Thorax 1983;38:632-634

Intrapulmonary teratoma 10 years after removal of a mediastinal teratoma in ayoung man

HW PRAUER, D MACK, R BABIC

From the Department of Thoracic Surgery and Institute ofPathology, Klinikum Rechts der Isar, TechnicalUniversity, Munich

Teratoma is a common tumour of the mediastinum but israrely found in the lung. The twentieth reported case waspublished in 1975 by Day and Taylor and four cases havebeen published since.'-5 A rare condition, not previouslyrecorded, was observed by us in a young male patient. Hewas found to have a mediastinal teratoma, which was fol-lowed 10 years later by the growth of an intrapulmonaryteratoma.

Case report

A 14-year-old boy was admitted to our thoracic unit forthe first time in 1970 with an opacity in the right pleuralcavity detected on a routine chest film. He had noticedintermittent pain in the right chest radiating to the arm anda non-productive cough. The chest radiograph showed asolid mass of varying density 10 cm in diameter just abovethe right diaphragm and adjacent to the right cardiac bor-der (fig la). Tomography showed calcification and cysticareas. Routine laboratory investigations and bronchoscopygave normal results. Posterolateral thoracotomy was per-formed and a firm mass with a knobbly surface was foundin the lower anterior mediastinum. The tumour waspedunculated, arising from the upper mediastinum andreceiving an additional blood supply from branches of theinternal mammary artery. It was excised without difficultyand the patient made an uneventful postoperative recov-ery.The operative specimen was a nodular tumour measur-

ing 13 x 12 x 7 cm. The cut surface showed several cavitiescontaining sebaceous material and hair, surrounded byareas of hyaline tissue, bone, and cartilage. On microscopicexamination some cystic cavities were seen to be lined bysquamous epithelium with sebaceous glands and hair folli-cles and others by respiratory and intestinal epithelium.Sections from various points showed connective tissue,hyaline cartilage, and fat tissue. In one nodular areaepithelial strands and tubuli suffounded by immaturemesenchyme were found. There was no evidence of malig-nant change in any part of the tumour. The diagnosis wasbenign cystic teratoma of the mediastinum.Ten years later the patient reappeared with a five-month

history of recurrent bronchopneumonia unresponsive to

Address for reprint requests: Dr HW Priuer, Chirurgische Klinikund Poliklinik der Technischen Universit5t Munchen, Ismaninger Fig 1 Chest radiographs showing (a) an opacity in theStrasse 22, D-8000 Mtinchen 80, W Germany.

right lower anterior mediastinum (1970) and (b) a cysticAccepted 9 February 1983 mass in the right upper lobe with focal calcifications (1980,

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Intrapulmonary teratoma 10 years after removal of a mediastinal teratoma in a young manantibiotics. Chest radiographs showed an opacity in theright upper lobe 9 cm in diameter with cystic areas andcalcification. The shadow was strikingly similar inconfiguration and structure to that found in 1970 (fig lb).The haemoglobin concentration was 14*8 g/dl, the whiteblood cell count 9.1 x 109/l, and the erythrocyte sedimen-tation rate 120 mm in the first hour. The concentration ofserum a-fetoprotein was raised (192 ng/ml) but that ofcarcinoembryonic antigen was normal. Bronchoscopyshowed purulent discharge from the orifice of the rightupper-lobe bronchus and a tuft of hair protruding into thelumen. Scans of liver, skeleton, and brain were normal.Intrapulmonary teratoma was suspected and anotherthoracotomy was performed. After partial extrapleural dis-section a solid tumour was palpable in the anterior segmentof the right upper lobe, densely adherent to the middlelobe and the pericardium. For complete excision bilobec-tomy (right upper and middle lobe) and partial pericardec-tomy had to be performed.The right upper lobe was occupied by an encapsulated,

partially cystic mass 8 cm in diameter. The cut surfaceshowed hair and sebaceous material within the cavities and

bony areas in the surrounding tissue (fig 2a). The middlelobe was collapsed.

Histologically well differentiated structures of epithelialand mesenchymal origin were present. There were cavitieslined with squamous epithelium and others with respirat-ory epithelium. Areas of smooth muscle, fat, cartilage,pancreatic acinar tissue, and fetal lung (fig 2b) could beidentified. The diagnosis was a predominantly mature butpartially immature intrapulmonary teratoma.The postoperative course was uncomplicated. The

ca-fetoprotein concentration has gradually returned tonormal and has remained so during a follow-up period of28 months.

Discussion

Several well-documented cases of intrapulmonaryteratoma have been published in recent years, and theclinicopathological features have been delineated and itspossible origins discussed.'7 We confine ourselves to

(b)Fig 2 (a) Cross-section of the operative specimen showing the well-encapsulated intrapulmonary teratoma. (b) Sectionfrom another part of the tumour showing fetal lung. (Haematoxylin and eosin, x 16.)

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drawing attention to some unusual aspects of the case we

describe.Firstly, this patient had teratomas developing both in the

mediastinum and in the lung. The second tumourdeveloped independently of the first, although a common

origin from aberrant tissue of the third pharyngeal pouchmay be assumed.6 Secondly, the bronchoscopic demonstra-tion of hair in the bronchial tree-equivalent to the rare

symptom of expectoration of hair (trichoptysis)-led to thepreoperative diagnosis of intrapulmonary teratoma.258Thirdly, the appearance of the second tumour was associ-ated with a raised serum a-fetoprotein concentration,which returned to normal after removal of the tumour.Evidence of its origin from tumour tissue was obtained byimmunocytochemical labelling (PAP reaction).9 Alpha-fetoprotein is typically produced in hepatocytes, yolk saccells, and probably also certain cells of the primitive gut.Recently a primary lung cancer producing a-fetoproteinwas observed.'0 To our knowledge an a-fetoprotein-producing intrathoracic teratoma has not been reportedpreviously. Our observations may be consistent with thetheory of the origin of thoracic teratoma from tissue of theupper primitive gut.

Prauer, Mack, Babic

References

'Day DW, Taylor SA. An intrapulmonary teratoma associatedwith thymic tissue. Thorax 1975;30:582-7.

2 Kravetz VM, Kamentsky MC, Efdaha PN, Ilivsky IP. Intrabron-chial teratoma. Kln Khir 1976;8:54-5.

3Holt S, Deverall PB, Boddy JE. A teratoma of the lung contain-ing thymic tissue. J Pathol 1978;126:85-9

4Eckert VM, Gerassimidis T. Intrapulmonales Teratom. Fall-bericht und Literaturlibersicht. Fortschr Med 1979;97:1051-4.

Jamieson MPG, McGowan AR. Endobronchial teratoma.Thorax 1982;37: 157-9.

6Bateson EM, Hayes JA, Woo-Ming M. Endobronchial teratomaassociated with bronchiectasis and bronchiolectasis. Thorax1968;23:69-76.

Gautam HP. Intrapulmonary malignant teratoma. Am RevRespir Dis 1969;100:863-5.

8 Laffitte MH. Embryome tdratoide intra-pulrnonaire. Exerbse enun temps. Mem Acad Chir 1937;63:1076-85.

9 Sternberger LA. Immunocytochemistry. 2nd ed. New York: JohnWiley and Sons, 1979:104-69.

'° Yasunami R, Hashimoto Z, Ogura T, Yamamura Y. Primarylung cancer producing alpha-fetoprotein. Cancer 1981;47:926-9.

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