Difference between revisions of "Paper - Some aspects of ovarian pregnancy - with report of a case (1918)"

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=Some Aspects of Ovarian Pregnancy - With Report of a Case=
 
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[[File:Arthur William Meyer.jpg|thumb|150px|alt=File:Arthur William Meyer.jpg|thumb|link=Embryology History - Arthur Meyer|alt=Arthur William Meyer (1873 – 1966)|Arthur William Meyer (1873 – 1966)]]
By A. W. Meyer and H. M. N. Wynne
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By [[Embryology History - Arthur Meyer|A. W. Meyer]] and H. M. N. Wynne
  
 
From the Department of Gyneeology of The Johns Hopkins Medical School, the Department of Embryology of the Carnegie Institution and the Department of Anatomy of Stanford University
 
From the Department of Gyneeology of The Johns Hopkins Medical School, the Department of Embryology of the Carnegie Institution and the Department of Anatomy of Stanford University
  
  
 
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==Introduction==
 
Although the first case of ovarian pregnancy under that heading in the Index Medicus is that of Kouwer ('97) (van Tnssenbroek, '99), careful scrutiny of the titles listed for the last decade reveals the fact that 5 cases of ovarian pregnancy were reported in 1908, 4 each in 1909 and 1910, 7 in 1911. 13 in 1912, 9 in 1913, 7 in 1914, 3 in 1915, 1 in 191G, and 5 in 1917. This makes a total of 58 cases apparently reported within this decade. Since the reports on some of the cases were published in three different Journals, these were, of course, counted merely as one, and although the authenticity of four of the cases must be questioned on the basis of the titles alone, the series, nevertheless, is a large one in spite of these facts and of a marked decline in the number reported during the war. Since Norris ('09) stated that only 19 certain cases, a])proximately only one-third as many as all cases Usted in the last decade, were reported in the decade between 1899 and 1909, it would seem that ovarian pregnancy not only is receiving increasing attention, but that a change in attitude probably is in progress. Tliis conclusion would seem to be justified even though a careful examination of the descriptions of the cases reported in the decade between 1908 and 1917 would reduce somewhat the number listed.
 
Although the first case of ovarian pregnancy under that heading in the Index Medicus is that of Kouwer ('97) (van Tnssenbroek, '99), careful scrutiny of the titles listed for the last decade reveals the fact that 5 cases of ovarian pregnancy were reported in 1908, 4 each in 1909 and 1910, 7 in 1911. 13 in 1912, 9 in 1913, 7 in 1914, 3 in 1915, 1 in 191G, and 5 in 1917. This makes a total of 58 cases apparently reported within this decade. Since the reports on some of the cases were published in three different Journals, these were, of course, counted merely as one, and although the authenticity of four of the cases must be questioned on the basis of the titles alone, the series, nevertheless, is a large one in spite of these facts and of a marked decline in the number reported during the war. Since Norris ('09) stated that only 19 certain cases, a])proximately only one-third as many as all cases Usted in the last decade, were reported in the decade between 1899 and 1909, it would seem that ovarian pregnancy not only is receiving increasing attention, but that a change in attitude probably is in progress. Tliis conclusion would seem to be justified even though a careful examination of the descriptions of the cases reported in the decade between 1908 and 1917 would reduce somewhat the number listed.
  
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The opinion that many, even if not all, cases of so-called hematocele, hematoma, apoplexy, blood cysts, and rupture of the ovaries, probably are nothing but cases of ovarian pregnancy in disguise, has been held by various investigators for some time. Hence, if hematocele of the ovaries repeats the history of hematosalpinx, it is not tmlikely that the near future will see a marked increase in the reported frequency of " a fact so curioiis and important in itself," as Granville aptly put it a century ago. This would seem to be true in spite of the fact reported by Norris and Mitchell ('08), that only a single case of ovarian pregnancy was found among 44 extra-uterine specimens and 58 hemorrhagic cysts contained in the collection of 1700 gynecological specimens at the hospital of the University of Pennsylvania. At any rate, a careful microscopic examination of all such eases would seem
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The opinion that many, even if not all, cases of so-called hematocele, hematoma, apoplexy, blood cysts, and rupture of the ovaries, probably are nothing but cases of ovarian pregnancy in disguise, has been held by various investigators for some time. Hence, if hematocele of the ovaries repeats the history of hematosalpinx, it is not tmlikely that the near future will see a marked increase in the reported frequency of " a fact so curioiis and important in itself," as Granville aptly put it a century ago. This would seem to be true in spite of the fact reported by Norris and Mitchell ('08), that only a single case of ovarian pregnancy was found among 44 extra-uterine specimens and 58 hemorrhagic cysts contained in the collection of 1700 gynecological specimens at the hospital of the University of Pennsylvania. At any rate, a careful microscopic examination of all such eases would seem to be indicated in the future in order to determine, if possible, which cases are, and which are not, conceptual in origin.
  
  
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Today it is no longer true, as stated by Freund and Thome ('06) and by Sencert and Arom as late as 1914, that authentic cases of ovarian pregnancy belong to the great rarities. Yet the fact that many of our states, as well as many large clinics, have not a single case on their records seems to suggest that the condition still is seldom recognized, a century after Granville observed his first case. Moreover, a nimaber of continental gynecologists and obstetricians, for a quarter of a century, have regarded the oceiu-rence of ovarian pregnancy as undoubted. Anderson ('17) stated that German writers began to report eases of ovarian pregnancy with some frequency after 1901, and Gilford ('01) also called attention to the fact that continental opinion long had accepted ovarian implantation not only as possible, but as proven. Gilfonl further referred to the often quoted opinion of Tait that ovarian pregnancy is as rare as "A blue lion or a swan with two necks," and in his article in 1899 also called attention to the opinion of Bland-Sutton, that ovarian pregnancy not only has no existence, but that it is impossible. These opinions are particularly interesting in view of the carefitl reports made by Granville (1830 and 1834), in connection with the two cases which he then and which others since have regarded a.< cases of undoubted ovarian pregnancy, in spite of the absence of microscopic examination. In view of this lack it is particularly fortunate that both of these reports of Granville are accompanied by splendid illustrations by Bauer, which also won his praise and admiration and which greatly strengthened his cases. It may be recalled in this connection that Werth ('01) accepted Granville's case recorded in 1820, but said nothing about his second more convincing instance reported in 1834.
  
to be indicated in the future in order to determine, if possible, which cases are, and which are not, conceptual in origin.
 
  
To-day it is no longer true, as stated by Freund and Thome ('06) and by Sencert and Arom as late as 1914, that authentic cases of ovarian pregnancy belong to the great rarities. Yet the fact that many of our states, as well as many large clinics, have not a single case on their records seems to suggest that the condition still is seldom recognized, a century after Granville observed his first case. Moreover, a nimaber of continental gynecologists and obstetricians, for a quarter of a century, have regarded the oceiu-rence of ovarian pregnancy as undoubted. Anderson ('17) stated that German writers began to report eases of ovarian pregnancy with some frequency after 1901, and Gilford ('01) also called attention to the fact that continental opinion long had accepted ovarian implantation not only as possible, but as proven. Gilfonl further referred to the often quoted opinion of Tait that ovarian pregnancy is as rare as " A blue lion or a swan with two necks," and in his article in 1899 also called attention to the opinion of Bland-Sutton, that ovarian pregnancy not only has no existence, but that it is impossible. These opinions are particularly interesting in view of the carefitl reports made by Granville (1830 and 1834), in connection with the two cases which he then and which others since have regarded a.< cases of undoubted ovarian pregnancy, in spite of the absence of microscopic examination. In view of this lack it is particularly fortunate that both of these reports of Granville are accompanied by splendid illustrations by Bauer, which also won his praise and admiration and which greatly strengthened his cases. It may be recalled in this connection that Werth ('01) accepted Granville's case recorded in 1820, but said nothing about his second more convincing instance reported in 1834.
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Although there is as yet no agreement as to what constitutes an authentic case, a review of the literature justifies the growing and apparently well-founded belief that in the past too much emphasis has been laid on certain criteria wliich later experience has shown to be partly inapplicable. It is becoming clear that some cases, formerly excluded for reasons regarded as sufficient, with our present knowledge could no longer be rejected. Moreover, it does not seem at all improl>able that some cases listed as tubal really were orarian in origin. Xor must it be forgotten that not even the entire absence of remnants of the conceptus can positively exclude a case from the category of true ovarian pregnancy. In a number of cases in the literature, and also in the present case, the clinical history and gross anatomic findings suggest the conclusion drawn by Scott ('01) on a priori grounds alone, that the conceptus may be completely resorbed. It may, of course, also be aborted and disintegrate completely. That such an assumption is justified is indicated by the lysis of the embryo or fetus in a large number t)f eases of ovarian jtrejrnancy, and also by the very degenerated condition of some of the vesieles and of the surrounding ovarian stroma. The possibility of such an occurrence is establislied also by similar events in tlic uterine and tubal pregnancies discussed elsewhere (Meyer. '19), and probably is illustrated by such cases as those of Anning and Littlewood {'01 ). in which no mention is made (if an embryonic disc in a translucent conccptus the size of "pea." Then, to be sure, there are the cases of unruiiturcil ovarian ])regiinncies containing villi only, as well as the rare case, probaltly of double ovarian pregnancy, of Holland ('11 ). Although one cannot be certain that embryonic tissue was removed from the left ovary with the blood-clot which was forcibly c.\]ielled at the time of operation, it is not at all improbable that the small jdasmodial masses found in the Icli ovary were the only remnants of the concejitus. 1 realize fully that the conclusion that young conceptuses may be wholly dissolved is fraught with great uncertainty, but I am quite sure that it is justified by the facts, and that it therefore is in the direction of truth. It could oidy fail to be so if every ovum that beconu's implanted within the ovaries were aborted or were removed by operation before lysis was possible.
  
Although there is as yet no agreement as to what constitutes an authentic case, a re\'iew of the literature justifies the growing and apparently well-founded belief that in the past too much emphasis has been laid on certain criteria wliich later experience has shown to be partly inapplicable. It is becoming clear that some cases, formerly excluded for reasons regarded as sufficient, with our present knowledge could no longer be rejected. Moreover, it does not seem at all improl>able that some cases listed as tubal really were orarian in origin. Xor must it be forgotten that not even the entire absence of remnants of the conceptus can positively exclude a case from the category of true ovarian pregnancy. In a number of cases in the literature, and also in the present case, the clinical history and gross anatomic findings suggest the conclusion drawn by Scott ('01) on a priori grounds alone, that the conceptus may be completely resorbed. It may, of course, also be aborted and disintegrate completely. That such an assumption is justified is indicated by the lysis of tlie emljiv" or fetus in a large number t)f eases of ovarian jtrejrnancy, and also by the very degenerated condition of some of the vesieles and of the surrounding ovarian stroma. The possibility of such an occurrence is establislied also by similar events in tlic uterine and tubal pregnancies discussed elsewhere (Meyer. '19), and probably is illustrated by such cases as those of Anning and Littlewood {'01 ). in which no mention is made (if an embryonic disc in a translucent conccptus the size of "pea." Then, to be sure, there are the cases of unruiiturcil ovarian ])regiinncies containing villi only, as well as the rare case, probaltly of double ovarian pregnancy, of Holland ('11 ). Although one cannot be certain that embryonic tissue was removed from the left ovary with the blood-clot which was forcibly c.\]ielled at the time of operation, it is not at all improbable that the small jdasmodial masses found in the Icli ovary were the only remnants of the concejitus. 1 realize fully that the conclusion that young concejjtuscs may be wholly dissolved is fraught with great uncertainty, but I am quite sure that it is justified by the facts, and that it therefore is in the direction of truth. It could oidy fail to be so if every ovum that beconu's implanted within the ovaries were al)orted or were removed by operation before lysis was possible.
 
  
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One cannot rightly refuse to recognize the possibility of the spontaneous disappearance of an ovarian pregnancy. Sinee implantation in the ovary occurs under such al)normal conditions, it would .seem that for this reason alone the great majority of such implantations inevitably must succumb. This would .>;eem probable wholly aside from considerations regarding the development of the corpus luteum, although lack of, or interference with, the development of the latter also woulil .'ieem to condition early death of the conceptus if the results of the long series of experiments on rabbits by Frankel ('0.3-'10) are indicative of the role iilaycd by the ••orpus luteum in early imidantations in nian also. It surely is didicult. if not impo.ssible, to see liow implantation witbii: the (Jraalian follicle, and esj)ecially the later development ol the conceptus, can fail to interfere with the development of ii normal corpus luteum. Ca.^es in the literature, and also in tlie present case, did not reveal the presence of any well-preserved or even true luteal cells at the time the pregnancy was terminated. Although this fact does not presu|)pose an entire lack of development of these cells in the earliest stages of the implantations, it undoubtedly does im|)ly a defective development, which in itself may have become responsible for tbi> death of the conceptus. \or should the jiossiide toxic effect upon the conceptus of luteal lells be forgotten in this i-onnection.
  
One cannot rightly refuse to recognize the possibility of the spontaneous disappearance of an ovarian pregnancy. Sinee implantation in the ovary occurs under such al)normal conditions, it would .seem that for this reason alone the great majority of such implantations inevitably must succumb. This would .>;eem probable wholly aside from considerations regarding the development of the corpus luteum, although lack of, or interference with, the development of the latter also woulil .'ieem to condition early death of the conceptus if the results of the long series of experiments on rabbits by Frankel ('0.3-'10) are indicative of the role iilaycd by the ••orpus luteum in early imidantations in nian also. It surely is didicult. if not impo.ssible, to see liow implantation witbii: the (Jraalian follicle, and esj)ecially the later development ol the conceptus, can fail to interfere with the development of ii normal corpus luteum. Ca.^es in the literature, and also in tlie present case, did not reveal the presence of any well-pre.'<er\eil or even true luteal cells at the time the pregnancy was terminated. Although this fact does not presu|)pose an entire lack of developnu'ut of these cells in the earliest stages of tlu' implantations, it undoubtedly does im|)ly a defective development, which in it.*elf nniy have become responsible for tbi> death of the conceptus. \or should the jiossiide toxic eti'ect upon the conceptus of luteal lells be forgotten in this i-onnection.
 
  
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I do not assume to be sure that the eliiiieal symptoms mikI signs alone should suflice finally to group a s|)ecinien as truly ovarian, but when these arc indicative of the pre.«ence of an ectopic gestation, and when undouiitcd intra-uterine decidual changes are present, in the absence of abdominal pregmincy or tubal involvement and a normal corpus luteum. and the presence of a blood-clot within the ovary, there would seem to he little reason for doubting the authenticity of the ovarian implantation even in the absence of embryonic remnant.
  
I do not assume to be sure that the eliiiieal symptoms mikI signs alone should suflice finally to group a s|)ecinien as truly ovarian, but when these arc indicative of the pre.«ence of an ectopic gestation, and when undouiitcd intra-uterine decidual changes are present, in the absence of abdominal pregmincy or tubal involvement and a normal corpus luteum. and the presence of a blood-dot within the ovary, there would seem to he little reason for doubting the authenticity of the ovarian implantation even in the absence of cnd)ryonic remmint.-.
 
  
  
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Siiu-e changes suggesting decidual reaction in the ovary have been reported so seldom it is doubtful whether nnich emphasis can be laid on them. One seems justified in saying this in spite of the fact that the presence of decidua in the ovary formed the only anatomic evidence upon which Kantorowic ('04) confidently classed his two rather atlvanced cases of ectopic pregnancy anumg the authentic. Moreover, if it be true, as stated by Webster ('04 ) that changes which cannot be distinguished from true decidual changes not infrequently occur in the ovary in connection with normal uterine pregnancies, then the ])rescnce of islands of decidual cells in an ovary surely cannot be regarded as indicative of ovarian pregnancy. I wonder, however, whether it woidd not be po.ssible to distinguish genuine decidual cells by modern histnchemical methods. In making this observation, 1 am fully aware that various criteria have been advanced from time to time by means of which to judge ovarian pregnancies, and that many of these have met with objection and have hence been modified. Such modifications would seem to be inevitable as long as there is progress in the sulutinii nf mm unsettli'd question.
  
Siiu-e changes suggesting decidual reaction in the ii\ary ha\e l)eeu reported so seldom it is doubtful whether nnich emphasi^ can be laid on them. One seems justified in saying this in spite of the fact that the presence of decidua in the ovary formed the only anatomic evidence U])on which Kantorowic/ ('04) confidently classed his two rather atlvanced cases of ectopic pregnancy anumg the authentic. Moreover, if it be true, as stated by Webster ('04 ) that changes which cannot be distinguished from true decidual changes not infrequently occur in the ovary in connection with normal uterine pregnancies, then the ])rescnce of islands of decidual cells in an ovary surely cannot be regarded as indicative of ovarian pregnancy. 1 wonder, however, whether it woidd not be po.ssible to distinguish genuine decidual cells by modern histnchemical methods. In making this observation, 1 am fully aware that various criteria have been advanced from time to time by means of which to judge ovarian pregnancies, and that many of these have met with objection and have hence been nu>dified. Such modifications would seem to be inevitable as long as there is progress in the sulutinii nf mm unsettli'd question.
 
  
 
The absence of the iCtus in inanv of the recorded cases in itself demonstrates tlie entire inapplicability of the criterion addeil by Jacobson ('(I.S). Moreover,' the histologiiappearance of the ovarian tissue around certain jiortions of the blood-clot in the present, ami also in some of the cases in tinliterature, would seem to suggest that it may be very diflicull to find remnants of ovarian tissue at several points in a casi' of pregnancy which has ailvanced far. Hence, this criterion of Spiegelbcrg ('7S) cannot be regarded as necessarily crui-ial. Whenever the implantation is developed at the outer instead of at the inner margin of a follicle, as in the ca.^e of Banks ('12), early destruction, even if not early rupture, of the overlying ovarian stroma and cajisule would seem to be inevitable. Indeed, whenever the layer of ovarian stroma overlying the pliuiiita is thin, very early death of the fetus would seem to lie inevitalde from defective nutrition alone. On the other hand, when jdaccntal development occurs in the region of the follicle directed toward the body of the ovary, great destruction of the ovarian stroma woulil seem to be unavoidable, even if something akin to normal decidual formation actually took ]dace. In the case of Kngelking ('lU), for example, not a trace of an ovary was found in an ovarian pregnancy which had become interstitial. Kven without assuming the complete aidhenticity of this rather ecpiivocal case, it W(uild seem highly probable that the presence of ovarian tissue later in the pri'gnaiiey probably is determined very largely by the location of the fertilized follicle within, or by the e.\act location of the im|ilanta1ion upon the ovary.
 
The absence of the iCtus in inanv of the recorded cases in itself demonstrates tlie entire inapplicability of the criterion addeil by Jacobson ('(I.S). Moreover,' the histologiiappearance of the ovarian tissue around certain jiortions of the blood-clot in the present, ami also in some of the cases in tinliterature, would seem to suggest that it may be very diflicull to find remnants of ovarian tissue at several points in a casi' of pregnancy which has ailvanced far. Hence, this criterion of Spiegelbcrg ('7S) cannot be regarded as necessarily crui-ial. Whenever the implantation is developed at the outer instead of at the inner margin of a follicle, as in the ca.^e of Banks ('12), early destruction, even if not early rupture, of the overlying ovarian stroma and cajisule would seem to be inevitable. Indeed, whenever the layer of ovarian stroma overlying the pliuiiita is thin, very early death of the fetus would seem to lie inevitalde from defective nutrition alone. On the other hand, when jdaccntal development occurs in the region of the follicle directed toward the body of the ovary, great destruction of the ovarian stroma woulil seem to be unavoidable, even if something akin to normal decidual formation actually took ]dace. In the case of Kngelking ('lU), for example, not a trace of an ovary was found in an ovarian pregnancy which had become interstitial. Kven without assuming the complete aidhenticity of this rather ecpiivocal case, it W(uild seem highly probable that the presence of ovarian tissue later in the pri'gnaiiey probably is determined very largely by the location of the fertilized follicle within, or by the e.\act location of the im|ilanta1ion upon the ovary.
  
Werth ('8T) is said to have collected 12 ca.srs. among which he regarded only that of Leopold ('S2) as authentic. Ix-opolil ('1)9) reported 14. (Jilford ('01), in a splendid succinct review of tlu' literature gave 28 cases. Hi of whi<h he regarded as nndoubteil and 12 as ]>robalile. Koclie {'02) accepted only 12 ca.<es. Filth {'02) accepted 21. Kantorowicz {'04), using the criteria of I.,eopold {'99), together with a microscopic examination as a basis, groujied the cases in the literature a
 
  
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Werth ('8T) is said to have collected 12 ca.srs. among which he regarded only that of Leopold ('S2) as authentic. Ix-opolil ('1)9) reported 14. (Jilford ('01), in a splendid succinct review of tlu' literature gave 28 cases. Hi of whi<h he regarded as nndoubteil and 12 as ]>robalile. Koclie {'02) accepted only 12 ca.<es. Filth {'02) accepted 21. Kantorowicz {'04), using the criteria of I.,eopold {'99), together with a microscopic examination as a basis, groujied the cases in the literature a certain, probable, and uncertain. He considered 17 as certain, 10 as probable, and 13 as uncertain. And to the 17 cases regarded as certain by him, Kantorowicz then added two of his own, basing his decision, however, mainly upon the presence of deeidua in the ovaries, thus making 19 cases regarded as authentic by him. Freund and Thome ('06) regarded 23 of all the cases reported up to that time as certain. Norris ami Jlitchell ('08) considered 16 as positive, 15 as probable and !) as fairly probable. Warbanoff ('09) collected 3-t cases and iSTorris ('09) regarded 19 of those contained in the literature of the previous decade as positive; but Williams ('10), from a critical review of the literature up to 1906, and upon tlic l)asis of the criteria of Spiegelberg, regarded only 13 as positive, 17 as highly probable, and 5 as probable. Mapes ('14) collected 30 cases, but wholly from secondary sources, and Lockyer ('17) 42, from the years 1910 to 1917. Of these eases Lockyer accepted 22 as authentic and 20 as questionable ami undecided from the evidence available to him.
  
  
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This short summary suffices to show that there is as yet no consensus of opinion as to what constitutes an ovarian pregnancy. Although this fact finds its explanation partly in our lack of sufficient knowledge, it is due also to the meagerness of some of the reports. Besides, if complete disintegration and lysis of intraovilrian conceptusps can occur, then it must always remain a. question of opinion in the future wliether some of the cases so rejiorted really were or were not true ovarian jiregnancies. This must remain true no matter how thorouuh the microscopic examination, unless the clinical history or changes in the maternal organism can afford us crucial tests in such cases.
  
certain, probable, and uncertain. He considered 17 as certain, 10 as probable, and 13 as uncertain. And to the 17 cases regarded as certain by him, Kantorowicz then added two of his own, basing his decision, however, mainly upon the presence of deeidua in the ovaries, thus making 19 cases regarded as authentic by him. Freund and Thome ('06) regarded 23 of all the cases reported up to that time as certain. Norris ami Jlitchell ('08) considered 16 as positive, 15 as probable and !) as fairly probable. Warbanoff ('09) collected 3-t cases and iSTorris ('09) regarded 19 of those contained in the literature of the previous decade as positive; but Williams ('10), from a critical review of the literature up to 1906, and upon tlic l)asis of the criteria of Spiegelberg, regarded only 13 as positive, 17 as highly probable, and 5 as probable. Mapes ('14) collected 30 cases, but wholly from secondary sources, and Lockyer ('17) 42, from the years 1910 to 1917. Of these eases Lockyer accepted 22 as authentic and 20 as questionable ami undecided from the evidence available to him.
 
  
This short summary suffices to show that there is as yet no consensus of opinion as to what constitutes an ovarian pregnancy. Although this fact finds its explanation partly in our lack of sufficient knowledge, it is due also to the meagerness of some of the reports. Besides, if complete disintegration and lysis of intraovilrian conceptusps can occur, then it must always remain a. question of opinion in the future wliether some of the cases so rejiorted really were or were not true ovarian jiregnancies. This must remain true no matter how thorouuh the microscopic examination, unless the clinical history or changes in the maternal organism can afford us crucial tests in such cases.
+
Anyone who reads far into the literature of ovarian pregnancy also nnist become aware of the fact that even very recently skepticism has been carried too far. Jacobson ('08), for example, placed the case of Ivouwer-van Tussenbroek (a ease which finally convinced Bland-Sutton) and that of Webster ('01) in the doubtful class! Furthermore, Jacobson also insisted upon the presence of an cndiryo or fetus as absolutely essential.
  
Anyone who reads far into the literature of ovarian pregnancy also nnist become aware of the fact that even very recently skepticism has been carried too far. Jacobson ('08), for example, placed the case of Ivouwer-van Tussenbroek (a ease which finally convinced Bland-Sutton) and that of Webster ('01) in the doubtful class! Furthermore, .Tacobson also insisted upon the presence of an cndiryo or fetus as absolutely essential.
 
  
 
It must be emphasized, however, that even a liberal attitude on the part of a reviewer would not justify him in accepting all cases reported as genuine upon the basis of the reports themselves, for they — especially the older ones — often are too meager to enable one to form a reliable opinion. This is illustrated also by such recent reports as those of (iarrard ('16), Martin ('17), Sweeney ('17), and of Mills ('17). Although it must he remembered that from the very nature of tilings it sometimes is impossible to make a report which in itself carries conviction, it is regrettable that in a niunber of relatively recent cases in which such a report apparently could have been made, this was not done. ^lills' case seems to have been an instance of ovarian implantation in a region other than the Graafian follicle, and hence recalls the first ease of Granville and the cases of Franz ('02), Norris ('09), Paucot et Debeyre ('13)?, and perhaps also that of Kouwer ('97) (van Tussenbroek, '99).
 
It must be emphasized, however, that even a liberal attitude on the part of a reviewer would not justify him in accepting all cases reported as genuine upon the basis of the reports themselves, for they — especially the older ones — often are too meager to enable one to form a reliable opinion. This is illustrated also by such recent reports as those of (iarrard ('16), Martin ('17), Sweeney ('17), and of Mills ('17). Although it must he remembered that from the very nature of tilings it sometimes is impossible to make a report which in itself carries conviction, it is regrettable that in a niunber of relatively recent cases in which such a report apparently could have been made, this was not done. ^lills' case seems to have been an instance of ovarian implantation in a region other than the Graafian follicle, and hence recalls the first ease of Granville and the cases of Franz ('02), Norris ('09), Paucot et Debeyre ('13)?, and perhaps also that of Kouwer ('97) (van Tussenbroek, '99).
  
From evideiu'e contained in the literature, it is clear that further re|)orts of single cases are not needed for the purpose of emphasizing the occurrence of ovarian pregnancy, yet such reports nevertheless may help in the determination of the relative frequency of this novel and sinister condition, and also throw further light upon its genesis and the finer relations of the implantations, as well as upon other matters. Moreover, since the cases which are accompanied by a careful histologic examination and wliich for this reason alone are wholly unequivocal from an anatomic standpoint still are relatively few, the report of an additional case would seem to be justified. The present specimen (Carnegie Collection, No. 1322) was donated by Wynne to the Department of Embryology of the Carnegie Institution of Washington, and the following clinical report furnished by him :
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From evideiu'e contained in the literature, it is clear that further reports of single cases are not needed for the purpose of emphasizing the occurrence of ovarian pregnancy, yet such reports nevertheless may help in the determination of the relative frequency of this novel and sinister condition, and also throw further light upon its genesis and the finer relations of the implantations, as well as upon other matters. Moreover, since the cases which are accompanied by a careful histologic examination and wliich for this reason alone are wholly unequivocal from an anatomic standpoint still are relatively few, the report of an additional case would seem to be justified. The present specimen (Carnegie Collection, No. {{CE1322}}) was donated by Wynne to the Department of Embryology of the Carnegie Institution of Washington, and the following clinical report furnished by him :
  
 
==Clinical History==
 
==Clinical History==
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Idem: 1917. Obstetrics; A text-book for the use of students and practitioners, N. Y. and Lond.
 
Idem: 1917. Obstetrics; A text-book for the use of students and practitioners, N. Y. and Lond.
  
Young. E. B., and L. J. Rhea: 1911'. Ovarian pregnancy. Report of a case. Bost. Med. and Surg. Jour.. CLXIV.
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Young. E. B., and L. J. Rhea: 1911. Ovarian pregnancy. Report of a case. Bost. Med. and Surg. Jour.. CLXIV.
  
 
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[[Category:Carnegie Embryo 1322]]

Revision as of 15:03, 15 February 2020

Meyer AW. and Wynne HMN. Some aspects of ovarian pregnancy - with report of a case. (1919) Johns Hopkins Hospital Bulletin 30:

Some Aspects of Ovarian Pregnancy - With Report of a Case

Arthur William Meyer (1873 – 1966)
Arthur William Meyer (1873 – 1966)

By A. W. Meyer and H. M. N. Wynne

From the Department of Gyneeology of The Johns Hopkins Medical School, the Department of Embryology of the Carnegie Institution and the Department of Anatomy of Stanford University


Introduction

Although the first case of ovarian pregnancy under that heading in the Index Medicus is that of Kouwer ('97) (van Tnssenbroek, '99), careful scrutiny of the titles listed for the last decade reveals the fact that 5 cases of ovarian pregnancy were reported in 1908, 4 each in 1909 and 1910, 7 in 1911. 13 in 1912, 9 in 1913, 7 in 1914, 3 in 1915, 1 in 191G, and 5 in 1917. This makes a total of 58 cases apparently reported within this decade. Since the reports on some of the cases were published in three different Journals, these were, of course, counted merely as one, and although the authenticity of four of the cases must be questioned on the basis of the titles alone, the series, nevertheless, is a large one in spite of these facts and of a marked decline in the number reported during the war. Since Norris ('09) stated that only 19 certain cases, a])proximately only one-third as many as all cases Usted in the last decade, were reported in the decade between 1899 and 1909, it would seem that ovarian pregnancy not only is receiving increasing attention, but that a change in attitude probably is in progress. Tliis conclusion would seem to be justified even though a careful examination of the descriptions of the cases reported in the decade between 1908 and 1917 would reduce somewhat the number listed.


Loekyer ('17) accepted as authentic only 22 cases of those reported between 1910 and 1917, but his review is only a partial one. Even so, it shows that there is a decided increase in the number of cases which have been regarded as genuine from decade to decade. The marked increase in the number of genuine cases re])orted in recent decades becomes still more evident if one recalls that Williams ('10) found only 13 positive cases up to 1906, whereas Norris found 19 positive cases in the single decade between 1899 and 1909. That is, ISTorris foimd more positive cases reported in this decade than had been reported in all previous medical history up to 1906. This surely is a significant fact.


The opinion that many, even if not all, cases of so-called hematocele, hematoma, apoplexy, blood cysts, and rupture of the ovaries, probably are nothing but cases of ovarian pregnancy in disguise, has been held by various investigators for some time. Hence, if hematocele of the ovaries repeats the history of hematosalpinx, it is not tmlikely that the near future will see a marked increase in the reported frequency of " a fact so curioiis and important in itself," as Granville aptly put it a century ago. This would seem to be true in spite of the fact reported by Norris and Mitchell ('08), that only a single case of ovarian pregnancy was found among 44 extra-uterine specimens and 58 hemorrhagic cysts contained in the collection of 1700 gynecological specimens at the hospital of the University of Pennsylvania. At any rate, a careful microscopic examination of all such eases would seem to be indicated in the future in order to determine, if possible, which cases are, and which are not, conceptual in origin.


Today it is no longer true, as stated by Freund and Thome ('06) and by Sencert and Arom as late as 1914, that authentic cases of ovarian pregnancy belong to the great rarities. Yet the fact that many of our states, as well as many large clinics, have not a single case on their records seems to suggest that the condition still is seldom recognized, a century after Granville observed his first case. Moreover, a nimaber of continental gynecologists and obstetricians, for a quarter of a century, have regarded the oceiu-rence of ovarian pregnancy as undoubted. Anderson ('17) stated that German writers began to report eases of ovarian pregnancy with some frequency after 1901, and Gilford ('01) also called attention to the fact that continental opinion long had accepted ovarian implantation not only as possible, but as proven. Gilfonl further referred to the often quoted opinion of Tait that ovarian pregnancy is as rare as "A blue lion or a swan with two necks," and in his article in 1899 also called attention to the opinion of Bland-Sutton, that ovarian pregnancy not only has no existence, but that it is impossible. These opinions are particularly interesting in view of the carefitl reports made by Granville (1830 and 1834), in connection with the two cases which he then and which others since have regarded a.< cases of undoubted ovarian pregnancy, in spite of the absence of microscopic examination. In view of this lack it is particularly fortunate that both of these reports of Granville are accompanied by splendid illustrations by Bauer, which also won his praise and admiration and which greatly strengthened his cases. It may be recalled in this connection that Werth ('01) accepted Granville's case recorded in 1820, but said nothing about his second more convincing instance reported in 1834.


Although there is as yet no agreement as to what constitutes an authentic case, a review of the literature justifies the growing and apparently well-founded belief that in the past too much emphasis has been laid on certain criteria wliich later experience has shown to be partly inapplicable. It is becoming clear that some cases, formerly excluded for reasons regarded as sufficient, with our present knowledge could no longer be rejected. Moreover, it does not seem at all improl>able that some cases listed as tubal really were orarian in origin. Xor must it be forgotten that not even the entire absence of remnants of the conceptus can positively exclude a case from the category of true ovarian pregnancy. In a number of cases in the literature, and also in the present case, the clinical history and gross anatomic findings suggest the conclusion drawn by Scott ('01) on a priori grounds alone, that the conceptus may be completely resorbed. It may, of course, also be aborted and disintegrate completely. That such an assumption is justified is indicated by the lysis of the embryo or fetus in a large number t)f eases of ovarian jtrejrnancy, and also by the very degenerated condition of some of the vesieles and of the surrounding ovarian stroma. The possibility of such an occurrence is establislied also by similar events in tlic uterine and tubal pregnancies discussed elsewhere (Meyer. '19), and probably is illustrated by such cases as those of Anning and Littlewood {'01 ). in which no mention is made (if an embryonic disc in a translucent conccptus the size of "pea." Then, to be sure, there are the cases of unruiiturcil ovarian ])regiinncies containing villi only, as well as the rare case, probaltly of double ovarian pregnancy, of Holland ('11 ). Although one cannot be certain that embryonic tissue was removed from the left ovary with the blood-clot which was forcibly c.\]ielled at the time of operation, it is not at all improbable that the small jdasmodial masses found in the Icli ovary were the only remnants of the concejitus. 1 realize fully that the conclusion that young conceptuses may be wholly dissolved is fraught with great uncertainty, but I am quite sure that it is justified by the facts, and that it therefore is in the direction of truth. It could oidy fail to be so if every ovum that beconu's implanted within the ovaries were aborted or were removed by operation before lysis was possible.


One cannot rightly refuse to recognize the possibility of the spontaneous disappearance of an ovarian pregnancy. Sinee implantation in the ovary occurs under such al)normal conditions, it would .seem that for this reason alone the great majority of such implantations inevitably must succumb. This would .>;eem probable wholly aside from considerations regarding the development of the corpus luteum, although lack of, or interference with, the development of the latter also woulil .'ieem to condition early death of the conceptus if the results of the long series of experiments on rabbits by Frankel ('0.3-'10) are indicative of the role iilaycd by the ••orpus luteum in early imidantations in nian also. It surely is didicult. if not impo.ssible, to see liow implantation witbii: the (Jraalian follicle, and esj)ecially the later development ol the conceptus, can fail to interfere with the development of ii normal corpus luteum. Ca.^es in the literature, and also in tlie present case, did not reveal the presence of any well-preserved or even true luteal cells at the time the pregnancy was terminated. Although this fact does not presu|)pose an entire lack of development of these cells in the earliest stages of the implantations, it undoubtedly does im|)ly a defective development, which in itself may have become responsible for tbi> death of the conceptus. \or should the jiossiide toxic effect upon the conceptus of luteal lells be forgotten in this i-onnection.


I do not assume to be sure that the eliiiieal symptoms mikI signs alone should suflice finally to group a s|)ecinien as truly ovarian, but when these arc indicative of the pre.«ence of an ectopic gestation, and when undouiitcd intra-uterine decidual changes are present, in the absence of abdominal pregmincy or tubal involvement and a normal corpus luteum. and the presence of a blood-clot within the ovary, there would seem to he little reason for doubting the authenticity of the ovarian implantation even in the absence of embryonic remnant.


Siiu-e changes suggesting decidual reaction in the ovary have been reported so seldom it is doubtful whether nnich emphasis can be laid on them. One seems justified in saying this in spite of the fact that the presence of decidua in the ovary formed the only anatomic evidence upon which Kantorowic ('04) confidently classed his two rather atlvanced cases of ectopic pregnancy anumg the authentic. Moreover, if it be true, as stated by Webster ('04 ) that changes which cannot be distinguished from true decidual changes not infrequently occur in the ovary in connection with normal uterine pregnancies, then the ])rescnce of islands of decidual cells in an ovary surely cannot be regarded as indicative of ovarian pregnancy. I wonder, however, whether it woidd not be po.ssible to distinguish genuine decidual cells by modern histnchemical methods. In making this observation, 1 am fully aware that various criteria have been advanced from time to time by means of which to judge ovarian pregnancies, and that many of these have met with objection and have hence been modified. Such modifications would seem to be inevitable as long as there is progress in the sulutinii nf mm unsettli'd question.


The absence of the iCtus in inanv of the recorded cases in itself demonstrates tlie entire inapplicability of the criterion addeil by Jacobson ('(I.S). Moreover,' the histologiiappearance of the ovarian tissue around certain jiortions of the blood-clot in the present, ami also in some of the cases in tinliterature, would seem to suggest that it may be very diflicull to find remnants of ovarian tissue at several points in a casi' of pregnancy which has ailvanced far. Hence, this criterion of Spiegelbcrg ('7S) cannot be regarded as necessarily crui-ial. Whenever the implantation is developed at the outer instead of at the inner margin of a follicle, as in the ca.^e of Banks ('12), early destruction, even if not early rupture, of the overlying ovarian stroma and cajisule would seem to be inevitable. Indeed, whenever the layer of ovarian stroma overlying the pliuiiita is thin, very early death of the fetus would seem to lie inevitalde from defective nutrition alone. On the other hand, when jdaccntal development occurs in the region of the follicle directed toward the body of the ovary, great destruction of the ovarian stroma woulil seem to be unavoidable, even if something akin to normal decidual formation actually took ]dace. In the case of Kngelking ('lU), for example, not a trace of an ovary was found in an ovarian pregnancy which had become interstitial. Kven without assuming the complete aidhenticity of this rather ecpiivocal case, it W(uild seem highly probable that the presence of ovarian tissue later in the pri'gnaiiey probably is determined very largely by the location of the fertilized follicle within, or by the e.\act location of the im|ilanta1ion upon the ovary.


Werth ('8T) is said to have collected 12 ca.srs. among which he regarded only that of Leopold ('S2) as authentic. Ix-opolil ('1)9) reported 14. (Jilford ('01), in a splendid succinct review of tlu' literature gave 28 cases. Hi of whi<h he regarded as nndoubteil and 12 as ]>robalile. Koclie {'02) accepted only 12 ca.<es. Filth {'02) accepted 21. Kantorowicz {'04), using the criteria of I.,eopold {'99), together with a microscopic examination as a basis, groujied the cases in the literature a certain, probable, and uncertain. He considered 17 as certain, 10 as probable, and 13 as uncertain. And to the 17 cases regarded as certain by him, Kantorowicz then added two of his own, basing his decision, however, mainly upon the presence of deeidua in the ovaries, thus making 19 cases regarded as authentic by him. Freund and Thome ('06) regarded 23 of all the cases reported up to that time as certain. Norris ami Jlitchell ('08) considered 16 as positive, 15 as probable and !) as fairly probable. Warbanoff ('09) collected 3-t cases and iSTorris ('09) regarded 19 of those contained in the literature of the previous decade as positive; but Williams ('10), from a critical review of the literature up to 1906, and upon tlic l)asis of the criteria of Spiegelberg, regarded only 13 as positive, 17 as highly probable, and 5 as probable. Mapes ('14) collected 30 cases, but wholly from secondary sources, and Lockyer ('17) 42, from the years 1910 to 1917. Of these eases Lockyer accepted 22 as authentic and 20 as questionable ami undecided from the evidence available to him.


This short summary suffices to show that there is as yet no consensus of opinion as to what constitutes an ovarian pregnancy. Although this fact finds its explanation partly in our lack of sufficient knowledge, it is due also to the meagerness of some of the reports. Besides, if complete disintegration and lysis of intraovilrian conceptusps can occur, then it must always remain a. question of opinion in the future wliether some of the cases so rejiorted really were or were not true ovarian jiregnancies. This must remain true no matter how thorouuh the microscopic examination, unless the clinical history or changes in the maternal organism can afford us crucial tests in such cases.


Anyone who reads far into the literature of ovarian pregnancy also nnist become aware of the fact that even very recently skepticism has been carried too far. Jacobson ('08), for example, placed the case of Ivouwer-van Tussenbroek (a ease which finally convinced Bland-Sutton) and that of Webster ('01) in the doubtful class! Furthermore, Jacobson also insisted upon the presence of an cndiryo or fetus as absolutely essential.


It must be emphasized, however, that even a liberal attitude on the part of a reviewer would not justify him in accepting all cases reported as genuine upon the basis of the reports themselves, for they — especially the older ones — often are too meager to enable one to form a reliable opinion. This is illustrated also by such recent reports as those of (iarrard ('16), Martin ('17), Sweeney ('17), and of Mills ('17). Although it must he remembered that from the very nature of tilings it sometimes is impossible to make a report which in itself carries conviction, it is regrettable that in a niunber of relatively recent cases in which such a report apparently could have been made, this was not done. ^lills' case seems to have been an instance of ovarian implantation in a region other than the Graafian follicle, and hence recalls the first ease of Granville and the cases of Franz ('02), Norris ('09), Paucot et Debeyre ('13)?, and perhaps also that of Kouwer ('97) (van Tussenbroek, '99).


From evideiu'e contained in the literature, it is clear that further reports of single cases are not needed for the purpose of emphasizing the occurrence of ovarian pregnancy, yet such reports nevertheless may help in the determination of the relative frequency of this novel and sinister condition, and also throw further light upon its genesis and the finer relations of the implantations, as well as upon other matters. Moreover, since the cases which are accompanied by a careful histologic examination and wliich for this reason alone are wholly unequivocal from an anatomic standpoint still are relatively few, the report of an additional case would seem to be justified. The present specimen (Carnegie Collection, No. Template:CE1322) was donated by Wynne to the Department of Embryology of the Carnegie Institution of Washington, and the following clinical report furnished by him :

Clinical History

Gyn. No. 22303. — The patient, an Italian woman of 37 years, was admitted to, the Gynecological Service of The Johns Hopkins Hospital July 12. 1916, complaining of pain in the lower abdomen, nausea and vomiting.

Family History. — Negative.

Past History. — General health good. She has never had any serious illness. For the past five years following a labor she has had recurring mild attacks of pain in the abdomen without nausea or vomiting.

Menstrual History. — Always regular every month except when pregnant or lactating. Duration four to five days; painless, moderate flow. Last period ,Iune 25, 1916. Last preceding period March 16, 1916. No intermenstrual bleeding before present illness.

Marital. — Married IS years; seven children, oldest 16, youngest born IVa years ago (died. 1915). Has had three miscarriages. History of labors and puerperia vague.

Present Illness. — Began five days ago (July 7, 1916) with sudden pain in lower abdomen, nausea and vomiting. She has had marked dysuria and painful defecation. For 12 hours after onset there was rather profuse bleeding from the vagina and there has been a bloody vaginal discharge since.

(The patient does not understand English and her husband acted as interpreter. )

Physical Examination.— T. 101.6° F. P. 96. R. 20. W. B. C. 8400. Hbg. 46 per cent.

The patient lies in bed grunting with pain. The skin is pale. The lips and raucous membranes are quite pale. There is a systolic blow heard at the apex and increasing toward the base, being loudest over the pulmonic area.

A drop of clear fluid was expressed from the right breast.

The abdominal respiratory movements are limited, although she does not complain of pain on deep inspiration. The flanks bulge somewhat. There is no demonstrable movable dulness. There is tenderness all over the abdomen, most marked over the lower left quadrant. There is increased resistance over the lower abdomen, but no muscle spasm. No masses can be made out.

There is a profuse bloody vaginal discharge. The cervix is pushed up behind the symphysis by a soft, exquisitely tender mass, filling the cul-de-sac. No crepitus is made out. Rectal examination confirms the vaginal. The fundus of the uterus is not felt.

July 13. Ifiia. — Ether examination.

There is a dark, bloody discharge from the vagina. There is no vaginal cyanosis. The cervix is lacerated, firm, and normal in size. The fundus of the uterus is about normal in size and is in anteposition. A boggy mass fills the cul-de-sac and to the right of the uterus a fairly firm mass, the size of a small orange, which is somewhat movable, can be felt. Definite blood-clot crepitus can be felt on rectal examination.

Pre-Operative Diagnosis. — Extra-uterine pregnancy, ruptured.

Operation (Dr. W. R. Holmes, Resident Gynecologist). — A free midline Incision was made below the umbilicus. The peritoneum was blood-stained. The abdomen contained 200 to 300 c. c. of dark fluid blood and clots, and a large clot filled the cul-de-sac. Active bleeding had ceased. The left tube and ovary were normal and free of adhesions. The ri,i;ht tube, which was quite normal in appearance, lay over a mass which had replaced the right ovary. This mass was roughly spherical. 5 to 6 cm. in diameter and semisolid in consistency. Over the surface there were six or eight nodular projections, about 1 cm. in diameter, .^t the top of one of these projections there was a very small opening, from which bloody fluid could be squeezed. The surface of this mass was white with spots of bluish-black discoloration.

The appendi.x was normal except at the lip. where it was adherent to a blood-clot.

The tumor was removed by clamping, tying, and cutting into the^ right iiifundibulo-pelvic ligament and the right utero-ovarian ligament. The right tube was not removed. The appendix was also removed and all blood and clots were cleaned out of the abdomen. The incision was closed without drainage. At the close of the operation, the uterus was curetted. The uterine cavity measured 7.5 cm. in length.

A subcutaneous salt solution infusion was started on the table and continued on the ward until 2000 c. c. had been absorbed.

The patient was in good condition at the end of the operation and made an uneventful recovery.

The urinr on admission contained red blood cells, white blood cells, no casts, acetone, a trace of albumin and no sugar. Several days after the operation it was negative, except for a faint trace of albumin.

August J, V.Hi't. — Discharged in good condition.

Oyn. Path.. \o. i^.?}'i". — Normal endometrium from curettage.

A letter from the patient dated February 12. 191S. said that she had remained in good health since the operation and had given birth to a full-term child January 29. 1918.

Several features in this cliuit-al history ikviTvo coninu'iit. First amoii<^ these is the menstrual a<;e as eoni|)ared witli the size of the chorionie vesiele. Since the eross-seetions of t!u' latter measure 15 x 18 mm. and since it and the amnion are degenerateil and devoid of an enihryo, it is evident that the latter must have <lied a jjood while before the time of o|)eriition. Hence, the menstrual period rei«)rted for June 25, l!)l(i. very evidently wa.s not the lii.it period before prejrnancy supervened, but the lirst jteriod which recurred after the death of the coiHej)tus. Consequently, tiiis prejrnmuy undoul)tedly dates from near April 13, the time of the first omitted period. Moreover, the conceptus must have died long enough before Jtine 25 to have niatie iidiiltition of the succeeding period impossible. It shoidd lie noted, however, that the original menstrual cycle n f) pure nil i/ was broken, for with the customary inter-menstruar period of 28 days, menstnuition normally would have fallen due on June 7 instead of .June 'i'>. Hence, the mainteimnce of the original cycle woidd have brought rufiture of this ovarian pregnancy, as indicated liy the symptoms, on July 7. in direct relation with the on.*et oi menstruation. Nor does it seem unlikely that the hyperemiii accompanying the return of menstruation on June 25, if sudi it really was, may have been jiartly responsii)le for the on.<el of a sufficiently large and persistent hemorrhage to cause tiie slight rujiture indicated by the symjitoms on July 7. It must also be rcmembered in this connection that cases of ovarian ()regiiancy have been reported in which menstruation was uninterrupted. But in the ca.«o of Chiene ('13), fi>r example, the death of the conceptus may have occurred .«o early that the succeeding period was not inhibited, and the same thing may be true of the case of Lea ('10).


Since the material from the curettage, done at the time of operation, showed the presence of a normal endometrium, the uterine deeidua as.sociated with this ])rcgnancy nnist have been shed some time jireviously. Such a conclusion also woulil seem justified iiy the condition of the conceptus. which apparently was unai)le to prevent a return to the normal. Tinabsence of deeidua at the time of operation al.^o suggests thai what was re|)orted as a return of imrmal menstruation on June 25 may have iiecii licniorrhage accompanying the expulsion of the deeidua.

Since, in the present ca.-:c, the .iKiriunic vesicle was so degenerated and .<o completely isolated in a large clot, and especially since no well-iin|)lanted villi were found in the sections and gross jiortions examined, it is not at all probahle that the hemorrhage that caused the rupture ova due to a contemporaneous invasion of the vessels by the fetal trophoblast, such as occurs in uterine and tubal im|ilantations, and as has been actually described also in ovarian implantations by Franz ('02) and by others. In the present, and in similar cases in the literature, it would strm that hemorrhage was made jiossihle also by degenerative changes in the highly va.«cular stroma of the ovary which ha<l been greatly com|ircssed and stretched by the proportituiately large blooddot, the organization of whieli would seem U> iiare been precluded by its size alone.

The fact that relatively few unruptured ovarian pregnancies are recorded suggests that the old tenet that rupture is le.>is likely the more advanced the pregnancy becomi-s. probably is open to .serious doubt, as suggested by Hanks ('12), who bclievetl that the lubes can accommoilatc themselves more readily than the ovary. Hanks stated that in the majority of cases of ovarian jiregnancy rupture occurred in the lirst two or three weeks, and Caturani ('11) also cxjinwed doubts regarding the dictum that rupture of the ovary is lc.«s likely the more advanced the pregnancy. .No one will di'ny, 1 prc.sume, that the .symiitoms of rupture nniy have been totally absent, as rejiortcd in the ca.^es of .N'nrris ('OK) ami (Jrimsdale ('13), but this does not imjily that the ovarian stroma or the germinal cjiithcliuni still surrounded the lull-term i-onceptus. Such an occurrence would be possible only if tlu' ovarian stronni and the overlying germinal c|iithcliinn undi-rwent an astonishing hyperplasia. Although such a thing is conceivable it is decidedly significant that no one has reported any such finding or observed the presence of mitotic figures.

Instead of undergoing hyperplasia the ovarian .stroma in this ca,«e is found invaded, stretched, compre.<.-<cd, anil degenerate, and the germinal epithelium is entirely absent. The fact that several observers have seen what they took for the fibrin layer of Xitabuch also shows that degenerative changes in the ovarian impbinlations may be extensive. Heur-e, it would seem to follow that the absence of symptoms of rupture merely nniy mean that the ovarian stroma and epithelium which ha|ipcned to overlie the fetal membranes gradually have died and degenerated before being forced apart by the expanding conceptus or the increasing hetnorrhage. That such a sequence of events is possible would seem to be undoubted, and merely distension of the ovarian stroma until it com])letely surrounded a full-term pregnancy ' is hardly conceivable ; whereas, the absence of pain upon the yielding of an exceedingly thin degenerate layer of ovarian stroma is quite conceivable.

That rupture may occur very early is exemplified also by the cases of Chiene ('13), Seedorff ('15), and especially by that of Aiming and Littlewood ('01) and of Holland ('11). In such curious instances as that of Grimsdale ('13) one can hardly assume that the ovarian tissue was preserved about the entire .•onceptus, and it is not at all unlikely that full-term ovarian ]iregnancies, which, according to WarbanofE, supplied a sur])risingly large percentage of all cases collected by him, will form a far smaller percentage in the statistics of the near future. Indeed, they already form a far smaller percentage of those reported up to the present, and the advances in diagnosis alone make it very imlikely that in the future many cases of ovarian pregnancy will advance far before being detected.

The present specimen, which had been hardened and cut before it came to my attention, is a firm, nodular, dark-colored mass, 26 X 16 X 11 cm., shown in Fig. 1. In the gross, it especially recalls the specimens of Freund and Thome, Giles ('14-'15), Jaschke ('15), and Lockyer's ('17) second case. The exterior is smooth though bosselated and formed by a rather injected layer which is extremely thin, showing tlie blood-clot beneath, aroimd the greater extent of the specimen. The surface layer is eroded over several small elevated areas in which the blood-clot imderneath is exposed. Hence, the capsule may have been ruptured in several or only in one ol' the areas as noted at the time of the operation. Xear the region of amjjutation through the mesovarium shown to the right in the figure and marked by the corrugations of the hemostat, the tissue overlying the clot becomes more opaque, thicker, and also more yellowish. Here it is studded with small cysts, the character of which in itself suggests ovarian tissue. The color of the area to the right also is suggestive, and the cysts later were foimd to contain a clear viscid fluid, so characteristic of cysts of the ovary. The location of the main portion of the ovarian stroma shows that implantation occurred near the mid-point of the free convex or posterior border of the ovary, and that the stroma forming the sides gradually was forced apart, not by the growing conceptus, except perhaps at the lieginning, but mainly by the liemorrhage itself.

The major ]iortion of the surface of the divided specimen shown in Fig. 2 is composed of blood-clot, the presence of which confirms the " blood-clot crepitus " spoken of in the history. This clot contains an empty vesicle, the wall of which is formed for the greater part of its extent by a thin fibrous layer, except near the proximal or lower portion in the figure, where a thicker portion of ovarian tissue hoods the clot. Approximately only about one-third of the ovarian stroma seems to be preserved.

Near the exterior of the thicker portions of the latter, a small cyst with clear contents is found directly beneath the surface, as indicated in Fig. 4. The distal or upper portion in the figure shows the clot to contain an empty, smoothwalled, degenerate chorionic vesicle, such as is frequently seen in tubal clots.

Examination of the cut surface with the binocular microscope shows the presence of only a few isolated, degenerated, and some hydropic villi scattered through the clot. Examination of the chorionic vesicle shows the presence of only a few attached villi and that the amnion is fused to the chorion. Since the entire sjjecimen obtained at operation is still covered by a layer of ovarian tissue which is unbroken save in a few very small areas, it is e^ddent that we are dealing with a very good example of an undoubted ovarian pregnancy in spite of the absence of an embryo. The latter undoubtedly did not escape through the small rupture in the capsule, for the chorionic vesicle seems entirely intact. Although the absence of a corpus luteum in the opposite or left ovary was not especially mentioned, lack of comment would seem to suggest that none was present, for the ovary very evidently was examined. Hence, this implantation probably took place within the Graafian follicle itself, and not in some other area of the ovary.

Celloidin sections of the excised portion show that the bloodclot contains no fibrin and that it is composed of relatively fresh and fairly well-preserved blood in the region near the main body of the ovarian stroma. The latter is quite normal although decidedly vascular, and contains ova. The layer of the ovarian stroma which surrounds the clot becomes thinner and thinner the nearer the free border is approached. It also becomes more trabeculated, hemorrhagic and degenerate. No overlying layer of smooth muscle, as mentioned by Yoimg and Rhea ('11) and also by Kantorowicz, was seen. Some infiltration with pohauorphonuclear leucocytes is noticeable. Degenerate villi are scattered about in the blood and a few others are apparently still attached to the equally degenerate ovarian stroma. Trophoblast is absent on these, although some of the villi that lie isolated in the clot possess a very evident epithelium and also are associated \\'ith a few small masses of very degenerate syncytium. Only a few degenerate nonvascular vilU are still seen on the chorion. Very little evidence of epithelial proliferation is present on these, despite the fact that the blood in which the vesicle is eml^edded is not very degenerate. This seems to suggest that the hemorrhage wliich caused the rupture of the ovary was comparatively recent, although the conceptus had been dead for some time. Some of the villi scattered about in the blood-clot are outlined by degenerate syncytium only, and nothing but small degenerate masses of the latter are seen on the chorionic membrane or lying about isolated in the degenerate blood foimd in other places. Nevertheless, the epithelium of the chorionic vesicle is thickened at several points. The amnion is fused -nith the chorion and both membranes are very degenerate and destroyed almost completely in several places. The surroimding ovarian tissue, which is markedly vascidar and degenerate, shows infiltration in jdaces, especially where it is stretched over the large clot. No fibrous layer bounds the implantation cavity, as in the case re]>orted by Seedorff. The ovarian stroma merely



Fig. 1. — External appearance of the reconstituted gross specimen (Carnegie Collection, No. 152'^). Natural size.


Fig. 2. — Appearance of cross-section of specimen shown in Fig. 1. X 1.


Fig. 3. — Photograph of a section from a part of the specimen showing the clot largely surrounded by ovarian stroma and containing the empty vesicle. The arrow points to the portion near which degenerate masses of what may have been lutein cells aro found. X 2.


Fig. 4. — Photograph of a section t;il<in from the thick portion of the ovarian stroma near the mesovarlum, showing a well-developed Graafian follicle. X 3.


is slightly condensed here ami thero. and in places contains areas of hyaline ilegeneratioii, tlie exact origin of which conld not be definitely determined. A few of these are found near tile thin bounding layer of the ovarian stroma, hut no lutein layer or even luteal cells could i)e recognized. Tiie only objects seen which might be regarded as ])ossibly luteal in origin arc two microscopic rhoniboidai areas which lie near a small depression upon tiie surface, indicated in Fig. 3. The.se areas, which were covered by a very thin layer of ovarian stroma only, were made up of parallel, ilegenerate, slightly separated cords consisting of a syncytium containing numerous rather pycnotic unequal-sized nuclei. No pigment was seen in these areas, and were it not for the arrangement of the syncytia! cords, one would not be reminded even remotely of a ])ossil)le luteal origin. Although the germinal epithelium was wholly absent in the areas examined, these questionable areas nevertheless may have had such an origin. In the absence of lutein cells the present ca.se corresponds to that of Freund and Thome luid others, and stands in marked contrast to the ca.se • if van Tussenbroek, Franz, Anning and Littlewood ('01 ) and Thompson ('0^). As in the tase of Seedorff, no decidua was jircsent and nothing suggestive of an attempt at decidual formation, as rejwrtcd by Franz, Webster ('04 and '07), and by Caturani and Kantomwicz, was seen in the jiortioiis examined.

In describing his case, Sccdorif declared that in some places "f c-ontact between the fetal and maternal tissues he could not discriminate between trophoblast and connective-ti.ssue cells which looked like decidual cells and lutein cells. It is interesting that Sudortf al.«o spoke of villi which were almost Idled with Langhans' cells, an observation which naturallv makes one wonder whether by any possibility these cmdd not have been Ilofbauer's cells.

The pre-served ovarian tissue which was found near the amputation stumji contained hemorrhagic follicles, as observed al.so by Mall and fuUen ('13). A Graafian follicle 3 mm. in section, shown in Fig. 4, protruded above the rest of the .-troma ajid was quite mature. The presence of this follicle might be taken as an evidence of the occurrence of ovulation during pregnancy, were it not for the fact that the pre'sence of so degenerate a conceptus shows that as far as any effect u|)oii the maternal organism was conc-erned, the pregnancy virtually had been terminated long before. That both ovulation and menstruation can and do return after the death of an ovarian conceptus, but before its removal from the ovary, is illustrated also by ca.ses in the literature, especially by that of Xorri^ ('09). One must assume, however, that few, if any, surviving fetal element.* can be present under these circumstances. This concliLsion also would seem to lie confirmed by the remarkable case of Sencert and .\ron ('14). These authors reported a case of ovarian pregnancy in which nothing but a portion of an umbilical cord ."> mm. long containing Wharton's jelly, two arteries and a vein, and what was regarded as a placenta, remained. The latter was said to be composed of a narrow layer of plasmodiuni and a much thicker layer of trabeculat..! syncytial trophoblast containing blood between the trabecula-.


Because of the singidar structure of this placenta and also because of the failure to find villi or any remnant of the membranes, the authors concluded that the chorionic vesicle therefore could not ha\e reached the villous sUige. How such a supposition can be reconciled with the survival of a portion of an umbilical cord entirely nornnd in structure, it is dilHcult to see. The ovary concerned was brown, of the size of a " large fresh walnut," and contained a tumor, apparently the socalled placenta, which was 2 cm. in diameter. Although these fetal remnants had brought about not only almost complete amenorrhea for two years and also atrophy of the ovary and uterus, ablation of the affected ovary was followed not onlv by a return of the menses, but by a normal pregnancy within .seven months.

A sec-ond instance of ovarian pregnancy of sjiecial interest was that of Giles. Xo fetus was found, although the jtregnancy was unrujitured, and Giles estimati'd that the conceptus had died in the third or fourth week. The operation was not done until five months after the onset of the pregnancy. What is particularly interesting in this case is that Giles s])eaks of the mucoid degeneration of the connective tissue of the villi. The latter were found to be large, much branched, and had ramified in the clot. Since Giles also spoke of one of the illustrations as showing a vascular state of some of the villi, it seems possible that this was a case of hydatiform degeneration, even though there were no signs of activity of the syncvtium. Since the fetal membranes were isolated in a bloodclot very much degenerated and the villi without a Langhans' layer, one scarcely could exi)cct to find much evidence of epithelial jiroliferation .so common (but not essential) in hydatiform degeneration. Giles estimated that four months had elapsed since death of the conccptu-s, and if this specimen really was a hydatiform degeneration, it is the first one observed in ovarian ])reginincy and hence of particular interest for this reason alone.

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