Case Reports Columnar Papilloma of the Bronchus Case Report and Literature Review1,2 STEPHEN BASHEDA, GORDON N. GEPHARDT, and JAMES K. STOLLER
Introduction Endobronchial papillomas are rare (1-3),the commonest of which are solitary squamous papillomas (1).This report presents a patient with a papillary columnar adenoma, the eighth such case to our knowledge.
Case Report A 74-yr-oldwhite man presented with recurrent hemoptysis. For severalyears he had produced chronic sputum consistent with chronic bronchitis. During the previous 2 yr, he had experienced several episodes of blood-streaked sputum. No other bleeding diathesis was evident. He denied any history of tuberculosis, thromboembolic disease, or pneumonia, but he continued to smoke one pack of cigarettes daily, as he had for 60 years. No occupational exposures were elicited. Theophylline and a steroid metered-dose inhaler controlled his symptoms of chronic obstructive pulmonary disease. His physical examination was normal. Laboratory data demonstrated a leukocytosis (20,800 WBC with a normal differential count). Arterial blood gases, prothrombin time, partial thromboplastin time, and platelet count were normal. Pulmonary function tests revealed a FEV 1 of 1.18 L (39070 predicted), a FVC of 3.38 L (85% predicted), and a FEV,/FVC demonstrating severe obstruction (ratio, 0.35). A chest radiograph demonstrated an abnormal density adjacent to the right heart border, better shown to be a right lower lobe collapse on chest CT (figure 1). Fiberoptic bronchoscopy demonstrated a white, glistening, friable mass arising from the superior segment of the right lower lobe (figure 2 ), which obstructed the bronchus to the basilar segments. As shown in figure 3, microscopic examination of biopsies fixed in Hollande's solution demonstrated multiple segments of a papilloma exhibiting a single layer of columnar cells without cilia. Most cells exhibited basal nuclei and clear cytoplasm, but neither mitotic figures nor mucicarmine-positive material were found. There was no evidence of atypia or invasion of the stroma by the neoplasm. Lymphocytesand plasma cellswere prominent with-
(Received in original form April 10, 1990 and in revised form June 10, 1991) , From the Departments of Pulmonary Disease and Pathology, The Cleveland Clinic Foundation, Cleveland, Ohio. 2 Correspondence and requests for reprints should be addressed to James K. Stoller, M.D., Department of Pulmonary Disease,A90, The Cleveland Clinic Foundation, One Clinic Center, Cleveland, OH 44195.
1400
SUMMARY Solitary papillomas are rare endobronchial neoplasms that are most commonly lined by a squamous epithelium (I.e., squamous papillomas). Papillomas exhibiting nonsquamous epithelium are even more unusual; only seven solitary papillomas lined by columnar epithelium (I.e., columnar papillomas) have been previously described. The current report presents the eighth patient with a columnar papilloma and demonstrates some features common to solitary papillomas (I.e., central location In the airways, associated atelectasis), but distinctive from squamous papilloAM REV RESPIR DIS 1991; 144:1400-1402 mas (I.e., absence of malignant potential).
Fig. 1. Chest CT scan demonstrating partial collapse (arrow) of right lower lobe by columnar papilloma.
Fig. 2. Bronchoscopicviewofcolumnar papilloma partially occluding basilar segments of right lower lobe.
1401
CASE REPORT
in the connective tissue stroma of the papill ary stalks. The undigested and digested periodic acidSchiff stains demons trat ed no evidence of neutral mucins. Imm unoperoxidase sta ining performed on sections from the paraffin-embedded material demonstrated no evidence of enolase, chromogranin or serotonin.
Fig . 3. Papillary columnar adenoma of the bronchus. At th is magn ificat ion , the column ar cells are well defined , as is the fib rovasc ular co re. Hema toxy lin-eosin stain ; magn ification: 260.
TABLE 1 SUMMARY OF AVAILABLE REPORTS OF ENDOBRONCHIAL PAPILLOMAS (n = 59) Type of Epithe lial Cell Lini ng Papilloma
Au thor (Date) Pollack (1938) Ashmore (1953 ) Ashley (196 3) Sm ith (1963) Dren nan (1965) Jac kson (1968) Laubsc her (1969) Assor (1971) Frea nt (1971) Spencer (1980) Rov iaro (1981) Hurt (1984) Maxwell (1985) Barz6 (1987) Trillo (1988)
Reference No. 5 6 7 8 4 9 10 2 11 1 3 12 13 14 15
Tota l
Pat ien ts
Total Squamous '
Total Cubo idal or Col umnar '
(n)
(n with dysplasia )
(n with dysplasia)
1 1 2
(O) (O) 8 1 2 1
1
2 1 1 5 21 4 1 1
5 7 3 1 1
1 (O) (O) (O) (O)
(1) (0)
(O) (O)
(0)
(0)
(O)
(O)
(0) 4 (0) 1 (0) (0) (0)
8 2
(4) (3) (0) (1) 2 (1) 2 (0)
59
33 (10)
1 0 1 0 0 0 0 1 0 10 0 0 0
(0) 1 (0)
(0)
8
Other/ Not Spec ifiedt
(O) (0)
6
7 (0)
19
0
• Number with dysplasia shown in parentheses. t Includes inflammatory polyps. squamous cell carci noma, cystadenomas, transitional cell polyps.
Discussion Papillomatous lesions of the endobronchial tree have been classified into thr ee groups (1-3): (1) multiple squamous papillomas, as seenin the childhood diseaseof juvenilelaryngotra cheobronchial papillomatosis; (2) inflammatory pol yps associa ted with an endobron chial foreign bod y, chronic bron chitis, or broncholithiasis; (3) solitary papillomas (4). Solitary papillomas of the endobronchial tree are rare neoplasms. As reviewed in table I, 59 well-documented cases (present case excluded) emerged from our literature search. Presen ting symptoms of all solitary papillomas reflect chronic airway obstruction and include produ ctive cough, hemopt ysis, wheezing, and dyspnea, often leading to misdiagnosis as asthma or chronic bronchitis. Patients may carry this diagnosis for mon ths to years before the diagnosis of endobronchial pap illoma is discovered. Commonly, radiogra phic findings of obstructive pneumonitis, abscess, atelectasis, or bronchiectasis are evident. Pathologically, these solitary pap illomas usually consist of squamous epithelium (squamou s papilloma), with approximatel y onethird demonstrating squamous dysplasia, carcinoma in situ, or invasive carcinoma (table 1). When feasible, conventiona l wisdom suggests that squamous papillomas should be surgically removed becau se of this malignant predispo sition. In contrast to squamous papillomas, columnar (or cuboidal) papillomas are much rarer, with seven cases previously reported to our knowledge (ta ble 2). As summarized in table 2, the eight available reports (current case included) suggest that columnar papillomas occur most commonly in middle age (mean age, 55 years; range, 26 to 74 years), show a male predom inance (5:2 in described cases) and show a right-sided predom inance (also 5:2), especially for the right lower lobe. The pa-
TAB LE 2 SUMMARY OF FEATURES OF AVAILABLE BRONCHIAL COLUMNAR PAPILLOMAS (n = 8)
Au thor (Date) Ashmore (1953) Ashle y (1963)
Refere nce No.
Roviaro (1981)
(n)
Sex
Location
51 62
F M
RLL RLL
Ciliated columnar Ciliated columnar
4 (1)
60
M
LLL
(2)
59
M
RLL
Cubo idal + mult ilayered nonsquamous Columnar + multil ayered nonsquamous
(3) (4) 1
26 57
6
7
Spen cer (196 0)
3
Cu rrent Case Definirion of abbr eviati ons : RLL
Ep ithelial Cell Typ e
Age n
74
No details ava ilable F LUL M RLL M
= right lower lobe; RML = right middle
RLL
Co lumnar + cuboidal Ciliated columnar/cuboidal
Treatm ent
Foll ow-up
Bronchoscopic rem oval
No recurrence afte r 18 month s
LLL lobectom y Bronchos copic re mo val
Lobec tomy Bilobectomy (RLU RML)
Simple columnar
lobe: LUL - left upper lobe; LLL
No recurrence after 2.5 y
= left lower lobe; numbe rs in parentheses reler
Lost to follow-up to case number in the Spencer series.
1402
tient presented in the current report is the oldest patient with a columnar papilloma described to date. Although follow-up information is limited, none of the available cases has demonstrated malignant transformation, again in contrast to the more common squamous papilloma. In the absence of demonstrable malignant potential, conservative management of columnar papillomas (i.e., with laser ablation or bronchoscopic debridement) seems justifiable, in contrast to surgical resection that is favored for squamous papillomas. In summary, the current report presents the eighth available patient with a solitary bronchial papilloma lined with columnar epithelium. In the small number of available cases, common clinical features include presentation in middle-aged men with endobronchial masses obstructing the right lower lobe. When possible, conservative management (e.g., endobronchial removal) seems justified by the absence of observed malignant potential in columnar bronchial papillomas. However, the
CASE REPORT
lack of long-term follow-up experience may be responsible for this failure to observe malignancy.
7. Ashley DJB, Banino EA, Davies HD. Bronchial polyps. Thorax 1963; 18:45-9. 8. Smith JF, Dexter D. Papillary neoplasms of the bronchus of low grade malignancy. Thorax 1963;
Acknowledgment The writers thank J. Schaaf, M.D., for referring the patient.
9. Jackson DA, Hatch HB. Solitary benign squamous papilloma of the bronchus. Report of two cases. Am Rev Respir Dis 1968; 97:699-705. 10. Laubscher FA. Solitary squamous cell papilloma of bronchial origin. Am J Clin Pathol 1969;
References I. Spencer H, Dail DH, Arneaud J. Non-invasive bronchial epithelial papillary tumors. Cancer 1980; 45:1486-97.
2. Assor D. A papillary transitional cell tumor of the bronchus. Am J Clin Pathol 1971; 55:761-4. 3. Roviaro GC, VarioliF, Pagnini CA. Is the solitary papilloma of the bronchus always a benign tumor? ORL J Otorhinolaryngol Relat Spec 1981; 43:301-8.
4. Drennan JM, Douglas AC. Solitary papilloma of a bronchus. J Clin Pathol 1965; 18:401-2. 5. Pollak BS,GnassiAM. Inflammatory bronchial tumors: report of a case and review of the literature. Arch Otolaryngol (Chicago) 1938; 27:426. 6. Ashmore PG. Papilloma of the bronchus. J Thorac Surg 1954; 27:293-4.
18:340-9.
52:599-603. 11. Freant LJ, Sawyers JL. Benign bronchial polyps and papillomas. Ann Thorac Surg 1971; 11:460-7. 12. Hurt R. Benign tumors of the bronchus and trachea, 1951-1981. Ann R Coli Surg Engl 1984; 66:22-6.
13. Maxwell RJ, Gibbons JR, O'Hara MD. Solitary squamous papilloma of the bronchus. Thorax 1985; 40:68-71. 14. Barzo P, Molnar L, Minik K. Bronchial papillomas of various origins. Chest 1987; 92:132-6. 15. Trillo A, Guha A. Solitary condylomatous papilloma of the bronchus. Arch Pathol Lab Med 1988; 112:731-3.
Introduction Endobronchial papillomas are rare (1-3),the commonest of which are solitary squamous papillomas (1).This report presents a patient with a papillary columnar adenoma, the eighth such case to our knowledge.
Case Report A 74-yr-oldwhite man presented with recurrent hemoptysis. For severalyears he had produced chronic sputum consistent with chronic bronchitis. During the previous 2 yr, he had experienced several episodes of blood-streaked sputum. No other bleeding diathesis was evident. He denied any history of tuberculosis, thromboembolic disease, or pneumonia, but he continued to smoke one pack of cigarettes daily, as he had for 60 years. No occupational exposures were elicited. Theophylline and a steroid metered-dose inhaler controlled his symptoms of chronic obstructive pulmonary disease. His physical examination was normal. Laboratory data demonstrated a leukocytosis (20,800 WBC with a normal differential count). Arterial blood gases, prothrombin time, partial thromboplastin time, and platelet count were normal. Pulmonary function tests revealed a FEV 1 of 1.18 L (39070 predicted), a FVC of 3.38 L (85% predicted), and a FEV,/FVC demonstrating severe obstruction (ratio, 0.35). A chest radiograph demonstrated an abnormal density adjacent to the right heart border, better shown to be a right lower lobe collapse on chest CT (figure 1). Fiberoptic bronchoscopy demonstrated a white, glistening, friable mass arising from the superior segment of the right lower lobe (figure 2 ), which obstructed the bronchus to the basilar segments. As shown in figure 3, microscopic examination of biopsies fixed in Hollande's solution demonstrated multiple segments of a papilloma exhibiting a single layer of columnar cells without cilia. Most cells exhibited basal nuclei and clear cytoplasm, but neither mitotic figures nor mucicarmine-positive material were found. There was no evidence of atypia or invasion of the stroma by the neoplasm. Lymphocytesand plasma cellswere prominent with-
(Received in original form April 10, 1990 and in revised form June 10, 1991) , From the Departments of Pulmonary Disease and Pathology, The Cleveland Clinic Foundation, Cleveland, Ohio. 2 Correspondence and requests for reprints should be addressed to James K. Stoller, M.D., Department of Pulmonary Disease,A90, The Cleveland Clinic Foundation, One Clinic Center, Cleveland, OH 44195.
1400
SUMMARY Solitary papillomas are rare endobronchial neoplasms that are most commonly lined by a squamous epithelium (I.e., squamous papillomas). Papillomas exhibiting nonsquamous epithelium are even more unusual; only seven solitary papillomas lined by columnar epithelium (I.e., columnar papillomas) have been previously described. The current report presents the eighth patient with a columnar papilloma and demonstrates some features common to solitary papillomas (I.e., central location In the airways, associated atelectasis), but distinctive from squamous papilloAM REV RESPIR DIS 1991; 144:1400-1402 mas (I.e., absence of malignant potential).
Fig. 1. Chest CT scan demonstrating partial collapse (arrow) of right lower lobe by columnar papilloma.
Fig. 2. Bronchoscopicviewofcolumnar papilloma partially occluding basilar segments of right lower lobe.
1401
CASE REPORT
in the connective tissue stroma of the papill ary stalks. The undigested and digested periodic acidSchiff stains demons trat ed no evidence of neutral mucins. Imm unoperoxidase sta ining performed on sections from the paraffin-embedded material demonstrated no evidence of enolase, chromogranin or serotonin.
Fig . 3. Papillary columnar adenoma of the bronchus. At th is magn ificat ion , the column ar cells are well defined , as is the fib rovasc ular co re. Hema toxy lin-eosin stain ; magn ification: 260.
TABLE 1 SUMMARY OF AVAILABLE REPORTS OF ENDOBRONCHIAL PAPILLOMAS (n = 59) Type of Epithe lial Cell Lini ng Papilloma
Au thor (Date) Pollack (1938) Ashmore (1953 ) Ashley (196 3) Sm ith (1963) Dren nan (1965) Jac kson (1968) Laubsc her (1969) Assor (1971) Frea nt (1971) Spencer (1980) Rov iaro (1981) Hurt (1984) Maxwell (1985) Barz6 (1987) Trillo (1988)
Reference No. 5 6 7 8 4 9 10 2 11 1 3 12 13 14 15
Tota l
Pat ien ts
Total Squamous '
Total Cubo idal or Col umnar '
(n)
(n with dysplasia )
(n with dysplasia)
1 1 2
(O) (O) 8 1 2 1
1
2 1 1 5 21 4 1 1
5 7 3 1 1
1 (O) (O) (O) (O)
(1) (0)
(O) (O)
(0)
(0)
(O)
(O)
(0) 4 (0) 1 (0) (0) (0)
8 2
(4) (3) (0) (1) 2 (1) 2 (0)
59
33 (10)
1 0 1 0 0 0 0 1 0 10 0 0 0
(0) 1 (0)
(0)
8
Other/ Not Spec ifiedt
(O) (0)
6
7 (0)
19
0
• Number with dysplasia shown in parentheses. t Includes inflammatory polyps. squamous cell carci noma, cystadenomas, transitional cell polyps.
Discussion Papillomatous lesions of the endobronchial tree have been classified into thr ee groups (1-3): (1) multiple squamous papillomas, as seenin the childhood diseaseof juvenilelaryngotra cheobronchial papillomatosis; (2) inflammatory pol yps associa ted with an endobron chial foreign bod y, chronic bron chitis, or broncholithiasis; (3) solitary papillomas (4). Solitary papillomas of the endobronchial tree are rare neoplasms. As reviewed in table I, 59 well-documented cases (present case excluded) emerged from our literature search. Presen ting symptoms of all solitary papillomas reflect chronic airway obstruction and include produ ctive cough, hemopt ysis, wheezing, and dyspnea, often leading to misdiagnosis as asthma or chronic bronchitis. Patients may carry this diagnosis for mon ths to years before the diagnosis of endobronchial pap illoma is discovered. Commonly, radiogra phic findings of obstructive pneumonitis, abscess, atelectasis, or bronchiectasis are evident. Pathologically, these solitary pap illomas usually consist of squamous epithelium (squamou s papilloma), with approximatel y onethird demonstrating squamous dysplasia, carcinoma in situ, or invasive carcinoma (table 1). When feasible, conventiona l wisdom suggests that squamous papillomas should be surgically removed becau se of this malignant predispo sition. In contrast to squamous papillomas, columnar (or cuboidal) papillomas are much rarer, with seven cases previously reported to our knowledge (ta ble 2). As summarized in table 2, the eight available reports (current case included) suggest that columnar papillomas occur most commonly in middle age (mean age, 55 years; range, 26 to 74 years), show a male predom inance (5:2 in described cases) and show a right-sided predom inance (also 5:2), especially for the right lower lobe. The pa-
TAB LE 2 SUMMARY OF FEATURES OF AVAILABLE BRONCHIAL COLUMNAR PAPILLOMAS (n = 8)
Au thor (Date) Ashmore (1953) Ashle y (1963)
Refere nce No.
Roviaro (1981)
(n)
Sex
Location
51 62
F M
RLL RLL
Ciliated columnar Ciliated columnar
4 (1)
60
M
LLL
(2)
59
M
RLL
Cubo idal + mult ilayered nonsquamous Columnar + multil ayered nonsquamous
(3) (4) 1
26 57
6
7
Spen cer (196 0)
3
Cu rrent Case Definirion of abbr eviati ons : RLL
Ep ithelial Cell Typ e
Age n
74
No details ava ilable F LUL M RLL M
= right lower lobe; RML = right middle
RLL
Co lumnar + cuboidal Ciliated columnar/cuboidal
Treatm ent
Foll ow-up
Bronchoscopic rem oval
No recurrence afte r 18 month s
LLL lobectom y Bronchos copic re mo val
Lobec tomy Bilobectomy (RLU RML)
Simple columnar
lobe: LUL - left upper lobe; LLL
No recurrence after 2.5 y
= left lower lobe; numbe rs in parentheses reler
Lost to follow-up to case number in the Spencer series.
1402
tient presented in the current report is the oldest patient with a columnar papilloma described to date. Although follow-up information is limited, none of the available cases has demonstrated malignant transformation, again in contrast to the more common squamous papilloma. In the absence of demonstrable malignant potential, conservative management of columnar papillomas (i.e., with laser ablation or bronchoscopic debridement) seems justifiable, in contrast to surgical resection that is favored for squamous papillomas. In summary, the current report presents the eighth available patient with a solitary bronchial papilloma lined with columnar epithelium. In the small number of available cases, common clinical features include presentation in middle-aged men with endobronchial masses obstructing the right lower lobe. When possible, conservative management (e.g., endobronchial removal) seems justified by the absence of observed malignant potential in columnar bronchial papillomas. However, the
CASE REPORT
lack of long-term follow-up experience may be responsible for this failure to observe malignancy.
7. Ashley DJB, Banino EA, Davies HD. Bronchial polyps. Thorax 1963; 18:45-9. 8. Smith JF, Dexter D. Papillary neoplasms of the bronchus of low grade malignancy. Thorax 1963;
Acknowledgment The writers thank J. Schaaf, M.D., for referring the patient.
9. Jackson DA, Hatch HB. Solitary benign squamous papilloma of the bronchus. Report of two cases. Am Rev Respir Dis 1968; 97:699-705. 10. Laubscher FA. Solitary squamous cell papilloma of bronchial origin. Am J Clin Pathol 1969;
References I. Spencer H, Dail DH, Arneaud J. Non-invasive bronchial epithelial papillary tumors. Cancer 1980; 45:1486-97.
2. Assor D. A papillary transitional cell tumor of the bronchus. Am J Clin Pathol 1971; 55:761-4. 3. Roviaro GC, VarioliF, Pagnini CA. Is the solitary papilloma of the bronchus always a benign tumor? ORL J Otorhinolaryngol Relat Spec 1981; 43:301-8.
4. Drennan JM, Douglas AC. Solitary papilloma of a bronchus. J Clin Pathol 1965; 18:401-2. 5. Pollak BS,GnassiAM. Inflammatory bronchial tumors: report of a case and review of the literature. Arch Otolaryngol (Chicago) 1938; 27:426. 6. Ashmore PG. Papilloma of the bronchus. J Thorac Surg 1954; 27:293-4.
18:340-9.
52:599-603. 11. Freant LJ, Sawyers JL. Benign bronchial polyps and papillomas. Ann Thorac Surg 1971; 11:460-7. 12. Hurt R. Benign tumors of the bronchus and trachea, 1951-1981. Ann R Coli Surg Engl 1984; 66:22-6.
13. Maxwell RJ, Gibbons JR, O'Hara MD. Solitary squamous papilloma of the bronchus. Thorax 1985; 40:68-71. 14. Barzo P, Molnar L, Minik K. Bronchial papillomas of various origins. Chest 1987; 92:132-6. 15. Trillo A, Guha A. Solitary condylomatous papilloma of the bronchus. Arch Pathol Lab Med 1988; 112:731-3.
