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Hydatidiform Mole with “Benign” Metastasis to Lung
HYDATIDIFORM MOLE WITH "BENIGN" METASTASIS TO LUNG Histological Evidence of Regressing Lesion in Lung FosTER
J.
jACOBSON,
MILWAUKEE,
:NLD.,
AND NoRBERT ENzER,
M.D.,
W1s.
(From the Department of Obstetrics and Gynecology and the Department of Pathology, Mt. Sinai Hospital)
properties of the normal trophoblast make normal placental INVASIVE histology an interesting study. When the trophoblast undergoes neoplastic growth, the very nature of the growth and the markedly variable pattern provide added problems and room for much speculation. Recognized authorities in the field have categorized epithelial neoplasms of the placenta into hydatidiform mole, chorioadenoma destruens (invasive mole), and choriocarcinoma and have outlined the characteristic features of each group. Helpful though this has been, only brief perusal of the literature proves that many cases do not fall into one classification and that the clinician and the pathologist may be confused by the oftentimes bizarre clinical and/or pathological features. Of particular importance along this line is the histologically benign hydatid mole which, after evacuation from the uterus, is followed by persistently positive biological tests for pregnancy and ''metastatic'' lesions in the lung. It is this particular group in which we are interested in this presentation. Many authors have reported "regressing" or disappearing lesions in the lungs, as evidenced by radiological studies. This has been demonstrated with ordinary hydatid mole,'· 5 as weli as with invasive moles 5 • 10 and the malignant counterpart, choriocarcinoma/• 8 after removal of the tumor and occasionally after x-ray therapy of the lungs. We believe, however, that few, if any, cases have been reported similar to the one presented here, in which a lung resection was performed for metastases from a benign hydatidiform mole, and which offers histological, in addition to radiological, evidence of regressing molar tissue in the lungs. 4
•
Case Report This 22-year-old primigravida was first seen in the office on April 28, 1955, for an initial prenatal visit. The last normal menstrual period was Feb. 27, 1955. Early signs of pregnancy were present and the uterus was 5 to 6 weeks' size as expected. She was next seen on May 16, 1955, 2% weeks later, with a history of brownish spotting for 10 days without pain but with persistent nausea. The uterus had grown much more rapidly than anticipated (instead of the expected 8 weeks' size, it had reached a level of 1 em. 868
Volume 78 Number 4
HYDATIDiFORM MOLE WITH "BENIGN" METASTASiS TO LUNG
869
below the umbilicus-nearly 4 months' size). Bilateral ovarian enlargement was also noted. Blood pressure and urinalysis were normal. Quantitative Aschheim.Zondek tests were performed on the suspicion of hydatidiform mole and were positive in dilutions up to 1:800. The patient was hospitalized on May 20, 1955. Quantitative pregnancy tests remained positive in dilutions up to 1:800. X-ray examination of the abdomen revealed a pelvic mass the size of a 41h to 5 months' gestation, -with no fetal parts visible.. X-ray examination of the lungs was interpreted as negative (Fig. 1). Under observation for 4 days in the hospital, the uterus had reached a level of 2 em. above the umbilicus. On May 24, 1955, an abdominal hysterotomy was performed and the uterine cavity evacuated of a hydatid mole. Bilateral theca-lutein cysts of the ovary were noted but were not removed. The microscopical diagnosis of the removed tissue was ''hydatid degeneration of placenta with focal necrosis.'' The immediate postoperative course was uneventful, but on May 31, the seventh postoperative day, the patient experienced a sudden severe hemorrhage from the uterus, estimated at 800 to 1,000 c.c. Blood replacement was instituted and dilute intravenous Pitocin given; the bleeding stopped spontaneously in 12 hours. The uterus was slightly enlarged and firm, and no hydatid tissue had been passed. An Aschheim·Zondek test on this date proved negative at full strength and at 1:10 and 1:100 dilutions. The patient was next seen 10 days later at the office, at which time there was still some minimal pinkish discharge, the uterus was firm, and the cervix remained soft in consistency. An Aschheim·Zondek test on June 23, 1955, proved positive but was negative in dilutions over 1:200. A persistently positive test on July 5, 1955, along with continued minimal bleeding led to the suspicion of a possible invasive mole or choriocarcinoma, and a dilatation and curettage was done on this date. Curenmgs were m1m· mal and microscopically interpreted as decidual tissue and reactive hyperplasia of the endometrium.
Fig. 1.
Fig. 2.
Fig. 1.-Negative lung findings at the time of surgical evacuation of the mole. Fig. 2.-Lung lesion (arrow), 11 weeks after evacuation of mole.
However, the pregnancy test on July 20, 1955, remained positive in dilutions up to On physical examination, the uterus was normal in size and firm to palpation, the cervix being slightly softened. Persistence of the positive tests for 2 more weeks led to rehospitalization on Aug. 6, 1955. Repeat chest x-ray at this time revealed a round density in the third interspace of the left lung, measuring about 1.5 to 2.5 em. in diam· AtA,. lli'iO' 2) 'J'h" rill'ht lunl! was normal. This was believed to be a metastatic focus, 0 ~~:·sibl; ~h;;ioc~r~ino~a. B;cause of the youthfulness of the patient and her lack of 1:200.
870
JACOBSON AND ENZER
Am.
J. Obst. &
Gynec. October. 1959
children, however, it was decided to do another curettage prior to considering hysterectomy. Curettage on August 10, like that one month before, yielded no evidence of invasive mole or choriocarcinoma, but showed only decidual reaction and reactive hyperplasia of the endometrium. At the request of the patient's family, she was seen at the Mayo Clinic on August 19. At that examination, the lesion in the left lung was noted to have remained the same, but for the first time in 3 months, the quantitative 24 hour urine specimen was entirely negative for chorionic gonadotropin. 'l'he sedimentation rate and complete blood count were within normal limits, as was the physical examination of the pelvic organs. Repetition of the chorionic gonadotropin titer was negative on Aug. 25, 1955, as was the pregnancy test at full strength and at all dilutions. Repeat chest x ·ray examination on August 25 revealed the lesion to be unchanged since the last films. The possibility of a malignancy with metastasis and even of primary tumor or granuloma of the lung was
Fig. 3.-Zone of necrotic and inflammatory exudate aujacent to granulation tissue and extending in an a rea of liquefaction oval homogeneous body with characters suggestive of placental villus. considered. A con~ulting thoracic surgeon f elt ~ urgical exploration was indicated, and on September 9 a segmental resection of the anterior segment of the left upper lobe and superior :;egment of the left lower lobe was p erformed.
Pathologic Desm·iption.- Th e specimen received for examination was a segment of lung tissue 7 em. in diameter. Except for the lesion s about to be described, the lung parenchyma was not remarkable. 'l'he areas calling for investigation consisted of several brown-red elevations in and under the pleura and in the lung parenchyma, all within a radius of about 3 em. The lesions ranged from 2 mm. to 10 mm. in diameter. Most of the lesions were brown, hemorrhagic, and slightly eleva t ed, with central zones of dis· integration. Microscopic studies revealed a rather complex pattern without evidence of :;pecific granuloma or tumor. Frozen sections revealed only inflammatory indices with necrosis and hemorrhage. Fragments were separated for culture and methods designed to recove r bacteria, fungi, and acid-fast bacilli; all were without avail. Sections stained for bac· teria., a.cid-fa.st organisms, and fungi failed to reveal any organisms. The nodular zones were characterized by a periphery of congestion an rl organizing pneumonitis lined by fibrous granulation tissue in which many phagocytes w e re trapped
Volume 78 Number 4
HYDATIDIFORM MOLE WITH "BENIGN" METASTASIS TO LUNG
871
which contained hemosiderin and lipid material staining positive with Sudan IV. Beyond this inflammatory process the lung tissue had an increased vascular pattern, deposits of hemosiderin and lipid material, occasional giant cells, and scattered mixed inflammatory cell infiltration. Of greatest interest were those fields containing the central necrotic core and the lining boundary of granulation tissue. Hemorrhage and necrosis predominated in these areas. In spite of the necrosis, it could be clearly demonstrated that there Fig. 4.
Fig. 5. Fig 4 -Irregularly outlined villus with central necrosis anu marginal distribution of trophobla:st:like cells (arrow), all surrounded by necrotic material and hemorrhage. Fig. 5.-High-power magnification of large cells at periphery of villus.
were patterns highly suggestive of chorionic villi, some almost completely infarcte~, others somewhat better preserved (Figs. 3-5). Surrounding these ''villi'' hyperchromatiC bizarre nucleated cells were attached, sometimes lying free and in several areas clinging to the inner surface of the above-mentioned fibrous membrane (Figs. 5 and 6). These
872
JACOBSON AND ENZER
Am.
J, Obst . & Gynec. October. JQ I 0
cells, in spite of variable morphologic eharacteristics, re~embled t~ophoblast typ es (Fig. 7) and, in add ition to this, a vaguely outline<'! syneyb al f ormatiOn of cells. :ou:d be identified strongly reminiscent of decidu al patterns. In nont> of the many sectiOns and manv fi elds t>xamin ed was th ere any evidence of tumor cell~. ·It was concluded that this ti~sue rep resented the n ec rotizing infarcted phase of embolic implantation in t.h c lung of placental villi and tropl10blast cells. 'l'he bizarre Fig, 6.
Fig. 7.
Fig. 6.-SyncyUum-like arrangement of necrotic cells surrounded bY irregularly distributed masses of necrotic material containing large irregular cells, trophoblasts. Fig. 7.-High-power m agnification of cells of trophoblast type.
and irregular pattern of the '• villi'' and the presence of tlw peripherally arran ged cells referred to previously seemed to justify the opinion that these were actually villi fro m a hydatid :placenta. In some places one could almost persuade one's self that remnants of these villi and these cells were present in distended veins, now occluded by a thrombotic organization (Fig. 8) . Much of our thinkin g with r espect to the cellular minutiae could, of course, be the result of a self-persuasion that all of the histological findings
Volume 78 I\' umber 4
HYDATIDIFO RM MOLE WITH "BENIGN" METASTASI S TO LUNG
873
represented various stages of tissue reaction and necrosis to emboli from the original placental area in the uterus. A definitive opinion about this on purely histological grounds is not warranted. One can offer these observations, findings, and opinions only for what they are worth in the light of the total clinical picture in this caae. At the time of the examination of the resected lung tissue a diagnosis was offered as follows: ''Multiple focal infarctions of the lung with secondary regional pneumonitis due to em· boli of decidual and probable placental villous tissue.'' The entire pattern of the microscopical findings in this case is certainly unlike any other type of inflammatory reaction in the lung that we have experienced and, since all efforts to uncover demonstrable etiological agents other than those indicated by our diag· nosis failed, it would appear that we have here good reason to believe that this case represents a rather striking and perhaps specific tissue reaction to the implantation in the lung, by means of emboli, of specific tiss11e elements, namely, fragments of the hydatid placenta. Postoperative ly the patient progressed very well. A pregnancy test on Sept. 16, 1955, was negative and remained negative thereafter. Menses were restored in October, 1955, and have remained regular and normal since. Follow-up chest x-ray examinations revealed a normal chest. Clinically, the patient has remained well and was last examined on Sept. 9, 1958.
Fig. 8.-Biand thrombotic occlusion in distended vein with pattern suggesting orga nizing
villus.
Comment 7 Since the very early publications on this ~ubject by M.archan
874
JACOBSON AND ENZER
Am.
J. Obst. & Gynec.
October. 1950
of supposedly proved choriocarcinoma with lung metastases, which have disappeared after hysterectomy with or without irradiation of pulmonary m~tas tases and so-called "cures" in these supposedly fatal cases. 1 • 8 Often the histological diagnosis has been questioned either by the authors themselves or by editorial comments. Although many questionable or speculative cases have been reported in the literature, we are going to mention only a :few typical reports from t~e more recent literature. Hunt and associates 4 regarded their series of hydatid mole cases as questionably malignant, and felt that roentgenologic~lly demonstrable pulmonary lesions which disappear after irradiation may have represented merely inflammatory lesions. Nolan and his group 8 reported 2 cases oE chorionepithelioma with pulmonary metastases with recovery, that is, regt·cssion of pulmonary lesions after hysterectomy plus irradiation to .lungs. (Novak,S commenting in the editor's remarks, says slides showed no evidence of choriocarcinoma but rather molar tissue and the lung x-rays probably represented inflammatory lesions or molar metastases.) Novak, 12 in a review in 1954, spoke of the regression of "metastatic" areas of trophoblastic tissue associated with chorioadenoma destruens following removal of the primary tumor. In a recent review, Logan and Motyloff5 reported 72 cases of hydatid mole, eight o:f which were unusual. Four of these latter cases revealed "benign" pulmonary metHstases with regression. Our case is unique in that not only was a pulmonary lesion demonstrated roentgenologically, but a segment of lung tissue was resected and offered histological evidence of "regressing" molar tissue in the lung. The histologica1 studies provided many questions, however, particularly, Could we justifiably call these areas in the lung "tissue of placental origin"' Did the necrotic and ~
Volume 78 Number 4
HYDATIDIFORM MOLE WITH "BENIGN" METASTASIS TO LUNG
""'" <''llu
demonstrated regressing metastatic hydatid molar tissue-offering histological evidence of such regression instead of radiological "speculation" of regreRsion heretofore presented in the literature. Many thanks to Dr. John I. Brewer for his kind assistance in reviewing slides and photomicrograpl" and for his valuable ~uggestions in preparation of this paper.
References L 2. 3. 4. 5. 6.
7. 8. 9. 10. 11. 12. 13.
Delfs, E.: Obst. & Gynec. 9: 1, 1957. Ewing, J. A.: Surg. Gynec. & Obst. 10: 366, 1910. Hertig, A. T., and Sheldon, W. E.: AM . •T. 0BST. & GYNEC. 53: 1, 1947. Hunt, S., Dockerty, M., and Randall, L.: Obst. & Gynec. 1: 6, 593, 1953. Logan, B. J., and Motyloff, L.: AM. J. 0BST. & GYNEC. 75: 5, 1134, 1958. Marchand, F.: Quoted by Ewing.2 Marchand, F.: Obst. & Gynaec. Brit. Emp. 4: 74, 1903. Nolan, J., Cook, E., Jr., and Dahlke, W.: West .•T. Surg. 63: 101, 1955. comment by E. Novak in Obst. & Gynec. Surv., February, 1956.) Novak, E.: AM. J. 0BST. & GYNEC. 59: 1355, 1950. Novak, E.: AM. J. 0BST. & GYNEC. 68: 387, 1954. Novak, E.: J. A.M. A. 78: 1771, 1922. Novak, E., and Seah, C. 8.: AM. J. OBST. & GYNEC. 67: 933, 1954. Williams, J. W.: Johns Hopkins Hosp. Rep. 4: 46, 1895.
(Editorial
J.
jACOBSON,
MILWAUKEE,
:NLD.,
AND NoRBERT ENzER,
M.D.,
W1s.
(From the Department of Obstetrics and Gynecology and the Department of Pathology, Mt. Sinai Hospital)
properties of the normal trophoblast make normal placental INVASIVE histology an interesting study. When the trophoblast undergoes neoplastic growth, the very nature of the growth and the markedly variable pattern provide added problems and room for much speculation. Recognized authorities in the field have categorized epithelial neoplasms of the placenta into hydatidiform mole, chorioadenoma destruens (invasive mole), and choriocarcinoma and have outlined the characteristic features of each group. Helpful though this has been, only brief perusal of the literature proves that many cases do not fall into one classification and that the clinician and the pathologist may be confused by the oftentimes bizarre clinical and/or pathological features. Of particular importance along this line is the histologically benign hydatid mole which, after evacuation from the uterus, is followed by persistently positive biological tests for pregnancy and ''metastatic'' lesions in the lung. It is this particular group in which we are interested in this presentation. Many authors have reported "regressing" or disappearing lesions in the lungs, as evidenced by radiological studies. This has been demonstrated with ordinary hydatid mole,'· 5 as weli as with invasive moles 5 • 10 and the malignant counterpart, choriocarcinoma/• 8 after removal of the tumor and occasionally after x-ray therapy of the lungs. We believe, however, that few, if any, cases have been reported similar to the one presented here, in which a lung resection was performed for metastases from a benign hydatidiform mole, and which offers histological, in addition to radiological, evidence of regressing molar tissue in the lungs. 4
•
Case Report This 22-year-old primigravida was first seen in the office on April 28, 1955, for an initial prenatal visit. The last normal menstrual period was Feb. 27, 1955. Early signs of pregnancy were present and the uterus was 5 to 6 weeks' size as expected. She was next seen on May 16, 1955, 2% weeks later, with a history of brownish spotting for 10 days without pain but with persistent nausea. The uterus had grown much more rapidly than anticipated (instead of the expected 8 weeks' size, it had reached a level of 1 em. 868
Volume 78 Number 4
HYDATIDiFORM MOLE WITH "BENIGN" METASTASiS TO LUNG
869
below the umbilicus-nearly 4 months' size). Bilateral ovarian enlargement was also noted. Blood pressure and urinalysis were normal. Quantitative Aschheim.Zondek tests were performed on the suspicion of hydatidiform mole and were positive in dilutions up to 1:800. The patient was hospitalized on May 20, 1955. Quantitative pregnancy tests remained positive in dilutions up to 1:800. X-ray examination of the abdomen revealed a pelvic mass the size of a 41h to 5 months' gestation, -with no fetal parts visible.. X-ray examination of the lungs was interpreted as negative (Fig. 1). Under observation for 4 days in the hospital, the uterus had reached a level of 2 em. above the umbilicus. On May 24, 1955, an abdominal hysterotomy was performed and the uterine cavity evacuated of a hydatid mole. Bilateral theca-lutein cysts of the ovary were noted but were not removed. The microscopical diagnosis of the removed tissue was ''hydatid degeneration of placenta with focal necrosis.'' The immediate postoperative course was uneventful, but on May 31, the seventh postoperative day, the patient experienced a sudden severe hemorrhage from the uterus, estimated at 800 to 1,000 c.c. Blood replacement was instituted and dilute intravenous Pitocin given; the bleeding stopped spontaneously in 12 hours. The uterus was slightly enlarged and firm, and no hydatid tissue had been passed. An Aschheim·Zondek test on this date proved negative at full strength and at 1:10 and 1:100 dilutions. The patient was next seen 10 days later at the office, at which time there was still some minimal pinkish discharge, the uterus was firm, and the cervix remained soft in consistency. An Aschheim·Zondek test on June 23, 1955, proved positive but was negative in dilutions over 1:200. A persistently positive test on July 5, 1955, along with continued minimal bleeding led to the suspicion of a possible invasive mole or choriocarcinoma, and a dilatation and curettage was done on this date. Curenmgs were m1m· mal and microscopically interpreted as decidual tissue and reactive hyperplasia of the endometrium.
Fig. 1.
Fig. 2.
Fig. 1.-Negative lung findings at the time of surgical evacuation of the mole. Fig. 2.-Lung lesion (arrow), 11 weeks after evacuation of mole.
However, the pregnancy test on July 20, 1955, remained positive in dilutions up to On physical examination, the uterus was normal in size and firm to palpation, the cervix being slightly softened. Persistence of the positive tests for 2 more weeks led to rehospitalization on Aug. 6, 1955. Repeat chest x-ray at this time revealed a round density in the third interspace of the left lung, measuring about 1.5 to 2.5 em. in diam· AtA,. lli'iO' 2) 'J'h" rill'ht lunl! was normal. This was believed to be a metastatic focus, 0 ~~:·sibl; ~h;;ioc~r~ino~a. B;cause of the youthfulness of the patient and her lack of 1:200.
870
JACOBSON AND ENZER
Am.
J. Obst. &
Gynec. October. 1959
children, however, it was decided to do another curettage prior to considering hysterectomy. Curettage on August 10, like that one month before, yielded no evidence of invasive mole or choriocarcinoma, but showed only decidual reaction and reactive hyperplasia of the endometrium. At the request of the patient's family, she was seen at the Mayo Clinic on August 19. At that examination, the lesion in the left lung was noted to have remained the same, but for the first time in 3 months, the quantitative 24 hour urine specimen was entirely negative for chorionic gonadotropin. 'l'he sedimentation rate and complete blood count were within normal limits, as was the physical examination of the pelvic organs. Repetition of the chorionic gonadotropin titer was negative on Aug. 25, 1955, as was the pregnancy test at full strength and at all dilutions. Repeat chest x ·ray examination on August 25 revealed the lesion to be unchanged since the last films. The possibility of a malignancy with metastasis and even of primary tumor or granuloma of the lung was
Fig. 3.-Zone of necrotic and inflammatory exudate aujacent to granulation tissue and extending in an a rea of liquefaction oval homogeneous body with characters suggestive of placental villus. considered. A con~ulting thoracic surgeon f elt ~ urgical exploration was indicated, and on September 9 a segmental resection of the anterior segment of the left upper lobe and superior :;egment of the left lower lobe was p erformed.
Pathologic Desm·iption.- Th e specimen received for examination was a segment of lung tissue 7 em. in diameter. Except for the lesion s about to be described, the lung parenchyma was not remarkable. 'l'he areas calling for investigation consisted of several brown-red elevations in and under the pleura and in the lung parenchyma, all within a radius of about 3 em. The lesions ranged from 2 mm. to 10 mm. in diameter. Most of the lesions were brown, hemorrhagic, and slightly eleva t ed, with central zones of dis· integration. Microscopic studies revealed a rather complex pattern without evidence of :;pecific granuloma or tumor. Frozen sections revealed only inflammatory indices with necrosis and hemorrhage. Fragments were separated for culture and methods designed to recove r bacteria, fungi, and acid-fast bacilli; all were without avail. Sections stained for bac· teria., a.cid-fa.st organisms, and fungi failed to reveal any organisms. The nodular zones were characterized by a periphery of congestion an rl organizing pneumonitis lined by fibrous granulation tissue in which many phagocytes w e re trapped
Volume 78 Number 4
HYDATIDIFORM MOLE WITH "BENIGN" METASTASIS TO LUNG
871
which contained hemosiderin and lipid material staining positive with Sudan IV. Beyond this inflammatory process the lung tissue had an increased vascular pattern, deposits of hemosiderin and lipid material, occasional giant cells, and scattered mixed inflammatory cell infiltration. Of greatest interest were those fields containing the central necrotic core and the lining boundary of granulation tissue. Hemorrhage and necrosis predominated in these areas. In spite of the necrosis, it could be clearly demonstrated that there Fig. 4.
Fig. 5. Fig 4 -Irregularly outlined villus with central necrosis anu marginal distribution of trophobla:st:like cells (arrow), all surrounded by necrotic material and hemorrhage. Fig. 5.-High-power magnification of large cells at periphery of villus.
were patterns highly suggestive of chorionic villi, some almost completely infarcte~, others somewhat better preserved (Figs. 3-5). Surrounding these ''villi'' hyperchromatiC bizarre nucleated cells were attached, sometimes lying free and in several areas clinging to the inner surface of the above-mentioned fibrous membrane (Figs. 5 and 6). These
872
JACOBSON AND ENZER
Am.
J, Obst . & Gynec. October. JQ I 0
cells, in spite of variable morphologic eharacteristics, re~embled t~ophoblast typ es (Fig. 7) and, in add ition to this, a vaguely outline<'! syneyb al f ormatiOn of cells. :ou:d be identified strongly reminiscent of decidu al patterns. In nont> of the many sectiOns and manv fi elds t>xamin ed was th ere any evidence of tumor cell~. ·It was concluded that this ti~sue rep resented the n ec rotizing infarcted phase of embolic implantation in t.h c lung of placental villi and tropl10blast cells. 'l'he bizarre Fig, 6.
Fig. 7.
Fig. 6.-SyncyUum-like arrangement of necrotic cells surrounded bY irregularly distributed masses of necrotic material containing large irregular cells, trophoblasts. Fig. 7.-High-power m agnification of cells of trophoblast type.
and irregular pattern of the '• villi'' and the presence of tlw peripherally arran ged cells referred to previously seemed to justify the opinion that these were actually villi fro m a hydatid :placenta. In some places one could almost persuade one's self that remnants of these villi and these cells were present in distended veins, now occluded by a thrombotic organization (Fig. 8) . Much of our thinkin g with r espect to the cellular minutiae could, of course, be the result of a self-persuasion that all of the histological findings
Volume 78 I\' umber 4
HYDATIDIFO RM MOLE WITH "BENIGN" METASTASI S TO LUNG
873
represented various stages of tissue reaction and necrosis to emboli from the original placental area in the uterus. A definitive opinion about this on purely histological grounds is not warranted. One can offer these observations, findings, and opinions only for what they are worth in the light of the total clinical picture in this caae. At the time of the examination of the resected lung tissue a diagnosis was offered as follows: ''Multiple focal infarctions of the lung with secondary regional pneumonitis due to em· boli of decidual and probable placental villous tissue.'' The entire pattern of the microscopical findings in this case is certainly unlike any other type of inflammatory reaction in the lung that we have experienced and, since all efforts to uncover demonstrable etiological agents other than those indicated by our diag· nosis failed, it would appear that we have here good reason to believe that this case represents a rather striking and perhaps specific tissue reaction to the implantation in the lung, by means of emboli, of specific tiss11e elements, namely, fragments of the hydatid placenta. Postoperative ly the patient progressed very well. A pregnancy test on Sept. 16, 1955, was negative and remained negative thereafter. Menses were restored in October, 1955, and have remained regular and normal since. Follow-up chest x-ray examinations revealed a normal chest. Clinically, the patient has remained well and was last examined on Sept. 9, 1958.
Fig. 8.-Biand thrombotic occlusion in distended vein with pattern suggesting orga nizing
villus.
Comment 7 Since the very early publications on this ~ubject by M.archan
874
JACOBSON AND ENZER
Am.
J. Obst. & Gynec.
October. 1950
of supposedly proved choriocarcinoma with lung metastases, which have disappeared after hysterectomy with or without irradiation of pulmonary m~tas tases and so-called "cures" in these supposedly fatal cases. 1 • 8 Often the histological diagnosis has been questioned either by the authors themselves or by editorial comments. Although many questionable or speculative cases have been reported in the literature, we are going to mention only a :few typical reports from t~e more recent literature. Hunt and associates 4 regarded their series of hydatid mole cases as questionably malignant, and felt that roentgenologic~lly demonstrable pulmonary lesions which disappear after irradiation may have represented merely inflammatory lesions. Nolan and his group 8 reported 2 cases oE chorionepithelioma with pulmonary metastases with recovery, that is, regt·cssion of pulmonary lesions after hysterectomy plus irradiation to .lungs. (Novak,S commenting in the editor's remarks, says slides showed no evidence of choriocarcinoma but rather molar tissue and the lung x-rays probably represented inflammatory lesions or molar metastases.) Novak, 12 in a review in 1954, spoke of the regression of "metastatic" areas of trophoblastic tissue associated with chorioadenoma destruens following removal of the primary tumor. In a recent review, Logan and Motyloff5 reported 72 cases of hydatid mole, eight o:f which were unusual. Four of these latter cases revealed "benign" pulmonary metHstases with regression. Our case is unique in that not only was a pulmonary lesion demonstrated roentgenologically, but a segment of lung tissue was resected and offered histological evidence of "regressing" molar tissue in the lung. The histologica1 studies provided many questions, however, particularly, Could we justifiably call these areas in the lung "tissue of placental origin"' Did the necrotic and ~
Volume 78 Number 4
HYDATIDIFORM MOLE WITH "BENIGN" METASTASIS TO LUNG
""'" <''llu
demonstrated regressing metastatic hydatid molar tissue-offering histological evidence of such regression instead of radiological "speculation" of regreRsion heretofore presented in the literature. Many thanks to Dr. John I. Brewer for his kind assistance in reviewing slides and photomicrograpl" and for his valuable ~uggestions in preparation of this paper.
References L 2. 3. 4. 5. 6.
7. 8. 9. 10. 11. 12. 13.
Delfs, E.: Obst. & Gynec. 9: 1, 1957. Ewing, J. A.: Surg. Gynec. & Obst. 10: 366, 1910. Hertig, A. T., and Sheldon, W. E.: AM . •T. 0BST. & GYNEC. 53: 1, 1947. Hunt, S., Dockerty, M., and Randall, L.: Obst. & Gynec. 1: 6, 593, 1953. Logan, B. J., and Motyloff, L.: AM. J. 0BST. & GYNEC. 75: 5, 1134, 1958. Marchand, F.: Quoted by Ewing.2 Marchand, F.: Obst. & Gynaec. Brit. Emp. 4: 74, 1903. Nolan, J., Cook, E., Jr., and Dahlke, W.: West .•T. Surg. 63: 101, 1955. comment by E. Novak in Obst. & Gynec. Surv., February, 1956.) Novak, E.: AM. J. 0BST. & GYNEC. 59: 1355, 1950. Novak, E.: AM. J. 0BST. & GYNEC. 68: 387, 1954. Novak, E.: J. A.M. A. 78: 1771, 1922. Novak, E., and Seah, C. 8.: AM. J. OBST. & GYNEC. 67: 933, 1954. Williams, J. W.: Johns Hopkins Hosp. Rep. 4: 46, 1895.
(Editorial