CLINICAL SECTION

SUPERIOR MEDIASTINAL TERATO-DERMOIDSA Report of Two Cases

By A. C. BREWER, F.R.C.S.Hon. Assistant Surgeon, Liverpool Royal Infirmary

andR. MARCUS, CH.M., F.R.C.S.

Hon. Surgical Clinical Assistant, Liverpool Royal Infirmary

Mediastinal terato-dermoids can be classifiedinto three different types, namely dermoid cysts,teratomata and a complicated pathological groupwhich does not fit into either of the first twogroups.The first example of a terato-dermoid of the

mediastinum was reported by J. A. Gordon inI823. Kerr and Warfield (1928) computed thetotal number of recorded cases at I38, but it isdoubtful if all the reported cases were in realitygenuine examples of intrathoracic terato-dermoids.Rusby (I944) critically analyzed the literature upto 1939 and estimated that 245 of the cases wereterato-dermoids and reported six further cases.In I945 Laipply independently arrived at the sametotal and added one case of his own. Since thisdate there have been several further cases reportedin the literature.

Terato-dermoids are usually situated in theanterior mediastinum. These tumours may belocated in orie or other side of the thorax.

It appears that the first successful operation forremoval of a mediastinal terato-dermoid was bySenn (1905). Since then several successful caseshave been reported, the most recent report, offour cases, being by Bradford, Mahon and Grow(1947)-

It has been possible to collect from the literature12 instances in which the first clue to the presenceof an intrathoracic terato-dermoid was furnishedby the appearance of a visible swelling in the neck.In at least three other cases, Gordon (1827),Spath (1836) and Poehm (1871), such a tumourbecame obvious later in the course of the diseasewhen other manifestations had brought the patientunder observation. The report of two furthercases which presented themselves with swelling inthe neck is of interest as they throw some light onthe aetiology of the condition.

Case iW.H., aet. 36, labourer. Complained of an un-

productive cough which commenced in June I946.In July I944 the sternum had been bruised by apiece of shrapnel. He noticed a painless swellingin the right side of the neck in May 1947. Therewas no dyspnoea, haemoptysis, loss of weight orimpairment of appetite.

Physical examination revealed a well-built,healthy man. A fixed mass 2 in. by i in. wassituated deep to the middle of the right stero-mastoid and extended from beneath the anteriormargin of the muscle. It did not pulsate, was notfluctuant, failed to transilluminate and did notmove on deglutition. Pressure on the swellingproduced an irritating cough. There was nolymph node enlargement in the neck or axillae.The trachea was displaced to the left, the rightpupil was slightly smaller than the left, but re-action to light and accommodation was normal.There was slight ptosis of the right upper eyelid.The blood pressure was equal in the two arms,110/70 mm. Hg. The remainder of the physicalexamination revealed nothing of note.

Investigations. W.R.-negative. Blood sedi-mentation rate 3 mm. in one hour (Wintrobe).X-ray showed a rounded shadow in the superiormediastinum extending to the right hilum. Themass was displacing the trachea but not causingany gross oesophageal displacement. There wasslight pulsation.

Operation; June 26, 1947. Under thiopentone,intratracheal gas, oxygen and ether, a collar in-cision was made in the neck one finger's breadthabove the suprasternal notch. The strap muscleswere divided and a cystic swelling was found ex-tending upwards from the superior mediastinumbehind the right lobe of the thyroid as far as theupper pole. Postero-medially it was related closely

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Ma/rch 1952 Clinical Sectiont I85

to the structures of the carotid sheath. It wascompletely encapsulated. The medial side of thecapsule was adherent to the carotid sheath. Theswelling was partly mobilized posteriorly by divid-ing its capsule, but it was not possible to insert afinger behind it into the superior mediastinum. Bya combination of traction on the pedicle from aboveand the index finger hooked in front and below themass it was delivered into the wound. Thevascular pedicle was then divided. ''he specimen(Fig. i) consisted of loose, very oedematous

lining or lymphocytic infiltration. It was con-sidered that this was a branchial cyst.Case 2

L.T., aet. 49. A housewife. Was admitted onAugust 5, I947, complaining of cough and sputumfor 14 months. Her past medical history was un-eventful and the family history contained nothingrelevant. She had no haemoptysis, stridor,hoarseness or dysphagia. Her weight wasstationary and the appetite good.

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areolar tissue containing numerous thin-walledvessels, some of which contained blood. Scatteredthroughout the section were several islands ofosteoid tissue not yet calcified. There was noevidence of thyroid tissue. The tumour re-sembled a benign terato-dermoid.On October 8, I947, the patient was re-admitted

for excision of the original swelling on the rightside of the neck. A vertical incision was made overthe middle third of the anterior border of sterno-mastoid. The swelling was situated deep tosterno-mastoid and extended from the level of thesixth cervical vertebra to the angle of the jaw. Itwas removed except for the portion attached tothe carotid sheath. The patient had an uneventfulconvalescence.

Section consisted of a cyst lined by granulationtissue in which there was no evidence of epithelial

Physical examination revealed a healthy womanwho had telangiectasis on the face and enlargedveins in the neck and right side of the upperthorax. In the region of the right lobe of thethyroid there was a large nodular mass which ex-tended into the superior mediastinum. It did notmove on deglutition and its lower part was hard.The trachea was displaced to the left. The re-mainder of the examination was normal.

Investigations. X-ray of the chest and neck(Figs. 2 and 3) revealed a large soft tissue shadowprojecting down into the mediastinum and morepronounced on the right side. There was de-pression and lateral displacement of the aorta.The trachea showed severe displacement to theleft and considerable narrowing. Hb. was 86 percent., white cells 8,900 per c.mm., neutrophils75 per cent., lymphocytes 21 per cent., monocvtcs

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i86 POSTGRADUATE LMEI)ICAL JOURNAI, March 19552

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FIG. 2.-(Case 2.) X-ray of chest.

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FIc.3.--(Casez.) Bariumswallow; lateralview.

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FIG. 4.--(Case 2.) 'I'he specimen removed.

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March 1952 Clinical Sectiolo IX7

2 per cent. and esoinophils i per cent. Electro-cardiogram was normal.

Operation; August 8, I947. The thyroid was ex-posed through a collar incision and found to benormal. A tense, encapsulated, pulsating swellingwas seen extending up from the superiormediastinum and lying on the pre-vertebralmuscles and cervical spine. The great vessels ofthe neck wexe situated postero-laterally. Thethyroid and trachea were displaced to the left onits postero-superior surface. The pedicle wasdivided and ligated. The capsule was then openedand separated from the smooth surface of the swell-ing which was removed as in the preceding case.The patient had an uneventful convalescence.

The tumour (Fig. 4) in the main part was com-posed of myxomatous fibrous tissue and vasculargranulation tissue. In addition there were manylarge blood-filled sinuses which gave the sectionthe appearance of a cavernous angioma. Scatteredthroughout the section were several islands ofosteoid tissue, not yet calcified. There was noevidence of malignancy.DiscussionThe pathogenesis of mediastinal terato-dermoids

has been reviewed by Rusby, who concludes thateach theory has its shortcomings. None fits all thefacts and the final elucidation of this problem mustbe awaited. The monogerminal theory is morereasonable than the bigerminal. The explanationpossibly lies in an abnormality of the third andfourth branchial arches, the abnormal cells beingcarried into the thorax by the normal descent ofthe heart and the great vessels. Cases i and 2appear to corroborate this view in being locatedpartly in the neck. The association of a branchialcyst and terato-dermoid in Case i appears to besignificant, as branchial cysts probably take originfrom abnormal remnants of the cervical sinus.The relationship between trauma and tumour isstill sub judice.The diagnosis of terato-dermoids from saccular

aneurysm, retro-sternal goitre and thymoma is notalways easy. Terato-dermoids may pulsate becauseof their close proximity to the heart and greatervessels. Sometimes they are extremely vascular.

Burvill-Holmes (1934), discussing this subject,attaches little importance to pulsation, and re-marks that in his experience pulsation of ananeurysmal sac is the exception rather than therule. The fluoroscopic visualization of the wholelength of the aorta, the clinical findings in the heartand the serological results of the blood examina-tion are useful aids in helping to establish thediagnosis.A retro-steinal goitre may so resemble a terato-

dermoid as to render differential diagnosis in-definite until operation. The characteristic wedge-shaped opacity on X-ray is not always recognizable,and the elevation on swallowing is not always de-tected nor is it pathognomonic of substernalgoitres. Calcification occurs in both and is nohelp in the diagnosis. Tracer studies with radio-active iodine will show the presence of a retro-sternal goitre provided it has not taken on malig-nant changes or been completely replaced by cysticdegeneration (Ansell and Rotblat, I948; Marcus,1950).Thymic tumours (Reid and Marcus, 1949) are

suspected when there are symptoms of myastheniagravis. Here a skiagram would show a shadowwhich, in the antero-posterior view, occupies themediastinum to one or other side of the heartshadow and on a lateral view occupies the superiormediastinum above the heart.The importance of terato-dermoids depends on

the symptoms they produce from pressure and onthe complications to which they are liable. Theymay, however, be symptomless and only be dis-covered on mass radiography or at post-mortem.Pressure symptoms resemble those of anymediastinal tumour. They are liable to secondaryinfection and may develop adhesions to neighbour-ing structures. Rupture may occur into abronchus, a blood vessel or the pleura. Finally,degeneration and malignancy may supervene.For these reasons attempt at removal in suitablecases should always be contemplated.SummaryTwo cases of superior mediastinal terato-

dermoids are reported. In one case the associatedpresence of a branchial cyst is of particular interest.

BIBLIOGRAPHY

ANSELL, G., and ROTBLAT, J. (1948), Brit. J. Rad., 21, 552.BRADFORD, M. L., MAHON, H. W., and GROW, J. B. (I947),

Surg. Gynaec. & Obst., 85, 467.BURVILL-HOLMES, E. (I934), Radiology, 23, 449.GORDON, J. A. (1827), Med. Chir. Clin. Trans., 13, I2.KERR, I. H., and WARFIELD, J. 0., junr. (1928), Ann. Sirg.,

88, 607.

LAIPPILY, T. C. (1945), Arclt. Path., 39, 153.MARCUS, R. (I95o), paper read at the Liverpool Medical In-

stitution on 'Tracer and Therapeutic Studies with Radio-Isotopes,' 30,/I1/50.

POEHM, H. (i871), Inaug. Dis.REID, H., and MARCUS, R. (1949), Brit. J. Surg., 36, 271.RUSBY, W L. (1944), J. Thorac. S¥urg., 13, 169.

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