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Transitional Hydatidiform Mole Associated with Extensive Hydatidi Degeneration of the Placenta and a Well Developed Fetus
TBAif&~ .Pti~IJI ...........ftD•.WJTII
EX'IWIIU
IJORENZO
R.
JI'I1MftD . . JIIJIIJtl . . . OJ'.'f8:P~A
.dD A WE.L-~ND 1"&708 Report of a Case
BERRY, :MAJOR, MC, USA, MARTIN A. BWERDLOW, CAP'fAIN, USA,* AND ALFRED EDWARDS, CAP'l'AIN,
MC,
MC,
USA
(From the 'Pathology Branch, Sixth Army Area Medical Laboratory, Fort Baker, Calif.)
HIS is a report of a case of a large hydatidiform mole associated with extensive hydatid degeneration of the placenta and a well-developed fetus. The occurrence of this combination is rare. GreenhiW has stated, ''A fetus is seldom found when a hydatidiform mole is present.'' A review of the literature over the past thirty-five years did not reveal a case with exactly identical features. Several cases were noted, however, which were similar. Bowles 2 reported a case of "Extensive Hydatidiform Mole Formation With Living Child," and he stated, "In 9,501 deliveries at the Queen's Hospital, Honolulu, from 1932 to 1941, there was only one recorded instance of a single ovum with coexisting fetus and hydatidiform mole.'' Cline" reported a case of '' Hydatidiform Degeneration of Placenta, Complete With Fetus.'' Mueller and Lapp 6 reported a case of "A Single Ovum Pregnancy, With Marked Molar Degeneration of the Placenta, 'l'erminating in Birth of a Living Child.'' Beltz and Hutchins 1 reported a ''Coexisting Hydatidiform Mole With a Living Child and Placenta Praevia.''
T
Clinical Course The patient was a 26-year-old pregnant white woman, para iii, gravida iv. Her pre· natal course was uneventful until approximately the sixteenth week of ge,station when she suddenly developed abdominal pain and. cramps. These symptoms abated with eonserva· tive management only to recur 48 hours ·later with profuse vaginal bleeding, shock, and finally the spontaneouH passage of a well-developed but nonviable fetus, foflowed by a placenta and approximately 1 gallon of fluid and grapelike vesicles. Uterine scrapings seven weeks after this episode revealed no residual molar tissue. Physical examination twelve weeks after the spontaneous abortion revealed a nor· mally regressed uterus and a decrease in size of the patient's previously cystically enlarged ovaries. Regular monthly quantitative F'riedman tests have been negative.
Pathological Examination.Fetus: The fetus was a well-developed male that measured 18 em. from crown to heel, 13 em. from crown to rump, and weighed 140 grams (Fig. 1). There were no external or internal abnormalities noted at necropsy and the fetal age was calculated as approximately sixteen weeks, in accordance with the outlines of Potter.7 •Present address, Menorah Medical Center, 4949 Rockhill Road, Kansas City 10, Mo. 912
Volume 69 Number 4
HYDATIDIFORM MOLE WITH WELL-DEVELOPED FETUS
Fig. 1.-Fetus with placenta attached.
Transparent and opaque cystic structures occupy large
areas of the plaC'f'nta.
Fig. 2.-Hydatidiform mole, expelled following the placenta in Fig. 1. noted between the vesides.
Decidua and fibrin are
!114
BERRY ,
~WERVLO\\ ',
AND
J<~D\\' A IW H
·\ n,.
J.
Oh>t. &Gy n
April , iCJ S<
Place nta: The placenta measured 12 em. in diameter and ~.onsi ste d of usual placenta admixed with adherent, variable-sized gray vesicular structnnls which wm·e filled with clea r, straw-colored finid. The microscopic appearan ce varied from the usual-appearing villi through varion ~ intermediar~· forms to huge edemato us cystic villi, many of wh ich were fenestrated . A striking anrl prominent feature was the absence of visible intravillous capillarie>< in th P cysti c villi. Fig. 3.
Fig. 4.
Fig. a.-Section of trans\tional a rea s of pla<:!enta showing· u8ual villi as well I.'S larger
edematous cystic villi. ( XlOO:; reduced l,f,. l Fig. 4.-Sectlon of avascular .villus, cystically rlistended with edema ftuid and covert>t1 by a thic k mantle of syncytial snd cytotrophoblastlc <'ells.
< X HO ; reduced I,!;.)
Volume 69 ~umber
4
J!YllA'flDll<'OHM :HOLE \Vl'I'Jl WELL-DEVELOPED FE1'US
!115
Hydatidiform mole: The molar tissue consisted of approximately 1 gallon of cystic grapelike structures hung together by hemorrhagic stalks of thin connective tissue. The vesicula structures measmed from less than 1 CHI, in diameter to over· 3 em, in diameter (Fig. 2). Histologically the vesicles were similar to t ho~e in the placenta except that here manr Yilli were eovere
Comment This appears to be a case of transitional hydatidiform mole. 'l'he term transitional is used in the same mannf'r as it was used by Hertig and Edrnonds 5 in reporting "The Genesis of Hydatidiform Molf'." These authors have described this type of mole as abortuses averaging sixteen and sixth-tenths weeks in duration. Grossly, they are defined as ''abortuses in which various portions of the ovum (chorion, villi, amnion, and embryo) can still Jw identified hut in which enough villi have sufficient hydatidiform degeneration to make the (liagnosis clearly evident from the gross specimen." Transitional stages were noted hetween early hydatidiform degeneration in the villi and classic hydatidiform mole in 15 pathologic ova described in their report. In our case transitional stages are noted between early hydatidiform degeneration of the villi of the placenta (Fig. 3) and the true hydatidiform mole (Fig. 4). The possibility exists that this was a twin pregnancy with the mole developing in the associated placenta with subsequent death and absorption of the fetus. This seems unlikely in this case as no remnants of a second placenta were noted and because of the transitional stages from normal villi to true hydatidiform mole seen in the placenta still attached to the fetus. It is interesting to speculate as to the contents and the histologic structure of the cysts or other enlargement present in each ovary. It is also of interest to speculate as to the hormonal relationship between the cystic ovaries and the hydatidiform mole. In our case the ovaries grew smaller after expulsion of the mole. This case supports the contention that a fetus can live while getting nourishment from only a portion of the placenta even though a part is hydatidiform. That this is the case has also been shown by others. 1 • 2 • s, 6 The rarity of this type of lesion is emphasized by Williams, 8 Greenhill, 4 mH1 otheri'i. Williams reported in 1918 that no instance of a viable fetus had been found together with hydatidiform mole formation of the placenta in 17,980 ohstetrical cases treated in ,Johns Hopkins Hospital.
Summary and Conclusion A ease of transitional hydatidiform mole characterized by extensive hydatidiform degeneration of the placenta and a well-developed fetus is presented. The dynamic sequence of events in this case appears to be that the fetus and placenta at first developed in the usual manner but that sometime during development a hydatidiform change gradually took place in the placenta. The fetus lived as long as there was enough functioning placenta to maintain it, nml, wht>n thf're W!'IR no 1ongel' Rufficient p1ar.\'nta. the fetuR diril and tht> pa-
916
BERRY, SWEHDLOW, .\ND EDWARDH
Arn ..T. Obst. & Gynec. Apr1l, JOSS
tient aborted. In addition to this, as a factor iu bl"ingiug about abortion, was the mechanical distention of the uterus hy the lar·ge amount of mole tissue.
References 1. Beltz, R. V., and Hutchins, C. L.: Ohio 'M • •T. 47: 832, 1951.
2. Bowles, H. E.: AM.•T. 0BST. & GYNEG. 46: 154, 1943. 3. Cline, A.: AM. J. OBBT. & GvNEC. 51: 1.3~, 1946.
4. Greenhill, .r. P.: Obstetrics in General Practice, Chicago, 1940, ~rhc Year Book Pultlishers, Inc., p. 92. 5. H!Jrtig, A. T., and Edmonds, H. \V.: Areh. Path. 30: 260, 1940. 6. Mueller, C. W., and Lapp, W. A.: New York .T. Med. 50: 1279, 1950. 7. Potter, E. L.: Pathology of the Fetu~ and the Newborn, Chicago, 1952, The Year Book Publishers, Inc., pp. 11-14. 8. Williams, J. W.: quoted by Curti~, A. H., editor: Obstetrics and Gynecology, Philadelphia, 1933, W. B. Saunders Company, vol. 2, p. fi63.
EX'IWIIU
IJORENZO
R.
JI'I1MftD . . JIIJIIJtl . . . OJ'.'f8:P~A
.dD A WE.L-~ND 1"&708 Report of a Case
BERRY, :MAJOR, MC, USA, MARTIN A. BWERDLOW, CAP'fAIN, USA,* AND ALFRED EDWARDS, CAP'l'AIN,
MC,
MC,
USA
(From the 'Pathology Branch, Sixth Army Area Medical Laboratory, Fort Baker, Calif.)
HIS is a report of a case of a large hydatidiform mole associated with extensive hydatid degeneration of the placenta and a well-developed fetus. The occurrence of this combination is rare. GreenhiW has stated, ''A fetus is seldom found when a hydatidiform mole is present.'' A review of the literature over the past thirty-five years did not reveal a case with exactly identical features. Several cases were noted, however, which were similar. Bowles 2 reported a case of "Extensive Hydatidiform Mole Formation With Living Child," and he stated, "In 9,501 deliveries at the Queen's Hospital, Honolulu, from 1932 to 1941, there was only one recorded instance of a single ovum with coexisting fetus and hydatidiform mole.'' Cline" reported a case of '' Hydatidiform Degeneration of Placenta, Complete With Fetus.'' Mueller and Lapp 6 reported a case of "A Single Ovum Pregnancy, With Marked Molar Degeneration of the Placenta, 'l'erminating in Birth of a Living Child.'' Beltz and Hutchins 1 reported a ''Coexisting Hydatidiform Mole With a Living Child and Placenta Praevia.''
T
Clinical Course The patient was a 26-year-old pregnant white woman, para iii, gravida iv. Her pre· natal course was uneventful until approximately the sixteenth week of ge,station when she suddenly developed abdominal pain and. cramps. These symptoms abated with eonserva· tive management only to recur 48 hours ·later with profuse vaginal bleeding, shock, and finally the spontaneouH passage of a well-developed but nonviable fetus, foflowed by a placenta and approximately 1 gallon of fluid and grapelike vesicles. Uterine scrapings seven weeks after this episode revealed no residual molar tissue. Physical examination twelve weeks after the spontaneous abortion revealed a nor· mally regressed uterus and a decrease in size of the patient's previously cystically enlarged ovaries. Regular monthly quantitative F'riedman tests have been negative.
Pathological Examination.Fetus: The fetus was a well-developed male that measured 18 em. from crown to heel, 13 em. from crown to rump, and weighed 140 grams (Fig. 1). There were no external or internal abnormalities noted at necropsy and the fetal age was calculated as approximately sixteen weeks, in accordance with the outlines of Potter.7 •Present address, Menorah Medical Center, 4949 Rockhill Road, Kansas City 10, Mo. 912
Volume 69 Number 4
HYDATIDIFORM MOLE WITH WELL-DEVELOPED FETUS
Fig. 1.-Fetus with placenta attached.
Transparent and opaque cystic structures occupy large
areas of the plaC'f'nta.
Fig. 2.-Hydatidiform mole, expelled following the placenta in Fig. 1. noted between the vesides.
Decidua and fibrin are
!114
BERRY ,
~WERVLO\\ ',
AND
J<~D\\' A IW H
·\ n,.
J.
Oh>t. &Gy n
April , iCJ S<
Place nta: The placenta measured 12 em. in diameter and ~.onsi ste d of usual placenta admixed with adherent, variable-sized gray vesicular structnnls which wm·e filled with clea r, straw-colored finid. The microscopic appearan ce varied from the usual-appearing villi through varion ~ intermediar~· forms to huge edemato us cystic villi, many of wh ich were fenestrated . A striking anrl prominent feature was the absence of visible intravillous capillarie>< in th P cysti c villi. Fig. 3.
Fig. 4.
Fig. a.-Section of trans\tional a rea s of pla<:!enta showing· u8ual villi as well I.'S larger
edematous cystic villi. ( XlOO:; reduced l,f,. l Fig. 4.-Sectlon of avascular .villus, cystically rlistended with edema ftuid and covert>t1 by a thic k mantle of syncytial snd cytotrophoblastlc <'ells.
< X HO ; reduced I,!;.)
Volume 69 ~umber
4
J!YllA'flDll<'OHM :HOLE \Vl'I'Jl WELL-DEVELOPED FE1'US
!115
Hydatidiform mole: The molar tissue consisted of approximately 1 gallon of cystic grapelike structures hung together by hemorrhagic stalks of thin connective tissue. The vesicula structures measmed from less than 1 CHI, in diameter to over· 3 em, in diameter (Fig. 2). Histologically the vesicles were similar to t ho~e in the placenta except that here manr Yilli were eovere
Comment This appears to be a case of transitional hydatidiform mole. 'l'he term transitional is used in the same mannf'r as it was used by Hertig and Edrnonds 5 in reporting "The Genesis of Hydatidiform Molf'." These authors have described this type of mole as abortuses averaging sixteen and sixth-tenths weeks in duration. Grossly, they are defined as ''abortuses in which various portions of the ovum (chorion, villi, amnion, and embryo) can still Jw identified hut in which enough villi have sufficient hydatidiform degeneration to make the (liagnosis clearly evident from the gross specimen." Transitional stages were noted hetween early hydatidiform degeneration in the villi and classic hydatidiform mole in 15 pathologic ova described in their report. In our case transitional stages are noted between early hydatidiform degeneration of the villi of the placenta (Fig. 3) and the true hydatidiform mole (Fig. 4). The possibility exists that this was a twin pregnancy with the mole developing in the associated placenta with subsequent death and absorption of the fetus. This seems unlikely in this case as no remnants of a second placenta were noted and because of the transitional stages from normal villi to true hydatidiform mole seen in the placenta still attached to the fetus. It is interesting to speculate as to the contents and the histologic structure of the cysts or other enlargement present in each ovary. It is also of interest to speculate as to the hormonal relationship between the cystic ovaries and the hydatidiform mole. In our case the ovaries grew smaller after expulsion of the mole. This case supports the contention that a fetus can live while getting nourishment from only a portion of the placenta even though a part is hydatidiform. That this is the case has also been shown by others. 1 • 2 • s, 6 The rarity of this type of lesion is emphasized by Williams, 8 Greenhill, 4 mH1 otheri'i. Williams reported in 1918 that no instance of a viable fetus had been found together with hydatidiform mole formation of the placenta in 17,980 ohstetrical cases treated in ,Johns Hopkins Hospital.
Summary and Conclusion A ease of transitional hydatidiform mole characterized by extensive hydatidiform degeneration of the placenta and a well-developed fetus is presented. The dynamic sequence of events in this case appears to be that the fetus and placenta at first developed in the usual manner but that sometime during development a hydatidiform change gradually took place in the placenta. The fetus lived as long as there was enough functioning placenta to maintain it, nml, wht>n thf're W!'IR no 1ongel' Rufficient p1ar.\'nta. the fetuR diril and tht> pa-
916
BERRY, SWEHDLOW, .\ND EDWARDH
Arn ..T. Obst. & Gynec. Apr1l, JOSS
tient aborted. In addition to this, as a factor iu bl"ingiug about abortion, was the mechanical distention of the uterus hy the lar·ge amount of mole tissue.
References 1. Beltz, R. V., and Hutchins, C. L.: Ohio 'M • •T. 47: 832, 1951.
2. Bowles, H. E.: AM.•T. 0BST. & GYNEG. 46: 154, 1943. 3. Cline, A.: AM. J. OBBT. & GvNEC. 51: 1.3~, 1946.
4. Greenhill, .r. P.: Obstetrics in General Practice, Chicago, 1940, ~rhc Year Book Pultlishers, Inc., p. 92. 5. H!Jrtig, A. T., and Edmonds, H. \V.: Areh. Path. 30: 260, 1940. 6. Mueller, C. W., and Lapp, W. A.: New York .T. Med. 50: 1279, 1950. 7. Potter, E. L.: Pathology of the Fetu~ and the Newborn, Chicago, 1952, The Year Book Publishers, Inc., pp. 11-14. 8. Williams, J. W.: quoted by Curti~, A. H., editor: Obstetrics and Gynecology, Philadelphia, 1933, W. B. Saunders Company, vol. 2, p. fi63.