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Pulmonary carcinosarcoma: A case study and review of the literature
Br.J.
Dis.
Chest
(1985)
79, 83
PULMONARY CARCINOSARCOMA: A CASE STUDY AND REVIEW OF THE LITERATURE A. CABARCOS,
M. GOMEZ DORRONSORO J. L. LOB0 BERISTAIN
The General Geriatric Hospital of the Jose Matia Caluo the Basque County, Facul& of Medicine of the University Campus
AND
Foundation, of Bilbao,
Uniuersig of San Sebastian
Summary A case report of pulmonary carcinosarcoma and a review of the literature is presented. This is the only case where diagnosis has been obtained by thin needle percutaneous biopsy. Introduction The pulmonary carcinosarcoma is a rare malignant tumour composed of two components; carcinoma and sarcoma. Its existence has been a matter of controversy for many years (Jenkins 1968; Rodriguez Mendez et al. 1975; Ludwigsen 1977; Edwards et al. 1979) but recently it has been generally accepted as a histopathological and clinical entity even though its histogenesis remains obscure. Compared with all pulmonary neoplasms it occurs with a frequency of 0.3% (Diaconita 1975; Co11 et al. 1981) and in Spain only six cases including this one have been reported. In this paper we present a detailed case report on a patient with pulmonary carcinosarcoma and review the clinical and histological parameters in the literature. Case Report The patient was a 72.year-old man who smoked 15 cigarettes per day until 4 years before he was admitted to hospital complaining of a progressive pain in his right scapular region. The pain had started after a severe trauma to this region 6 weeks before he was admitted. The pain radiated down the thoracic wall to the right hypochondrium. Apart from this, the patient showed a progressive disinterest in his surroundings as well as asthenia, anorexia and a weight loss of 6 kg. Physical examination did not reveal anything definitive except that percussion of the lower third of the right hemithorax was dull and there were inspiratory crepitations with a small increase in the transmission of vocal vibrations in this area. Investigations showed a mild hypoalbuminaemia with a polyclonal hypergammaglobulinaemia. The sedimentation rate was 76 mm/h. The ;\/lantoux (5 U PPD) was 20 mm and, bacteriological testing and sputum culture in Liiwenstein’s medium were both negative. Chest radiographs and tomograms revealed the presence of a mass in the right inferior pulmonary lobe that was 6 cm in diameter, homogeneous and with irregular borders. Obliteration Address Bidebieta-2,
for
correspondence: San Sebastian-9
Antonio (Espana).
Cabarcos
MD,
Plaza
de 10s Cazadores
no 63”
B, Ayete
A. Cabarcos, A4. Gomez Dorronsoro and J. L. Lobo Beristain of the costophrenic angle was observed along with a pleural thickening and a local elevation of the diaphragm. There was no evidence of cavitation nor of calcification or adenopathy of the mediastinal or hilar lymph nodes. Bronchoscopy did not show evidence of an endobronchial growth nor bronchial compression by extrabronchial tumour. Cytological examination of the bronchial washings along with five separate sputum examinations showed a complete absence of malignant cells in the inflammatory exudate. Transbronchial biopsy demonstrated foci of squamous metaplasia together with an intense oedema and chronic inflammation. The thin needle percutaneous, aspiration showed evidence of a haemorrhagic liquid that contained neoplastic cells and anaerobic, aerobic and Lowenstein cultures
Fig. 1. The (H
malignant
epithelial
component
merges
with
the
malignant
mesenchymal
component.
& E x320)
were all negative. Additional studies such as a barium swallow, cerebral scan, bone scan, studies for bone marrow metastasis among others did not reveal significant abnormalities. The diagnosis was reached by the histopathological analysis of the thin needle percutaneous aspiration which clearly contained two distinct malignant cellular components. The first one consisted of a polygonal cell with clear squamous differentiation without glandular formation, The second, intimately combined with the first, was composed of sarcomatous fusiform cells of a fibroblastic type without myoid or chondroid differentiation (Fig. 1). This double cellular pattern was demonstrated using Snook’s staining technique for reticulin (Fig. 2). During the patient’s stay in the hospital the tumour proved to be highly aggressive, growing very rapidly and eventually infiltrating the thoracic wall and destroying a rib. The patient died and a necroscopy was not performed.
Pulmonav
a5
Carcinosarcoma
Discussion The first description of this pulmonary tumour as a bicellular histopathological entity, according to Bergmann et al. (1951), was made by Kika (cited in Herxheimer & Reinke 1912) in 1908. Since then six cases have been published in Spain and a total of 42 in the rest of the world (Table I). Incidence according to sex and age The tumour has a clear prevalence in the male (Stackhouse et al. 1969; Bull & Grimes 1974; Ludwigsen 1977; Edwards et al. 1979; Co11 et al. 1981) as 40 of the
Fig. 2. The carcinomatous
sarcomatous element
element is unstained.
has produced (Snook’s reticulin
abundant X 160)
reticulin
fibres
in
contrast,
the
48 publications deal with male patients. That gives a male to female incidence of 5: 1. As can be seen in Fig. 3, the incidence with respect to age shows three peaks. The most frequent is between the sixth and seventh decade followed by the fifth and sixth and the seventh and eighth respectively. Summing up all the cases presented in the three decades stated, it can be observed that a total of 41 or 85% occurred between the ages of 50 and 80 years. This age range of presentation combined with the fact that the authors are at the only geriatric hospital in the 7*
45 F
M M M M
74 71 59 46
64 M
Moore
70 M
Stackhouse
et al. 1969
81 M 73 F
Jenkins 1968 Stackhouse et al. 1969
1961
R.U.L. R.U.L. R.M.Br. L.M.Br.
M M M M
51 56 69 57
Bergmann et al. 1951 Bergmann et al. 1951 Taylor & Rae 1952 Taylor & Rae 1952 Cavallero 1956 Peabody 1959 Drury & Stirland 1959 Drury & Stirland 1959 Prive et al. 1961
R.L.L. L.M.Br.+ L.u.L.+ L.L.L. R.U.L.
R.L.L.
L.U.L. R.U.L. R.L.L. L.U.L.
L.U.L.
L.U.L.
R.U.L.
R.U.L. R.U.L.
L.L.L.
R.L.L.
55 F
et al. 1949
1939
Weber
Hochberg
61 M
1938
Fischer
77 M
43 M -70 M
Selye 1928 Geschelin 1928 Ogawa 1929
1915
Frank
&
Age, sex Location
35 F
andyear
Kika 1908 Cited in Herxheimcr Reinke 1912) Saltikow 1914
Author
cm
-
7X4X3
5
3
1 10 2X1X1
-
4X1X1 2x1
3X1
6~4x4
11X10X5
10 cm
10 cm
15 cm
10 cm
10X8
14 cm
Size
Surgery
Autopsy surgery
Surgery
Surgery Surgery Surgery Autopsy Autopsy Surgery Autopsy Autopsy Surgery
Surgery
Autopsy
Autopsy
Autopsy Autopsy Autopsy
Autopsy
Autopsy
Diqnostic
Table I. Cases
published
in world
Yes
Not Not
Not
Yes Not Yes Not
Not Not Not Not
Yes
NO1
Not Yes
Not
Yes Not Not Yes
Not Not Not Not
Yes
Yes
Not
Yes
Yes
Not Yes
Yes
Metastasis
Yes
Local invasion
Epidermoid Undifferentiat
Epidermoid
Epidermoid
Epidermoid Epidermoid Epidermoid
Epidermoid Epidermoid Epidermoid Epidermoid -
Epidermoid keratinizing Epidermoid keratinizing, Adenocarcinoma, Undifferentiat Epidermoid
Adenocarcinoma
Epidermoid
Undifferentiat. Epidermoid Adenopapillary Ca.
of comjwnent
Carcinomatous
Nature
literature
Fibrosarcoma+ Chondrosarc
Fibrosarcoma
Fibrosarcoma Fibrosarcoma \Chondrofibrosarcoma Fibrosarcomat osteoid+ rhabdomyobla.
Fibrosarcoma Fibrosarcoma Fibrosarcoma Fibrosarcoma
Fibrosarcoma
Spindle cell+ pleomorphic Small spindle cell Pleomorphic Small spindle cell Large spindle cell + pleomorphic Spindle cell
-
Sarcomatous
Lobectomy+ Radiation
Not
Lobectomy+ radiation
Pneumonec. Pneumonec. Pneumonec. Not Not Lobectomy Not Not Pneumonec.
Lobectomy
Not
Not
Not Not Not
Not
Not
-
Treatment
+3Y
+3 m
+
Alive
2 y
+postoperative Alive 6 y Alive 2 y Alive 3 y + + +3 m +6 m +l m Alive 2 y
+
+
+ + +
+
+
Evolution
<
tz, g, 3
s-
P t?
s
s 2 2
n & Q 2 s
b
co -.
Mendez
Rodriguez 1975
Case
72 M
21 F 52 M
al. 1981 & Deshotels
1982
55 M 62 M 74 M
et al. 1976 Barca et al. 1977 Valerio et al.
report
Anaya Lopez Loscos 1981 Co11 et Sarma
71 M
1977
Ludwigsen
71 M
R.U.L. L.U.L. L.L.L.
66 M 62 M 56 M
Davis et al 1972 Kaik et al. 1973 Bull & Grimes 1974
et al.
L.M.Br
61 F
et al. 1972
Davis
K.L.1,.
L.L.L. L.L.L.
I,.TJ.L. R.L.L. R.U.L.
L.L.L.
R.U.L.
R.U.L.
65 M
et al. 1972
Davis
R.U.L.
L.U.L. R.U.L. R.U.L.
60 F
61 M 67 M 58 M
et al. 1969
Stackhouse
74 M
Razzuk et al. 1971 Weaver et al. 1971 Davis et al. 1972
et al. 1969
Stackhouse
R.L.L. L.U.L. R.U.L.+ M.Br. R.U.L.
L.U.L. L.L.L. L.U.L. L.L.L. L.U.L.
et al. 1969
Stackhouse
70 M 69 M 54 M
M M M M F
et al. 1969
Stackhouse
L.U.L.
68 65 69 72 66
et al. 1969
Stackhouse
52 M
Stackhouse et al. 1969 Chaudhuri 1971 Chaudhuri 197 1 Kakos et al. 1971 Razzuk et al. 1971
et al. 1969
Stackhouse
10
8 3
1x1 8x8 8
2
9~8x6
3x2
2X1X1
3
4 5X2x2
3~2x2
3X2x3 10x 10x6 1x1
6X5X5
6X5X5
12X5X6 5X4X3 --
3X4X3
Biopsy
Surgery Autopsy
Surgery Surgery
surgery
Autopsy
Surgery Surgery Surgery
Surgery
Autopsy
Surgery Surgery Surgery
Surgery Surgery Surgery Surgery Surgery
Surgery
Surgery
Surgery Surgery Surgery
Surgery
~
Yes
YCS -
Plot Yes
Yes
Not
-
-
~~ ~ ~-
~ ~ ~-
-
-
~-
Not
-~
xot
Yes
Not
Not No1 Yes
Yes
X0t
Yes Yes Yes
ITot
Not
Yes
YtX xot
Not Yes Not Yes YCS
YCS
Adenocarcinoma Epidermoid+ adenoid q. Epidermoid
Epidermoid Epidermojd Epidermoid
Epidermoid
Epidermoid
Adenocarcinorna+ Undiffercntiat. Epidermoid Undilrerentiat. Epidermoid+ tubular Epidermoid+ cubic Epidermoid+ Adenocarcinoma Epidermoid Epidermoid Epidermoid
Adenocarcinoma+ Undiffcrcntiat. Epidermoid Epidermoid Epidermoid
Epidermoid
Adenocarcinoma Adcnocarcinoma Epidermoid
Yes Yes YCS YCS
Epidermoid
Yes
cell
SpindIp
cell
Spindle cell Osteosarcoma
Fibrosarcorna+ osteoid Osteosarcorna+ rabdomyosar. fibrosarcoma Spindle cell Fibrosarcoma Spindle cell
Spindle cell Leiomyosarc. Spindle cell+ blastoma Spindle cell+ blastoma Spindle cell+ blastoma Spindle cell Spindle cell Spindle cell
Spindle
Fibrosarcoma Pibrosarcoma Spindle cell
Fibrosarcoma+ ostroid Fibrosarcoma
Fibrosarcoma +osteoid Fibrosarcoma Fibrosarcoma Fibrosarcoma
+
+
m
m
Not
+I5 +19
Pneurnonec. Pneumonec.
+2 *n
d d
+
2 y
3 m
+ postop. +5 m
Alive
4
+9 m +2 m
+9
Alive
+ postop.
Alive15m 1-18 m
+21
+6 d +3 m +2 d Alive 6 m +10 m
m
4 y
Pncumoncc. Pneumonec. Not
Not
Radiation
PIEUIIlOIlCC.
Pneurnoncc. Pneumonec.
Pneumoncc.
Pneumonec.
Lobectomy Lobectomy
PIl~U~OIlCC.
Pneumonec. Pneumoncc. Lobectomy Pncumonec. Lobectomy
+11
+6 m
Pneumonec. Lobectomy
+2 m +3 m
-
Alive
Lobectomy Lobectomy Radiation
Lobectomy
2
2
iz v Q a ? 8’ 8 e 2
88
A. Cabarcos, M. Gomez Dorronsoro and J. L. Lobo Beristain
country have been the deciding observe one of these rare cases.
factors
that
have given
us the opportunity
to
Clinical presentation The semiological data are presented in Fig. 4. From’ this it can be observed that the most frequent symptoms are a cough, thoracic chest pain and dyspnoea. Thus, it is quite clear that the disease has non-specific signs and symptoms and to establish a definitive diagnosis, additional analytical techniques must be employed.
N=
CASES
25 N=19
r
20
15 N=12 10
N=lO
5 0 N=41 I
I
I
0
10
20
,
30
1
40
-TV-,
-7
50
60
AGE
(85%) ---r~--I
70
I
80
90
100
Fig. 3. Age of presentation
Location and type of growth The majority of the-cases (~~32, 69%) show the tumour affecting the superior lobe and/or the principal bronchi, while it is infrequent in the lower lobes. The patient presented in this paper was affected in the most unusual location: the right lower lobe (Fig. 5). With respect to the type of growth, Moore, in 1961 distinguished two different histopathological variations of the carcinosarcoma: endobronchial and peripheral. The endobronchial type appears as a peduncular mass with limited infiltration into the surrounding parenchyma and late regional and systemic metastasis. This form of growth frequently presents with obstructive pulmonary symptoms. The peripheral type frequently presents as a large mass with parietal infiltration affecting the mediastinum as well as intrapulmonary vessels; because of this it is sometimes referred to as peripheral-invasive. Metastasis is frequently present in this type at the time of diagnosis. This combined with the fact that it is a much more aggressive tumour (Stackhouse et al. 1969; Chaudhuri 1971; Co11 et al.
Pulmonary Carcinosarcoma
N=19
89
COUGH I
N=16
COSTAL
PAIN 1
'N=121
ITT-l
N=9J
-LT-EEq
N=4
EXPECTORATION
I 3 N=
2
ASTHENIA,ANOREXIA
5
*
.
*
10
15
20
Fig. 4. Semiological
data
1981; Sarma & Deshotels 1982) gives a poor prognosis and a much shorter survival time. Many of the publications in the literature do not classify the carcinosarcoma in two subgroups and therefore it is hard to make conclusions. The case presented
Fig. 5. Localization
8
of pulmonary
carcinosarcoma
90
A. Cabarcos, M. Gomel Dorronsoro and J. L. Lobo Beristain
in this paper was clearly tumour was extraordinary,
in a peripheral location and the growth doubling in size in less than a month.
rate of the
Tumour size Given the large variation of sizes reported in the literature, the authors decided to classify them in the following form: (+) for O-2 cm in diameter, (+ +) for 2-6 cm in diameter and (+ ++) for greater than 6 cm in diameter. With this classification only five cases reported (+), 17 reported (+ +) and 16 reported (++ +). It should be noted with respect to this last group that many of the diameters reported were far in excess of 6 cm as occurred in the present patient whose tumour was approximately 10 cm in diameter. Diagnostic techniques
It has already been pointed out that the only definitive way to establish the diagnosis is through a histopathological study of the tumour tissue (Bergmann et al. 1951; Kakos et al. 1971; Razzuk et al. 1971; Bull & Grimes 1974). Of the 48 cases published, 31 obtained the tumour sample by a thoracotomy and 15 from the necroscopic analysis. The results from many of the other biopsy techniques have proved in general to be inconclusive mainly because the sample obtained consisted of solely necrotic tissue or the small size of the sample permitted diagnosis of only one of the malignant cell components (Kakos et al. 1971; Anaya et al. 1976; Edwards et al. 1979). For example, only one case reported in the literature arrived at the diagnosis using a sample obtained with a fibreoptic bronchoscope (Loscos Valerio et al. 1981). The present case is the only one reported at present to have arrived at the diagnosis of a carcinosarcoma using a thin needle percutaneous biopsy. Metastasis
Metastasis was not discussed in 11 of the 48 cases reported in the literature and of the other 37 cases, 26 (70%) reported the presence of distant metastasis either during the initial diagnosis (n=22) or later in the course of the disease (n=4). Direct invasion was reported in 11 cases. The aggressiveness and rapid fatal prognosis of this mixed pulmonary tumour can be clearly seen in these data. The histopathology of the pulmonary carcinosarcoma
It is well accepted at present that the carcinosarcoma is a mixed pulmonary tumour composed of two histopathological components: epithelial and sarcomatous. Fig. 6 presents the most frequent types of cellular differentiation of each of the two components. From this it can be seen that in the majority of the cases reported (n=35), the epithelial component presented an epidermoid differentiation. In 10 of the cases a glandular differentiation was seen (adenocarcinoma) and in six cases the epithelial component was undifferentiated. The sarcomatous cellular component was reported to have a libromatous differentiation (fibrosarcoma) in 21 of the publications even though these
Pulmonary Carcinosarcoma
91
presented a fusocellular morphology and in nine publications such diverse differentiations as osteogenic, chondrogenic and leiomyomatous were presented. In the present case the epithelial component was epidermoid and the sarcomatous component was undifferentiated with a fusocellular morphology. It was impossible to make any relation between the smoking habit (Chaudhuri 1971; Lopez Barea et al. 1977) and the epidermoid differentiation because only eight of the publications presented these data. All six smokers had an epidermoid differentiation and of the two non-smokers, one had epidermoid and the other an adenocarcinoma.
DIFFERENTIATION
N=35 N=21 N=19 N=lO
n UNDIFFERENT.
ADENOCARC.
EPITHELIAL
F&. 6. Tumoral
In N=9
EPIDERMOID
FIBROSARC.
COMPONENT
cellular
UNDIFFERENT.
SARCOMATOUS
differentiations
in components
Correlations between the anatomopathological distinct parameters, such as age, sex, mortality, sive.
- epithelial
OSTEOID...
COMPONENT
and sarcomatous
type of tumour and other etc. were statistically inconclu-
Histogenesis Regardless of the many ultrastructural and microscopic studies done, the histogenesis of the pulmonary carcinosarcoma remains controversial. Meyer (Bergmann et al. 1951; Anaya et al. 1976) defined the theory of a ‘collision tumour’ in which both a carcinoma and a sarcoma begin simultaneously (Chaudhuri 1971). Herxheimer on the other hand is of the opinion that a carcinoma induces a malignant change in the stroma (Herxheimer & Reinke 1912; Drury & Stirland 1959; Jenkins 1968; Bull & Grimes 1974). Other authors
92
A. Cabarcos, M. GomeL Dorronsoro and J. L. Lobo Beristain
question the malignancy of the stroma and see its response to the carcinoma as an overabundant fibrous proliferation (Jenkins 1968; Edwards et al. 1979). Prive et al. (1961) describe the theory of a hamartoma that turned malignant, but the cases in the literature do not report evidence of a previous lesion. Virchow (1864) considered the tumour a result of the differentiation of a pluripotential stem cell and mesenchymal with the capacity to differentiate in both an epithelial direction. This presents the possibility on a purely conceptual level that the pulmonary carcinosarcoma consists of a metaplastic carcinoma, that is, a tumour solely of epithelial origin consisting of a carcinoma and a metaplastic sarcoma. The metaplasia would have the fusiform morphology and the observed reticular pattern explaining the observed images of transition of the cellular components in these tumours. The existence of this tumour has been recognized in different organs such as the breast, cervix, endometrium, ureter, etc. (Chaudhuri 1971; Bull & Grimes 1974; Anaya et al. 1976). However, Sarma & Deshotels (1982) have pointed out the,similarity that exists be’tween the pulmonary carcinosarcoma and those of the larynx, oesophagus, palate and tongue, and suggest that these represent a heterogeneous group of epithelial tumours that undergo a sarcomatous transformation. Ultrastructural techniques and tissue cultures (Rubin 1959; Anaya et al. 1976; Sarma & Deshotels 1982) have not helped a great deal in characterizing the histogenesis of the carcinosarcomas. However, the new techniques using immunoperoxidase with monoclonal antibodies (cytokeratin) may be able to be adapted to the study of the pulmonary carcinosarcoma, as has been done with other tumours of unknown histogenesis (Seifert 1983). Treatment
Given the turnour’s large size, it was not treated specifically in 16 of the 48 cases. In the other 32 cases surgery and/or radiotherapy was done. A pneumonectomy was done in 19 of the cases, in 12 a lobectomy, in four surgery and radiotherapy and in one case only radiotherapy was done. The patient in this case was unfit for surgery and refused radiotherapy. Evolution
and Prognosis
as stated previously is quite poor and it appears that the The prognosis, peripheral invasive form progresses more rapidly than the endobronchial type. Of the 48 cases in the literature, only 26 reported the time between diagnosis and death. Of these 26, death occurred within days in 16 and within 3 months in eight. In three cases the patient survived between 3 and 6 months and in seven cases the patient survived more than 6 months. References Anaya,
and Bibliography
A., Menendez, J. & Figuera, D. (1976) Carcinosarcoma caracteristicas morfobiol6gicas peculiares. Med. Clin. 66, 140-145.
de
pulmh
Un
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de
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G., Harazim, H. & Kaik, B. (1973) Uber Karzinosarkome der Lunge. Prux. Pneumol. 27, 604-616. Kakos, G. S., Williams, Jr., T. E., Assor, D. & Vasko, J. S. (1971) Pulmonary carcinosarcoma. Etiologic, therapeutic and prognostic considerations. J. thorac. cardiovasc. Surg. 61, 777-783. Lopez Barea, F., Garcia Villanueva, M., Fuejo Lago, D., Lopez Pujol, J. & Lago Viguera, J. (1977) Carcinosarcoma pulmonar: presentation de un case y revision de la literatura. Rev. Quir. Esp. 4, 137-141. Loscos Valerio, J. M., Gutierrez de1 Olmo, A. & Ramirez Armengol, J. A. (1981) Carcinosarcoma de pulmon. Arch. Bronconeumol. 17, 41-44. Ludwigsen, E. (1977) Endobronchial carcinosarcoma: a case with osteosarcoma of pulmonary invasive part and a review with respect to prognosis. Virchoms Archs. A. Path. Anat. und Histol. 373, 293-302. Moore T. C. (1961) Carcinosarcoma of the lung. Surgery 50, 886-893. Ogawa, K. (1929) Uber einen Fall vom sehr setterien primaren Karzionsarkom der Lunge, mit besonderer Beriicksichtigung der histologischen Untersuchugen. J. Orient Med. 11, 133-134. (Cited by Bergmann et al. 1951.) Peabody, C. N. (1959) Carcinoma of the lung of peripheral origin. J. thorac. cardiovasc. Surg. 37, 766. Prive, L., Tellem, M., Meranza, D. R. & Chodooff, R. D. (1961) C arcinosarcoma of the lung. Archs Path. 72, 351-357. Razzuk, M. A., Urschel, H. C., Race, G. J., Arndt, J. A. & Paulson, D. L. (1971) Carcinosarcoma of the lung. Report of two cases and review of the literature. J. thorac. cardiovasc. Surg. 61, 541-546.
94
A. Cabarcos, M. Gomez Dorronsoro and J. L. Lobo Beristain
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W. & Gruenagel, E. (1968) Kollisionstumoren der Lunge. Deutsch. Med. Wschr. 93, 1583. Mendez, F., Baro, X. & Reventos, J. (1975) El carcinosarcoma de pulmon. Arch.
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Rubin, A. (1959) Histogenesis of carcinosarcoma as revealed by tissue culture studies. Am. J. Obstet. Gynec. 77, 269. Saltikow, S. (1914) Beitrage zur Kenntnis des Karzinosarkoms. Verhandl. d. Deutsch. path. Gessellsch. 17, 351-363. (Cited by Razzuk et al. 1971.) Sarma, D. P. & Deshotels, S. J. (1982) C arcinosarcoma of the lung. J. Surg. Oncol. 19, 216-218. Seifert, G. (1983) Clasificacion de tumores orales y faringeos por medio de marcadores tumorales. Conferencia
en Las I Jornadas
de Anatomia
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de Basurto,
Bilbao,
7 octubre,
1983.
Selye, H. (1928) Uber zwei bemerkenswerthe Falle von Karzinosarkom. Med. Klin. 24, 1197-l 198. Stackhouse, E. M., Harrison, E. G. & Ellis, F. H. (1969) P rtmary mixed malignancies of the lung: carcinosarcoma and blastoma. J. thorac. cardiovasc. Surg. 57, 385-399. Taylor, H. E. & Rae, M. V. (1952) Endobronchial carcinosarcoma. J. thorac. cardiovasc. Surg. 24, 93-100. Virchow, R. (1864) Die Krankhaften Geschwulste, Vol. 2. Berlin: August Hirschwald. (Cited by Bergmann et al. 1951.) Weaver, P. C., Blanfoot, A. C. & Hargrove, R. L. (1971) A n endobronchial carcinosarcoma treated by lobectomy. J. thorac. cardiovasc. Surg. 61, 3OG303. Weber, F. (1939) Ein Karzinosarkom der Lunge. Zentralbl. f: allg. Path. u. path. Anat. 72, 113-l 17. (Cited by Razzuk et al. 1971.)
Dis.
Chest
(1985)
79, 83
PULMONARY CARCINOSARCOMA: A CASE STUDY AND REVIEW OF THE LITERATURE A. CABARCOS,
M. GOMEZ DORRONSORO J. L. LOB0 BERISTAIN
The General Geriatric Hospital of the Jose Matia Caluo the Basque County, Facul& of Medicine of the University Campus
AND
Foundation, of Bilbao,
Uniuersig of San Sebastian
Summary A case report of pulmonary carcinosarcoma and a review of the literature is presented. This is the only case where diagnosis has been obtained by thin needle percutaneous biopsy. Introduction The pulmonary carcinosarcoma is a rare malignant tumour composed of two components; carcinoma and sarcoma. Its existence has been a matter of controversy for many years (Jenkins 1968; Rodriguez Mendez et al. 1975; Ludwigsen 1977; Edwards et al. 1979) but recently it has been generally accepted as a histopathological and clinical entity even though its histogenesis remains obscure. Compared with all pulmonary neoplasms it occurs with a frequency of 0.3% (Diaconita 1975; Co11 et al. 1981) and in Spain only six cases including this one have been reported. In this paper we present a detailed case report on a patient with pulmonary carcinosarcoma and review the clinical and histological parameters in the literature. Case Report The patient was a 72.year-old man who smoked 15 cigarettes per day until 4 years before he was admitted to hospital complaining of a progressive pain in his right scapular region. The pain had started after a severe trauma to this region 6 weeks before he was admitted. The pain radiated down the thoracic wall to the right hypochondrium. Apart from this, the patient showed a progressive disinterest in his surroundings as well as asthenia, anorexia and a weight loss of 6 kg. Physical examination did not reveal anything definitive except that percussion of the lower third of the right hemithorax was dull and there were inspiratory crepitations with a small increase in the transmission of vocal vibrations in this area. Investigations showed a mild hypoalbuminaemia with a polyclonal hypergammaglobulinaemia. The sedimentation rate was 76 mm/h. The ;\/lantoux (5 U PPD) was 20 mm and, bacteriological testing and sputum culture in Liiwenstein’s medium were both negative. Chest radiographs and tomograms revealed the presence of a mass in the right inferior pulmonary lobe that was 6 cm in diameter, homogeneous and with irregular borders. Obliteration Address Bidebieta-2,
for
correspondence: San Sebastian-9
Antonio (Espana).
Cabarcos
MD,
Plaza
de 10s Cazadores
no 63”
B, Ayete
A. Cabarcos, A4. Gomez Dorronsoro and J. L. Lobo Beristain of the costophrenic angle was observed along with a pleural thickening and a local elevation of the diaphragm. There was no evidence of cavitation nor of calcification or adenopathy of the mediastinal or hilar lymph nodes. Bronchoscopy did not show evidence of an endobronchial growth nor bronchial compression by extrabronchial tumour. Cytological examination of the bronchial washings along with five separate sputum examinations showed a complete absence of malignant cells in the inflammatory exudate. Transbronchial biopsy demonstrated foci of squamous metaplasia together with an intense oedema and chronic inflammation. The thin needle percutaneous, aspiration showed evidence of a haemorrhagic liquid that contained neoplastic cells and anaerobic, aerobic and Lowenstein cultures
Fig. 1. The (H
malignant
epithelial
component
merges
with
the
malignant
mesenchymal
component.
& E x320)
were all negative. Additional studies such as a barium swallow, cerebral scan, bone scan, studies for bone marrow metastasis among others did not reveal significant abnormalities. The diagnosis was reached by the histopathological analysis of the thin needle percutaneous aspiration which clearly contained two distinct malignant cellular components. The first one consisted of a polygonal cell with clear squamous differentiation without glandular formation, The second, intimately combined with the first, was composed of sarcomatous fusiform cells of a fibroblastic type without myoid or chondroid differentiation (Fig. 1). This double cellular pattern was demonstrated using Snook’s staining technique for reticulin (Fig. 2). During the patient’s stay in the hospital the tumour proved to be highly aggressive, growing very rapidly and eventually infiltrating the thoracic wall and destroying a rib. The patient died and a necroscopy was not performed.
Pulmonav
a5
Carcinosarcoma
Discussion The first description of this pulmonary tumour as a bicellular histopathological entity, according to Bergmann et al. (1951), was made by Kika (cited in Herxheimer & Reinke 1912) in 1908. Since then six cases have been published in Spain and a total of 42 in the rest of the world (Table I). Incidence according to sex and age The tumour has a clear prevalence in the male (Stackhouse et al. 1969; Bull & Grimes 1974; Ludwigsen 1977; Edwards et al. 1979; Co11 et al. 1981) as 40 of the
Fig. 2. The carcinomatous
sarcomatous element
element is unstained.
has produced (Snook’s reticulin
abundant X 160)
reticulin
fibres
in
contrast,
the
48 publications deal with male patients. That gives a male to female incidence of 5: 1. As can be seen in Fig. 3, the incidence with respect to age shows three peaks. The most frequent is between the sixth and seventh decade followed by the fifth and sixth and the seventh and eighth respectively. Summing up all the cases presented in the three decades stated, it can be observed that a total of 41 or 85% occurred between the ages of 50 and 80 years. This age range of presentation combined with the fact that the authors are at the only geriatric hospital in the 7*
45 F
M M M M
74 71 59 46
64 M
Moore
70 M
Stackhouse
et al. 1969
81 M 73 F
Jenkins 1968 Stackhouse et al. 1969
1961
R.U.L. R.U.L. R.M.Br. L.M.Br.
M M M M
51 56 69 57
Bergmann et al. 1951 Bergmann et al. 1951 Taylor & Rae 1952 Taylor & Rae 1952 Cavallero 1956 Peabody 1959 Drury & Stirland 1959 Drury & Stirland 1959 Prive et al. 1961
R.L.L. L.M.Br.+ L.u.L.+ L.L.L. R.U.L.
R.L.L.
L.U.L. R.U.L. R.L.L. L.U.L.
L.U.L.
L.U.L.
R.U.L.
R.U.L. R.U.L.
L.L.L.
R.L.L.
55 F
et al. 1949
1939
Weber
Hochberg
61 M
1938
Fischer
77 M
43 M -70 M
Selye 1928 Geschelin 1928 Ogawa 1929
1915
Frank
&
Age, sex Location
35 F
andyear
Kika 1908 Cited in Herxheimcr Reinke 1912) Saltikow 1914
Author
cm
-
7X4X3
5
3
1 10 2X1X1
-
4X1X1 2x1
3X1
6~4x4
11X10X5
10 cm
10 cm
15 cm
10 cm
10X8
14 cm
Size
Surgery
Autopsy surgery
Surgery
Surgery Surgery Surgery Autopsy Autopsy Surgery Autopsy Autopsy Surgery
Surgery
Autopsy
Autopsy
Autopsy Autopsy Autopsy
Autopsy
Autopsy
Diqnostic
Table I. Cases
published
in world
Yes
Not Not
Not
Yes Not Yes Not
Not Not Not Not
Yes
NO1
Not Yes
Not
Yes Not Not Yes
Not Not Not Not
Yes
Yes
Not
Yes
Yes
Not Yes
Yes
Metastasis
Yes
Local invasion
Epidermoid Undifferentiat
Epidermoid
Epidermoid
Epidermoid Epidermoid Epidermoid
Epidermoid Epidermoid Epidermoid Epidermoid -
Epidermoid keratinizing Epidermoid keratinizing, Adenocarcinoma, Undifferentiat Epidermoid
Adenocarcinoma
Epidermoid
Undifferentiat. Epidermoid Adenopapillary Ca.
of comjwnent
Carcinomatous
Nature
literature
Fibrosarcoma+ Chondrosarc
Fibrosarcoma
Fibrosarcoma Fibrosarcoma \Chondrofibrosarcoma Fibrosarcomat osteoid+ rhabdomyobla.
Fibrosarcoma Fibrosarcoma Fibrosarcoma Fibrosarcoma
Fibrosarcoma
Spindle cell+ pleomorphic Small spindle cell Pleomorphic Small spindle cell Large spindle cell + pleomorphic Spindle cell
-
Sarcomatous
Lobectomy+ Radiation
Not
Lobectomy+ radiation
Pneumonec. Pneumonec. Pneumonec. Not Not Lobectomy Not Not Pneumonec.
Lobectomy
Not
Not
Not Not Not
Not
Not
-
Treatment
+3Y
+3 m
+
Alive
2 y
+postoperative Alive 6 y Alive 2 y Alive 3 y + + +3 m +6 m +l m Alive 2 y
+
+
+ + +
+
+
Evolution
<
tz, g, 3
s-
P t?
s
s 2 2
n & Q 2 s
b
co -.
Mendez
Rodriguez 1975
Case
72 M
21 F 52 M
al. 1981 & Deshotels
1982
55 M 62 M 74 M
et al. 1976 Barca et al. 1977 Valerio et al.
report
Anaya Lopez Loscos 1981 Co11 et Sarma
71 M
1977
Ludwigsen
71 M
R.U.L. L.U.L. L.L.L.
66 M 62 M 56 M
Davis et al 1972 Kaik et al. 1973 Bull & Grimes 1974
et al.
L.M.Br
61 F
et al. 1972
Davis
K.L.1,.
L.L.L. L.L.L.
I,.TJ.L. R.L.L. R.U.L.
L.L.L.
R.U.L.
R.U.L.
65 M
et al. 1972
Davis
R.U.L.
L.U.L. R.U.L. R.U.L.
60 F
61 M 67 M 58 M
et al. 1969
Stackhouse
74 M
Razzuk et al. 1971 Weaver et al. 1971 Davis et al. 1972
et al. 1969
Stackhouse
R.L.L. L.U.L. R.U.L.+ M.Br. R.U.L.
L.U.L. L.L.L. L.U.L. L.L.L. L.U.L.
et al. 1969
Stackhouse
70 M 69 M 54 M
M M M M F
et al. 1969
Stackhouse
L.U.L.
68 65 69 72 66
et al. 1969
Stackhouse
52 M
Stackhouse et al. 1969 Chaudhuri 1971 Chaudhuri 197 1 Kakos et al. 1971 Razzuk et al. 1971
et al. 1969
Stackhouse
10
8 3
1x1 8x8 8
2
9~8x6
3x2
2X1X1
3
4 5X2x2
3~2x2
3X2x3 10x 10x6 1x1
6X5X5
6X5X5
12X5X6 5X4X3 --
3X4X3
Biopsy
Surgery Autopsy
Surgery Surgery
surgery
Autopsy
Surgery Surgery Surgery
Surgery
Autopsy
Surgery Surgery Surgery
Surgery Surgery Surgery Surgery Surgery
Surgery
Surgery
Surgery Surgery Surgery
Surgery
~
Yes
YCS -
Plot Yes
Yes
Not
-
-
~~ ~ ~-
~ ~ ~-
-
-
~-
Not
-~
xot
Yes
Not
Not No1 Yes
Yes
X0t
Yes Yes Yes
ITot
Not
Yes
YtX xot
Not Yes Not Yes YCS
YCS
Adenocarcinoma Epidermoid+ adenoid q. Epidermoid
Epidermoid Epidermojd Epidermoid
Epidermoid
Epidermoid
Adenocarcinorna+ Undiffercntiat. Epidermoid Undilrerentiat. Epidermoid+ tubular Epidermoid+ cubic Epidermoid+ Adenocarcinoma Epidermoid Epidermoid Epidermoid
Adenocarcinoma+ Undiffcrcntiat. Epidermoid Epidermoid Epidermoid
Epidermoid
Adenocarcinoma Adcnocarcinoma Epidermoid
Yes Yes YCS YCS
Epidermoid
Yes
cell
SpindIp
cell
Spindle cell Osteosarcoma
Fibrosarcorna+ osteoid Osteosarcorna+ rabdomyosar. fibrosarcoma Spindle cell Fibrosarcoma Spindle cell
Spindle cell Leiomyosarc. Spindle cell+ blastoma Spindle cell+ blastoma Spindle cell+ blastoma Spindle cell Spindle cell Spindle cell
Spindle
Fibrosarcoma Pibrosarcoma Spindle cell
Fibrosarcoma+ ostroid Fibrosarcoma
Fibrosarcoma +osteoid Fibrosarcoma Fibrosarcoma Fibrosarcoma
+
+
m
m
Not
+I5 +19
Pneurnonec. Pneumonec.
+2 *n
d d
+
2 y
3 m
+ postop. +5 m
Alive
4
+9 m +2 m
+9
Alive
+ postop.
Alive15m 1-18 m
+21
+6 d +3 m +2 d Alive 6 m +10 m
m
4 y
Pncumoncc. Pneumonec. Not
Not
Radiation
PIEUIIlOIlCC.
Pneurnoncc. Pneumonec.
Pneumoncc.
Pneumonec.
Lobectomy Lobectomy
PIl~U~OIlCC.
Pneumonec. Pneumoncc. Lobectomy Pncumonec. Lobectomy
+11
+6 m
Pneumonec. Lobectomy
+2 m +3 m
-
Alive
Lobectomy Lobectomy Radiation
Lobectomy
2
2
iz v Q a ? 8’ 8 e 2
88
A. Cabarcos, M. Gomez Dorronsoro and J. L. Lobo Beristain
country have been the deciding observe one of these rare cases.
factors
that
have given
us the opportunity
to
Clinical presentation The semiological data are presented in Fig. 4. From’ this it can be observed that the most frequent symptoms are a cough, thoracic chest pain and dyspnoea. Thus, it is quite clear that the disease has non-specific signs and symptoms and to establish a definitive diagnosis, additional analytical techniques must be employed.
N=
CASES
25 N=19
r
20
15 N=12 10
N=lO
5 0 N=41 I
I
I
0
10
20
,
30
1
40
-TV-,
-7
50
60
AGE
(85%) ---r~--I
70
I
80
90
100
Fig. 3. Age of presentation
Location and type of growth The majority of the-cases (~~32, 69%) show the tumour affecting the superior lobe and/or the principal bronchi, while it is infrequent in the lower lobes. The patient presented in this paper was affected in the most unusual location: the right lower lobe (Fig. 5). With respect to the type of growth, Moore, in 1961 distinguished two different histopathological variations of the carcinosarcoma: endobronchial and peripheral. The endobronchial type appears as a peduncular mass with limited infiltration into the surrounding parenchyma and late regional and systemic metastasis. This form of growth frequently presents with obstructive pulmonary symptoms. The peripheral type frequently presents as a large mass with parietal infiltration affecting the mediastinum as well as intrapulmonary vessels; because of this it is sometimes referred to as peripheral-invasive. Metastasis is frequently present in this type at the time of diagnosis. This combined with the fact that it is a much more aggressive tumour (Stackhouse et al. 1969; Chaudhuri 1971; Co11 et al.
Pulmonary Carcinosarcoma
N=19
89
COUGH I
N=16
COSTAL
PAIN 1
'N=121
ITT-l
N=9J
-LT-EEq
N=4
EXPECTORATION
I 3 N=
2
ASTHENIA,ANOREXIA
5
*
.
*
10
15
20
Fig. 4. Semiological
data
1981; Sarma & Deshotels 1982) gives a poor prognosis and a much shorter survival time. Many of the publications in the literature do not classify the carcinosarcoma in two subgroups and therefore it is hard to make conclusions. The case presented
Fig. 5. Localization
8
of pulmonary
carcinosarcoma
90
A. Cabarcos, M. Gomel Dorronsoro and J. L. Lobo Beristain
in this paper was clearly tumour was extraordinary,
in a peripheral location and the growth doubling in size in less than a month.
rate of the
Tumour size Given the large variation of sizes reported in the literature, the authors decided to classify them in the following form: (+) for O-2 cm in diameter, (+ +) for 2-6 cm in diameter and (+ ++) for greater than 6 cm in diameter. With this classification only five cases reported (+), 17 reported (+ +) and 16 reported (++ +). It should be noted with respect to this last group that many of the diameters reported were far in excess of 6 cm as occurred in the present patient whose tumour was approximately 10 cm in diameter. Diagnostic techniques
It has already been pointed out that the only definitive way to establish the diagnosis is through a histopathological study of the tumour tissue (Bergmann et al. 1951; Kakos et al. 1971; Razzuk et al. 1971; Bull & Grimes 1974). Of the 48 cases published, 31 obtained the tumour sample by a thoracotomy and 15 from the necroscopic analysis. The results from many of the other biopsy techniques have proved in general to be inconclusive mainly because the sample obtained consisted of solely necrotic tissue or the small size of the sample permitted diagnosis of only one of the malignant cell components (Kakos et al. 1971; Anaya et al. 1976; Edwards et al. 1979). For example, only one case reported in the literature arrived at the diagnosis using a sample obtained with a fibreoptic bronchoscope (Loscos Valerio et al. 1981). The present case is the only one reported at present to have arrived at the diagnosis of a carcinosarcoma using a thin needle percutaneous biopsy. Metastasis
Metastasis was not discussed in 11 of the 48 cases reported in the literature and of the other 37 cases, 26 (70%) reported the presence of distant metastasis either during the initial diagnosis (n=22) or later in the course of the disease (n=4). Direct invasion was reported in 11 cases. The aggressiveness and rapid fatal prognosis of this mixed pulmonary tumour can be clearly seen in these data. The histopathology of the pulmonary carcinosarcoma
It is well accepted at present that the carcinosarcoma is a mixed pulmonary tumour composed of two histopathological components: epithelial and sarcomatous. Fig. 6 presents the most frequent types of cellular differentiation of each of the two components. From this it can be seen that in the majority of the cases reported (n=35), the epithelial component presented an epidermoid differentiation. In 10 of the cases a glandular differentiation was seen (adenocarcinoma) and in six cases the epithelial component was undifferentiated. The sarcomatous cellular component was reported to have a libromatous differentiation (fibrosarcoma) in 21 of the publications even though these
Pulmonary Carcinosarcoma
91
presented a fusocellular morphology and in nine publications such diverse differentiations as osteogenic, chondrogenic and leiomyomatous were presented. In the present case the epithelial component was epidermoid and the sarcomatous component was undifferentiated with a fusocellular morphology. It was impossible to make any relation between the smoking habit (Chaudhuri 1971; Lopez Barea et al. 1977) and the epidermoid differentiation because only eight of the publications presented these data. All six smokers had an epidermoid differentiation and of the two non-smokers, one had epidermoid and the other an adenocarcinoma.
DIFFERENTIATION
N=35 N=21 N=19 N=lO
n UNDIFFERENT.
ADENOCARC.
EPITHELIAL
F&. 6. Tumoral
In N=9
EPIDERMOID
FIBROSARC.
COMPONENT
cellular
UNDIFFERENT.
SARCOMATOUS
differentiations
in components
Correlations between the anatomopathological distinct parameters, such as age, sex, mortality, sive.
- epithelial
OSTEOID...
COMPONENT
and sarcomatous
type of tumour and other etc. were statistically inconclu-
Histogenesis Regardless of the many ultrastructural and microscopic studies done, the histogenesis of the pulmonary carcinosarcoma remains controversial. Meyer (Bergmann et al. 1951; Anaya et al. 1976) defined the theory of a ‘collision tumour’ in which both a carcinoma and a sarcoma begin simultaneously (Chaudhuri 1971). Herxheimer on the other hand is of the opinion that a carcinoma induces a malignant change in the stroma (Herxheimer & Reinke 1912; Drury & Stirland 1959; Jenkins 1968; Bull & Grimes 1974). Other authors
92
A. Cabarcos, M. GomeL Dorronsoro and J. L. Lobo Beristain
question the malignancy of the stroma and see its response to the carcinoma as an overabundant fibrous proliferation (Jenkins 1968; Edwards et al. 1979). Prive et al. (1961) describe the theory of a hamartoma that turned malignant, but the cases in the literature do not report evidence of a previous lesion. Virchow (1864) considered the tumour a result of the differentiation of a pluripotential stem cell and mesenchymal with the capacity to differentiate in both an epithelial direction. This presents the possibility on a purely conceptual level that the pulmonary carcinosarcoma consists of a metaplastic carcinoma, that is, a tumour solely of epithelial origin consisting of a carcinoma and a metaplastic sarcoma. The metaplasia would have the fusiform morphology and the observed reticular pattern explaining the observed images of transition of the cellular components in these tumours. The existence of this tumour has been recognized in different organs such as the breast, cervix, endometrium, ureter, etc. (Chaudhuri 1971; Bull & Grimes 1974; Anaya et al. 1976). However, Sarma & Deshotels (1982) have pointed out the,similarity that exists be’tween the pulmonary carcinosarcoma and those of the larynx, oesophagus, palate and tongue, and suggest that these represent a heterogeneous group of epithelial tumours that undergo a sarcomatous transformation. Ultrastructural techniques and tissue cultures (Rubin 1959; Anaya et al. 1976; Sarma & Deshotels 1982) have not helped a great deal in characterizing the histogenesis of the carcinosarcomas. However, the new techniques using immunoperoxidase with monoclonal antibodies (cytokeratin) may be able to be adapted to the study of the pulmonary carcinosarcoma, as has been done with other tumours of unknown histogenesis (Seifert 1983). Treatment
Given the turnour’s large size, it was not treated specifically in 16 of the 48 cases. In the other 32 cases surgery and/or radiotherapy was done. A pneumonectomy was done in 19 of the cases, in 12 a lobectomy, in four surgery and radiotherapy and in one case only radiotherapy was done. The patient in this case was unfit for surgery and refused radiotherapy. Evolution
and Prognosis
as stated previously is quite poor and it appears that the The prognosis, peripheral invasive form progresses more rapidly than the endobronchial type. Of the 48 cases in the literature, only 26 reported the time between diagnosis and death. Of these 26, death occurred within days in 16 and within 3 months in eight. In three cases the patient survived between 3 and 6 months and in seven cases the patient survived more than 6 months. References Anaya,
and Bibliography
A., Menendez, J. & Figuera, D. (1976) Carcinosarcoma caracteristicas morfobiol6gicas peculiares. Med. Clin. 66, 140-145.
de
pulmh
Un
tumor
de
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93
Aykan,
T. B. (1956) Considerations on the pathogenesis of carcinosarcoma and allied tumours in the light of experimental investigations. Oncologia (Basel) 9, 418-425. Barrett, H. D. & Barnard, W. G. (1945) Some unusual thoracic tumours. Br. J. Surg. 32, 447-457. Bergmann, M., Ackerman, L. V. & Kemler, R. L. (1951) C arcinosarcoma of the lung. Review of the literature and report of two cases treated by pneumonectomy. Cancer (Philad.) 4, 919-929. Bull, J. C. & Grimes, 0. F. (1974) Pulmonary carcinosarcoma. Chest 65, 9-12. Cavallero, G. (1956) Contributo allo studio dei tumori misti maligni de1 polmone di tipo bidermico (carcinosarcomi) Pathologica (Genova) 48, 2 13-226. (Cited by Moore 196 1.) Coll, R., Alberola, C., Padilla, J., Mayayo, E. & Marco, V. (1981) Un case de carcinosarcoma de pulmon. Revision de la literature. Arch Bronconeumol. 17, 63-66. Chaudhuri, M. R. (1971) Bronchial carcinosarcoma, J. thorac. cardiovasc. kg. 61, 319-323. Davis, P. W., Briggs, V. C., Seal, R. M. E. & Storring, F. K. (1972) Benign and malignant mixed tumours of the lung. Thorax 27, 657-673. Diaconita, G. (1975) Bronchopulmonary carcinosarcoma. Thorax 30, 682-686. Drury, R. A. B. & Stirland, R. M. (1959) C arcinosarcomatous tumours of the respiratory tract. J. Path.
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Kaik,
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G., Harazim, H. & Kaik, B. (1973) Uber Karzinosarkome der Lunge. Prux. Pneumol. 27, 604-616. Kakos, G. S., Williams, Jr., T. E., Assor, D. & Vasko, J. S. (1971) Pulmonary carcinosarcoma. Etiologic, therapeutic and prognostic considerations. J. thorac. cardiovasc. Surg. 61, 777-783. Lopez Barea, F., Garcia Villanueva, M., Fuejo Lago, D., Lopez Pujol, J. & Lago Viguera, J. (1977) Carcinosarcoma pulmonar: presentation de un case y revision de la literatura. Rev. Quir. Esp. 4, 137-141. Loscos Valerio, J. M., Gutierrez de1 Olmo, A. & Ramirez Armengol, J. A. (1981) Carcinosarcoma de pulmon. Arch. Bronconeumol. 17, 41-44. Ludwigsen, E. (1977) Endobronchial carcinosarcoma: a case with osteosarcoma of pulmonary invasive part and a review with respect to prognosis. Virchoms Archs. A. Path. Anat. und Histol. 373, 293-302. Moore T. C. (1961) Carcinosarcoma of the lung. Surgery 50, 886-893. Ogawa, K. (1929) Uber einen Fall vom sehr setterien primaren Karzionsarkom der Lunge, mit besonderer Beriicksichtigung der histologischen Untersuchugen. J. Orient Med. 11, 133-134. (Cited by Bergmann et al. 1951.) Peabody, C. N. (1959) Carcinoma of the lung of peripheral origin. J. thorac. cardiovasc. Surg. 37, 766. Prive, L., Tellem, M., Meranza, D. R. & Chodooff, R. D. (1961) C arcinosarcoma of the lung. Archs Path. 72, 351-357. Razzuk, M. A., Urschel, H. C., Race, G. J., Arndt, J. A. & Paulson, D. L. (1971) Carcinosarcoma of the lung. Report of two cases and review of the literature. J. thorac. cardiovasc. Surg. 61, 541-546.
94
A. Cabarcos, M. Gomez Dorronsoro and J. L. Lobo Beristain
Remmele, Rodriguez
W. & Gruenagel, E. (1968) Kollisionstumoren der Lunge. Deutsch. Med. Wschr. 93, 1583. Mendez, F., Baro, X. & Reventos, J. (1975) El carcinosarcoma de pulmon. Arch.
Bronconeumol:
11, 15 I-154.
Rubin, A. (1959) Histogenesis of carcinosarcoma as revealed by tissue culture studies. Am. J. Obstet. Gynec. 77, 269. Saltikow, S. (1914) Beitrage zur Kenntnis des Karzinosarkoms. Verhandl. d. Deutsch. path. Gessellsch. 17, 351-363. (Cited by Razzuk et al. 1971.) Sarma, D. P. & Deshotels, S. J. (1982) C arcinosarcoma of the lung. J. Surg. Oncol. 19, 216-218. Seifert, G. (1983) Clasificacion de tumores orales y faringeos por medio de marcadores tumorales. Conferencia
en Las I Jornadas
de Anatomia
Patol&ca
de1 Hospital
de Basurto,
Bilbao,
7 octubre,
1983.
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