ON CYSTS OF THE PREPUCE AND RAPHE, WITH AN ILLUSTRATIVE CASE*

By GEO. HENRY EDINGTON, M.D., M.R.C.S. Eng., Surgeon to the Central Dispensary, and Extra Surgeon to the Dispensary of the Western Infirmary, Glasgow.

Cysts of the prepuce receive but scant notice in the general text-books of surgery, and it is partly on this account that I now record the following case. I have been led, however, to do so also from the fact that recently cysts in this region have been engaging the minds of some writers in connection with their probable origin in a congenital abnormality. I shall first notice a case which has come under my own observation, and then refer to the literature of the subject. Willie D., aged 1 year, was brought to the Central Dispensary on 27th October, 1897, on account of his being the subject of " phimosis, accompanied by a small lump" at the distal extremity of the prepuce. This lump was first observed when he was 3 months old, and it increased in size till he reached the age of 6 months, since when no alteration in dimension had been noticed. It was stated, however, that, after ceasing to enlarge, the swelling had become harder than when first noticed. The prepuce (Fig. 1, p. 423), which was long, presented on inspection a spherical swelling on the under aspect of the free margin, with the antero-posterior vertical meridian corres- * Paper read and specimen shown at the meeting of the Glasgow Pathological and Clinical Society, 11th April, 1898. Dr. Edington?Cysts of the Prepuce and Baphe. 423 ponding to the raphe. The skin over the upper surface of the swelling was apparently much thinned, and there was some translucency apparent, although this was not tested with artificial light. On palpation there was some tenseness, but the impression conveyed was that we were dealing with a cyst, probably sebaceous, and not a solid tumour. The growth was purely preputial in its connections, and moved freely with

Fig. 1. natural size. Parts removed by circumcision ; natural size. the , being without attachment to the glans. The preputial orifice was contracted so that the glans could not be uncovered, but the cyst took no part in preventing retraction of the foreskin. The child was otherwise normally formed, but was small for its age. Circumcision was performed on account of the phimosis, and the part removed was placed to harden in spirit, the swelling being left unopened. After hardening, the specimen was opened up along the raph^, and on opening into the swelling

Fiq.Fig. 2. of material shown on Specimen opened up from below in line of raph?.raph6. Irregular deposit of fatty material shown on raw subcutaneous surface of the surface of cyst wall. Behind the cyst is the raw subcutaneous surface of the prepuce, also a fringe of the mucous layer.

it was found to be a cyst, situated in the skin portion of the prepuce and abutting behind and slightly above on the under surface of the mucous portion (Fig. 2). The diameter of the cyst was *8 cm. The cavity was empty in great part, and on the inner wall was a somewhat irregular deposit of fatty material. This presented an uneven surface, the deposit being thicker in some parts than in others, and where the wall of the cyst was visible it had a smooth, almost glistening, appearance. The 424 Dr. Edington?Cysts of the Prejpnce and RapM. emptiness of the cyst was due evidently to absorption of the fluid part of the contents during the hardening process. The solid contents were examined microscopically, and were seen to consist of squamous epithelial cells and a few leucocytes, all more or less showing fat-droplets in their substance, also free fat-droplets. The epithelial nuclei were easily stained with carmalum, but the leucocytes did not take up the colouring matter. No crystals were observed.

Fig. 3. Portion of cyst wall, from proximal part of specimen, showing (a) loose subcutaneous tissue firmer of of with large vessels ; (ft)(b) firmer layer of connective tissue forming part of wall; (c) epithelial of detritus in of a lining cyst; (d) detritus in cyst cavity (desquamated layers of epithelium). (From a micro-photograph taken by Mr. Archibald Young, M.B.)

A portion of the cyst was embedded in paraffin, and sections were cut comprising the complete circumference in the antero- posterior vertical direction. They were stained in the following, different ways :?(a) with acid hsemalum; (b) carmalum and picric alcohol; and (c) by Gram's method. After staining, the sections were cleared with xylol, and mounted in Canada balsam. Microscopic examination.?With Zeiss objective AA and No. 3 eyepiece, the cyst was seen to possess a lining mejnbranp, Dr. Edington?Cysts of the Prepuce and Rapht. 425 separated from the preputial epidermis by a layer?varying in thickness and being much thinned out at the distal portion of the cyst?of fibrous tissue, corresponding to the dermis. No layer of true subcutaneous tissue was present, save at proximal part, i.e., between cyst and meatus, and here there were many vessels in the large loose fibrous tissue (Fig. 3, p. 424). The lining membrane was very distinctly stained, and its appear- ance suggested an epithelial structure. With D objective, the lining membrane was seen to consist of several layers of flattened epithelium. Externally the stained nuclei were seen closely packed, but as one passed towards the cyst cavity they became much fewer, and the superficial layers (next the cyst cavity) assumed a homogeneous yellowish appearance, con- trasting with the deeply-stained stratum corneam of the epidermis. The nuclei were elliptical and were arranged with long axis parallel to the surface of the wall. On the surface of the cyst wall was seen fatty debris peeling off in layers. The wall presented no prominences, and no papillae were present in the surrounding fibrous tissue. The above description is from a specimen stained by hsemalum alone; the carmalum and picric alcohol showed innermost layers of cyst wall as a deep yellow homogeneous structure. Examination of section stained by Gram's method demonstrated the presence of eleidin-holding cells. These cells were irregularly situated, and did not form a continuous stratum. Literature. One finds little mention made of the condition in the general surgical text-books. Erichsen1 states that "sebaceous cysts are occasionally met wTith, usually on the under surface of the are rare have or prepuce. Dermoid cysts very ; they been found in the raphe, on the under surface of the penis, which the two halves unite to close the ." along " Henry Morris2 writes, Sebaceous cysts occasionally occur

. . . with the in the prepuce. Cysts connected glands behind the corona have been met with, sometimes single and in as large as a hen's egg; sometimes multiple and varying size from a shot to a horse-bean." In Heath's Dictionary, Jacobson3 mentions sebaceous tumours " as being occasionally met with on the penis. On looking into works dealing specially with affections of are the the genito-urinary organs, references made to subject under notice somewhat as follows:? Jacobson4 mentions that sebaceous cysts are occasionally 426 Dr. Edington?Cysts of the Prepuce and Raphe. met with in the long prepuce of boys. The duct of a sebaceous follicle may readily become blocked owing to irritation accom- panying a phimosis. He removed two by circumcision, each on the under surface of the penis, with the raph^ running over it. One was the size of an olive, the other of a small pea. He also quotes Cruveilhier5 as to the occurrence of cysts developed from the modified sebaceous glands behind the corona. Podrazki6 simply remarks that new formations occur in the prepuce not at all seldom; that sebaceous cysts are to be observed fairly often, singly or in groups, pedunculated or sessile. Kocher7 describes cysts with fluid contents, and also athero- matous cysts. He considers both forms to be regarded as retention cysts, and mentions the raphd as being their common situation. Kaufmann8 states that atheromatous cysts are more common in connection with Tyson's glands than with those of skin of penis. The smaller cysts are usually multiple;. the larger, solitary. He mentions four cases of large cysts, one of them observed by himself. Bland Sutton9 mentions that sebaceous cysts "are not uncommon in the skin of the penis and ." On turning up the recorded cases, one finds that the cystic formations in this region may be divided into four groups:? 1. Sebaceous. 2. Mucous. 3. Congenital (this last may include the former two). 4. Traumatic epithelial (implantation). 1. Sebaceous arise in connection with two sets of glandular structures?(a) those on the inner aspect of the prepuce (Tyson's), and (b) those on the outer aspect. (a) From Tysons glands.?Instances of these are reported by Cruveilhier,5 who mentions a cyst the size of a small hen egg being found in a cadaver. The cyst lay between the mucous and cutaneous portions of the prepuce, and, in his opinion, originated in one of the follicles on the inner surface of the foreskin. Dunlop10 reports a case in which several small nut-like bodies occurred on the corona. These bodies were hard and to the touch. On pressure, one of them discharged pus. horny" Growths" had been removed two years before, and then reappeared. On removing the recurrent nodules they were found to be cysts containing sebaceous matter undergoing change. Fano11 mentions the occurrence of a cyst in a case of Dr. Edington?Cysts of the Prepuce and Raphe. 427

in a phimosis child aged 3 years. After the skin was removed by circumcision, the mucous layer of prepuce was reflected from glans on the right side without difficulty, but on the left it was adherent, and on being forcibly raised a sebaceous cyst was found between it and the glans. The cyst had circumscribed smooth walls.* (b) Those ai'ising in connection with the glands on the outer aspect of the prepuce.?Fano11 reports a case of a child, set. years, with four small cysts on prepuce. H. J. " Bigelow12 records the removal of a wen of the prepuce." The tumour was congenital, had increased slowly, and was the size and shape of an acorn. It was fluctuant. Particulars as to the relation of the parts not given. W. Fairlie Clarke's case13 was that of a tumour, the size of a walnut, which hung in a pendulous manner from the free border of the prepuce. The patient was aged 39, was married, and the father of one child. The tumour had been present ever since he could remember, and had recently been growing rapidly. It con- tained semifluid substance of a fawn colour, which on micro- scopic examination was found to be composed of cholesterine plates, oil globules, and granular matter. The patient had also a large sebaceous tumour on the scalp. Mettenheimer14 mentions an appearance as of milium at the preputial orifice. He also instances a case of a pedunculated sebaceous cyst on the under surface of the prepuce of a child aged 7f years, and looks on the condition as of uncommon occurrence in a child. The cyst was observed at or a few days after birth as a small lump, which had since grown. The prepuce was long, but could be retracted easily. Griffon and Segall15 report a case of cyst on the upper aspect of the penis, from a lad 18 years of age. Present since early in infancy. The lining membrane consisted of several layers of stratified epithelium. On surface were flat cells, some with, others without, nuclei, and projecting into cavity of cyst as scales; the next layer was composed of cells corresponding to mucous layer of Malpighi, and the next of cylindrical epithelium. Brownish pigment infiltrated middle In no w ere there and deep layers. part glands ^ The authors incline to the belief that the case is one of dermoid cyst. 2. Mucous cysts are mentioned by Morgan16 as being com- These parable to similar formations occurring on the lips. * I think that it is questionable whether this was a true cyst or merely in a case of adherent a collection of sebaceous matter occurring prepuce. 428 Dr. Edington?Cysts of the Prepuce and Raphe.

are apparently not so commonly met with as sebaceous cysts, as, in a paper by Mermet,17 only four occur in a list of twenty, fourteen being sebaceous, and two fistulous, cysts. They have mucous contents, and are lined with epithelium which approxi- mates to the mucous type. One case of Redard's18 showed cylindrical cells lining the cyst cavity. 3. Congenital cysts of the genito-perineal raphe.?These arise in connection with the embryological development of the parts, and the following sketch of the development, summarised from Mermet's paper, may serve to explain their genesis:? At the end of the first month of intra-uterine life, the genito-urinary and intestinal tracts open at the hinder end of the embryo by a common cloaca. Towards the middle of the second month, the separation of the genito-urinary from the alimentary tract is brought about either by the spur (eperon) between allantois and hindgut growing downwards (eper on perineal) or by the ingrowth of two lateral folds (])lis cloacaux). As a result, the cloaca is subdivided into an anterior uro-genital and a posterior rectal cavity, with corres- ponding openings on the surface. In the course of the third month secondary folds are developed bounding these two. orifices, and in front of the anus they unite in the middle line to form the . From the sixth week an eminence (genital tubercle) is situated in front of the uro-genital opening, and grooved on its under surface by the urethral furrow. A raised fold (bourrelet genital) passes backwards from the eminence and surrounds the uro-genital opening. The edges of the furrow (urethral folds) approach one another towards the end of the third month, and by their coalescence complete the furrow to form the urethral tube. The junction is indicated externally by the raphe, and "congenital cysts" may be formed from the vestigial epithelium at the line of junction of the folds. The coming together of the lateral portions of the bourrelet forms the scrotum, with the scrotal raphe indicating the line of junction. The epithelium lining the urethral tube assumes mucous character, and, according to the situation of the vestigial epithelium?deep, near the urethra, or superficial, near the skin?you may have mucous or dermoid cysts formed. Besides cysts of globular form, one sometimes meets with long closed tubular cavities running parallel to the urethra and raph^, and sometimes described as urethral pouches, and giving rise to the condition known as double urethra. These are often of moniliform type. It follows from the above that the number of cysts present Dr. Edington?Cijsts of the Prepuce and Raphe. 429

may vary. More have, a and when " usually you single cyst, multiple" cysts are said to exist they are, as a rule, but subdivisions of a moniliform cavity. Of necessity their position corresponds with the middle line. The wall may be irregular, but is most often smooth and of whitish colour. The mucous cysts especially present an irregular surface on the inner wall. The character of the contents depends, of course, on the nature of the lining cells of the cyst (dermoid or mucoid). Sebaceous contents do not show hairs, teeth, &c., but cholesterine plates are present in more or less abundance. The sebaceous matter may undergo liquefaction, and this is instanced, according to Mermet (loc. cit., p. 397), in an observation Bauchet " by 'entitled Mucous Cyst in a Sebaceous Follicle." " During the present year, Thole,19 in a paper on Congenital Cysts of the Genito-PerimBl Raphe," relates a case, and also reviews the work of former observers. He founds the assumption of the congenital origin of a cyst on its site and microscopic characters, and not on its alleged time of appear- ance. If the cyst can be explained only on anomalies of development, then it is congenital (loc. cit., p. 448). He lays stress on the relation of the cyst to the raphe. His own case showed the cyst to lie somewhat to the right of the middle line, but this was associated with a similar deviation of the raphe. The cyst was moniliform, and occurred in a case of slight hypospadias. Tranverse serial sections showed the upper wall of the cyst to have a cylindrical epithelial lining, that of the lower being formed by flattened epithelium, which in places had a direct connection with the epidermis in the middle line. The lower wall showed epidermal pigment; this was absent from the upper wall, as also was cornification. The cylindrical cells were derived from those of urethra; the The was a squamous from those of epidermis. hypospadias further evidence of developmental error. A case reported by Cestan20 may be mentioned here. This was a mucous cyst of glans on the right lip of the meatus. The author asks if it should not be classed among the con- genital cysts. Shattock,21 in a recent number of the Journal of Pathology and Bacteriology, descants on the significance of eleidin- containing cells in a cyst wall. He demonstrates their presence by Gram's method of staining, and he declares that they confirm the origin of the cyst from epidermis, as they are in the stratum He states that these contained granulosum. " in cells line the hair-follicles to their deepest limits; hence, 430 Dr. Edington?Cysts of tite Prepuce and Raphe. sebaceous cysts ... a stratum granulosum is found" (loc. cit., p. 127). He mentions the case of a cyst in the neck with a lining of stratified squamous epithelium, and containing clear flattened epithelial cells, fatty leucocytes, and a little free fat. There was no stratum granulosum present, and he therefore classified the cyst as mucosal. This contrasts with Mermet's description of the epithelial lining of a dermoid cyst as having a thin stratum mucosum, stratum granulosum furnished with eleidin, and a horny layer whose most superficial cells desquamate to form a debris in cyst cavity (loc. cit^ p. 402). 4. Traumatic epithelial cysts.?Thole quote Trzebicky's two cases of cyst occurring in Jews, and lying to left of middle line. Trzebicky suggests a causal relation between their formation and the ritual circumcision, in which case they would fall to be classified as traumatic or implantation epithelial cysts. P^reire22 reports a case of sebaceous cyst of the prepuce, which had been present since birth, in a subject aged 40 years. The cyst was situated on the inferior aspect of the prepuce, and was covered by the expanded frsenum. The patient wished the growth removed, as it interfered with the sexual act. Pereire insists on the rarity of these tumours. Micro- scopic examination was made by Pilliet,23 and showed the cyst to be lined by several layers of epithelium,the deeper polyhedral, while the more superficial cells were flattened, without corneous transformation, and preserving their nuclei at the same time that the cytoplasm was charged with fat. Their desquamation formed the sebaceous contents of the cyst. In no part were crypts or Tyson's glands observed. There was a round-cell exudation in the substance of the dermic papillae, and, on account of this, Pilliet suggests the theory of the cyst being formed by traumatic epithelial inclusion.

Note.?I saw my patient on 27th March, 1898, and observed a small bud on the left lip of the meatus. It seemed solid, and was covered by the epithelium of the glans. It may have been present originally, but was not noticed at the time of the operation (cf. supra, Cestan).

Conclusions.

I have come to the following conclusions with regard to the case detailed at the commencement of this paper :? Dr. Edington?Cysts of the Prepuce and Raphe. 431

1. The flattened layers of epithelium, small quantity of solid contents, and absence of cholesterine crystals are quite in keeping with the theory of the cyst being of mucosal origin. This view is supported by the translucency in the fresh state. 2. The growing scantiness of stained nuclei in the superficial layers of epithelium of the cyst wall leans more to the view that the cyst is one of the sebaceous class, and this is determined by the presence of a stratum granulosum as shown by the eleidin-liolding cells. The paucity of solid contents is parallelled by Bauchet's case, quoted by Mermet (loc. cit., Obs. iv). 3. The relation of the cyst to the raphe is suggestive of a congenital origin. 4. There is no evidence of its being a traumatic epithelial (inplantation) cyst.

REFERENCES.

1 Erichsen, Science and Art of Surgery, tenth edition, 1895, vol. ii, p. 1225. 2 Morris, in Treves' System of Surgery, 1896, vol. ii, p. 1014. 3 Heath, Dictionary of Practical Surgery, 1886, vol. ii, p. 184. 4 Jacobson, Diseases of the Male Organs of Generation, 1893, p. 702. 6 Cruveilhier, Traite d'Anat. Path. Generate, Paris, 1856, vol. iii, p. 334. 6 Podrazki, Chirurgie, v. Pitha u. Billroth, Bd. iii, 2 abth., 8 Lfg., p. 29. 7 Kocher, Deutsche Chirurgie, Billroth u. Luecke, Lfg. 50b, p. 28. 8 Kaufmann, Deutsche Chirurgie, Billroth u. Luecke, Lfg. 50a, 1886, p. 259. 9 Bland Sutton, Tumours, Innocent and Malignant, London, 1893, p. 235. 10 Dunlop, Medical Press and Circular, vol. ii, 1882, p. 372. 11 Fa no, Gaz. des Hopitaux, 1867, p. 488. 12 Bigelow, Boston Medical and Surgical Journal, 1868, vol. 78, N. S. vol. i, p. 103. 13 W. F. Clarke, Transactions of the Pathological Society, London, 1868-9, vol. xx, p. 247. 14 Mettenheimer, Jahrbuch fiir Kinderheilk. u. Phys. Erziehg., N.F. Bd. 37, Leipsig, 1894. 15 Griffon et Segall, Bidl. de la Soc. Anat., Paris, 5me serie, tome xi, 1897, p. 536. ? 16 Morgan, Medical Times and Gazette, 1885, vol. ii, p. 43. 17 Mermet, Revue de Chirurgie, Paris, 1895, vol. xv. 18 Redard, Revue Mens, des'Mai. de VEnfance, Paris, 1890, p. 115. 19 Thole, Bruns Beitrage zur Klin. Chir., Bd. 20, Heft, Tubingen,

1898- , ? ? 20 Cestan, Bull, de la Soc. Anat., Paris, 5?e s^rie, tome xi, 1897, p. 126. 21 Shattock, Journal of Pathology and Bacteriology, London, 1898, vol. v,

p. 120. , . . 22 tome 317. Pereire, Bull, de la Soc. Anat., Paris, 5? s6rie, xi, 1897, p. 423. 23 Pilliet, Bull, de la Soc. Anat., Paris, 5?e sdrie, tome xi, 1897, p.