Bilateral MUltiple Sclerosing Hemangiomas of the Lung* Shiang-Tai Lee. M.D.; limg-Cltk lce, M.D ., FC .C.P.; Chth-Y« Hsu, M.D .• FC.C .P.; lI/1(/ Chi-Chum; Lin , M.D., FC .C.P.
A Chinese woman had bilateral coin lesions of the lung, which grew slowly. At pathologic examination, the lesions were considered to be multiple sclerosing hemangiomas. Multiplicity is rare, and bilaterality has not been previously reported. (Chest 1992; 101:572-73)
S
cle rosillJ.!: hemangioma is generally considered to be a solitary benign lesion , Multifocality is rare .' ..1 We report the case of a Chinese woman with bilateral coin lesions of the IUIIJ.!:, which were proved to be bilateral multiple sclerosing hemangiomas. CASE R":I'ORT
A 48-yt'ar-old Chinese woman was followed up fi,r three years with serial radiographs and computed tomographic (C'T) scans. The hilatl>rallung masses grew slowly. A hOlllogl'nt'OUs, 5-cm round l"in
*From the
Departments of Surgery (Drs. Lee and Lee) and Clinical Patholoh'Y (Dr. Lin), National Taiwan University Hospital, Taipei ; and till' Department ofInternal Medicine, Cathy Generaillospital (Dr. Hsu), Taipei, Taiwan , Republic of China. Reprint requests: Dr. I.....'e, National uliwlIn Unil)('rsity Hospital, 1, Chang-tv Street , TlIipei, TlIiwlIn. Republic of China
lesion in the left lower lohe was accompanied by two smaller lesions in the right lung. A C'T scan demonstrated a fourth lesion in the lingula of the left lung (Fig 1). At hilateral anteroluteral thoracotomy; three wedge sections were obtained , and a left lower basal segmentectomy was performed. The left lung contained a 5 x 5-slj em anterior busul lower lohe tumor and a 0.8 x 0 .7-slj em inferior basal hngular tumor. The right lung contained a 1.8 x 1.5-slj em lateral middle lobe mass and a 0.8 x 0.6-slj ern anterior basal lower lobe mass (Ftg 2). Each tumor was firm, elastic, and yellow, with focal hemorrhage. At microscopic examination, all four tumors were considered to be sclerosing hemangiomas composed of polygonal cells arranged in solid or papillary pallerns. FIle'll sclerosis and foamy histiocytes were present (Fig 3). DISCUSSION
The rubric "sclerosing hemangioma" proposed by Liebow and H ubbel' defines a subset of'benign tumors and tumorlike lesions of the lung, This subset diJfers from plasma cell granuloma and pseudolymphoma and may include cases of xanthomatous pseudotumor, postinflammatory pseudotumor, fibrous histiocytoma. alveolar angioblastoma, and the recently described pneumocytoma. The cell of origin was initially considered to be endothelial and thus the tumor to be akin to a tumor of the dermis of the skin . Subsequent electron microscopic studies suggested ' either alveolar pneurnocytes or mesothelium as the origin. Multiple authors utilizing immunologic studies have sup-
FIGllRE 1. Right, Chest x-ray film showed three coin-shaped tumor masses in bilateral lung fields, which were homogeneous in density (lIrrows). Left, on chest CT scan , another small lesion was found in the lingula (double lIrrows). U
c
\
FIGllRE 2. Gross specimens demonstrate the ball-like appearance of the tumors, which were found in the lateral basal segment of the left lower lobe (A), the inferior segment of the lingula (B), the anterior basal segment of the right lower lobe (C), and the lateral segment of the right middle lobe (D).
572
Bilateral Multiple SClerosing Hemangiomas (Lee
et Ill)
Chylous Transport of Amiodarone* Charli e Strange, M.D ., EC .C.P.; Dacid P. Nicolau , Pharm . D.; and Scott R. Drqzer; M.D .
A patient receiving amiodarone for longstanding ventricular dysrhythmias presented with idiopathic chylothorax, During drainage of chylothorax for pleurodesis, serial plasma amiodarone concentrations declined while pleural fluid concentrations remained stabile. Chylous transport of amiodarone and other lipid-bound drugs should be recognized to avert complications during chylothorax drainage. (Chest 1992; 101:573-74)
of amiodarone remain incomT hepletelypharmacokinetics understood . Following an oral dose, low bioaFIGURE 3. Histologic appearance of sclerosing hemangioma in this case . Tumor is composed of polygonal cells arranged in solid or papillary pattern. Focal sclerosing areas and aggregates of foamy histocytes are also observed (hematoxylin-eosin stain , original magnification x 400).
ported the hypothesis that both bronchiolar cells and alveolar type II pneumocytes are simultaneously the cells of origin .5 Thus, "pneumocytoma" is proposed as the appropriate term . Clinically, this tumor is asymptomatic and is an incidental x-ray finding. Chest pain, hemoptysis, and cough may result from impingement of a large tumor on adjacent structures of the bronchovascular ray. Fewer than ten cases of multifocal sclerosing hemangioma
vailability with interindividual variation has been attributed to intestinal mucosal cell dealkylation of amiodarone before it reaches the portal venous system .' Because of extensive protein binding within the circulation," a delay has been noted before amiodarone is distributed into tissue . Preferential uptake into adipose liver, and pulmon ary tissue has been noted with a steady state partition coefficient at least 100fold above plasma ."?The plasma concentration threshold for antiarrhythmic activity has been estimated to be between 0.5 to 1.0 Ilog per ml." We recently encountered a patient with idiopathic chylothorax which allowed investigation of the chylous content of amiodarone . Because of arniodarones lipid affinity, we questioned whether amiodarone would be removed during chest tube drainage.
have been reported in the English-language literature . ' ·3
CASE REPORT
This case is unique in that different lobes of the lung were
A 76-yea r-old white man presented with a three-month history of large right-sided pleural effusions. Serial thoracentesis had demon strated milky white Auid with triglyceride concentration = 127 mg/dl consistent with chylothorax . After chest and abdominal CT scanning and pleural fluid cytology had failed to demonstrate a malignancy, a chest-tube was placed for tetracycline pleurodesis. Malignant ventricular arrh ythmia s associated with coronary artery disease had been stabil ized one year previou sly with amiodarone 400 mg qd . No sign of amiodarone toxicity had been noted . After chest-tube placement, drainage of 9 L of chylous pleural effusion was accompli shed over a six-day interval. Although daily evaluation of electrolytes had been performed without change, the patient experienced sudden death while walking across his hospital room. No autopsy was performed . Pleural Auid and serum arniodarone and desethylarniodarone levels were performed on initial evaluation and near the time of death. Data appear in Table 1.
involved and in that the distribution was bilateral. One reported case is stated to have metastasized to local nodes ." However, prognosis is still excellent. Distant hematogenous spread has not been recorded . Since intrapulmonic metastases cannot be separated from a multifocal origin , this patient will be closely monitored during her postoperative course. REFERENCE S
1 Joshi K, Shankar SK, Gopinath N. Multipl e sclerosing hemangiomas of the lung, Postgrad Med J 1980; 56:50-3 2 Katzenstein AA, Gmelich ]T, Carrington CB. Sclerosing hemangioma of the lung: a clinicopathologtc study of 51 cases. Am J Surg PathoI1980; 4:343-56 3 Masayuki N, Tetsuro K, Shoujiro M. Multiple sclerosing hemangioma of the lung. Am J Surg Patholl986; 10:429-35 4 Liebow AA, HuOOIl OS. Sclerosing hemangioma (histiocytoma , xanthoma) of the lung. Cancer 1956; 9:53-75 5 Nagata N, Dairaku M, Sueishi K. Sclerosing hemangioma of the
*From the Division of Pulmonary and Critical Care Medicine, Department of Medicine and Department of Pharmacy, Medical University of South Carolina, Charleston. Reprint request s: Dr. Strange, Medical University of South Carolina , Charleston 29425
Table I-Pleural Fluid and Plasma Concentration Before and After Chylothorax Drainage
lung: an epithelial tumor composed of immunohi stochemically heterogenous cells. Am J Clin Patholl987; 88:552-59 6 lsao A, Masaharu I, Yasuo M. A case ofpneumocytoma (so called sclerosing hemangioma) with lymph node metastasis. Jpn J Clin Oncoll986; 16:77-86
Pleural fluid, day 1 Plasma, day 1 Pleural fluid. day 5 Plasma, day 6
Amiodarone
Desethylamiodarone
2.10 ILWml 1.30 ILwml
0.371LWml 0.821LWml
2 .30 ILwml O.86ILWm l
0 .30 ILWml 0 .60 ILWml
CHEST I 101 12 I FEBRUARY, 1992
573
A Chinese woman had bilateral coin lesions of the lung, which grew slowly. At pathologic examination, the lesions were considered to be multiple sclerosing hemangiomas. Multiplicity is rare, and bilaterality has not been previously reported. (Chest 1992; 101:572-73)
S
cle rosillJ.!: hemangioma is generally considered to be a solitary benign lesion , Multifocality is rare .' ..1 We report the case of a Chinese woman with bilateral coin lesions of the IUIIJ.!:, which were proved to be bilateral multiple sclerosing hemangiomas. CASE R":I'ORT
A 48-yt'ar-old Chinese woman was followed up fi,r three years with serial radiographs and computed tomographic (C'T) scans. The hilatl>rallung masses grew slowly. A hOlllogl'nt'OUs, 5-cm round l"in
*From the
Departments of Surgery (Drs. Lee and Lee) and Clinical Patholoh'Y (Dr. Lin), National Taiwan University Hospital, Taipei ; and till' Department ofInternal Medicine, Cathy Generaillospital (Dr. Hsu), Taipei, Taiwan , Republic of China. Reprint requests: Dr. I.....'e, National uliwlIn Unil)('rsity Hospital, 1, Chang-tv Street , TlIipei, TlIiwlIn. Republic of China
lesion in the left lower lohe was accompanied by two smaller lesions in the right lung. A C'T scan demonstrated a fourth lesion in the lingula of the left lung (Fig 1). At hilateral anteroluteral thoracotomy; three wedge sections were obtained , and a left lower basal segmentectomy was performed. The left lung contained a 5 x 5-slj em anterior busul lower lohe tumor and a 0.8 x 0 .7-slj em inferior basal hngular tumor. The right lung contained a 1.8 x 1.5-slj em lateral middle lobe mass and a 0.8 x 0.6-slj ern anterior basal lower lobe mass (Ftg 2). Each tumor was firm, elastic, and yellow, with focal hemorrhage. At microscopic examination, all four tumors were considered to be sclerosing hemangiomas composed of polygonal cells arranged in solid or papillary pallerns. FIle'll sclerosis and foamy histiocytes were present (Fig 3). DISCUSSION
The rubric "sclerosing hemangioma" proposed by Liebow and H ubbel' defines a subset of'benign tumors and tumorlike lesions of the lung, This subset diJfers from plasma cell granuloma and pseudolymphoma and may include cases of xanthomatous pseudotumor, postinflammatory pseudotumor, fibrous histiocytoma. alveolar angioblastoma, and the recently described pneumocytoma. The cell of origin was initially considered to be endothelial and thus the tumor to be akin to a tumor of the dermis of the skin . Subsequent electron microscopic studies suggested ' either alveolar pneurnocytes or mesothelium as the origin. Multiple authors utilizing immunologic studies have sup-
FIGllRE 1. Right, Chest x-ray film showed three coin-shaped tumor masses in bilateral lung fields, which were homogeneous in density (lIrrows). Left, on chest CT scan , another small lesion was found in the lingula (double lIrrows). U
c
\
FIGllRE 2. Gross specimens demonstrate the ball-like appearance of the tumors, which were found in the lateral basal segment of the left lower lobe (A), the inferior segment of the lingula (B), the anterior basal segment of the right lower lobe (C), and the lateral segment of the right middle lobe (D).
572
Bilateral Multiple SClerosing Hemangiomas (Lee
et Ill)
Chylous Transport of Amiodarone* Charli e Strange, M.D ., EC .C.P.; Dacid P. Nicolau , Pharm . D.; and Scott R. Drqzer; M.D .
A patient receiving amiodarone for longstanding ventricular dysrhythmias presented with idiopathic chylothorax, During drainage of chylothorax for pleurodesis, serial plasma amiodarone concentrations declined while pleural fluid concentrations remained stabile. Chylous transport of amiodarone and other lipid-bound drugs should be recognized to avert complications during chylothorax drainage. (Chest 1992; 101:573-74)
of amiodarone remain incomT hepletelypharmacokinetics understood . Following an oral dose, low bioaFIGURE 3. Histologic appearance of sclerosing hemangioma in this case . Tumor is composed of polygonal cells arranged in solid or papillary pattern. Focal sclerosing areas and aggregates of foamy histocytes are also observed (hematoxylin-eosin stain , original magnification x 400).
ported the hypothesis that both bronchiolar cells and alveolar type II pneumocytes are simultaneously the cells of origin .5 Thus, "pneumocytoma" is proposed as the appropriate term . Clinically, this tumor is asymptomatic and is an incidental x-ray finding. Chest pain, hemoptysis, and cough may result from impingement of a large tumor on adjacent structures of the bronchovascular ray. Fewer than ten cases of multifocal sclerosing hemangioma
vailability with interindividual variation has been attributed to intestinal mucosal cell dealkylation of amiodarone before it reaches the portal venous system .' Because of extensive protein binding within the circulation," a delay has been noted before amiodarone is distributed into tissue . Preferential uptake into adipose liver, and pulmon ary tissue has been noted with a steady state partition coefficient at least 100fold above plasma ."?The plasma concentration threshold for antiarrhythmic activity has been estimated to be between 0.5 to 1.0 Ilog per ml." We recently encountered a patient with idiopathic chylothorax which allowed investigation of the chylous content of amiodarone . Because of arniodarones lipid affinity, we questioned whether amiodarone would be removed during chest tube drainage.
have been reported in the English-language literature . ' ·3
CASE REPORT
This case is unique in that different lobes of the lung were
A 76-yea r-old white man presented with a three-month history of large right-sided pleural effusions. Serial thoracentesis had demon strated milky white Auid with triglyceride concentration = 127 mg/dl consistent with chylothorax . After chest and abdominal CT scanning and pleural fluid cytology had failed to demonstrate a malignancy, a chest-tube was placed for tetracycline pleurodesis. Malignant ventricular arrh ythmia s associated with coronary artery disease had been stabil ized one year previou sly with amiodarone 400 mg qd . No sign of amiodarone toxicity had been noted . After chest-tube placement, drainage of 9 L of chylous pleural effusion was accompli shed over a six-day interval. Although daily evaluation of electrolytes had been performed without change, the patient experienced sudden death while walking across his hospital room. No autopsy was performed . Pleural Auid and serum arniodarone and desethylarniodarone levels were performed on initial evaluation and near the time of death. Data appear in Table 1.
involved and in that the distribution was bilateral. One reported case is stated to have metastasized to local nodes ." However, prognosis is still excellent. Distant hematogenous spread has not been recorded . Since intrapulmonic metastases cannot be separated from a multifocal origin , this patient will be closely monitored during her postoperative course. REFERENCE S
1 Joshi K, Shankar SK, Gopinath N. Multipl e sclerosing hemangiomas of the lung, Postgrad Med J 1980; 56:50-3 2 Katzenstein AA, Gmelich ]T, Carrington CB. Sclerosing hemangioma of the lung: a clinicopathologtc study of 51 cases. Am J Surg PathoI1980; 4:343-56 3 Masayuki N, Tetsuro K, Shoujiro M. Multiple sclerosing hemangioma of the lung. Am J Surg Patholl986; 10:429-35 4 Liebow AA, HuOOIl OS. Sclerosing hemangioma (histiocytoma , xanthoma) of the lung. Cancer 1956; 9:53-75 5 Nagata N, Dairaku M, Sueishi K. Sclerosing hemangioma of the
*From the Division of Pulmonary and Critical Care Medicine, Department of Medicine and Department of Pharmacy, Medical University of South Carolina, Charleston. Reprint request s: Dr. Strange, Medical University of South Carolina , Charleston 29425
Table I-Pleural Fluid and Plasma Concentration Before and After Chylothorax Drainage
lung: an epithelial tumor composed of immunohi stochemically heterogenous cells. Am J Clin Patholl987; 88:552-59 6 lsao A, Masaharu I, Yasuo M. A case ofpneumocytoma (so called sclerosing hemangioma) with lymph node metastasis. Jpn J Clin Oncoll986; 16:77-86
Pleural fluid, day 1 Plasma, day 1 Pleural fluid. day 5 Plasma, day 6
Amiodarone
Desethylamiodarone
2.10 ILWml 1.30 ILwml
0.371LWml 0.821LWml
2 .30 ILwml O.86ILWm l
0 .30 ILWml 0 .60 ILWml
CHEST I 101 12 I FEBRUARY, 1992
573
