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Intrathoracic Osteosarcoma Diagnosed by CT Scan and Pleural Biopsy
Intrathoracic Osteosarcoma Diagnosed by CT Scan and Pleural Biopsy* John P. Connolly, M .D.;t Curtis A. McGuyer, M .D.;t W Scott Sageman , M .D. ;t and Harry Bailey, M.D .§
Oste osa rc oma r arely presents as a primary lesion in th e ches t, whe re as pulmona ry metastases are common. The diagnosi s of primar y intrathoracic osteosarcoma has invariably been by thoracotom y or autopsy. We present a case of a densely calcifie d, primary intrathoracic osteosarcoma whe re diagnosis was mad e ante mor tem by pleural biopsies and computed tom ogr aphy scan . (Chest 1991; 100:265-67)
P
rimary intrathoracic osteosarcoma is rare . Extraosseous osteosarco ma originating in the lung is 500 tim es less common th an ca rcino ma of the lung . I Sarcoma originating from the bony ele me nts of th e chest wall is even rarer, and few re ports of th is entity can be found ." We present th e case of an elde rly patient, who was not a can didate for diagnostic th oracotomy, in whom an expanding, dens ely calcified, int rathoracic mass was demonstrated to be an ost eosarcoma by pleural biop sies and com puted tomography scan. CASE REPOHT
A 93-year-old F ilipino man was fou nd to have asymptomatic , leftuppe r-lobe , calcific lun g d e nsities on chest x-ray film in December 1987 (F ig 1). In retrospect, a chest x-ray film of Apr il 1987 also show ed a ca lcified, left-upper-lobe , lun g abnormality, whic h appeared somewhat mor e p rominen t in th e int erval. Hi s ches t x-ray film was normal in Novembe r 1986 . Th e patien t deni ed che st pain, cough , fever, weight loss, or cha nge in his bas eli ne mild dyspn ea and fe lt he was in good health . He had no p rior h istor y of pn eumonia or can cer. He did not smo ke , and he denied knowledge of tubercu losis exposure . Ph ysical examination an d scr ee ni ng lab orat ory tests were unremarkable . Serial chest x-ray films documented a continue d e n large me nt of the mass (Fig 2). Evalua tion incl ud ed plan e tomography show ing several small nod ular d ensities wit h irr egu lar calcification in th e left upper lu ng fie ld adjace nt to the posterior pleura. A purifi ed prote in derivative test was po sit ive at 20 mm induration . F ive sputum samp les were negative for acid-fast bacilli and oth er pathoge ns, as were b ron choscopy sp ecimen s. A ches t CT scan disclosed a 2 X 2 X 4 cm ma ss ab utting th e pleu ra , wh ich was den sel y calcified . No evidence of unde rlyin g rib dest ruction was see n. D ue to th e patient's advanced age and poor pu lmonar y reser ve , it was felt he was not a candidate for surgical explora tion. Two transth oracic needle aspira tions und e r lIuoroscopic and CT sca n guid an ce re vea led on ly calciu m and nondiagnostic ce llular e le me nts. No pathoge ns were appare nt on sm ear or cultu re . The mass again appeared lar ge r on CT images , which wer e don e for th e pu rpose of biop sy (F ig 3). He was follow ed with serial ches t x-ray examinations.
*Fro m th e D epart me nts of tInte rn al Medi cin e (Pulmonary Division), tLabora to ry Med icin e , §Radio lo!,'Y, and Clinical In vestigation, Naval Hospital, San Diego , Californ ia. Th e work reported herein was pe rformed under the Navy Clinical In vestigat ion Program , case report no. 84-16-1968-148 . Th e views exp res sed in thi s articl e are those of the au thors and do not rell ect th e official policy of the Department of th e Navy, Departm ent of Defen se , nor th e U .S . Governme nt . Rep rint requests: D r. Connolly, Naval Hospital, San Diego 921345000
FIGUHE 1. C he st x-ray film on p rese n tation , Dece mber 1987 . In March 1989, he was fou nd to have a new, la rge , but still asym p tom atic , left pleural effusion (F ig 4). T horacentesis was pe rformed , whi ch revea led a he morr hagic exudative effusio n with negative cytologic examination. A bon e scan revea led mod erate up take in the righ t foot ar ea and inte nse upt ake in the reg ion of his left hemi thorax. X-ray films of the right foot revea led a hea ling compressio n fracture of the talu s hu t nothi ng to sug gest a ma lignant proc ess. His chest x-ray films wer e fe lt to repres ent an e nlarging, left uppe r lohe lu ng mass with exte nsive ple ural stu dd ing an d effusio n. He und erwent repeat thoracentesi s with mu ltip le ple u ral biop sies per formed with the Abra ms needl e . Th ese reveal ed ske le tal mu scle , adi pose tissue , and loci of a ma lignan t neoplasm . T here wer e ar eas man ifestin g a predom inant spind led patt e rn , as we ll as malignant osteohla stic-app earing ce lls admixed with osteo id . T he pathologic findin gs wer e fe lt to he consiste nt wit h a d iagnos is of ost eogen ic sarcoma, although a fibrous mesot helioma with osseous metaplasia was also conside red . Review of th e pa tie nt's clinica l course and CT scan findings was fe lt to he most cons iste nt with osteosarcoma. T he patient d ied at hom e in Dec embe r 1989 ,
F IGUHE 2. En larging mass in August 1988 . CHEST / 100 / 1 / JULY, 1991
265
FI G UII E 3. C hes t CT sec tion fro m December 1988 , don e for CT dir ect ed needle aspira te , show ing den sel y calcified , left-uppe r-lobe
mass.
app roxima te ly 2.5 yea rs afte r first ab norma l che st x-ray film findings. Aut opsy was refused by the family.
DISC USSIO N
Th e most com mon p resentation of oste osarcoma in th e ches t is as a metastatic lesion. In th ese cas es, multipl e lesions are th e norm and th e primary extrathoracic lesion is seldom asy mptomatic . Of th e primary pulmon ar y sar comas, fibrosarcomas and leiomyosar comas see m to be th e most common , I but th e re are a few cas e reports in th e literature of osteos arco ma pres enting as a p rima ry pulmonary lesion .?" Osteo sar coma ma y also arise from the bon y elem ents of the chest wall (eg, ribs). Our patient presented with an expanding, densely calcified mass abutting th e pleura th at was asym ptomatic until afte r it had clearl y exte nde d to th e ple ural space . No evide nce of rib des tructi on or int ramedullary tumor was obse rved on multiple CT scan s. However, th e differential diagno sis of thi s lesi on m ust include parosteal osteosarcoma of th e rib , a les ion that has onl y been reported onc e to our knowledg e ? Roe nt ge nographically, lobular paro steal osteo sarcomas ten d to be den sely ca lcified , lobular masses
adjacent to bon e , with discret e attachme nts to th e underlying cortex as well as a radi olu cent cleavage plane betw een tumor and cortex.!" Slow tumor growth and a more favorable pro gno sis than th at with conve ntiona l osteosar coma s are cha rac teristic of parosteal osteos ar com as." In th e present ca se , th e density of th e calcificati on and th e peripheral location of th e tumor favor th e diagnosis of parosteal ost eosarcoma, although a clear association with th e underl ying ribs was never demonstrated. A second diagno stic pos sibility cons idered was fibr ous mesothelioma with osseous metaplasia. However, th e pattern of calcification an d th e othe r CT findings were not cha racte ristic. Patterns of calcification seen in mesoth elioma include associated ple ur al plaques (often being ab sorbed by tumor) and irregular minimal calcifications along tumor margins." Oth er CT findings of mesotheli om a, including pleural th icke ning, contralate ral pleural abnormalities, par en ch ym al evidence of asb estosis, decrea se in hem ithorax size , and early presentation of pleural effusion, were not seen in th e present case . A th ird, and perhaps most likely possibility is th at this lesion is an ext raosseous osteo sarcoma of lun g tissu e . As a primary lesion in th e lung, ost eos ar coma may ari se from the connect ive tissue eleme nts of th e bronchial wall and present with symptoms of obstruction (eg, wh eezing). It may also arise in th e pulmonary paren chyma and be asymptomatic until it reaches a larg e size or invad es th e adjace nt st ruc ture s. Up to 50 percent of pati e nt s with primary pulmona ry sarcoma s may be asymptomatic at prese ntation," as was our pati ent. The re are no roentgen ographic characte ristics diagnostic of extraosseous pulmonary osteosarcom a, although a large periphe ral, well-circumscribed mass is suggestive of th e diagnosis." Pathologic diagnosis of osteo sarcom a has mo st often been by examination of op en biopsy or by autopsy mater ial providing lar ge histologic specim en s. The use of per cutaneous cutt ing, fine-needle biop sy to diagno se oste osar comas of bon e without th e neces sit y for op en biop sy has been descri bed , and thi s method is wid el y used .P-!" Th e fineneedle aspiration technique , when applied to oste osarcomas, ha s two impo rtant limitations. First, th e den sely osteoblastic tumors yield poorly ce llular aspirates, and sec ond , it is difficult to identify osteoid confide ntly on cytologic smears.10 In th e pres ent case, th e exte nsion of th e mali gnanc y to th e pleural space provid ed th e opportunity to confirm the diagn osis of ost eosarcoma with pleural biopsies without pro ceed ing to high -risk thoracotomy in thi s pati ent. Autopsy was unfort un atel y refus ed , elim inating th e po ssibilit y of further histologic confirmation. A bone sca n showing no evide nce oft umor outside th e ches t confirms its intrath oracic ori gin . In sum mary, we have pres ented a case of an unusual, densely calcified , intrathoracic ost eo sarcoma, likely of pulmonary ori gin , and diagnosed by pleural bi op sies and CT scan review. Diagnosis of intrathoracic ost eos ar coma short ofthoracotomy or autop sy has not been previously described. R E FERE NCES
F IG U HE
266
4 . C hest x-ray film on pr esen tat ion , March 1989.
Gucc ion JG , Rosen SII . Bron chopu lmonary le iomyosarcoma and fibro sarcom a: a stud y of 32 case s and review of the literature . Cance r 1972; 30:836-47 2 Kawa S, Kobayashi S, II an zawa S, \ Vada G , Kimura II , Okamoto Intrathoracic Osteo sarcoma (Connollyet al)
3 4 5 6 7 8 9 10
K. Computed tomography of parosteal osteosarcoma of the rib. ] Comput AssistTomogr 1985; 9:269-73 Nascimento AG, Unni KK, Bernatz PE . Sarcomas of the lung. MayoClin Proc 1982; 57:355-59 Reingold 1M, Amromin GD. Extraosseous osteosarcoma of the lung. Cancer 1971; 28:491-98 Nosanchuk ]S, Weatherbee L. Primary osteogenic sarcoma in the lung: report of a case. ] Thorac Cardiovasc Surg 1969; 58: 242-47 Lindell MM, Shirkhoda A, RaymondAK, Murray ]A, Harle TS. Parosteal osteosarcoma: radiologic-pathologic correlation with emphasis on CT. A]R 1987; 148:323-28 Campanacci M, Pieci P, Gherlinzoni F, Guera A, Bertoni F, Neff]R. Parosteal osteosarcoma. ] Bone Joint Surg 1984; 66B: 313-21 Kreel L. Computed tomography in mesothelioma. Semin Oncol 1981; 8:302-12 de Santos LA, Murray]A, AyalaAG. The value of percutaneous needle biopsy in the management of primary bone tumors. Cancer 1979;43:735-44 White VA, Fanning Cv, AyalaAG, Raymond AK, Carrasco CH, Murray ]A. Osteosarcoma and the role of the fine needle aspiration: A study of 51 cases. Cancer 1988; 62:1238-46
Laceration of the Left Pulmonary Artery during Removal of a Bronchogenic Cyst by Right Thoracotomy· A. Addison Barman , M.D. , F.C.C .P.; Ahamed S. Moid.een, M.D ., F.C.C .P.; Saqib S. Chaudhry, M.D ., F.C .C .P.; and Deborah Reich , B.A.
Major vascular involvement by a bronchogenic cyst is rare; most large cysts cause respiratory symptoms. We present a case in which a large bronchogenic cyst was adherent to the left pulmonary artery. During removal of the cyst by right thoracotomy, the artery lacerated. Repair of the artery in such a situation taxes the surgeon's ingenuity and decision-making process. (Chest 1991; 100:267-68)
T ace ration of the left pulmonary artery during excision of L a bronchogenic cyst by right thoracotomy presents a formidable surgical challenge. Inflow occlusion of both venae cavae provides a bloodless field but only allows a short time for repair of the pulmonary artery. To the best of our knowledge, this is the first reported case where this particular set of circumstances occurred. CASE REPORT
A32-year-old patient with chronic renal failure who was receiving hemodialysis was hospitalized in December 1989 with the chief complaints of chest pain. cough, and some difficulty in breathing. He was known to have a "cyst of the lung" noted on chest roentgenogram two years ago for which surgery was recommended but refused. Routine chest roentgenogram as compared with the previous one showed considerable enlargement of the cystic mass. This was confirmed with a computed tomographic (CI) scan (Fig 1).
*From the Department of Surgery, Flushing Hospital Medical Center. Flushing, New York.
Reprint requests : Dr. Barman, 146-01 45th Avenu e, Flushing , SY
11355
FIGl 'R E 1. Computed tomography shows large bronchogenic cyst located to right of midline. Because of symptoms and the increased size of the cyst, surgery was recommended . A right posterolateral thoracotomy was used to approach the cyst that was freed with great difficulty from the carina, right main bronchus. and the esophagus. While attempting to free the cyst from the left lateral aspect structure s, brisk hemorrhage ensued that proved to be a laceration of the left pulmonary artery. Since we were in the right side of the chest, even with single lung anesthesia with a double lumen tube that allowed for a complete collapse of the right lung, repair of the pulmonary artery in the depth s of the mediastinum was not feasible. With digital compression of the laceration controlling the hemorrhage, the pericardium was widely opened anter ior to the right phrenic nerve and encircling loops were placed around both venae cavae. After inflow occlusion, the operative field was sufficiently dry to allowfor repair of the left pulmonary artery by continuous polypropylene suture . The inflow occlusion time was approximately 1'/. minutes. The cyst was removed piecemeal and a remaining piece of the wall had its lining ablated by electrocautery. The patient had an uneventful postoperative course. DISCUSSIO:'ll
Most of the symptoms of bronchogenic cysts are due to compression and irritation of the airway. 1 Although compression of vascular structures has been previously descnbed.v' to the best of our knowledge , a lacerated left pulmonary artery during removal of a bronchogenic cyst by right thoracotomy has not been reported. There are several lessons to be learned from this case : (1) Bronchogenic cyst s in an adult are liable to be adherent to neighboring structures because of intense inflammatory reaction. (2) Total removal may be hazardous and one should opt for partial excision and ablation of the lining by electrocautery. (3) If CT shows adherence to the left pulmonary artery, one should remember that the artery is anterior to the left main bronchus and hence, quite inaccessible by right thoracotomy whe n the subcarinal location makes such an incision appropriate (4) One-lung ventilation provides ideal exposure by collapse of the right lung. (5) Tear of the left pulmonary artery is a formidable problem. Should this occur, the sur geon should immediately proceed to inflow occlusion by snaring both venae cavae after generous pericardiotomy. REFERENCES
Strivella S, F1>n! WB. Zikria EM. Miller \\'11. Samadani SR. Sullivan ME. Foregut cysts of the medinxtinum: results in 20 consecutive surgically treated cases. J Thorne Cardiovasc Sun; 1985: 90:;;6-82 CHEST I 100 I 1 I JULY, 1991
267
Oste osa rc oma r arely presents as a primary lesion in th e ches t, whe re as pulmona ry metastases are common. The diagnosi s of primar y intrathoracic osteosarcoma has invariably been by thoracotom y or autopsy. We present a case of a densely calcifie d, primary intrathoracic osteosarcoma whe re diagnosis was mad e ante mor tem by pleural biopsies and computed tom ogr aphy scan . (Chest 1991; 100:265-67)
P
rimary intrathoracic osteosarcoma is rare . Extraosseous osteosarco ma originating in the lung is 500 tim es less common th an ca rcino ma of the lung . I Sarcoma originating from the bony ele me nts of th e chest wall is even rarer, and few re ports of th is entity can be found ." We present th e case of an elde rly patient, who was not a can didate for diagnostic th oracotomy, in whom an expanding, dens ely calcified, int rathoracic mass was demonstrated to be an ost eosarcoma by pleural biop sies and com puted tomography scan. CASE REPOHT
A 93-year-old F ilipino man was fou nd to have asymptomatic , leftuppe r-lobe , calcific lun g d e nsities on chest x-ray film in December 1987 (F ig 1). In retrospect, a chest x-ray film of Apr il 1987 also show ed a ca lcified, left-upper-lobe , lun g abnormality, whic h appeared somewhat mor e p rominen t in th e int erval. Hi s ches t x-ray film was normal in Novembe r 1986 . Th e patien t deni ed che st pain, cough , fever, weight loss, or cha nge in his bas eli ne mild dyspn ea and fe lt he was in good health . He had no p rior h istor y of pn eumonia or can cer. He did not smo ke , and he denied knowledge of tubercu losis exposure . Ph ysical examination an d scr ee ni ng lab orat ory tests were unremarkable . Serial chest x-ray films documented a continue d e n large me nt of the mass (Fig 2). Evalua tion incl ud ed plan e tomography show ing several small nod ular d ensities wit h irr egu lar calcification in th e left upper lu ng fie ld adjace nt to the posterior pleura. A purifi ed prote in derivative test was po sit ive at 20 mm induration . F ive sputum samp les were negative for acid-fast bacilli and oth er pathoge ns, as were b ron choscopy sp ecimen s. A ches t CT scan disclosed a 2 X 2 X 4 cm ma ss ab utting th e pleu ra , wh ich was den sel y calcified . No evidence of unde rlyin g rib dest ruction was see n. D ue to th e patient's advanced age and poor pu lmonar y reser ve , it was felt he was not a candidate for surgical explora tion. Two transth oracic needle aspira tions und e r lIuoroscopic and CT sca n guid an ce re vea led on ly calciu m and nondiagnostic ce llular e le me nts. No pathoge ns were appare nt on sm ear or cultu re . The mass again appeared lar ge r on CT images , which wer e don e for th e pu rpose of biop sy (F ig 3). He was follow ed with serial ches t x-ray examinations.
*Fro m th e D epart me nts of tInte rn al Medi cin e (Pulmonary Division), tLabora to ry Med icin e , §Radio lo!,'Y, and Clinical In vestigation, Naval Hospital, San Diego , Californ ia. Th e work reported herein was pe rformed under the Navy Clinical In vestigat ion Program , case report no. 84-16-1968-148 . Th e views exp res sed in thi s articl e are those of the au thors and do not rell ect th e official policy of the Department of th e Navy, Departm ent of Defen se , nor th e U .S . Governme nt . Rep rint requests: D r. Connolly, Naval Hospital, San Diego 921345000
FIGUHE 1. C he st x-ray film on p rese n tation , Dece mber 1987 . In March 1989, he was fou nd to have a new, la rge , but still asym p tom atic , left pleural effusion (F ig 4). T horacentesis was pe rformed , whi ch revea led a he morr hagic exudative effusio n with negative cytologic examination. A bon e scan revea led mod erate up take in the righ t foot ar ea and inte nse upt ake in the reg ion of his left hemi thorax. X-ray films of the right foot revea led a hea ling compressio n fracture of the talu s hu t nothi ng to sug gest a ma lignant proc ess. His chest x-ray films wer e fe lt to repres ent an e nlarging, left uppe r lohe lu ng mass with exte nsive ple ural stu dd ing an d effusio n. He und erwent repeat thoracentesi s with mu ltip le ple u ral biop sies per formed with the Abra ms needl e . Th ese reveal ed ske le tal mu scle , adi pose tissue , and loci of a ma lignan t neoplasm . T here wer e ar eas man ifestin g a predom inant spind led patt e rn , as we ll as malignant osteohla stic-app earing ce lls admixed with osteo id . T he pathologic findin gs wer e fe lt to he consiste nt wit h a d iagnos is of ost eogen ic sarcoma, although a fibrous mesot helioma with osseous metaplasia was also conside red . Review of th e pa tie nt's clinica l course and CT scan findings was fe lt to he most cons iste nt with osteosarcoma. T he patient d ied at hom e in Dec embe r 1989 ,
F IGUHE 2. En larging mass in August 1988 . CHEST / 100 / 1 / JULY, 1991
265
FI G UII E 3. C hes t CT sec tion fro m December 1988 , don e for CT dir ect ed needle aspira te , show ing den sel y calcified , left-uppe r-lobe
mass.
app roxima te ly 2.5 yea rs afte r first ab norma l che st x-ray film findings. Aut opsy was refused by the family.
DISC USSIO N
Th e most com mon p resentation of oste osarcoma in th e ches t is as a metastatic lesion. In th ese cas es, multipl e lesions are th e norm and th e primary extrathoracic lesion is seldom asy mptomatic . Of th e primary pulmon ar y sar comas, fibrosarcomas and leiomyosar comas see m to be th e most common , I but th e re are a few cas e reports in th e literature of osteos arco ma pres enting as a p rima ry pulmonary lesion .?" Osteo sar coma ma y also arise from the bon y elem ents of the chest wall (eg, ribs). Our patient presented with an expanding, densely calcified mass abutting th e pleura th at was asym ptomatic until afte r it had clearl y exte nde d to th e ple ural space . No evide nce of rib des tructi on or int ramedullary tumor was obse rved on multiple CT scan s. However, th e differential diagno sis of thi s lesi on m ust include parosteal osteosarcoma of th e rib , a les ion that has onl y been reported onc e to our knowledg e ? Roe nt ge nographically, lobular paro steal osteo sarcomas ten d to be den sely ca lcified , lobular masses
adjacent to bon e , with discret e attachme nts to th e underlying cortex as well as a radi olu cent cleavage plane betw een tumor and cortex.!" Slow tumor growth and a more favorable pro gno sis than th at with conve ntiona l osteosar coma s are cha rac teristic of parosteal osteos ar com as." In th e present ca se , th e density of th e calcificati on and th e peripheral location of th e tumor favor th e diagnosis of parosteal ost eosarcoma, although a clear association with th e underl ying ribs was never demonstrated. A second diagno stic pos sibility cons idered was fibr ous mesothelioma with osseous metaplasia. However, th e pattern of calcification an d th e othe r CT findings were not cha racte ristic. Patterns of calcification seen in mesoth elioma include associated ple ur al plaques (often being ab sorbed by tumor) and irregular minimal calcifications along tumor margins." Oth er CT findings of mesotheli om a, including pleural th icke ning, contralate ral pleural abnormalities, par en ch ym al evidence of asb estosis, decrea se in hem ithorax size , and early presentation of pleural effusion, were not seen in th e present case . A th ird, and perhaps most likely possibility is th at this lesion is an ext raosseous osteo sarcoma of lun g tissu e . As a primary lesion in th e lung, ost eos ar coma may ari se from the connect ive tissue eleme nts of th e bronchial wall and present with symptoms of obstruction (eg, wh eezing). It may also arise in th e pulmonary paren chyma and be asymptomatic until it reaches a larg e size or invad es th e adjace nt st ruc ture s. Up to 50 percent of pati e nt s with primary pulmona ry sarcoma s may be asymptomatic at prese ntation," as was our pati ent. The re are no roentgen ographic characte ristics diagnostic of extraosseous pulmonary osteosarcom a, although a large periphe ral, well-circumscribed mass is suggestive of th e diagnosis." Pathologic diagnosis of osteo sarcom a has mo st often been by examination of op en biopsy or by autopsy mater ial providing lar ge histologic specim en s. The use of per cutaneous cutt ing, fine-needle biop sy to diagno se oste osar comas of bon e without th e neces sit y for op en biop sy has been descri bed , and thi s method is wid el y used .P-!" Th e fineneedle aspiration technique , when applied to oste osarcomas, ha s two impo rtant limitations. First, th e den sely osteoblastic tumors yield poorly ce llular aspirates, and sec ond , it is difficult to identify osteoid confide ntly on cytologic smears.10 In th e pres ent case, th e exte nsion of th e mali gnanc y to th e pleural space provid ed th e opportunity to confirm the diagn osis of ost eosarcoma with pleural biopsies without pro ceed ing to high -risk thoracotomy in thi s pati ent. Autopsy was unfort un atel y refus ed , elim inating th e po ssibilit y of further histologic confirmation. A bone sca n showing no evide nce oft umor outside th e ches t confirms its intrath oracic ori gin . In sum mary, we have pres ented a case of an unusual, densely calcified , intrathoracic ost eo sarcoma, likely of pulmonary ori gin , and diagnosed by pleural bi op sies and CT scan review. Diagnosis of intrathoracic ost eos ar coma short ofthoracotomy or autop sy has not been previously described. R E FERE NCES
F IG U HE
266
4 . C hest x-ray film on pr esen tat ion , March 1989.
Gucc ion JG , Rosen SII . Bron chopu lmonary le iomyosarcoma and fibro sarcom a: a stud y of 32 case s and review of the literature . Cance r 1972; 30:836-47 2 Kawa S, Kobayashi S, II an zawa S, \ Vada G , Kimura II , Okamoto Intrathoracic Osteo sarcoma (Connollyet al)
3 4 5 6 7 8 9 10
K. Computed tomography of parosteal osteosarcoma of the rib. ] Comput AssistTomogr 1985; 9:269-73 Nascimento AG, Unni KK, Bernatz PE . Sarcomas of the lung. MayoClin Proc 1982; 57:355-59 Reingold 1M, Amromin GD. Extraosseous osteosarcoma of the lung. Cancer 1971; 28:491-98 Nosanchuk ]S, Weatherbee L. Primary osteogenic sarcoma in the lung: report of a case. ] Thorac Cardiovasc Surg 1969; 58: 242-47 Lindell MM, Shirkhoda A, RaymondAK, Murray ]A, Harle TS. Parosteal osteosarcoma: radiologic-pathologic correlation with emphasis on CT. A]R 1987; 148:323-28 Campanacci M, Pieci P, Gherlinzoni F, Guera A, Bertoni F, Neff]R. Parosteal osteosarcoma. ] Bone Joint Surg 1984; 66B: 313-21 Kreel L. Computed tomography in mesothelioma. Semin Oncol 1981; 8:302-12 de Santos LA, Murray]A, AyalaAG. The value of percutaneous needle biopsy in the management of primary bone tumors. Cancer 1979;43:735-44 White VA, Fanning Cv, AyalaAG, Raymond AK, Carrasco CH, Murray ]A. Osteosarcoma and the role of the fine needle aspiration: A study of 51 cases. Cancer 1988; 62:1238-46
Laceration of the Left Pulmonary Artery during Removal of a Bronchogenic Cyst by Right Thoracotomy· A. Addison Barman , M.D. , F.C.C .P.; Ahamed S. Moid.een, M.D ., F.C.C .P.; Saqib S. Chaudhry, M.D ., F.C .C .P.; and Deborah Reich , B.A.
Major vascular involvement by a bronchogenic cyst is rare; most large cysts cause respiratory symptoms. We present a case in which a large bronchogenic cyst was adherent to the left pulmonary artery. During removal of the cyst by right thoracotomy, the artery lacerated. Repair of the artery in such a situation taxes the surgeon's ingenuity and decision-making process. (Chest 1991; 100:267-68)
T ace ration of the left pulmonary artery during excision of L a bronchogenic cyst by right thoracotomy presents a formidable surgical challenge. Inflow occlusion of both venae cavae provides a bloodless field but only allows a short time for repair of the pulmonary artery. To the best of our knowledge, this is the first reported case where this particular set of circumstances occurred. CASE REPORT
A32-year-old patient with chronic renal failure who was receiving hemodialysis was hospitalized in December 1989 with the chief complaints of chest pain. cough, and some difficulty in breathing. He was known to have a "cyst of the lung" noted on chest roentgenogram two years ago for which surgery was recommended but refused. Routine chest roentgenogram as compared with the previous one showed considerable enlargement of the cystic mass. This was confirmed with a computed tomographic (CI) scan (Fig 1).
*From the Department of Surgery, Flushing Hospital Medical Center. Flushing, New York.
Reprint requests : Dr. Barman, 146-01 45th Avenu e, Flushing , SY
11355
FIGl 'R E 1. Computed tomography shows large bronchogenic cyst located to right of midline. Because of symptoms and the increased size of the cyst, surgery was recommended . A right posterolateral thoracotomy was used to approach the cyst that was freed with great difficulty from the carina, right main bronchus. and the esophagus. While attempting to free the cyst from the left lateral aspect structure s, brisk hemorrhage ensued that proved to be a laceration of the left pulmonary artery. Since we were in the right side of the chest, even with single lung anesthesia with a double lumen tube that allowed for a complete collapse of the right lung, repair of the pulmonary artery in the depth s of the mediastinum was not feasible. With digital compression of the laceration controlling the hemorrhage, the pericardium was widely opened anter ior to the right phrenic nerve and encircling loops were placed around both venae cavae. After inflow occlusion, the operative field was sufficiently dry to allowfor repair of the left pulmonary artery by continuous polypropylene suture . The inflow occlusion time was approximately 1'/. minutes. The cyst was removed piecemeal and a remaining piece of the wall had its lining ablated by electrocautery. The patient had an uneventful postoperative course. DISCUSSIO:'ll
Most of the symptoms of bronchogenic cysts are due to compression and irritation of the airway. 1 Although compression of vascular structures has been previously descnbed.v' to the best of our knowledge , a lacerated left pulmonary artery during removal of a bronchogenic cyst by right thoracotomy has not been reported. There are several lessons to be learned from this case : (1) Bronchogenic cyst s in an adult are liable to be adherent to neighboring structures because of intense inflammatory reaction. (2) Total removal may be hazardous and one should opt for partial excision and ablation of the lining by electrocautery. (3) If CT shows adherence to the left pulmonary artery, one should remember that the artery is anterior to the left main bronchus and hence, quite inaccessible by right thoracotomy whe n the subcarinal location makes such an incision appropriate (4) One-lung ventilation provides ideal exposure by collapse of the right lung. (5) Tear of the left pulmonary artery is a formidable problem. Should this occur, the sur geon should immediately proceed to inflow occlusion by snaring both venae cavae after generous pericardiotomy. REFERENCES
Strivella S, F1>n! WB. Zikria EM. Miller \\'11. Samadani SR. Sullivan ME. Foregut cysts of the medinxtinum: results in 20 consecutive surgically treated cases. J Thorne Cardiovasc Sun; 1985: 90:;;6-82 CHEST I 100 I 1 I JULY, 1991
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