Thorax (1968), 23, 434.

Endometriosis of the lungA case report and brief review of the literature

TATIANA JELIHOVSKY AND A. F. GRANT

From the Departmenit of Pathology and the Page Chest Pavilion, Royal Prince Alfred Hospital, Camperdown,New South Wales, Australia

A case is described of endometriosis of the lung in a symptomless 54-year-old woman. This andthe 11 previously recorded cases of histologically examined pulmonary endometriosis have beendivided into two groups. Six of these women had died during pregnancy or shortly after delivery,and their lungs contained deposits of decidua but no endometrial glands; the remaining sixpatients, at least five of whom had had previous pregnancies, had surgically resected lungspecimens showing larger single lesions with endometrial glands. The possible relationship betweenthese two groups is discussed.

Endometriosis of the lung is rare. Although thecondition has been diagnosed clinically in patientswith a history of recurrent catamenial haemo-ptyses, a search of the literature revealed only 11cases in which endometrial tissue was found onhistological examination of the lungs. These fellinto two distinct groups. In the first, small islandsof decidua were found at necropsy in the lungs ofsix subjects who had died either during pregnancyor shortly after delivery (Table I). In the second,larger single lesions with endometrial glands werefound in surgically resected specimens (Table II).

One of these was from a post-menopausal woman(Sturzenegger, 1960), and it is the purpose of thispaper to present a second such case.

CASE HISTORY

A 54-year-old woman, born in Hungary but living inAustralia during the past eight years, was found onmass miniature radiography to have a shadow in theright lung and was referred for further investigation.She gave a history of exertional dyspnoea for the pastfew years, during which she had gained weight. At thetime of examination her weight was 200 lb. (90 7 kg.).

TABLE IDECIDUA FOUND IN THE LUNG AT POST-MORTEM EXAMINATION

Author Time of Death Lung Remarks

Park (1954) 4 hours after delivery One focus ofdecidua 250 I across, 6 mm. beneath the pleura,not lying in blood vessels and with no evidence of a largevessel having been eroded. Large capillaries within and atmargin of plaque

Hartz (1956) Previously unpublished case ofDr. Park

Hartz (1956) 4th month of pregnancy One focus of decidua 500 p across, lying directly beneath thepleura. In one section a thin-walled vessel was seenbetween the cells

Lattes et al. During delivery Multiple small clusters of decidual cells apparently emboliz- Previously unpublished case of(1956) ing capillaries of the interalveolar septa Dr. Godman similar to that

reported by Dr. Hartz

Cameron and After delivery Multiple islands of decidua up to 580 p across, well estab-Park(1965) lished and growing in the walls of blood vessels andCase 1 alveoli

Cameron and 35th week of pregnancy; One plaque of decidua 300 p across, lying 5 mm. beneathPark(1965) undelivered the pleuraCase 2

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Endometriosis of the lung

TABLE II

ENDOMETRIAL GLANDS FOUND IN RESECTED LUNG SPECIMEN

ReproductiveAuthor Status Past History Presenting Features Lung Lesion

Lattes et al. Pregnant Caesarean and curet- Cyclical haemoptyses following curet- Nodule of decidua and endometrial(1956) tage tage. Radiographic shadow became glands, 18 mm. diam., with a poly-

obvious during last pregnancy poid projection into bronchus

Sturzenegger Post-menopausal Panhysterectomy at Radiographic opacity Cyst, 4 cm. diam., lined by endomet-(1960) age 46 rium with a polypoid projection

Rodman and Pre-menopausal Abortion Cyclical haemoptyses following abor- Mural nodule of endometriosis pro-Jones(1962) tion. Radiographic opacity appearing jecting into bronchiole and contain-

only at time of haemoptysis ing a branch of a bronchial artery

Mobbs and Pregnant Pregnancy terminated Radiographic opacity during 2nd preg- Nodule of decidua and endometrialPranner by surgical induc- nancy. No cavitation glands, 2-5 cm. across(1963) tion at 20 weeks

Kovarik and Pre-menopausal Gravida 3, para 3 Recurrent pneumothorax occurring Collapsed cysts lined by low cuboidalToll (1966) during first 2 days of menstrual epithelium similar to that in the

period. Began after birth of first endometriotic lesion of the dia-child phragm. The pulmonary lesion is

not in itself diagnostic of endo-i metriosis

Present case Post-menopausal Normal deliveries 35 Radiographic opacity Thin-walled cyst with polypoid projec-and 14 years pre- tionviously

There was no history of cough, haemoptysis, or chestpain. She had had two uncomplicated pregnancies, 35and 14 years previously, and her menopause hadoccurred in 1964 with no subsequent bleeding. Shegave no history of any operation or serious illness.

INVESTIGATION A radiograph of the chest (Fig. I)showed a thin-walled cystic lesion in the right mid-zone with a little irregularity of the lower wall of thecyst. A tomogram showed the lesion to extend fromthe 8 cm. to the 11 cm. cut and to have the appear-ance of a multilocular cyst. The Mantoux test wasnegative at 1/1,000 O.T. Lung function studies werewithin normal limits. The vital capacity was 2-55 1.,F.E.V.1 2-25 1., F.E.V.1/V.C. 88%, and the indirectM.B.C. 83 I./min. The haemoglobin was 13-7 g./100ml. and white cell count 6,400/c.mm. (neutrophils71%, lymphocytes 28%, monocytes 1%). The erythro-cyte sedimentation rate was 21 mm./hour (Wester-gren). No carcinoma cells or acid-fast bacilli werefound in the sputum.On 30 August 1965 a right thoracotomy was

performed and a cystic lesion was removed from theposterior region of the upper lobe.On 12 October 1965 she was referred to a

gynaecologist, who found a fibroid tumour of theuterus.On 3 August 1966 the chest radiograph was normal.

PATHOLOGY

Gross The specimen was a wedge of lung withpleura on one surface. It measured 2-5 x 2-3 cm. and FIG. 1. Tomogram of chest showing a cystic lesion in thecontained a collapsed cyst 2 cm. in diameter. The right mid-zone.

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46Tatiana Jelihovsky and A. F. Grant

FIG. 2. Subpleural endometrialcyst (H. and E. x9).

wall of the cyst was thin, but on the side nearest thepleura a polypoid nodule projected into the lumen.

Microscopic (Figs 2 and 3) The lesion was welldemarcated but not encapsulated. It lay immediatelyunder the pleura, which was slightly thickened byfibrosis. In the polypoid portion of the lesion therewere glandular spaces which varied from small tubulesto large dilated cysts and were lined by dark-staining,columnar epithelium with prominent elongatednuclei and occasional wider cells with pale-stainingcytoplasm and round nuclei. The stroma was very

x

FIG. 3. Part of the polypoidprojection showing thick-walledartery (H. and E. x 50).

cellular. It was more fibrous and contained largerblood vessels than does normal endometrial stroma.This and the structure of the glands gave the lesionthe appearance of an endometrial polyp. At the tipof the polypoid projection there was a small muscularartery with an internal and an external elastic laminaand a prominent intima. In its thinner portion thecyst wall was lined by a single layer of low columnarto flattened epithelial cells resting upon a loose con-nective tissue stroma. Unfortunately, the connexionwith the bronchus was not demonstrated in thesections, although a large bronchiole was seen imme-diately outside the cyst wall.

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DISCUSSION

This case resembles that described by Sturzenegger(1960) in that the patient was post-menopausal andgave no history of haemoptysis. The absence ofhaemoptysis in our patient is surprising in view ofthe large number of vessels and the prominentartery lying immediately under the surface of thepolypoid projection.The theories of pathogenesis have been covered

in several previous papers (Hartz, 1956; Lattes,Shepard, Tovell, and Wylie, 1956; Mobbs andPfanner, 1963; Park, 1954). Although pleuralendometriosis can be explained by coelomic meta-plasia or spread through pleuroperitoneal sinuses,a similar explanation for endometriosis within thelung is more difficult to accept, especially in thepresence of a circulation which is known totransport other fragmentary material such astrophoblast. The possibility of vascular spread hasbeen shown experimentally in rabbits by Hobbsand Bortnick (1940) but is more difficult to provein the human. At least two of the cases listed inTable I (Dr. Godman's case reported by Latteset al. (1956) and Cameron and Park's first case(1965) ) showed decidual tissue in blood vessels, andin none of these was the decidual tissue in contactwith the pleura. It is likely that the lesions wereembolic in origin. In the second group, on theother hand, most of the lesions were directlybeneath the pleura. The only exception is the casedescribed by Lattes et al. (1956) in which theproximity of the lesion to the pleura was notmentioned, although the illustration of the grossspecimen suggests that it may have been. Theproximity of the lesions to the pleura makes itdifficult to exclude a pleural origin with subsequentgrowth into the lung substance. If both groups oflesions are embolic in origin, why are there glands

in all the larger and probably older lesions (TableII) but not in the small, apparently recent depositsof decidua (Table I) ? In at least three of the casesin the first group (Park, 1954; Cameron and Park,1965), the decidual cells appear to have beengrowing in the lung parenchyma. Whether theywould have persisted after the pregnancy is notknown. Neither is it known whether the glandsseen in the second group had been transported asglands or whether they appeared later by meta-plasia and if so from which cells. Most of thepatients in the second group had had previouspregnancies (no history is given in Sturzenegger'scase (1960)). If their lung lesions had resultedfrom metastases during those pregnancies, itwould be interesting to know whether cells otherthan differentiated decidual cells were present inthe emboli.

We are very grateful to Dr. V. J. McGovern,Director of Fairfax Institute of Pathology, RoyalPrince Alfred Hospital, for his help in the preparationof this paper.

REFERENCES

Cameron, H. M., and Park, W. W. (1965). Decidual tissue within thelung. J. Obstet. Gynaec. Brit. Cwlth, 72, 748.

Hartz, P. H. (1956). Occurrence of decidua-like tissue in the lung.Report of a case. Amer. J. clin. Path., 26, 48.

Hobbs, J. E., and Bortnick, A. R. (1940). Endometriosis of the lung;an experimental and clinical study. Amer. J. Obstet. Gynec., 40,832.

Kovarik, J. L., and Toll, G. D. (1966). Thoracic endometriosis withrecurrent spontaneous pneumothorax. J. Amer. med. Ass.. 196,595.

Lattes, R., Shepard, F., Tovell, H., and Wylie, R. (1956). A clinicaland pathologic study of endometriosis of the lung. Surg. Gynec.Obstet., 103, 552.

Mobbs, G. A., and Pfanner, D. W. (1963). Endometriosis of the lung.Lancet, 1, 472.

Park, W. W. (1954). The occurrence of decidual tissue within thelung: report of a case. J. Path. Bact., 67, 563.

Rodman, M. H., and Jones, C. W. (1962). Catamenial hemoptysisdue to bronchial endometriosis. New Engl. J. Med., 266, 805.

Sturzenegger, H. (1960). Lungenendometriose unter dem Bild desRundschattens. Schweiz. Z. Tuberk., 17, 259.

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