpansion. Pathologic examination of the intrapleural debris showed organizing blood clots, granulation tissue, and fibrohyaline/ collagenous tissue. The pleural showed fibrosis and chronic inflammatory changes. Pleural fluid cultures were negative for bacteria, fungi, and acid-fast
104 | www.bronchology.com
J Bronchol Intervent Pulmonol
Volume 21, Number 1, January 2014
FIGURE 1. Preoperative computed tomography scan showing loculated right pleural effusion and marked pleural thickening. Intrapleural air from recent thoracentesis.
bacilli. His postoperative course was complicated by an episode of hospital-acquired pneumonia. Exertional dyspnea improved progressively after discharge. YNS is a rare lymphatic disorder of unknown etiology. It is characterized by the triad of deformed yellow nails, lymphedema, and respiratory manifestations. These most commonly include pleural effusions, bronchiectasis, sinusitis, and recurrent pneumonia.1 Women are affected twice as often as men.2 The characteristic nail findings are present in 89% of patients2 but are not necessary for diagnosis. The complete triad is seen in only about 30% of cases3 and the manifestations may become apparent over a considerable span of years.2 The pleural effusions are bilateral in 50% of cases. The fluid is most often a yellow exudate with lymphocytic predominance, and normal glucose level.2 The pathophysiology seems to result from hypoplasia or functional blockage of the lymphatic system.4 Previous literature on YNS demonstrates resolution of pleural effusions through thor-
acentesis,4 pleuroperitoneal shunting,5 pleurovenous shunting,6 and mechanical or talc slurry pleurodesis as a last resort.1 In our patient, extensive parietal and visceral pleural thickening resulted in a trapped lung. To the best of our knowledge, this has been previously reported only once before in YNS. In that case, the patient was managed with open thoracotomy and decortication of the parietal pleura.7 In our patient, parietal and visceral pleural decortication was accomplished exclusively through VATS. The patient’s clinical presentation was also atypical. It is rare for YNS to present in childhood.4 In one of the largest series of patients with YNS reported, the median age at diagnosis was 61 years.1 Our patient presented at the age of 5 with peripheral edema, pericardial effusion, and otitis media. In addition, although familial predisposition has not been reported,3 our patient had an extensive family history of lymphedema suggesting a possible hereditary component in YNS. Videoscopic surgical r
2014 Lippincott Williams & Wilkins
J Bronchol Intervent Pulmonol
Volume 21, Number 1, January 2014
decortication seems to have benefit in this systemic disease. Jessie Rosenberg, Steven D. Herman, Peter R. Smith, Verena Liu, Joshua Rosenberg,
MD* MDw MDw MDw MDz
*University of Maryland Medical Center College Park, MD wDivisions of Thoracic Surgery and Pulmonary Medicine SUNY-Downstate University Hospital of Brooklyn at Long Island College Hospital, and SUNY-Downstate Medical Center Brooklyn, NY zThe Brooklyn Hospital Center New York, NY
REFERENCES 1. Maldonado F, Tazelaar H, Wang CW, et al. Yellow nail syndrome: analysis of 41 consecutive patients. Chest. 2008;134: 375–381. 2. Light R, Lee G. Textbook of Pleural Disease. 2nd ed. Boca Raton Florida: CRC Press; 2008. 3. Banta DP, Dandamudi N, Parekh HJ, et al. Yellow nail syndrome following thoracic surgery: a new association? J Postgrad Med. 2009; 55:270–271. 4. Maldonado F, Ryu JH. Yellow nail syndrome. Curr Opin Pulm Med. 2009;15:371–375.
Letters to the Editor
5. Brofman JD, Hall JB, Scott W, et al. Yellow nails, lymphedema, and pleural effusion. Treatment of chronic pleural effusion with pleuroperitoneal shunting. Chest. 1990; 97:743–745. 6. Tanaka E, Matsumoto K, Shindo T, et al. Implantation of a pleurovenous shunt for massive chylothorax in a patient with yellow nail syndrome. Thorax. 2005;60: 254–255. 7. Lewis M, Kallenbach J, Zaltzman M, et al. Pleurectomy in the management of massive pleural effusion associated with primary lymphoedema: demonstration of abnormal pleural lymphatics. Thorax. 1983; 38:637–639.
13th INTERNATIONAL CONGRESS ON PEDIATRIC PULMONOLOGY The 13th International Congress on Pediatric Pulmonology (CIPP XIII) will take place in Bruges, Belgium, from June 26 through 29, 2014. As the only global congress devoted entirely to pediatric pulmonology, CIPP XIII offers a unique opportunity for state-of-the-art reviews of advances in this field, debates on controversial issues, unique clinical sessions and cuttingedge clinical science. For more information about the congress, please visit the Web site: www.cipp-meeting.org.
r
2014 Lippincott Williams & Wilkins
www.bronchology.com |
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Volume 21, Number 1, January 2014
histoplasmosis being the most common etiology in the United States and tuberculosis in the rest of the world. However, primary endobronchial lesions, such as calcified endobronchial infection and aspirated foreign body, are responsible for broncholithiasis in a small portion of patients, as illustrated in this case.2 The chemical composition of a broncholith is similar to that of bone, consisting of 85% to 90% of calcium phosphate and 10% to 15% of calcium carbonate.3 The most common presenting symptom of broncholithiasis is nonproductive cough although fever, hemoptysis, localized wheezing, chest pain, and lithoptysis (stone expectoration) can also be present. Its complications, including recurrent pneumonia, massive hemoptysis, and fistula formation between the bronchi and adjacent mediastinal structures, are infrequent but had also been reported.4 Lithoptysis, the most common presenting symptoms in the prebronchoscopy era, was considered as pathognomonic sign of broncholithiasis but it is nowadays rare.1 Plain chest x-ray often fails to show calcification of broncholith though sign of obstruction, such as atelectasis and bronchiectasis, can be prominent. Computed tomography of chest usually provides more useful information. Presence of endobronchial or peribronchial calcified nodules with associated findings of bronchial obstruction is very suggestive for the diagnosis.2 Bronchoscopy is diagnostic when broncholiths are revealed although sometimes it is difficult to distinguish broncholith from tumor base on the gross appearance. r
2014 Lippincott Williams & Wilkins
Letters to the Editor
FIGURE 1. A, Chest x-ray revealed right upper lobe atelectasis. B, Chest computed tomography showed a small soft tissue with peripheral calcification in the right upper lobe bronchus (arrow). C, Bronchoscopy revealed an easily movable yellow mass in the right upper lobe bronchus. D, Microscopic examination of the mass demonstrated calcified degenerated vegetable matter surrounded by leukocyte and gram-positive filamentous bacteria.
Therapeutic options include observation, bronchoscopic removal, and surgery as spontaneous broncholith expectoration may occur. However, bronchoscopic treatment or surgery should be exploited in symptomatic patient to avoid its potentially life-threatening complications. Bronchoscopic treatment can be considered in an uncomplicated case with easily movable broncholith, as in this case, otherwise surgical resection including segmentectomy, lobectomy, or pneumectomy, depending upon the size and location of broncholith is preferred.2,4 Patompong Ungprasert, Narat Srivali, Michael A. Bauer, Lee C. Edmonds,
MD MD MD MD
Department of Internal Medicine Bassett Medical Center Columbia University College of Physicians and Surgeons Cooperstown, NY
REFERENCES 1. Anwer M, Venkatram S. Broncholithiasis: “incidental finding during bronchoscopy”—case report and review of the literature. J Bronchol Intervent Pulmonol. 2011;18:181–183. 2. Seo JB, Song KS, Lee JS, et al. Broncholithiasis: review of the causes with radiologic-pathologic correlation. Radiographics. 2002;22:S199–S213. 3. Dixon GF, Donnerberg RL, Schonfeld SA, et al. Advances in the diagnosis and treatment of broncholithiasis. Am Rev Respir Dis. 1984;129:1028–1030. 4. De S, De S. Broncholithiasis. Lung India. 2008;25:152–154.
Video-assisted Thoracoscopic Decortication of Trapped Lung in a Patient With Yellow Nail Syndrome Disclosure: There is no conflict of interest or other disclosures.
www.bronchology.com |
103
Letters to the Editor
To the Editor: A 26-year-old Yemenite male presented with complaints of chronic shortness of breath, dyspnea, pleural effusions, and recurrent respiratory infections. At age 5, he developed lymphedema and was diagnosed with yellow nail syndrome (YNS). He underwent pericardial window at age 10 for pericardial effusion and myringotomy at age 19 to drain an effusion from the middle ear. He has several cousins with chronic lymphedema. In the 2 years before the current admission, he had undergone rightsided thoracentesis twice for recurrent pleural effusions. Pleural fluid analysis on both occasions revealed a serosanguinous, lymphocyte-predominant exudate. Cultures for bacteria, mycobacteria, and fungi were negative. Cytologic examination was negative for malignant cells. On the current admission, CT scan (Fig. 1) showed a large, loculated right and a moderate left pleural effusion. The patient underwent VATS and the right parietal and visceral pleura were found to be fibrotic with areas of calcification. A large amount of debris and approximately 100 mL of fluid were removed. The parietal pleura, which was 3 to 4 mm thick in some areas, was removed entirely down to the chest wall. The lung remained trapped by a dense visceral pleural peel. This was excised permitting almost complete reexpansion. Pathologic examination of the intrapleural debris showed organizing blood clots, granulation tissue, and fibrohyaline/ collagenous tissue. The pleural showed fibrosis and chronic inflammatory changes. Pleural fluid cultures were negative for bacteria, fungi, and acid-fast
104 | www.bronchology.com
J Bronchol Intervent Pulmonol
Volume 21, Number 1, January 2014
FIGURE 1. Preoperative computed tomography scan showing loculated right pleural effusion and marked pleural thickening. Intrapleural air from recent thoracentesis.
bacilli. His postoperative course was complicated by an episode of hospital-acquired pneumonia. Exertional dyspnea improved progressively after discharge. YNS is a rare lymphatic disorder of unknown etiology. It is characterized by the triad of deformed yellow nails, lymphedema, and respiratory manifestations. These most commonly include pleural effusions, bronchiectasis, sinusitis, and recurrent pneumonia.1 Women are affected twice as often as men.2 The characteristic nail findings are present in 89% of patients2 but are not necessary for diagnosis. The complete triad is seen in only about 30% of cases3 and the manifestations may become apparent over a considerable span of years.2 The pleural effusions are bilateral in 50% of cases. The fluid is most often a yellow exudate with lymphocytic predominance, and normal glucose level.2 The pathophysiology seems to result from hypoplasia or functional blockage of the lymphatic system.4 Previous literature on YNS demonstrates resolution of pleural effusions through thor-
acentesis,4 pleuroperitoneal shunting,5 pleurovenous shunting,6 and mechanical or talc slurry pleurodesis as a last resort.1 In our patient, extensive parietal and visceral pleural thickening resulted in a trapped lung. To the best of our knowledge, this has been previously reported only once before in YNS. In that case, the patient was managed with open thoracotomy and decortication of the parietal pleura.7 In our patient, parietal and visceral pleural decortication was accomplished exclusively through VATS. The patient’s clinical presentation was also atypical. It is rare for YNS to present in childhood.4 In one of the largest series of patients with YNS reported, the median age at diagnosis was 61 years.1 Our patient presented at the age of 5 with peripheral edema, pericardial effusion, and otitis media. In addition, although familial predisposition has not been reported,3 our patient had an extensive family history of lymphedema suggesting a possible hereditary component in YNS. Videoscopic surgical r
2014 Lippincott Williams & Wilkins
J Bronchol Intervent Pulmonol
Volume 21, Number 1, January 2014
decortication seems to have benefit in this systemic disease. Jessie Rosenberg, Steven D. Herman, Peter R. Smith, Verena Liu, Joshua Rosenberg,
MD* MDw MDw MDw MDz
*University of Maryland Medical Center College Park, MD wDivisions of Thoracic Surgery and Pulmonary Medicine SUNY-Downstate University Hospital of Brooklyn at Long Island College Hospital, and SUNY-Downstate Medical Center Brooklyn, NY zThe Brooklyn Hospital Center New York, NY
REFERENCES 1. Maldonado F, Tazelaar H, Wang CW, et al. Yellow nail syndrome: analysis of 41 consecutive patients. Chest. 2008;134: 375–381. 2. Light R, Lee G. Textbook of Pleural Disease. 2nd ed. Boca Raton Florida: CRC Press; 2008. 3. Banta DP, Dandamudi N, Parekh HJ, et al. Yellow nail syndrome following thoracic surgery: a new association? J Postgrad Med. 2009; 55:270–271. 4. Maldonado F, Ryu JH. Yellow nail syndrome. Curr Opin Pulm Med. 2009;15:371–375.
Letters to the Editor
5. Brofman JD, Hall JB, Scott W, et al. Yellow nails, lymphedema, and pleural effusion. Treatment of chronic pleural effusion with pleuroperitoneal shunting. Chest. 1990; 97:743–745. 6. Tanaka E, Matsumoto K, Shindo T, et al. Implantation of a pleurovenous shunt for massive chylothorax in a patient with yellow nail syndrome. Thorax. 2005;60: 254–255. 7. Lewis M, Kallenbach J, Zaltzman M, et al. Pleurectomy in the management of massive pleural effusion associated with primary lymphoedema: demonstration of abnormal pleural lymphatics. Thorax. 1983; 38:637–639.
13th INTERNATIONAL CONGRESS ON PEDIATRIC PULMONOLOGY The 13th International Congress on Pediatric Pulmonology (CIPP XIII) will take place in Bruges, Belgium, from June 26 through 29, 2014. As the only global congress devoted entirely to pediatric pulmonology, CIPP XIII offers a unique opportunity for state-of-the-art reviews of advances in this field, debates on controversial issues, unique clinical sessions and cuttingedge clinical science. For more information about the congress, please visit the Web site: www.cipp-meeting.org.
r
2014 Lippincott Williams & Wilkins
www.bronchology.com |
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