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The Breus mole
The 8 reus mole Report of two cases
CLYDE V. VON DER AHE, M.D. Beverly Hills, California
A N u N u s u A L complication of pregnancy was originally described by the Austrian gynecologist Carl Breus, in 1892, as tuberous subchorial hematoma of the decidua on the basis of 5 cases observed by him. These cases presented the usual signs of early pregnancy, followed by symptoms of threatened and missed abortion, with subsequent spontaneous expulsion of the mole, in variable periods of time up to 11 months after the onset of symptoms. In each case the placenta demonstrated diffuse intervillous hemorrhage with deposition of fibrin subchorially, thus raising the chorionic plate into tuberous folds and mounds limited by the attachment of the subchorionic septa. Embryos were present in all 5 cases. They were malformed and their growth stunted. Relatively little has been written recently concerning the Breus mole. Reports published by European authors 60 to 70 years ago dealt primarily with the pathology of the Breus mole. Taussig/ wrote a classic monograph entitled, "The Hematom-Mole; Its Clinical and Pathological Characteristics and Relation to Early Hydramnios." This monumental work was read before the Chicago Gynecological Society, in 1904. One of
the more recent reports has been that of Linthwaite 6 who, in 1963, reported on 2 cases of moles observed by him. We are presenting 2 additional cases of the Breus mole. One is unusual in that the Breus mole recurred in a subsequent pregnancy. Taussig 7 has reported a similar occurrence. Case reports Case No. 1. The patient was seen by the attending physician in 1958, in the emergency room of the Centinella Valley Community Hospital, because of an abortion at 6 weeks' gestation. The specimen consisted of a placenta that measured approximately 7.0 x 4.5 x 2.5 em. in its greatest dimension. When the amniotic sac was extended, there was noted a large subchorionic hematoma which was arranged in ridges. An abortive embyro was present measuring 0.4 em. in total length. These findings were typical of the so-called Breus mole. Microscopic sections showed placental tissue with a large subchorionic hematoma. The diagnosis was embryo and placenta (hematomole or Breus's mole). The patient conceived again in the spring of 1961. The uterus did not grow past 11 weeks' size and in September, 1961, she had an abortion of another Breus's mole. The specimen consisted of an oval piece of reddish gray placental tissue measuring 10.0 x 6.0 x 2.5 em. with a cystic space measuring 9.0 em. across, containing amber fluid in which the umbilical cord or fetus was not grossly recognizable. Cut section of the placental tissue showed it to measure up to 0.6 em. in thickness and showed multiple hemorrhages throughout. Gross findings were compatible with the Breus mole. Microscopic sections of placenta show multiple hemorrhages, chiefly subchorionic in location, along with
From the Department of Obstetrics and Gynecology. University of Southern California School of Medicine, and St. John's Hospital. Presented as a part of a Symposium of Interesting and Unusual Cases, at the Thirty-first Annual Meeting of the Pacific Coast Obstetrical and Gynecological Society, Santa Barbara, California, Nov. 4-7, 1964.
699
700
july 1 196.-, •\rn. J. Oh>t. I; Gvneo·.
Von der Ahe
rather extensive hyalinization and focal areas of necrosis with acute inflammatory cell infiltrations. The diagnosis was Breus's mole, placenta. Case No. 2. A 28-year-old gravida iii, para ii was seen on March 31, 1964, with the complaint of early pregnancy. The last normal menses was Jan. 10, 1964. From February 14 to February 19, she noticed a dark brownish discharge. Vaginal spotting recurred on March 17, and from March 28 until March 31. The patient also noted painful breasts and urinary frequency. A pelvic examination revealed a normal healthy vagina with a soft cyanotic cervix. The uterine corpus was questionably enlarged. The right adnexa contained an ovarian cyst 3 x 4 em. The left adnexal area was surgically absent. A pregnancy test was positive. On April 21, the patient stated that she had been spotting for one week. Bed rest was ordered. Spotting did not improve, and continued daily until May 3, 1964. This was described as a dark brown spotting with a foul odor and an occasional gush of bright red blood. Examination on May II, !964, revealed that the cervix was closed and firm. The uterus was anterior and normal in size. A diagnosis of a spontaneous abortion was made. Seventeen days later she began bleeding heavily and was admitted to St. John's hospital for dilatation and curettage. At operation the cervix was dilated 4 em. Protruding through the external os was a large g-lobular mass of placental tissuP. The uterus was anterior in position and no larger than normal size. There wcre no palpable tumors. In the right adnexal area then~ was a large cystic mass 5 ern. in diameter. The left adnexal arPa was surgically absent. Obtained from the uterine cavity was a roughly oval to spherical shaggy mass of tissue 6 em. in diameter. On section, the tissue surrounded a chorionic vesicle measuring 3 em. in greatest dimension, containing a macerated embryonic structure measuring 12 mm. crown-rump length. The wall of the sac was convertPd into a multinodular grapelike blue hemorrhagic structun·, individual nodules of hemorrhage measuring from 0.5 to 2.5 em. in diameter, suggestive of the Breus mole. Sections demonstrated the retained chorionic vesicle in which there were mounds of clotted blood elevating the chorionic membrane from the surface of the underlying placental villi and decidual tissue. Placental villi appeared partially degenerated, but islands of trophoblast appeared viable. Decidua was hemorrhagic and partially autolyzed. The diagnosis
was Breus's mole with degenerated nonviablefetus. The postoperative course was unewntful. The patient was discharged from the hospital on the second postoperative day. In suhs<·quenl office follow-up visits, her normal health '' itl> regular menses has returned. Comment
In his original article, Breus divided these moles into two categories depending upon whether or not the amniotic cavity was obliterated. Those cases in which the amniotic cavity was completely obliterated were referred to as fleshy or carneous moles. Cases of incomplete obliteration of the amniotic cavity have been called bv various names such as tuberous moles, tuberous fleshy moles, ova tuberculosa, hematoma mole, ami thrBreus mole. Grossly, the moles present as a large rounded mass, up to lO em. in diameter, with a dark currant jelly appearance. When the mole is opened, bluish discoloration under the chorionic plate is immediatelv apparent. Amniotic fluid may or may not be present. Occasionally, it is turbid or hazelcolored in appearance. An embryo may !:w present. Its size, however, is inconsistent with the duration of pregnancy. Gottschalk ratht't' arbitrarily set 17 mm. as the upper limit for these mole rmbryos.' There is a high mctdence of fetal malformation associated with the Breus mole. Microscopically diffuse hemorrhage occurs under the amnion and chorion. Fibrin is present. Organized thrombi may be present if the hemorrhage is of long duration. Inflammatory cells and placental villi an' also present. Of great interest is the method of formation of hematomata beneath the chorionic plate. Breus 3 believed that intervillous hemorrhage occurred following death of the embryo primarily with continued growth of tlw amnion and chorion. As a result, the chorion was pushed up into folds causing the appearance typical of the mole. Other authors have disagreed, feeling that the death of the embryo was secondary to the intervillous hemorrhage.
Volume 92
Breus mole
701
Number:)
Taussig' stated that after the death of the embryo in the first or second month an increase in volume of the amniotic fluid and fetal membranes occurred causing a hydrarnnios. This continued up to a certain
point and then was followed by gradual absorption of amniotic fluid. By the negative pressure thus produced, folds or invaginations of the amnion and chorion occurred. These became filled with blood from the intervillous spaces resulting in the formation of the hematoma of the tuberous mole. While the exact mechanism of the forma-
tion of the tuberous mole is at present unknown, certainly the mechanics of placental circulation as elaborated by Ramsay, and the well-known fact that fetal membranes and nl::JcPnt>l .._, fetal o!!TOW followin!! ---------- to r---------- continue death as in hydatidiform mole, lend plausibility to Taussig's theory. Torpin 8 is of the opinion that subchorial hematoma mole, placenta circumvallate, and placenta membranacea arise from excessively deep implantation of the fertilized ovum into the uterine decidua.
REFERENCES
I. Benirschke, K.: Obst. & Gynec. 18: 309, 1961. 2. Benirschke, K.: AM. ]. 0BST. & GvNEC. 84:
1595, 1962. Breus, C.: F. Deuticke Leipzig & Wien, 1892. 4. Faulkner, R. L., and Douglas, M.: Essentials of obstetrical and gynecological pathology, ed. 2, St. Louis, 1949, The C. V. Mosby Company, p. 311. ~>.
5. Huber, C. P., Carter, J. E., and Vellios, F.: AM. ]. 0BST. & GYNEC. 81: 560, 1960. 6. Linthwaite, R. F.: ]. A. M. A. 186: 867, 1964. 7. Taussig, F. ].: Am. ]. Obst. 50: 456, 1904. 8. Torpin, R.: J. Obst. & Gynaec. Brit. Emp. 57: 990, 1960.
CLYDE V. VON DER AHE, M.D. Beverly Hills, California
A N u N u s u A L complication of pregnancy was originally described by the Austrian gynecologist Carl Breus, in 1892, as tuberous subchorial hematoma of the decidua on the basis of 5 cases observed by him. These cases presented the usual signs of early pregnancy, followed by symptoms of threatened and missed abortion, with subsequent spontaneous expulsion of the mole, in variable periods of time up to 11 months after the onset of symptoms. In each case the placenta demonstrated diffuse intervillous hemorrhage with deposition of fibrin subchorially, thus raising the chorionic plate into tuberous folds and mounds limited by the attachment of the subchorionic septa. Embryos were present in all 5 cases. They were malformed and their growth stunted. Relatively little has been written recently concerning the Breus mole. Reports published by European authors 60 to 70 years ago dealt primarily with the pathology of the Breus mole. Taussig/ wrote a classic monograph entitled, "The Hematom-Mole; Its Clinical and Pathological Characteristics and Relation to Early Hydramnios." This monumental work was read before the Chicago Gynecological Society, in 1904. One of
the more recent reports has been that of Linthwaite 6 who, in 1963, reported on 2 cases of moles observed by him. We are presenting 2 additional cases of the Breus mole. One is unusual in that the Breus mole recurred in a subsequent pregnancy. Taussig 7 has reported a similar occurrence. Case reports Case No. 1. The patient was seen by the attending physician in 1958, in the emergency room of the Centinella Valley Community Hospital, because of an abortion at 6 weeks' gestation. The specimen consisted of a placenta that measured approximately 7.0 x 4.5 x 2.5 em. in its greatest dimension. When the amniotic sac was extended, there was noted a large subchorionic hematoma which was arranged in ridges. An abortive embyro was present measuring 0.4 em. in total length. These findings were typical of the so-called Breus mole. Microscopic sections showed placental tissue with a large subchorionic hematoma. The diagnosis was embryo and placenta (hematomole or Breus's mole). The patient conceived again in the spring of 1961. The uterus did not grow past 11 weeks' size and in September, 1961, she had an abortion of another Breus's mole. The specimen consisted of an oval piece of reddish gray placental tissue measuring 10.0 x 6.0 x 2.5 em. with a cystic space measuring 9.0 em. across, containing amber fluid in which the umbilical cord or fetus was not grossly recognizable. Cut section of the placental tissue showed it to measure up to 0.6 em. in thickness and showed multiple hemorrhages throughout. Gross findings were compatible with the Breus mole. Microscopic sections of placenta show multiple hemorrhages, chiefly subchorionic in location, along with
From the Department of Obstetrics and Gynecology. University of Southern California School of Medicine, and St. John's Hospital. Presented as a part of a Symposium of Interesting and Unusual Cases, at the Thirty-first Annual Meeting of the Pacific Coast Obstetrical and Gynecological Society, Santa Barbara, California, Nov. 4-7, 1964.
699
700
july 1 196.-, •\rn. J. Oh>t. I; Gvneo·.
Von der Ahe
rather extensive hyalinization and focal areas of necrosis with acute inflammatory cell infiltrations. The diagnosis was Breus's mole, placenta. Case No. 2. A 28-year-old gravida iii, para ii was seen on March 31, 1964, with the complaint of early pregnancy. The last normal menses was Jan. 10, 1964. From February 14 to February 19, she noticed a dark brownish discharge. Vaginal spotting recurred on March 17, and from March 28 until March 31. The patient also noted painful breasts and urinary frequency. A pelvic examination revealed a normal healthy vagina with a soft cyanotic cervix. The uterine corpus was questionably enlarged. The right adnexa contained an ovarian cyst 3 x 4 em. The left adnexal area was surgically absent. A pregnancy test was positive. On April 21, the patient stated that she had been spotting for one week. Bed rest was ordered. Spotting did not improve, and continued daily until May 3, 1964. This was described as a dark brown spotting with a foul odor and an occasional gush of bright red blood. Examination on May II, !964, revealed that the cervix was closed and firm. The uterus was anterior and normal in size. A diagnosis of a spontaneous abortion was made. Seventeen days later she began bleeding heavily and was admitted to St. John's hospital for dilatation and curettage. At operation the cervix was dilated 4 em. Protruding through the external os was a large g-lobular mass of placental tissuP. The uterus was anterior in position and no larger than normal size. There wcre no palpable tumors. In the right adnexal area then~ was a large cystic mass 5 ern. in diameter. The left adnexal arPa was surgically absent. Obtained from the uterine cavity was a roughly oval to spherical shaggy mass of tissue 6 em. in diameter. On section, the tissue surrounded a chorionic vesicle measuring 3 em. in greatest dimension, containing a macerated embryonic structure measuring 12 mm. crown-rump length. The wall of the sac was convertPd into a multinodular grapelike blue hemorrhagic structun·, individual nodules of hemorrhage measuring from 0.5 to 2.5 em. in diameter, suggestive of the Breus mole. Sections demonstrated the retained chorionic vesicle in which there were mounds of clotted blood elevating the chorionic membrane from the surface of the underlying placental villi and decidual tissue. Placental villi appeared partially degenerated, but islands of trophoblast appeared viable. Decidua was hemorrhagic and partially autolyzed. The diagnosis
was Breus's mole with degenerated nonviablefetus. The postoperative course was unewntful. The patient was discharged from the hospital on the second postoperative day. In suhs<·quenl office follow-up visits, her normal health '' itl> regular menses has returned. Comment
In his original article, Breus divided these moles into two categories depending upon whether or not the amniotic cavity was obliterated. Those cases in which the amniotic cavity was completely obliterated were referred to as fleshy or carneous moles. Cases of incomplete obliteration of the amniotic cavity have been called bv various names such as tuberous moles, tuberous fleshy moles, ova tuberculosa, hematoma mole, ami thrBreus mole. Grossly, the moles present as a large rounded mass, up to lO em. in diameter, with a dark currant jelly appearance. When the mole is opened, bluish discoloration under the chorionic plate is immediatelv apparent. Amniotic fluid may or may not be present. Occasionally, it is turbid or hazelcolored in appearance. An embryo may !:w present. Its size, however, is inconsistent with the duration of pregnancy. Gottschalk ratht't' arbitrarily set 17 mm. as the upper limit for these mole rmbryos.' There is a high mctdence of fetal malformation associated with the Breus mole. Microscopically diffuse hemorrhage occurs under the amnion and chorion. Fibrin is present. Organized thrombi may be present if the hemorrhage is of long duration. Inflammatory cells and placental villi an' also present. Of great interest is the method of formation of hematomata beneath the chorionic plate. Breus 3 believed that intervillous hemorrhage occurred following death of the embryo primarily with continued growth of tlw amnion and chorion. As a result, the chorion was pushed up into folds causing the appearance typical of the mole. Other authors have disagreed, feeling that the death of the embryo was secondary to the intervillous hemorrhage.
Volume 92
Breus mole
701
Number:)
Taussig' stated that after the death of the embryo in the first or second month an increase in volume of the amniotic fluid and fetal membranes occurred causing a hydrarnnios. This continued up to a certain
point and then was followed by gradual absorption of amniotic fluid. By the negative pressure thus produced, folds or invaginations of the amnion and chorion occurred. These became filled with blood from the intervillous spaces resulting in the formation of the hematoma of the tuberous mole. While the exact mechanism of the forma-
tion of the tuberous mole is at present unknown, certainly the mechanics of placental circulation as elaborated by Ramsay, and the well-known fact that fetal membranes and nl::JcPnt>l .._, fetal o!!TOW followin!! ---------- to r---------- continue death as in hydatidiform mole, lend plausibility to Taussig's theory. Torpin 8 is of the opinion that subchorial hematoma mole, placenta circumvallate, and placenta membranacea arise from excessively deep implantation of the fertilized ovum into the uterine decidua.
REFERENCES
I. Benirschke, K.: Obst. & Gynec. 18: 309, 1961. 2. Benirschke, K.: AM. ]. 0BST. & GvNEC. 84:
1595, 1962. Breus, C.: F. Deuticke Leipzig & Wien, 1892. 4. Faulkner, R. L., and Douglas, M.: Essentials of obstetrical and gynecological pathology, ed. 2, St. Louis, 1949, The C. V. Mosby Company, p. 311. ~>.
5. Huber, C. P., Carter, J. E., and Vellios, F.: AM. ]. 0BST. & GYNEC. 81: 560, 1960. 6. Linthwaite, R. F.: ]. A. M. A. 186: 867, 1964. 7. Taussig, F. ].: Am. ]. Obst. 50: 456, 1904. 8. Torpin, R.: J. Obst. & Gynaec. Brit. Emp. 57: 990, 1960.