OVARIAN
PREGNANCY,
PRIMARY
M.D., F.A.C.S.,
BERNARD MANN,
L)AVID
OR SECONDARY?” M.D.,
R. MERANZE,
BENJAMIN LEFF, M.D., PHILADELPHIA, (From the Gynecologic Service of the Mount Sinai
AXD
PA. Hospital)
PRIMARY, or true, ovarian pregnancy, is one in which the ovum, remaining in the follicle, is there fertilized, develops a sac from the contiguous ovarian tissue, and continues its growth in the follicle. In contrast, a secondary ovarian pregnancy is one in which the ovum is fertilized in tissues, tubal or uterine, outside of the ovary, develops its sac from this extraovarian tissue, and after developing its sac, becomes detached from its parent soil and secondarily attaches itself to the ovary. In the case of primary ovarian pregnancy, the sac if not completel> ovarian should be so preponderantly. In secondary ovarian pregnancy, the sac is likely to consist of an admixture of both varieties of tissue, ovarian and non ovarian, but there should be a predominance of tissues from the site of primary fecundation. The possible mechanism of primary or true ovarian pregnancy is at the present time a matter for speculation. Caturanii predicates as its basis a perioophoritis. -4 second theory postulates the rupture of a centrally placed follicle into a previously vacated surface follicle.:! Another theory assumes the retention of Miillerian tissue in the ovary.3 Sutton” formulates a theory based on Sampson’s work on endometriosis. To whatever theory we ultimatel? subscribe, we must accept the existence of two constant factors operating in its etiology, t,hat the ovum remains imprisoned within the cav-it.v of the ruptured follicle, and that the sperm cell finds its way to this particular ovum. Therefore, one may justly conclude that primary ovarian pregnancy is a rare phenomenon. Many Cases reported as primary ovarian pregnanc>es are excludell by careful reviewers. Thus, Schumann,5 in 192I, quoted only 43 cases; Sutton,4 in 1924, accepted only 47 cases; Frank,6 in 1927, accepted 64, while Wollner,7 in 1932, discarded 39 of 87 reported cases. More recently, in 1934, Dodeks referred to only 52 cases of true ovarian pregnancies. Young and Hawks report one case in a series of 118 cases of eetopic pregnancies, over a period of sixteen years, an incidence of 0.i per cent. Since then there have been a few reports of additional isolated cases. .-\t our hospital, the case about to be reported is the first noted in a period of over twenty-five years.
A
(!,.4SF 1 REPORT H. K., married, aged 31 years, was admittetl to Gynecologic Service No. 2 of the Mount Sinai Hospital on March 6, 1936, with vaginal bleeding and severe pain in the lower abdomen. Her present illness dates from January 30 of the same year, when after missing a regular period (last regular period Dec. IS, 1935), she was seized suddenly with severe abdominal pain and slight vaginal bleeding. This attack was accompanied by syncope. She was admitted to a neighboring hospital, where a Friedman modification of the Aschheim-Zondek test was positive. A diag nosis of threatened abortion, with a living fetus, was made at that institution. She was given one week of expectant treatment and discharged to the care of her family physician. However, intermittent cramplike pains continued. After four days at home, she returned to the same institution, remaining four additional days under observation, following which she was again discharged. Spproximately two weeks later, she was admitted to the Mount Sinai Hospital. On admission, the patient was found to be well nourished and comparatively comfortable, though complaining of pain and vaginal bleeding. Her temperature was 99.2” F., pulse 100 and of good volume. Tenderness was present over the ‘Read
at
& meeting
of
the
Obstetrical
Society
of
Philadelphia,
Pa.,
May
2, 1940.
MANX
ET
AI,. :
OVARIIIR‘
PREGNASCY
323
entire lower abdomen, more marked over the left lower quadrant. No rigidity or distention was noted. Bimanual examination revealed a soft cervix with a closed external OS. The uterus was enlarged to about the size of a two months’ pregnancy, partially movable, with tenderness to the right. A tentative diagnosis of intrauterine pregnancy, complicated by pelvic cellulitis and inevitable abortion was made. The following day, on. exploration of the uterus, a small amount of well-preserved decidua was found. Later a mass was palpated closely adherent to and to the right of the uterus. Hemoglobin was 68 per cent (Sahli) ; red cells, 3,680,OOO; white cells, 11,900; polymorphonuclear cells, 77 per cent. The urine was essentially negative, Because of the continued pain and increasing size of the pelvic mass, a laparotomy was performed on March 25, 1936. The uterus and adnexa were adherent to the large and small bowels. A tuboovarian abscess was found on the right side. On the left side the tube was adherent to and encircled a large, round, bluish, cystlike structure, occupying the region of the left ovary, measuring approximately 12 cm. in. diameter. A supravaginal hysterectomy with a bilateral salpingo-oophoreetomy was done. The patient made a complete recovery. PATHOLOGIC REPORT Macroscopic.--L’ The operatively removed specimen amputated uterus and a.dnexa. The uterus measures in consistency throughout. Its wall is intact at all is thin and moderately injected.
Fig.
l.-Showing
chorionic
villi.
consists of a supravaginally 8 by 5 by 4 cm. It is firm areas. The endometrial layer
X 60.
“To the right of the uterus is a tuboovarian abscess. The tube is greatly thickened, measuring 7 by 3 cm. Attached to the left side of the uterus and occupying the site of the left ovary is a large roughly globular, dark plum-colored and grayish mass measuring 11 by 8 by 6 cm. A thin layer of clotted blood is present over a small area of its surface. The surface bulges irregularly, particularly over an area 5.5 by 3.5 cm., the latter bulge being caused by a small thinwalled cyst distinct from the major mass. The major mass, on sectioning, consists of a cystic structure, with a moderately thickened wall. It appears on section to be composed of two portions. The lower smaller portion measures 2 by 6 cm. and consists in part of a large corpus luteum, 19 mm. in length. The remainder of this lower portion is a nondescript tissue, inundated with blood. “The upper portion is separated from the lower by a thin layer of compressed grayish tissue. This upper portion. measures 8 by 6 cm. Lying free within its cavity is a fully formed, well-preserved fetus, 8 cm. in length, to which is attached Plastered against the lateral, anterior, and posterior a narrow umbilical cord. walls of this cavity and occupying about three-fourths of its volume are firm,
bluish and grayish masses resembling l~lacental tissue. No membrauea ran be seen about the fetus nor (‘an the site of attachment of the umbilical cord to the placental structures 1~ determined at this time. The course of t,he Fallopian tube on the left, side has l,een sharply distorted, passing downward and anteriorly across the anterior surface of the large mass lust described. The tube is moderxtely thickened lrnt appwrs intwt throughout. 1 ts fimbria art) c’ongcstetl. ’’
Fig.
3.-Showing
wrtion
of ~a11
of sac
with
epithelis.l-lined
M~o~osco~~c.--” The ut,erine endometrium shows any decidual reaction. Both Fallopian tubes show esses, involving especially the mucosal layers aml muscle coats. No decidual reaction is noted in the and grayish tissue resembling placenta, observed in left-sided ovarian mass already desrrihed, are many villi, with Langhans and syncytial cell layers (Fig. and hemorrhagic. The corpus luteum described lutein cells (Fig. 2). In some of the early sections
tissue.
xX0.
no attached chorionic villi nor subchronic suppurative procthe innermost portions of the Fallopian tubes. In the bluish the upper portion of the large typical well-preserved chorionic 1). Other villi are necrotic grossly is composed of large taken from widely separated
MBNN
ET
AI,.
:
OVARIAN
PREGNANCY
325
portions of the wall of the sac enclosing the fet,us and placental tissue, are collections of ovarian stroma, occasionally containing a small follicle cyst. Graafian follicles are scanty in this ovarian stroma. The latter shows a decidua-like reaction. However, in sections taken later from the wall of the sac there is observed, ofteu in contact with necrotic tissue containing ehorionic villi, layers of tissue lined by a low columnar cell epithelium, resembling Fallopian tube epithelium or parovarian epithelium. In some areas this epithelium covers small papillary projections (Fig. 3). In the underlying wall no distinct muscle layers are present, but there are strands of tissue resembling muscle cells. This epithelial-lined tissue is infiltrated with inflammatory cells. It should be stressed that this tissue eonstitutes a portion of the wall enclosing the products of conception and that these latter products are apparently compressing it and its papillary projeetions (Fig. 3).” COMMEKT
Obviously, the case reported is that of some form of ovarian pregnaney. The uterine cavity is empty, the left tube is intact, the sac containing a fetus occupies the position where the ovary should be, and the sac is attached to the uterus by the utero-ovarian ligament. Since these gross criteria of Spiegelberg were fulfilled and since a preliminary microscopic report stated that ovarian tissue was found in different and widely separated sections taken from the sac wall, a prima facie case of true ovarian pregnancy appeared established. Furthermore, from the microscopic studies the additional criteria of both Williams and Norris appeared satisfied. In its entirety, then, this case met not only all the established criteria of ovarian pregnancy but in addition a fetus was found intact in the sac. According to Hunter as quoted by Thro,12 an embryo was recovered in only 19 instances out of 43 cases. Having met all the requirements, we were then placed in the anomalous position of having to dispute or, at least, cast a shadow of doubt on the diagnosis of true ovarian pregnancy, because of the presence microscopically in later sections of the epithelial tissue found in different portions of the sac wall as has already been described in the pathologic report. If this he a primary ovarian pregnancy, the question arises, Why was this epithelial tissue found scattered in different portions of the sac wall’+’ The possibilities are fourfohl : contact with the Fallopian tube, contact with an accessory tube, contact with some parovarian tissue, and an endometriosis of the ovary. We are inclined to consider the possibilities to rest between contact with an accessory tube or contact with some parovarian tissue. Here then we have a case meeting all the approved criteria of an ovarian pregnancy, and yet there are sufficient anomalous findings which make impossible its unequivocal acceptance as such. The interesting speculation arises, How conclusive are the great majority of eases of so-called primary ovarian pregnancy? Would, as in our case, a more thorough investigation by means of many microscopic sections, have also revealed disturbing findings in many of these reported cases?
(1) caturani, M.: Am. J. Obst. 69: 409, 1914. (2) Leopold: Arch. f. Gynak. 19: 210, 1882. (Quoted by Schumann.) (3) Likes, Lanning E.: Surg. Gynec. Obst. 55: 643, 1932. (4) Sutton, Lyle A.: AM. J. OBST. & GYNEC. 7: 1, 1924. (5) SchuPregnancy, Gynecologic and Obstetric Monographs, mann, Edward A.: Extrauterine New York, 1921, D. Appleton-Century Co., p, 73. (6) Frank, R. T.: Gynecologic Pathology, ed. 2, New York, 1932, D. Appleton-Century Co., p. 443. (7) Wollner, A.: AK J. OBST. & GYNEC. 23: 262, 1932. (8) Dodek, Samuel M.: Ibid. 28: 268, 1934. (9) Young, A. J., and Hawk, G. M.: Ibid. 26: 97, 1933. (10) Spiegelberg: Arch. f. GynHk. 13: 73, 1878. (Quoted by Schumann.) (11) Quoted by Schumann. (12) T’hro, William C.: AK J. OBST. C GYNEC. 29: 4517, 1935.
2019
PINE
STREET
DISCUSSION DR. criteria
EDWARD concerning
A. SCHUMANN.-It ovarian pregnancy
is extremely because so often
difficult to satisfy the walls of the
all the sac have
326
AMERICAN
been subjected and partially
JOURSAI,
to pressure destroyed.
and
OF
ORSTETRI(!S
to penetration
2ZSl)
l)y
(~YSECOI~OGI
blood,
so that
they
are
necrotic
I belong to the school of thought which was inaugurated by Webster of Chicago Bowand feel that pregnancy cannot occur in the absence of Miillerian tissue. ever, the discovery of the presence of endometrial cells and groups of uterine glands in the ovarian tissue itself, has cleared up this matter, so that it is wholly possible for a pregnancy to occur in an ovary, provided a little endometriosis is present. In the case now under discussion I should agree with Dr. Mann that he is dealing with an ovarian pregnancy.
THE EFFECT STILBESTROL LAMAN
(From
A.
GRAY,
the Departme&
OF DIETHYLSTILBESTROL AND DIETHYLDIPROPIONATE ON POSTMENOPAUSAL VAGINITIS AND SYMPTOMS M.D.,
AND
of
JOHK
D.
M.D.,
GORDINIER,
Obstetrvks and Gynecology, Vedical School)
Uniiwers~ty
LOUISVILLE,
of
KY.
Louisville
T IS to be noted that many American writers have found the stillbestrol compounds quite effective as estrogenic agents and giving excellent relief for menopausal symptoms. The so-called toxic effects of nausea and vomiting have almost entirely followed the oral preparations. In contrast to the opinion of Schorr, Robinson and Papanicolaoul t.hey have been much less frequent and have extremely rarely followed intramuscular injections. According to the Lancet” extensive use of stillbestrol in England “does not reveal a high proportion of unpleasant side effects. ’ ’
I
In the present study 111 women have been treated for menopausal symptoms and atrophic vaginitis. Sixty-two were given intramuscular injections of diethylstilbestrol dipropionate (Winthrop). Sixty-eight were given diethylstilbestrol by mouth (in 32 eases, Winthrop’s uncoated tablets : in 23, Parke Davis plain capsules; in 8, Lilly’s uncoate
*Because of toxic results reported by SO~X observers, given. The materials have been generously supplied by pany. Parke, Davis & Company and Eli Lilly Company. tImprovement was graded from one to four plus.
the the
origin Winthrop
of each material is Chemical Corn-