Repeat hydatidiform moles

Repeat hydatidiform of seven cases

Report

CHIEN-TIEN

HSU,

CHUNG-HSUAN

M.D.

CHANGCHIEN,

BIN-CHUAN Taipei,

M.D.

CHANGCHIEN Taiwan,

A L T H o u G H

M.D.

China.

hydatidiform

disease,

individual

M.D. LAI,

CHE-LANG

infrequent

its

mole

repetition

is extremely

rare.

is not

in After

the

only

43

cases

graphic

studies

proved

that

this

in Asia

than

in

theless,

so far

of

repeat

are

from

on

lesion the

as

western

from

In

Japan,

described

tidiform reports

have

7 cases

are

Case

In ever

added

part

of

MiyakeS3

cases

been to the

Neverreports the

world

has reported

hydatidiform while Abel Taiwan

have

common

determine,

this

single

mole.

more

countries.

can

Chesto colGeo-

mo1es44

is far

we

5 cases of repeat moles) among 222, have

literature.

hydatidiform

moles

meager.

the

The fifth pregnancy terminated in the delivery of a full-term living infant on March 18, 1960. At the age of 37, on March 19, 1962, the patient noted vaginal bleeding after amenorrhea for 70 days. Her blood pressure was 188/120 mm. Hg. Marked leg edema and proteinuria were noted. Vaginal examination revealed a uterus the size of an orange. Numerous vesicles were evacuated by curettage. The patient is still living and well. Case 2. Mrs. C. Y. N., aged 36, was married at 23. In 1951, after amenorrhea for 5 months, a dilatation and curettage was performed for hydatidiform mole. In 1952, after amenorrhea for 2 months, hydatidiform mole was diagnosed and, subsequently

an

same

a thorough

search for case reports of repeat moles, ley, Cosgrove, and Preecer’ were able lect

moles

and

of

mole (2 Her?

repeat no

made.

In

hyda-

such this

proved by dilatation and curettage. In January, 1953, after amenorrhea for 2 months, dilatation and curettage was performed and a typical hydatidiform mole was removed. On Oct. 3, 1953, the patient underwent dilata-

case paper

literature.

report

tion and curettage for hydatidiform mole, the diagnosis of which was suspected on the basis of vaginal bleeding, goose egg-sized uterus, and mild toxemia after amenorrhea for 52 days, and finally confirmed by the remove1 of numerous vesicles. On March 13, 1954, she again underwent dilatation and curettage for hydatidiform mole with a goose egg-sized uterus after amenorrhea of 53 days. A frog test was negative on April 23, 1954. On Oct. 18, 1954, a diagnostic curettage was performed because of vaginal bleeding after amenorrhea for 38 days. Numerous typical vesicles were evacuated from a slightly enlarged uterus. After dilatation and curettage three successive intravenous injections of nitrogen mustard (Nitol) , 5 mg., and methyl-bis-P-cholorethyl-

Case 1. Mrs. L. C. C. H., aged 37, was married at 27. Her first three pregnancies between 1953 and 1956, terminated in the delivery of fullterm living infants. On March 2, 1959, the patient complained of genital bleeding after amenorrhea for 3 months. A blood pressure of 132/70 mm. Hg and slight proteinuria indicated mild toxemia. The uterus was the size of a cantaloupe. At dilatation and curettage a large mass of vesicles was evacuated. From the Department of Obstetrics and Gynecology, Provincial Taipei Hospital, and Taipei ‘Medical College. Presented before the Fifty-fifth Annual Meeting of the Formosan Medical Association on Nov. 25, 1962. 543

544

Hsu

et a/.

amine-N-oxide (Nitromin) 5tJ mg., were given for the prevention of choriocarcinoma. On May 4, 195.5, dilatation and curettage was performed bccausc of vaginal bleeding after amenorrhea for 64 days. The uterus was goose egg-sized and numerous typical vrsiclt,s of hydatidiform mole were evacuated. On Nov. 10, 1955, dilatation and curettage was performed for her eighth hydatidiform mole because of vaginal bleeding after amenorrhea for 40 days. Typical molar vesiclrs confirmed the diagnosis. Simple total hysterectomy was performed on Jan. 17, 1956, because of longstanding intermittent vaginal bleeding and persistent positive frog tests after dilatation and curettage. Pathological examination revealed hydatidiform mole and adenomyosis in the uterus. Grossly, the uterus was enlarged and measured 10 by 8 by 4 cm. The cervix was normal except for a slight erosion. The utcrihe body was filled with hydropic degenerated villi, about 1.3 cm. in diameter. Microscopically, a typical picture of hydatidiform molt was revealed. There was, however, a moderate degree of trophohlastic proliferation and superficial trophoblastic invasion of the myometrium. A few endometrial glands in the myometrium indicated adenomyosis. The patient is living and well. Case 3. Mrs. H. Y. S., aged 27, had been married for 5 years. In 1958, at the age of 23, the patient gave birth to a full-term living baby after a normal prenatal course. In the second pregnancy she developed a hydatidiform mole, proved by dilatation and curettage, which was performed because of vaginal bleeding after amenorrhea for 3 months. In 1960 and 1961 she was delivered of full-term living infants. The fifth pregnancy \vas complicated by a hydatidiform mole proved by dilatation and curettage performed on Nov. 15, 1962, because of vaginal bleeding after amenorrhea for 3 months. She is still living and well. Case 4. Mrs. C. T., 33, married at age 24. The first pregnancy at the age of 25 terminated in the delivery of a full-Lerm living baby boy. The second pregnancy was complicated by hydatidiform mole, proved by dilatation and curettage. The third, fourth, and fifth pregnancies all ran normal prenatal courses and ended in normal full-term deliveries. The sixth pregnancy was complicated I)y a hydatidiform mole which was proved by dilatation and curettage performed on Dec. 9, 1958, because of vagina1 hlprding after amenorrhea for ahout 80 days. Pathological examination of the molt revealed

no marked trophoblastic proliferation. Al)out .1 months after the curettage, a growing mass trf thumb sine was noted in the right inguinal disclosed c,horioadyregion. Excisional bi opsy noma destruens with young trophoblastic ~11s infiltrating the adenoid structure of the inguinal lymph nodes. No villi were found. Frog tests on the follow-up examinations reverted to negati\,e :! weeks after the curettage. Normal delivery of a full-term living baby took place in Frbruary, 1960. She is still living and well. Case 5. Mrs. L. C. C., aged 32, married at 18. The first two pregnancies, in I!).50 and 1952, ended in full-term deliveries of living infants. In the third pregnancy, a hydatidiform mole was found at 4 months’ gestation with associated mild toxemia. Dilatation and curettage was performed on Aug. 16, 1953, when the uterine fundus was at the level of the umbilirus. The fourth and fifth pregnancies, in 1954 and 1955, terminated in normal deliveries of living infants. In the sixth pregnancy a hydatidiform mole was diagnosed at 6 months’ gestation with associated severe toxemia. Subtotal hysterectomy was done on April 25, 1958 (when she was 27 years old), when the uterine fundus was 2 fingerhreadths above the umbilicus. The patient is living and well. Case 6. Mrs. K. L. M. T., aged 21, married at 16. The first pregnancy, at 17, was associated with hydatidiform mole of 2 months’ gestational age. The uterus, grapefruit sized, was curettpd. The second pregnancy, at 18, terminated in a normal full-term delivery. The third pregnancy, at 19, was associated with a hydatidiform mole of 3 months’ gestational age. Curettage was performed in May. The uterus was cantaloupe sized. On Dec. 28, 1955, she noted irregular vaginal bleeding, which incrcascd on April 19, at which time a frog test taken was positive (250 F. LJ.). She underwent simple panhysterectomy with adnexectomy 011 May 11, 1956, and extirpation of peanut-sized nodules on the posterior vagina1 wall on May 21. She was discharged on June 2, at which time a small nodule was palpated on the left pelvic wall. Three days after discharge she went into a sudden shock and died. Pathological examination revralrd choriocarcinoma. Case 7. Mrs. L. Y. C., aged 35, was married at 20. The first pregnancy at age 21, terminated in a normal delivery of a full-term living baby girl. ‘I% srcond pregnancy was complicated l,y a

Volume Kumber

H7 4

Table

Repeat

I. Interspersion

of molar

pregnancies

with

normal No.

Author

1 1 2

,4be1 Hori25 Chesley. Cosgrove, PreecelO Harkin?? Kohn’g Rosenthaldl Baz&l* Bottirolir Our series Case 1 Case 3 Case 4 Case 5 Case 6 Case 7 “I’

=

Normal

and

1 3

1 4

/ 5

MPPPPPPPM PMPMPMM M P P

P

M

P

P

M

M

CC)

2

moles

545

pregnancies type

1 7

of pregnancy*

1 8

1 9

/ 10 Ill

1 12 1 13 1 14 / 15

M P M PPMPPPPPPPPM PPPPPPPPPPPMPPM PPPMMPPM PPPPMMPM PPPMPM P M PMPPPMP PPMPPM M P P MMP pregnancy;

hi

=

PM

Hydatidiform

mole;

C

Table II. Repeat hydatidiform moles according to the number of repetitions No. of repetitions of hydatidiform mole

and

1 6

hydatidiform

No. of patients

Authors

31 or 33 Chesley, Cosgrove and Preece,lO Abe, 1 Our series (5 casks), van Deurs,ls Bloch,s Puech (2 cases) ,40 von Winckel,ds Williamson,47 Findley,l’ EssenMijller,ls Muggia,ss Cipriani (doubtful case) ,11 Guiroy,ls Pascale, Mathieu ( 1 or 2 cases),32 Brew,* Sureau,43 Holman,24 Jeffreys and Graffagnino,27 Harkin, Kohn,sg and Rosenthal41 Miyakess (5 cases) Steinberger,bs Haase,21 Depaul,ls Warman, Buzzoni,g Haas,* Morosi,s* Thomas,45 Jeffreys and GrafFagnino,27 Ba&n,4 Bottiroli,? and Authors

3

12

4

4

Osborn,37p onnet,l’

ti

2

Lemaire,sO

7

1

Muggiass

8

1

Ourselves,

18

2

Mack and

ss Fritch,*s and Hori*s and

Enders”’

Case

2

and Catherwood,“l Essen-MGllerl6

Dig-

=

Choriocarcinoma.

hydatidiform mole of 4 months’ gestational age in a cantaloupe-sized uterus. Dilatation and curettage was performed on July 15, 1951. In the third pregnancy, at 24, hydatidiform mole was diagnosed at 4 months’ gestational age, with the uterine fundus reaching the umbilicus. Dilatation and curettage was performed on May 10, 1952. The fourth pregnancy ended in the spontaneous delivery of a full-term living baby girl. The fifth pregnancy terminated in an abortion at 2 months’ gestation completed by curettage. The sixth pregnancy, at 30, was associated with hydatidiform mole of 4% months’ gestational age, with an orange-sized uterus and a negative frog test. Simple panhysterectomy was

done in June, 1958. She is doing well. Comment

Anyone interested in the individual case reports of “recurrence” of hydatidiform moles should refer for details to Chesley, Cosgrove, and Preece’s paperlo in which a thorough survey of the literature was given. The repetition of hydatidiform moles can be classified in two main patterns, i. e., interspersion of molar pregnancies between normal pregnancies and successive repetitions of molar pregnancies. Interspersion of molar pregnancies between normal pregnancies. According to Chesley, Cosgrove, and Preece’O this pattern is unusual. They have been able to collect

546

Hsu et al.

only 5 cases of women who had normal pregnancies occurring between moles. Despite the absence of definite statistics, this pattern should be encountered oftener than the other in view of the preponderance of normal pregnancies over molar, the latter being merely an accidental anomaly of embryonal development. According to Dorland, Gerson, Kehrer, and SchweizeP the ratio of hydatidiform moles in the nullipara to that in the multipara is 28 to 63, 20 to 67, and 18 to 82, respectively. Acosta-Sison”, 3 has also observed the preponderant occurrence of this lesion in older women and multiparas. Kakuz8 has described the relative predilection of molar pregnancies among older women of high parity. Miyake33 has observed that from two thirds to three fourths of the molar pregnancies occur in multiparas and emphasized that although the number of normal pregnancies in the age group of from 45 to 49 dropped abruptly to one tenth of that in the age group of from 40 to 44, (which consisted of 5 per cent of the total pregnancies), the incidence of molar pregnancies in the former group was higher than that in the latter group (8.1 per cent to 5.9 per cent). Our unpublished dataz6 arc roughly in agreement with those of Novak and Seah,3G in that a large proportion of the moles were found in nulliparas or primiparas; the absolute incidence however, should be corrected following Miyake’s or Acosta-Sison’s treatment. In this connection, it will be of interest to observe the pattern of interspersion of molar pregnancies between normal pregnancies.

Authentic cases in the literature are shown in Table I. Consecutive occurrence of hydatidiform moles. Essen-MijlleP cited Hermont as ha\,ing had a patient with six consecutivre moles. and Madame Boivin” referred to a case of Bonus’ in whom three consecutive moles might have occurred within a period of a few months. In the United States six consecutive moles in a single patient were reported by Enders.lj We have seen a patient with eight consecutive moles in a period from 1951 to 1955. It is particularly noteworthy that in this patient two consecutive moles occurred in a year, in 1953, 1954, and 1955, respectively. It is unusual that such an anomaly in development in the embryonal stage could have occurred consecutively and so many times in the same individual in a short period of time. Such an occurrence might shed light on the etiological obscurity of hydatidiform mole. The cases of repetitive moles and the authors of the case reports above are summarized in Table II. Summary

Repetition of hydatidiform moles in the same individual is extremely rare. We have reported 6 cases in which there were normal pregnancies interspersed with hydatidiform moles (5 cases with two moles and one case with three moles), and one case in which eight consecutive moles were seen in 5 years. Six patients are free from malignant degeneration and still living and well. One patient died of choriocarcinoma.

REFERENCES

1. 2. 3. 4. 5.

6.

Abe, M.: Kinki Fujinkagakkai Zassi 11: 866, 1928. Acosta-Sison, H.: Obst. & Gynec. 12: 205, 1958. Acosta-Sison, H.: AM. J. ORST. & GYNEC. 81: 715, 1961. Bazin, J.: Semana, med. 33: 157, 1926. Bloch. M.: Die Blasenmole in wissenschaftlither’ und praktischer Beziehung, Freiburg, 1869, Wagner. Boivin. M. A. V. G.: Nouvelles recherches sur 1’ +origine, la nature et le traitement de la mole vesiculaire ou grossesse hydatique, Paris, 1827, Ches. Mequignon 1’ AinC, P&e.

7. 8. 9. 10. I I. 1’.

13.

Bottiroli, E.: Bol. Sot. de obst. y ginec. de Buenos Aires 20: 847, 1941. Brews, A.: J. Obst. & Gynaec. Brit. Emp. 46: 813, 1939. Buzzoni, R.: Arte ostet. Milan0 27: 353, 1913. Chesley, L. C., Cosgrove, S. A., and Preece, J.: AM. J. OBST. & GYNEC. 52: 311, 1946. Cipriani, F.: Riv. ostet. e ginec. prat. 7: 207, 1925. Depaul, J. A. H.: Lecon de clinique obstetricale. Paris. 1872-1876. V. Adrien Delahave et Cie, p. 280. van Deurs: Ztschr. Geburtsh. u. Gynak. 13: 456, 1843.

Repeat

l-t. 15. 16. 17. 18. 19. 20. 21.

22. 23.

24. 25. 26.

27. 28. 29. 30. 31.

Digonnet, L.: Bull. Sot. roy. belge gynec. et obst. 28: 400, 1939. Enders, R. J.: AM. J. OBST. & GYNEC. 81: 711, 1961. Essen-Moller, E.: Studien iiber die Blasenmole, Wiesbaden, 1912, J. F. Bergmann. Findlev. P.: Am. T. M. SC. 125: 486. 1903. Fritscd,‘H.: Ze&albl. GynPk. 16: 354, 1892. Guiroy, A. J.: Semana med. 34: 1484, 1927. Haas, A.: Med. Klin. 21: 811, 1925. Haase: In Chesley, L. C., C&grove, S. A., and Preece, J.: Ztschr. Geburtsh. u. Gynak. 11: 257, 1841. Harkin, A.: Dublin J. M. SC. 64: 330, 1877. Hitchmann, F.: Halban-Seitz Biologie und Pathologie des Weibes vol. 7, Berlin, 1928, Urban & Schwarzenberg, p. 483. Holman, A.: West. J. Surg. 50: 319, 1942. Hori, H.: Kinki Fujinkagakkai Zassi 1’7: 1439, 1934. Hsu, C. T., Yang, C. C., Changcheng, C. H., Tung, P. H., Lai, C. H., and Chen, C. C.: Unpublished data. Jeffreys, E. M., and Graffagnino, P.: West J. Surg. 52: 29, 1944. Kaku, M.: Textbook of Obstetrics, Vol. 2 ed. 1, Tokyo, Japan, 1955, Nanzando, p. 148. Kohn, H.: Allg. Wien. med. Ztg. 31: 445, 1886. Lemaire: Bull. Sot. d’ obst. et de gynCc. Par. 14: 117, 1911. Mack, H. C., and Catherwood, A. E.: AM. J. OBST. & GYNEC. 20: 670, 1930.

32. 33. 34. 35. 36. 37. 38. 39. 40. 41. 42. 43. 44.

45. 46. 47. 48.

hydatidiform

moles

547

Mathieu, A.: Surg. Gynec. & Obst. 64: 1021, 1937. Miyake, H.: Nippon Fujinkagakkai Zassi 32: 1565, 1937. Morosi, G.: Arch. ostet. e ginec. 20: 750, 1933. Muggia, V.: Ann. ostet. e ginec. 42: 19, 1920. Novak, E., and Seah, C. S.: AM. J. OBST. & GYNEC. 68: 378, 1954. Osborn, A. G.: Brit. M. J. 2: 390, 1864. Osborn, A. G.: Brit. M. J. 2: 180, 1865. Pascale, M.: Riv. ostet. e ginec. prat. 16: 122, 1934. Puech, A.: Gaz. d’ obst. Par. 8: 177, 1879. Rosenthal, E.: J. A. M. A. 10: 255, 1888. Steinberger: Ztschr. Geburtsh. u. GynHk. 2: 96, 1834. Sureau: Bull. Sot. roy. belge gynCc. et obst. 28: 402, 1939. The Joint Project for Study of Choriocarcinema and Hydatidiform Mole in Asia, Ann. New York Acad. SC. 80: 178, 1959. Thomas, F.: Thitse de Paris, 1934. (Cited by Chesley et a1.10) Warman, N.: Zentralbl. Gynak. 16: 353, 1892. Williamson, H.: Tr. Obst. Sot. Lond. 41: 303, 1899. von Winckel, F. C. L. W.: A Textbook of Obstetrics, Philadelphia, 1890, P. Blakiston, Son & Company. p. 304. P. 0. Box Taipei Taipei, Taiwan

No. 12110 China