Internal endometriosis (adenomyosis) of the urinary bladder

INTERNAL

ENDOMETRIOSIS URINARY

(ADENOMYOSIS) BLADDER

MARTIN L. DRETFUSS, M.D.," (From

the

Departmen?

of

OF’ THE

NEW YORK, N. Y.

Laboratories

of

Beth

Israel

Hospital)

TARR JUDD, in 1921, described the first ease of endometriosis of the bladder. s Ottow, in 1927, succeeded in making the first correct preoperative diagnosis In the same year, Plaut made the diagnosis in the operating by cystoscopy. Since that time, the number of room and confirmed it by rapid frozen section. cases reported has increased slowly but steadily. After v. Mikulicz’ survey in 1936, further cases have been described by Perlmann (1934), Weijtlandt (1934), Erle (1935), Chauvin (19361, Mark (19:37), Adams, Stoeckel, Homma (1938), and Reynolds (1939). The clinical picture of endometriosis of the bladder is well defined today. The The case which I am same cannot be said for its histogenesis and pathogen&s. reporting offers, in my opinion, several interesting features which may help to clarify some mooted points, particularly concerning histogenesis. CASE.+--C. B., a Z&year-old female, was admitted to Beth Israel Hospital with a diagnosis of acute cystitis on Oct. 26, 1938. (Admission No. 106153.) Her chief complaint was suprapubic pain on urination. Menstrual history: 14 x 3-T ‘r 1’3. The patient had considerable dysmenorrhea, two days before and on the first day of menstruation. She had never missed a period. The flow was moderate. The patient was unmarried and had never been pregnant. One and one-half years ago the patient was operated upon at another hospital. At that time a simple ovarian cyst was removed together with one tube and the vermiform appendix. There were no unusual findings. Three days before admission, simultaneously with the onset of her menses, the patient noticed a sharp suprapubic pain and burning on urination. There was also an increase in frequency. No pain in back, no fever, no gross hematuria were noticed. These symptoms had been occurring off and on for the past year, their onset coinciding with that of the menses although they often lasted longer than the menstrual period. Vaginally, a firm tumor on the right side of the uterus was felt anteriorly. Otherwise the clinical examination was negative.

Urine.-(Catheter specimen.) Specific gravity 1024, acid, bloody, albumin l-plus, white blood count S-10 per high power field, a few squamous epithelial cells. Blood Count.-3.89 million red hlood cells, 8,000 white blood cells. Slight anisocytosis. IntmTenous Urography.-There was a large filling defect in the basal portion of the right side of the bladder. The filling defect was also seen in the cystogram. Cystoscopy.-There was a bulging tumor on the right side extending from the sphincter margin backward to the posterior wall. The mucosa was not ulcerated. The tumor was the size of a quarter coin. Biopsy.-Inflamed bladder mucosa with large epithelial nests of Brunn. Preoperative Diag?&osis.-Neoplasm. The pathologist (Dr. A. Plaut) on the basis of the history and cystoscopia findings, mentioned the possibility of an endometriosis of the bladder. OperaGo%-(Dr. 8. Wilhelm,) The peritoneum was opened, no implants were found. The bladder was opened and the tumor excised. It was round, elastic and situated in the right lateral wall, extending from the sphincter margin backwards

'Dora Paul Cancer tThanks

are

clue

Dr.

Research E. A.

Fellow. Horowitz

for

336

permission

to

use

the

clinical

data.

DR.EYFUSS

:

INTERNAL

337

ENDOMETRIOSIS

to about 1.5 cm. from the right ureteral orifice. It was covered by a thickened mucosa. The trigone was normal. Gross Specimen.-Ellipsoid specimen 3.5 by 3.5 by 2.5. Part of the surface On the surface a square of 3 cm. was formed by deep red bladder appeared charred. mucosa in which three dark brownish red spots, about 6 mm. in diameter, were On bisecting, thin brownish fluid, suggestive of altered blood, came situated. out. The cut surface looked grayish, with streaked markings. There were, notably near the periphery, a few small indistinct cavities from which the fluid exuded.

Microscopic Description.-The surface, one slice 1 mm. thick The stains slide was stained. mucicarmin.

Fig. Figs.

structures

1 to 4.-The within the

Fig. l.-The stroma cylindrical. Fig. 2.-The stratifled single layered cylindrical what altered by edema.

tumor was bisected at right angle to the bladder cut in serial sections, the rest at levels. Every tenth used were hematoxylin-eosin, v. Gieson, elastica and

1.

Fig.

photomicrographs bladder wall. has

the

epithelium epithelium

represent

characteristics

small of

of the bladder of the gland.

portions

cytogenic

tissue.

surface The

aspect

2,

of the The

endometrioid epithelium

is continuous of the stroma

is

with the is some-

The bladder epithelium extended with deep folds into the submucous layer. It was somewhat thickened. Many nests of Brunn were seen. Some of them, when followed in serial sections, appeared separated entirely from the surface epithelium. Others were continuous with it and represented the deepest parts of the folds. They were in part solid with occasional intraepithelial vacuoles, in part they had a lumen and were somewhat glandlike with their inner epithelial layer formed by cylindrical cells. The mucin reaction in these cells was negatiye. No secretion was found in the lumina. &Some of them communicated with the lumen of the bladder, others had no demonstrable opening. The submucous layer was highly edematous and inflamed. In one area the surface epithelium was entirely missing (site of biopsy). The muscle coat was thick, but otherwise not remarkable. There was no evidence of new formation of muscle. The edema and inflammation extended into the internal muscle layer. In the subserosa occasional inconspicuous accumulations of lymphocytes were seen. The serosal cells were poorly preserved.

‘I’lle most important, findings were numerous glandular atlueturcs WI1 ich occupirll Thry were present ~1~0 in 1.11~ the submucosa and branched tovxctl the muscle coat. Somhere did thrbu reac.11 muxle, more in the internal than in the rskrnal layers. They were linecl ty a single layer of colunrnar, partlyc:ili:~tvrl. the subserosa. membrane. some of the epithelium. The epithelium rested on :I basement The epithelium OF thesr I*gsticz glands W:L~ flat glandular structures were cystif,. The lumen often contained well-preserved, or cuboidal, in some places cylindrical. 111 :, few. or degenerated erythracytea, cellular detritus, and amorphous matter. large accumulations of polymorphonuclear leucocytes were present. Some 11f 111~ Only traces of cytogenic tissue vould larger glands had papillary protrusions. be found around the glandular structures in the submucosa, while it was more Brownish. granular pigmr~r~t W:LL abundant around the glands in t.ho muscle coat. ‘I’llt~lYt situated near the glands, lloth between and within connective tissue cells. was no new formation of mus(~le, nor was the fibrous tisnle increased.

Big. 3 Fig. -1. Fig. 3.-The transitional epithcliuml of the Flad&r IIILIC~S:I is continuous with rhal,act&&c mutinous epitheliutn. Fig. 4.-Ciliated epithelium anti nlucinous epitheliuru touching exch other in one 01 the endometrioid glands.

In one area, in the center of the lesion, the surface epithelium reached particularly far into the submucosa, forming thus deep crypts, and the nest,s of Brunn were numerous and large. Here the glands opened into thtb crypts 01 into the cystic nests. The transitional surface epithelium was continuous with the columnar epithelium of the glands. The transition between the two types was rather sudden. In one ramification, the transitional epithelium rhanged into characteristic mucous epithelium which gave a positive intracellular mucicarmin stain. In another ramification; near the muscle coat and away from the surface, the continuity of the cylindrical epithelium was interrupted by the presence of of mucous epithelium for a short distance. Diagnosis.-Internal endometrioais of the urinary bladder. COMMENT R.

Meyer

1. Internal,

has

distinguished

originating

three probably

types from

of bladder

endometriosis epithelium.

of

the

bladder:

2. External,

originating

3. Collision,

resulting

from from

serosal

cells,

the combination

of 1 and 2.

It is evident from the literature that most of the cases belong to Group 2. Rclgardless of the mechanism involved, whether there be implantation (Sampson), penetration (Haselhorst). or metaplasia, all these cases have one principal characteristic in common: the bladder is invaded from without by the endometrioid structures. Therefore, these cases must be considered as external endometriosis of the bladder. Regarding internal endometriosis of the bladder (Group l), the various authors are not in accord as to the criteria for establishing such an entity. Erle lists 21 cases out of 30 as “primaryl:’ Weijtlandt 3 out of 35. According to Stoeckel there are no cases of true internal endometriosis of the bladder at all. Mark expresses himself in similar fashion. Adams considers as L‘primary” only those cases in which there is no continuity with the sex organ and no peritoneal involvement, and in which previous operations have not caused a trauma to the bladder. The fact that, in many cases of “primary” endometriosis, the process starts eviclently from without detracts from the usefulness of this term. It is much more accurate to continue the distinction of external and internal endometriosis. The latter term then would include those cases in which the origin can be traced to the bladder epithelium. Tt must, however, be kept in mind that the endometrioid structures may penetrate up to the bladder epithelium in cases of external endometriosis as well. The direction in which the glands ramify may then be helpful for a correct interpretation. It seems surprising at first that endometrioid structures should arise in the bladder euithelium. Some authors as Erie, Oehleeker, Mueller and Frommolt thought that undifferentiated coelomic cells, or some other dysontogenetic rests in the bladder, gave rise to the endometriosis. The bladder, in fact, in the embryo, is lined originally by a single layer of cylindrical epithelium. This epithelium assumes its definite appearance only in embryos of 55 to 60 mm. length. On the other hand, the presence of glands in the normal bladder is not generally admitted. Outside the trigone where sometimes aberrant urethral glands may be found, glandular structures are not considered a normal finding, although Moellendorf, Lendorf, and others have described rudimentary glands in the bladder. It is well known, however, that the bladder epithelium may form glands under pathologic conditions. The transformation of the epithelial nests of Brunn into glands has been shown recently by Patch and Rhea (cystitis glandularis). Nests of Brunn and gland formation are usually but not exclusively found in the inflamed bladder (Putschar). Another proof for the potentialities inherent to the bladder epithelium is the formation of mucous epithelium. It has been found in chronic cystitis and particularly in exstrophy of bladder. Mucus-producing adenocarcinoma of the bladder has also been observed occasionally (Hueckel). Homma, Mueller, and Frommolt described mucous epithelium in their cases of endometriosis of the bladder. According to Homma, it is due to a metaplasia of the bladder epithelium. Mueller and Frommolt considered it as evidence of a dysontogenetic origin and thought tfmt it was derived from aberrant rests of the primary gut. True internal endometriosis of the bladder is rare. Only the cases of Mueller (1927, Case l), Frommolt (1929, Case l), Oehlecker (1930) can be considered as true internal endometriosis of the bladder. A probable case of internal endometriosis is that of Erle. There is no microscopic description but the illustration is highly suggestive. In other cases (Weijtlandt and Reynolds) no biopsy was taken and therefore no conclusive judgment is possible. It is only by microscopic evidence that we can decide whether we are dealing with internal or external endometriosis. Pathogenesis : It is evident from the cyclic character of symptoms and the cyclic change in size and appearance which can be observed with the cystoscope, that these structures are under harmonic and especially under ovarian influenre. Phillips descrihd different func+onnI picstnres in two subsequent hiopsies. R. Meyer,

111 llll,st c’:,>,‘s. in one case, observe11 decidual r.e:t~tion in the endometrial stxi’turcs. however, the only ana.tomii> sign whieli c~ould be refcrre~l to ~~yclil~ ~:ll:nlgc~ i? 1 tl!* presence of old and fresh blootl. I’articularly impressive are t.he clinical observations of Mueller who war able 1.0 follow by serial cyatoscopic examinations, the efPect of pregnancy, x-ix!, am! hormone treatment upon the endometriosi~. These facts are definitely iu favor of :r dependence upon harmonic stimuli which most probably are ovarian. But there is no evidence, whatsoever, that endometriosis is caused by t,he action of llormont~a. Endometriosis might very well originate in an entirely different way and still come under hormonal influence secondarily. once cannot ignore the fact that, in many cases, the onset apparently follows mechani~~al trauma as operation, childbirth, curettage or some inflammatory condition in the pelvis or in the abdomen. In the case described by Plaut, a simple appendectom! It is difficult to imagine that this may have caused a only had been performed. hormonie imbalance. Even less r:onceivablr. iu sue11 il I’:w’, is an inlpl:intat.ion 111 endometrium. E‘or the cases of internal endometrioais of t,he urinary bladder, I would rather think that mechanical or inflammatory stimuli cause multipotent cells of the bladder epithelium to differentiate and that perhaps the direction in wlrieh they develop is influenced by hormones.

The origin of the endometrial structures strated in a case of internal endometriosis The causative role of ovarian hormones bladder is unproved. Once established, connected with the ovary.

from t,he bladder epithelium is demonof the bladder. in the genesis of endometriosis of the however, endometriosis is I’unc~tionally

REFERENCES

40’ * .% ‘1W ) lY:<% Chaticin, ?K.: Bull. SW. franc:. tl’urol., Adams, P. 8. : J Urol p. 318, 1936. Erie, n.: 3. A. M. A. 104: 1401, 1935. Ilomcmr, K.: Zentralbl. f. Gynlk. 62: 2115, 1938. Hueckel, R.: In Henke Lubarsch, Handbuch cl. path. Anat. 6: 2, 1934. Lendorf, A.: Anat. Anz. 17: 55- 1901. Xark, E’. G.: J. Urol. 37: 799, 1937. Mickuliw-Radecki, P. I*. : Zentrslbl. f. Gynak. 60: 2530, 1936. Patch, P. S., M. A. J. 33: 597. 1935. Perlmanq S.: TJrol & Cutan. amd Rhea, L. J. : Canad. Rev. 38: 76, 1934. l’hiZIi&~, R. B.: .J. Obst. & Gynaec,. Brit. Emp. 41: I65, 1934. Plant, A. : Zentralbl. f. Gyniik. 63: 8358, 1929. Y~tsclwlr, II’.: In Henke Lubarsch, Handbuch d. path. Anat. 6: Part 2, 333, 1934. Reyndb, L. R.: J. TJrol. 41: 157, 1939. Stoeckel, W.: Gynaekol. Urologic in Stoeckel ‘s Iiandbuch der (;ynarkologic 10: Part 1, 1938. JI~ei,jtZan.dt, J. ‘1. : Proc*. R.oy. Sot. Mml. 19: 15. 193-t.

Synephias: d’obst.

Spinal Anesthesia With et de gym%. 28: 137, 1939.

In the Strasbourg Maternity, 67 anesthesia. No serious complications in spite of its well-known dangers tractility, diminishes bleeding during is harmless to the child. Local anesthesia is excellent, but women who suffer violent uterine co1 cult to carry out when perf~~:mi:.~: of the uterus.

Pe; ::.:-I(: for Cesarean

Section,

cesurcan sections were performed were t,bserved. This anesthetic beacse it preserves satisfactory opcro tion, prevents postoperative

Bull.

SW.

under spinal was employed uterine constony, and

it .akes much more time and proves painful in !:actions. Furthermore, local anesthesia is diffiit cervical cesarean section with exteriorization J. P. GKEENIIIIL