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Basaloid carcinoma, a rare primary lung neoplasm: Report of a case and review of the literature
Lung Cancer 35 (2002) 335– 338
www.elsevier.com/locate/lungcan
Case report
Basaloid carcinoma, a rare primary lung neoplasm: Report of a case and review of the literature Christopher N. Foroulis a,*, Kosmas H. Iliadis a, Panayiotis M. Mauroudis b, Paris A. Kosmidis c a
Department of Thoracic Surgery, Diagnostic and Therapeutic Center of Athens ‘Hygeia’, Kifisias A6enue and 4, Er. Sta6rou Street, 15123 Maroussi, Athens, Greece b Department of Chest Medicine, Diagnostic and Therapeutic Center of Athens ‘Hygeia’, Kifisias A6enue and 4, Er. Sta6rou Street, 15123 Maroussi, Athens, Greece c 2nd Department of Clinical Oncology, Diagnostic and Therapeutic Center of Athens ‘Hygeia’, Kifisias A6enue and 4, Er. Sta6rou Street, 15123 Maroussi, Athens, Greece Received 11 June 2001; received in revised form 1 October 2001; accepted 2 October 2001
Abstract Basaloid carcinoma of the lung is a rare primary neoplasm, first described in 1992. Basaloid carcinoma is an aggressive subtype of Non small cell lung cancer, with poor 5-year survival, even in stage I and II resected tumors. Differential diagnosis from small cell, Neuroendocrine large cell and poorly differentiated squamous cell carcinoma is difficult to be made. We report a patient with lung basaloid carcinoma, initially diagnosed and treated as small cell carcinoma. Thoracotomy and resection of the tumor following chemotherapy, established the correct diagnosis. © 2002 Elsevier Science Ireland Ltd. All rights reserved. Keywords: Basaloid carcinoma of the lung; Uncommon malignant primary lung neoplasms; Rare lung neoplasms
1. Introduction Basaloid carcinomas (BC) have been first described in carcinomas of the tongue, hypopharynx, larynx and upper aerodigestive tract in 1986 [1]. Basaloid carcinomas of the lung, as a distinct entity, have been described by Brambilla et al. in 1992 [1 – 4]. BC may be considered as a variety of non small cell, non-endocrine lung carcinomas and may exist in pure form or mixed with other non small cell lung carcinoma patterns in 50% of cases [1,5]. These rare neoplasms, in their pure form, are histologically similar to the skin basal cell carcinoma and are distinct from any of the four types of lung carcinoma, described in the previous WHO classification (1981) [6]. This specific entity is now
* Corresponding author. Tel.: +30-1-6779693; mobile: +30-944910343; fax: + 30-1-6845089. E-mail address: [email protected] (C.N. Foroulis).
quoted in the actual WHO classification of lung tumors (1999) [7]. Misdiagnosis of the lung BC as small cell carcinoma (SCLC) may occur, especially when fine needle aspiration (FNA) and small bronchial biopsies are the way to establish cell type diagnosis of the tumor [5,8].
2. Case report A 58-year old, Caucasian male, with history of chronic obstructive pulmonary disease (COPD) and arterial hypertension, heavy smoker and cessation of smoking 7 years ago, was admitted to the hospital with pulmonary infection and exacerbation of COPD symptoms 1 year ago. Chest X-ray and computed tomography scan at that time identified a coin lesion, located at the periphery of the superior segment of the right lower lobe, 2× 3 cm in size, in close relation to the parietal pleura. Fiberoptic bronchoscopy was negative for intra-
0169-5002/02/$ - see front matter © 2002 Elsevier Science Ireland Ltd. All rights reserved. PII: S 0 1 6 9 - 5 0 0 2 ( 0 1 ) 0 0 4 2 7 - 5
336
C.N. Foroulis et al. / Lung Cancer 35 (2002) 335–338
luminal disease, as well as cytologic bronchial brushing and washing specimens. Computed tomography guided FNA of the lesion followed and the cytologic diagnosis was SCLC of the lung. Clinical staging of the neoplasm was T1N0M0/Stage I A. Surgical therapy was not suggested to the patient at that time, because of poor respiratory function associated with COPD [FVC: 2340 ml (58%), FEV1: 994 ml (31%), Tiffenau: 42.5%, mild response to bronchodilation]. The patient received 6 cycles of chemotherapy with carboplatin and etoposide every 3 weeks. New staging of the tumor after 3 chemotherapy cycles demonstrated partial response to chemotherapy. The lesion diminished in size (50%) and was separated from the underlying pleura (Fig. 1a). After the completion of chemotherapy no further response of the tumor and no evidence of metastasis elsewhere were observed. Because of poor respiratory function, surgical therapy or radiotherapy was not suggested to the patient and he remained under close observation. Three months after chemotherapy cessation, the tumor size measured the pre-chemotherapy dimensions, with no evidence of metastasis. Further increase in tumor dimensions (2.5× 4.5 cm) was observed in chest CT 3 months later (Fig. 1b). The
unusual clinical course of the patient in contrast to the moderate response to chemotherapy, which was not in accordance to the diagnosis of SCLC, suggested a possible different diagnosis. Intensive therapy with bronchodilators in addition to high dose of corticosteroids and intensive chest physiotherapy followed, aiming for the improvement of the patient’s respiratory condition and to further surgical treatment. Two weeks after the induction of intensive treatment, spirometry results were compatible with a limited pulmonary resection. [FEV1: 1290 ml (40.5%), FVC: 3150 ml (78%), Tiffenau: 41%]. Arterial blood oxygen saturation varied from 94 to 96% at rest, with no alternation in mild exercise. The patient underwent thoracotomy and resection of the superior segment of the right lower lobe. He had a 6 days, uncomplicated postoperative course. Histopathologic examination of the specimen established the diagnosis of BC of the lung (Fig. 2). The post-thoracotomy staging was T2N0M0/Stage I B. Despite the early stage of the tumor, liver metastases were observed 7 months after the operation and the patient died 3 months later, not responding to the chemotherapy administration.
Fig. 1. CT of the chest: (a) the tumor after three cycles of chemotherapy with Carboplatin and Etoposide treatment; (b) the tumor 3 months after chemotherapy cessation.
C.N. Foroulis et al. / Lung Cancer 35 (2002) 335–338
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Fig. 2. H&E stain × 400: solid and microtubular nests showing peripheral palisading of small tumor cells.
3. Discussion Basaloid carcinoma is mainly located in the major and moderate bronchi (85% of referred cases) [1]. Diagnosis of lung BC is difficult to establish preoperatively, particularly when a small number of cells are available for microscopic examination, as in material obtained from FNAs, bronchial brushings and washings and small bronchial biopsies also. Differential diagnosis of the pure form of BC from ‘intermediate type’ of SCLC and large cell neuroendocrine carcinoma is difficult to be made [1,6]. Immunohistochemical detection of neuroendocrine markers and/or electron microscopy can be used to detect or not the expression of neurosecretory granules [2,5]. Immunohistochemical positivity in one or less often two neuroendocrine markers is observed in BC, but BC never expresses three neuroendocrine markers, which is the acceptable definition for neuroendocrine differentiation in lung cancer [2]. Misdiagnosis as SCLC in FNA material is frequent, as reported by Vesoulis (1998), in lung and cervical lymph node metastasis from primary laryngeal BC [8]. Cytologic differentiation between BC and poorly differentiated squamous cell carcinoma is not always possible, with the 14 cytomorphologic parameters introduced by Dugam and biopsy confirmation may be a necessary diagnostic step [5]. Basaloid carcinoma has characteristic histopatholologic features, which are the lobular growth pattern of small tumor cells, with peripheral palisading, scant but visible cytoplasm, moderately hyperchromatic nuclei
without prominent nucleoli and frequent mitoses [1,2]. The characteristic pattern is evident in surgical samples [1,2,5]. Basaloid carcinoma is a rare subtype of non-small cell lung carcinoma (NSCLC) group, with an aggressive behavior. In a recent publication, Mai et al. (1999) describe a questionable case of lung BC with bronchioalveolar cell differentiation, features of a low grade tumor, with low mitotic rate and a favorable clinical course [9]. However, the features of a low-grade tumor, with low mitotic rate described in the previous mentioned case, are not in agreement with the consistent feature of BC with the high mitotic index; adenoid-cystic carcinoma with partial solid pattern could be included in the differential diagnosis [1,2]. In the published study by Moro et al. in 1994, in 37 cases of primary BC of the lung, the 5-year survival for stage I and II resected tumors was 15%, compared to 47% 5-year survival, for stage I and II resected poorly differentiated squamous cell carcinomas [2]. The difference in mean survival in stage III and IV tumors was not statistically significant in the same study [2]. Surgical therapy is not sufficient for BC, according to the previously mentioned study results [2]. Chemotherapy alone was also not effective treatment for our patient with BC. A combination of surgical and adjuvant therapy may constitute the optimal treatment, but randomized trials comparing surgery and combined treatment are needed to prove it. The small number of reported BC of the lung, make difficult the establishment of such studies.
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C.N. Foroulis et al. / Lung Cancer 35 (2002) 335–338
References [1] Brambilla E, Moro D, Veale D, Brichon PY, Paramelle B, Brambilla C. Basal cell (Basaloid) carcinoma of the lung: a new morphologic and phenotypic entity with separate prognostic significance. Hum Pathol 1992;23(9):993 –1003. [2] Moro D, Brichon P, Brambilla E, Veale D, Labat F, Brambilla C. Basaloid bronchial carcinoma. A histologic group with a poor prognosis. Cancer 1994;73:2734 –9. [3] Shields TW. Pathology of carcinoma of the lung. In: Shields TW, LoCicero J, Ponn RB, editors. General Thoracic Surgery, 5th ed. Philadelphia: Lippincot Williams and Wilkins, 1999:1249 –62. [4] Robinson PG, Shields TW. Uncommon primary malignant tumors of lung. In: Shields TW, LoCicero J, Ponn RB, editors. General Thoracic Surgery, 5th ed. Philadelphia: Lippincot Williams and Wilkins, 1999:1533 –53.
[5] Dugam JM. Cytologic diagnosis of basal cell (basaloid) carcinoma of the lung. A report of two cases. Acta Cytol 1995;39(3):539 – 42. [6] World Health Organization. Histological typing of lung tumors. 2nd ed. Am J Clin Pathol 1982;77(2):123 – 136. [7] Travis WD, Colby TV, Corrin B, Shimosato Y, Brambilla E. In Collaboration with Sobin LH and Pathologists from 14 Countries. Histological Typing of Lung and Pleural tumors. World Health Organization. International Histological Classification of tumors. 3rd ed. Berlin: Springer, 1999. [8] Vesoulis Z. Metastatic laryngeal basaloid squamous cell carcinoma simulating primary small cell carcinoma of the lung on fine needle aspiration lung biopsy. A case report. Acta Cytol 1998;42(3):783 – 7. [9] Mai KT, Perkins DG, Veinot JP, Shamji FM. Basaloid carcinoma of the lung with bronchiolo-alveolar cell differentiation. A case report. Tumori 1999;85(3):207 – 10.
www.elsevier.com/locate/lungcan
Case report
Basaloid carcinoma, a rare primary lung neoplasm: Report of a case and review of the literature Christopher N. Foroulis a,*, Kosmas H. Iliadis a, Panayiotis M. Mauroudis b, Paris A. Kosmidis c a
Department of Thoracic Surgery, Diagnostic and Therapeutic Center of Athens ‘Hygeia’, Kifisias A6enue and 4, Er. Sta6rou Street, 15123 Maroussi, Athens, Greece b Department of Chest Medicine, Diagnostic and Therapeutic Center of Athens ‘Hygeia’, Kifisias A6enue and 4, Er. Sta6rou Street, 15123 Maroussi, Athens, Greece c 2nd Department of Clinical Oncology, Diagnostic and Therapeutic Center of Athens ‘Hygeia’, Kifisias A6enue and 4, Er. Sta6rou Street, 15123 Maroussi, Athens, Greece Received 11 June 2001; received in revised form 1 October 2001; accepted 2 October 2001
Abstract Basaloid carcinoma of the lung is a rare primary neoplasm, first described in 1992. Basaloid carcinoma is an aggressive subtype of Non small cell lung cancer, with poor 5-year survival, even in stage I and II resected tumors. Differential diagnosis from small cell, Neuroendocrine large cell and poorly differentiated squamous cell carcinoma is difficult to be made. We report a patient with lung basaloid carcinoma, initially diagnosed and treated as small cell carcinoma. Thoracotomy and resection of the tumor following chemotherapy, established the correct diagnosis. © 2002 Elsevier Science Ireland Ltd. All rights reserved. Keywords: Basaloid carcinoma of the lung; Uncommon malignant primary lung neoplasms; Rare lung neoplasms
1. Introduction Basaloid carcinomas (BC) have been first described in carcinomas of the tongue, hypopharynx, larynx and upper aerodigestive tract in 1986 [1]. Basaloid carcinomas of the lung, as a distinct entity, have been described by Brambilla et al. in 1992 [1 – 4]. BC may be considered as a variety of non small cell, non-endocrine lung carcinomas and may exist in pure form or mixed with other non small cell lung carcinoma patterns in 50% of cases [1,5]. These rare neoplasms, in their pure form, are histologically similar to the skin basal cell carcinoma and are distinct from any of the four types of lung carcinoma, described in the previous WHO classification (1981) [6]. This specific entity is now
* Corresponding author. Tel.: +30-1-6779693; mobile: +30-944910343; fax: + 30-1-6845089. E-mail address: [email protected] (C.N. Foroulis).
quoted in the actual WHO classification of lung tumors (1999) [7]. Misdiagnosis of the lung BC as small cell carcinoma (SCLC) may occur, especially when fine needle aspiration (FNA) and small bronchial biopsies are the way to establish cell type diagnosis of the tumor [5,8].
2. Case report A 58-year old, Caucasian male, with history of chronic obstructive pulmonary disease (COPD) and arterial hypertension, heavy smoker and cessation of smoking 7 years ago, was admitted to the hospital with pulmonary infection and exacerbation of COPD symptoms 1 year ago. Chest X-ray and computed tomography scan at that time identified a coin lesion, located at the periphery of the superior segment of the right lower lobe, 2× 3 cm in size, in close relation to the parietal pleura. Fiberoptic bronchoscopy was negative for intra-
0169-5002/02/$ - see front matter © 2002 Elsevier Science Ireland Ltd. All rights reserved. PII: S 0 1 6 9 - 5 0 0 2 ( 0 1 ) 0 0 4 2 7 - 5
336
C.N. Foroulis et al. / Lung Cancer 35 (2002) 335–338
luminal disease, as well as cytologic bronchial brushing and washing specimens. Computed tomography guided FNA of the lesion followed and the cytologic diagnosis was SCLC of the lung. Clinical staging of the neoplasm was T1N0M0/Stage I A. Surgical therapy was not suggested to the patient at that time, because of poor respiratory function associated with COPD [FVC: 2340 ml (58%), FEV1: 994 ml (31%), Tiffenau: 42.5%, mild response to bronchodilation]. The patient received 6 cycles of chemotherapy with carboplatin and etoposide every 3 weeks. New staging of the tumor after 3 chemotherapy cycles demonstrated partial response to chemotherapy. The lesion diminished in size (50%) and was separated from the underlying pleura (Fig. 1a). After the completion of chemotherapy no further response of the tumor and no evidence of metastasis elsewhere were observed. Because of poor respiratory function, surgical therapy or radiotherapy was not suggested to the patient and he remained under close observation. Three months after chemotherapy cessation, the tumor size measured the pre-chemotherapy dimensions, with no evidence of metastasis. Further increase in tumor dimensions (2.5× 4.5 cm) was observed in chest CT 3 months later (Fig. 1b). The
unusual clinical course of the patient in contrast to the moderate response to chemotherapy, which was not in accordance to the diagnosis of SCLC, suggested a possible different diagnosis. Intensive therapy with bronchodilators in addition to high dose of corticosteroids and intensive chest physiotherapy followed, aiming for the improvement of the patient’s respiratory condition and to further surgical treatment. Two weeks after the induction of intensive treatment, spirometry results were compatible with a limited pulmonary resection. [FEV1: 1290 ml (40.5%), FVC: 3150 ml (78%), Tiffenau: 41%]. Arterial blood oxygen saturation varied from 94 to 96% at rest, with no alternation in mild exercise. The patient underwent thoracotomy and resection of the superior segment of the right lower lobe. He had a 6 days, uncomplicated postoperative course. Histopathologic examination of the specimen established the diagnosis of BC of the lung (Fig. 2). The post-thoracotomy staging was T2N0M0/Stage I B. Despite the early stage of the tumor, liver metastases were observed 7 months after the operation and the patient died 3 months later, not responding to the chemotherapy administration.
Fig. 1. CT of the chest: (a) the tumor after three cycles of chemotherapy with Carboplatin and Etoposide treatment; (b) the tumor 3 months after chemotherapy cessation.
C.N. Foroulis et al. / Lung Cancer 35 (2002) 335–338
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Fig. 2. H&E stain × 400: solid and microtubular nests showing peripheral palisading of small tumor cells.
3. Discussion Basaloid carcinoma is mainly located in the major and moderate bronchi (85% of referred cases) [1]. Diagnosis of lung BC is difficult to establish preoperatively, particularly when a small number of cells are available for microscopic examination, as in material obtained from FNAs, bronchial brushings and washings and small bronchial biopsies also. Differential diagnosis of the pure form of BC from ‘intermediate type’ of SCLC and large cell neuroendocrine carcinoma is difficult to be made [1,6]. Immunohistochemical detection of neuroendocrine markers and/or electron microscopy can be used to detect or not the expression of neurosecretory granules [2,5]. Immunohistochemical positivity in one or less often two neuroendocrine markers is observed in BC, but BC never expresses three neuroendocrine markers, which is the acceptable definition for neuroendocrine differentiation in lung cancer [2]. Misdiagnosis as SCLC in FNA material is frequent, as reported by Vesoulis (1998), in lung and cervical lymph node metastasis from primary laryngeal BC [8]. Cytologic differentiation between BC and poorly differentiated squamous cell carcinoma is not always possible, with the 14 cytomorphologic parameters introduced by Dugam and biopsy confirmation may be a necessary diagnostic step [5]. Basaloid carcinoma has characteristic histopatholologic features, which are the lobular growth pattern of small tumor cells, with peripheral palisading, scant but visible cytoplasm, moderately hyperchromatic nuclei
without prominent nucleoli and frequent mitoses [1,2]. The characteristic pattern is evident in surgical samples [1,2,5]. Basaloid carcinoma is a rare subtype of non-small cell lung carcinoma (NSCLC) group, with an aggressive behavior. In a recent publication, Mai et al. (1999) describe a questionable case of lung BC with bronchioalveolar cell differentiation, features of a low grade tumor, with low mitotic rate and a favorable clinical course [9]. However, the features of a low-grade tumor, with low mitotic rate described in the previous mentioned case, are not in agreement with the consistent feature of BC with the high mitotic index; adenoid-cystic carcinoma with partial solid pattern could be included in the differential diagnosis [1,2]. In the published study by Moro et al. in 1994, in 37 cases of primary BC of the lung, the 5-year survival for stage I and II resected tumors was 15%, compared to 47% 5-year survival, for stage I and II resected poorly differentiated squamous cell carcinomas [2]. The difference in mean survival in stage III and IV tumors was not statistically significant in the same study [2]. Surgical therapy is not sufficient for BC, according to the previously mentioned study results [2]. Chemotherapy alone was also not effective treatment for our patient with BC. A combination of surgical and adjuvant therapy may constitute the optimal treatment, but randomized trials comparing surgery and combined treatment are needed to prove it. The small number of reported BC of the lung, make difficult the establishment of such studies.
338
C.N. Foroulis et al. / Lung Cancer 35 (2002) 335–338
References [1] Brambilla E, Moro D, Veale D, Brichon PY, Paramelle B, Brambilla C. Basal cell (Basaloid) carcinoma of the lung: a new morphologic and phenotypic entity with separate prognostic significance. Hum Pathol 1992;23(9):993 –1003. [2] Moro D, Brichon P, Brambilla E, Veale D, Labat F, Brambilla C. Basaloid bronchial carcinoma. A histologic group with a poor prognosis. Cancer 1994;73:2734 –9. [3] Shields TW. Pathology of carcinoma of the lung. In: Shields TW, LoCicero J, Ponn RB, editors. General Thoracic Surgery, 5th ed. Philadelphia: Lippincot Williams and Wilkins, 1999:1249 –62. [4] Robinson PG, Shields TW. Uncommon primary malignant tumors of lung. In: Shields TW, LoCicero J, Ponn RB, editors. General Thoracic Surgery, 5th ed. Philadelphia: Lippincot Williams and Wilkins, 1999:1533 –53.
[5] Dugam JM. Cytologic diagnosis of basal cell (basaloid) carcinoma of the lung. A report of two cases. Acta Cytol 1995;39(3):539 – 42. [6] World Health Organization. Histological typing of lung tumors. 2nd ed. Am J Clin Pathol 1982;77(2):123 – 136. [7] Travis WD, Colby TV, Corrin B, Shimosato Y, Brambilla E. In Collaboration with Sobin LH and Pathologists from 14 Countries. Histological Typing of Lung and Pleural tumors. World Health Organization. International Histological Classification of tumors. 3rd ed. Berlin: Springer, 1999. [8] Vesoulis Z. Metastatic laryngeal basaloid squamous cell carcinoma simulating primary small cell carcinoma of the lung on fine needle aspiration lung biopsy. A case report. Acta Cytol 1998;42(3):783 – 7. [9] Mai KT, Perkins DG, Veinot JP, Shamji FM. Basaloid carcinoma of the lung with bronchiolo-alveolar cell differentiation. A case report. Tumori 1999;85(3):207 – 10.