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Struma ovarii with ascites and hydrothorax
Struma ovarii with ascites and hydrothorax HIROSHI KAWAHARA, M.D. Akita City, Japan
On pelvic examination following paracentesis, it was impossible to determine the exact origin of the tumor, which filled the true pelvis. The adnexa could not be clearly delineated, but the tumor seemed to be attached to the uterine fundus. The pelvic tumor did not apparently increase in size during these 7 years. Approximately a week prior to admission, the patient complained of cough and dyspnea for the first time. Physical examination revealed dullness in the right lung base posteriorly with diminution of breath sounds and voice sound upon auscultation. The left side of the thorax was essentially normal on physical examination. Chest x-ray on admission showed fluid in the right side of the chest (Fig. 1). Thoracocentesis was performed and about 2,000 mi. of yellow fluid was obtained. Neither the pleural nor the abdominal fluid was found to contain any malignant cells. Results of the laboratory examinations of the chest and abdominal fluids are shown in Table II. The icteric index was 4 units. The total serum protein was 6.3 Gm. per 100 mi. The cephalin flocculation liver test was recorded as 2-plus. The alkaline phosphatase was 3 Bodansky units. The thymol turbidity test was 3.5, zinc sulfate test was 14.2, bromsulphthalein test was 17.5 per cent, Gros reaction was reported as 3-plus, glutamic oxalacetic transaminase was 24 units per cubic centimeter per minute, and glutamic pyruvic transaminase was 20 units per cubic centimeter per minute. Urinalysis was negative. The phenolsulfonphthalein kidney test was recorded as 26 per cent by 15 minutes, 43.5 per cent by 30 minutes, 61.5 per cent by 60 minutes, 95.0 per cent by 120 minutes. A clinical diagnosis was made of Meigs' syndrome. On Jan. 11, 1962, under general anesthesia, laparotomy through a mid-line incision was
I N 1 9 3 7 Meigs and Cass 1 reported 7 cases of ascites and hydrothorax with ovarian fibromas that were cured after removal of the fibroma. This condition was named Meigs' syndrome by Rhoads and Terrell. 2 The incidence of struma ovarii was found to be 0.3 per cent in a series of 1,000 solid ovarian tumors, according to Higuchi and Kato. 3 Only 6 previous cases of struma ovarii associated with ascites and hydrothorax have been reported (Table I). About 12 per cent of struma ovarii are functioning/0 and there is a small group of cases in which the thyroid tumor of the ovary has produced clinical evidences of hyperthyroidismY· 12 ' 13 Report of case
Mrs. H. K., an 83-year-old woman, para ii, was admitted to the Nakadori Hospital on Dec. 13, 1961, with a chief complaint of cough, dyspnea, and ascites. She gave a history of being well until about 7 years previously, when she complained of soreness in the lower abdomen. Ascites and a pelvic tumor were detected by a doctor whom she consulted. Although an operation was advised, she at first did not agree to this on account of her age. Subsequently, paracentesis yielded 2,000 mi. of yellow fluid. Paracentesis was required every 2 to 3 weeks, approximately 1,000 to 2,000 mi. of yellow fluid being removed on each occasion. Paracenteses were then repeated for 7 years until her admission to the hospital. No malignant cells were found in the abdominal fluid, but a huge globoid mass, firm, nontender, and movable on palpation, was evident, extending from the pelvis to a level about 2 inches below the umbilicus. From the Department of Obstetrics and Gynecology, Nakadori Hospital.
85
86
Januan' l)
Kawahara
196:~
Am. J. Ohst. & Gyncc,
Table I. Summary of 7 cases of struma o\·arii with ascites and hydrothorax --------
< -~~------------
---·-·-
!.ora-
lion
Case Jl/o.
Author
Dote :Date of of A.~e Mari-! z,;o. pubtal ojJof of \{(]pachi!era- lie at;on t'"n . tient /U.I , dren
/,0((1-
lion
of Complaints
tumor !
of hid1 otho0 peratwn
Jax
1944 1947
·11
M
?
Marked mcrease lil Right Right Total hysterecto my including S!ZC of abdomen right adnPxa as well as left
2
Baskin and 1950 1951 ( ~ounst>ller 0
46
M
2
Cough, dyspnea. Pnlargement of abdomen
Left
3
E"slcy"
1954
1955
•}8
M
2
.\scites and hydrothorax
Right Right
4
Schuldcs 7
1955 1956
·!9
M
0
Enlargement of ab- Left domen and pain Ill right sidP of chest
5
N
1959
52
M
8
Enlargement of abdomen and dyspnea
Right Bilat- Bilateral salpingooophorectomy era!
6
Zellner;'
1961
ti:)
M
4
Enlargement of abdomen
Left
7
Kawahara
M
2
Cough and dyspnea Left
Calmenson r•t ~]!
1961
Wl
----·----------·----·- --------------
Fig. l. Right hydrothorax.
-----
------------- ··-----
Right Same
Right Bilateral oophorectomy
Right Same Right Left salpinguoophorectomy ----
------------·--·-
carried out. A large lobular, solid and 'ystic tumor, about 1::l em. in diameter, was found in the leftnt's poor g<"nt'ral condition, only a left salpingo-oophorectomy was performed. The specinwn was a lobulated finn and cystic mass measuring 15 by 1:l by R ern and weighing 600 grams (Fig. 2 \. The solid tissne was found to he pn·d••minantly of the fetal adenoma typ•~ of thyroid tumor, the so-called struma ovarii. In mor" solid areas where the epithelial elements Wf'l'<" arranged in closely packed cords, small glandular structures were found to contain little colloid (Fig. 3). An occasional larger acinus contained n~cognizahle colloid (Fig. 4). Malignant changes were nowhere evident. The patient's convalescence was uneventful. She had had no recurrence of pleural fluid or ascites, and roentgenogram of the thorax on January 25 showed no evidence of abnorrnalitv. ShP has gained weight and is asymptomatic
Volume 85 Number 1
Struma ovar11 with ascites and hydrothorax
87
Fig. 2. The left ovarian tumor is lobular and cystic. On section, lobulated cysts are encountered which contained mucus and somewhat gelatinoid material.
,..
Fig. 3. Closely packed, small glandular structures contained little colloid.
Fig. 4. An occasional large acinus contained colloid.
88
Jauuar; 1, 1963 Am, J. Obst. & Gvnee.
Kawahara
Table II. Laboratory findings m chest and abdominal fluid -----------c---------.-----Specific gravity Rivalta reaction Protein Sugar Pseudo-
Abdominal fluid
Chest fluid
1_020
1.022
+
+ 4.4 Gm./100 mi. 92 mg./100 mi.
4.8 Gm./100 mi. 100 mg./100 mL
illllClll
Comment
Meigs 11 • 15 has redefined and limited the syndrome to include cases showing ( 1) a fibroma-like tumor of the ovary (fibroma, thecoma, granulosa cell tumor, and Brenner tumor); (2) ascites; (3) hydrothorax; and ( ·l! cure following removal of the tumor. Yet there is some confusion in this definition. Since a certain number of the theca cell and granulosa cell tumors are malignant, as Kimbrough 16 said, their inclusion seems illogicaL On the other hand, various ovarian tumors associated with ascites and hydrothorax, even malignant ovarian tumors without peritoneal and pleural metastasis, have been reported. Some authors 17 - 20 have included such malignant ovarian tumors with ascites and hydrothorax in the Meigs' syndrome group. Kimbrough 16 has offered two alternatives in the definition of Meigs' syndrome with which I am in agreement--first, that the syndrome might include only fibroma of the ovary with associated ascites and hydrothorax as originally defined; second, perhaps more logically, the syndrome should include all ovarian tumors both solid and cystic
REFERENCES
1. Meigs, J. V., and Cass, J. W.: AM. J. OssT. & GYNEC. 33: 249, 1937. 2. Rhoads, J. E., and Terrell, A. W.: J. A. M. A. 109: 1684, 1937. 3. Higuchi, K., and Kato, T.: Sanfuginkanosekai (Japanese) 12: 729, 1960. t. Calmenson, M., Dockerty, M. B., and Bianco, J. J.: Surg. Gynec. & Obst. 84: 181, 1947.
with associated ascites and hydrothorax, provided there is no evidence of metastasis in the peritoneal and pleural cavities Lemon and Higgins 21 have demonstrated experimentally the pathway hv ,vhich lymphatic absorption of particulate matter proceeds through the diaphragm. Meigs, Armstrong, and Hamilton 22 were able to test this tht'ory on 2 patients with Meigs' syndrome. This experiment is suggestiYc evidence that the abdominal fluid arriws in the chest by the same pathway as thf' India ink particles There are also many theories and speculations as to why the fluid occurs in the abdomen with benign ovarian tumors. Some have attempted to explain the origin of ascites on the basis of Selye's 23 alarm reaction. Schenck and Eis 24 offered an interesting hypothesis derived from Selye's alarm reaction with the added factor of lymphatic obstruction by the compressing effect of the tumor. One of the more recent and more nearly adequate explanations of the etiologic basis for the ascitic fluid is that offered by Dockerty and Masson, 25 who believed the significant factor in the production of ascites was edema in the tumor itself or in its pedicle. Summary
1. A case of struma ovarii associated with ascites and hydrothorax is reported. 2. Since there is some inconsistency in the definitions of Meigs' syndrome it is proposed that either ( 1) only fibromas or {2) all ovarian tumors both benign and malignant should be included, provided there is no evidence of metastasis in the peritoneal or pleural cavities.
5. Baskin, R. H., and Counseller, \T. S.: Proc. Staff. Meet. Mayo Clin. 20: 60, 1951. 6. Easley, C. M.: Obst. & Gynec. 6: 630, 1955. 7. Schuldes, H.: Geburtsh. u. Frauenh. 16: 450, 1956. 8. Nakasato, T.: Sanfuginkanosekai (]apanesei 11: 1620, 1959. 9. Zellner, K.: Acta obst. et gynec. scandinav. 40: 40, 1961.
Volume 85 Number 1
Struma ovarii with ascites and hydrothorax 89
10. Anderson, W. A. D.: Pathology, ed. 3, St. Louis, 1957, The C. V. Mosby Company, p. 1081. 11. Moench, G. L.: Surg. Gynec. & Obst. 49: 150, 1929. 12. Kleine, H.: Arch. Gynak. 158: 62, 1934. 13. Kovacs, F.: Arch. Gynak. 122: 766, 1924. 14. Meigs, J. V.: AM. J. OssT. & GYNEC. 67: 962, 1954. 15. Meigs, J. V.: Obst. & Gynec. 3: 471, 1954. 16. Kimbrough, R. A.: Discussion of Meigs.14 17. Beresford, 0. D., and Aidin, R.: Lancet 2: 211, 1950.
18. Neilson, K., and Dennison, C. W.: Ann. Int. Med. 34: 1055, 1951. 19. Deacon, A. L.: Brit. M. J. 1: 317, 1954. 20. Mokrohisky, J. F.: Radiology 70: 578, 1958. 21. Lemon, W. S., and Higgins, G. M.: Am. J. M. Sc. 181: 697, 1931. 22. Meigs, J. V., Armstrong, S. H., and Hamilton, H. H.: AM. J. OssT. & GYNEC. 46: 19, 1943. 23. Selye, H.: Nature 138: 32, 1936. 24. Schenck, S. B., and Eis, B. M.: AM. J. OssT. & GYNEC. 38: 327, 1939. 25. Dockerty, M. B., and Masson, J. C.: AM. J. OssT. & GYNEC. 47: 741, 1944.
On pelvic examination following paracentesis, it was impossible to determine the exact origin of the tumor, which filled the true pelvis. The adnexa could not be clearly delineated, but the tumor seemed to be attached to the uterine fundus. The pelvic tumor did not apparently increase in size during these 7 years. Approximately a week prior to admission, the patient complained of cough and dyspnea for the first time. Physical examination revealed dullness in the right lung base posteriorly with diminution of breath sounds and voice sound upon auscultation. The left side of the thorax was essentially normal on physical examination. Chest x-ray on admission showed fluid in the right side of the chest (Fig. 1). Thoracocentesis was performed and about 2,000 mi. of yellow fluid was obtained. Neither the pleural nor the abdominal fluid was found to contain any malignant cells. Results of the laboratory examinations of the chest and abdominal fluids are shown in Table II. The icteric index was 4 units. The total serum protein was 6.3 Gm. per 100 mi. The cephalin flocculation liver test was recorded as 2-plus. The alkaline phosphatase was 3 Bodansky units. The thymol turbidity test was 3.5, zinc sulfate test was 14.2, bromsulphthalein test was 17.5 per cent, Gros reaction was reported as 3-plus, glutamic oxalacetic transaminase was 24 units per cubic centimeter per minute, and glutamic pyruvic transaminase was 20 units per cubic centimeter per minute. Urinalysis was negative. The phenolsulfonphthalein kidney test was recorded as 26 per cent by 15 minutes, 43.5 per cent by 30 minutes, 61.5 per cent by 60 minutes, 95.0 per cent by 120 minutes. A clinical diagnosis was made of Meigs' syndrome. On Jan. 11, 1962, under general anesthesia, laparotomy through a mid-line incision was
I N 1 9 3 7 Meigs and Cass 1 reported 7 cases of ascites and hydrothorax with ovarian fibromas that were cured after removal of the fibroma. This condition was named Meigs' syndrome by Rhoads and Terrell. 2 The incidence of struma ovarii was found to be 0.3 per cent in a series of 1,000 solid ovarian tumors, according to Higuchi and Kato. 3 Only 6 previous cases of struma ovarii associated with ascites and hydrothorax have been reported (Table I). About 12 per cent of struma ovarii are functioning/0 and there is a small group of cases in which the thyroid tumor of the ovary has produced clinical evidences of hyperthyroidismY· 12 ' 13 Report of case
Mrs. H. K., an 83-year-old woman, para ii, was admitted to the Nakadori Hospital on Dec. 13, 1961, with a chief complaint of cough, dyspnea, and ascites. She gave a history of being well until about 7 years previously, when she complained of soreness in the lower abdomen. Ascites and a pelvic tumor were detected by a doctor whom she consulted. Although an operation was advised, she at first did not agree to this on account of her age. Subsequently, paracentesis yielded 2,000 mi. of yellow fluid. Paracentesis was required every 2 to 3 weeks, approximately 1,000 to 2,000 mi. of yellow fluid being removed on each occasion. Paracenteses were then repeated for 7 years until her admission to the hospital. No malignant cells were found in the abdominal fluid, but a huge globoid mass, firm, nontender, and movable on palpation, was evident, extending from the pelvis to a level about 2 inches below the umbilicus. From the Department of Obstetrics and Gynecology, Nakadori Hospital.
85
86
Januan' l)
Kawahara
196:~
Am. J. Ohst. & Gyncc,
Table I. Summary of 7 cases of struma o\·arii with ascites and hydrothorax --------
< -~~------------
---·-·-
!.ora-
lion
Case Jl/o.
Author
Dote :Date of of A.~e Mari-! z,;o. pubtal ojJof of \{(]pachi!era- lie at;on t'"n . tient /U.I , dren
/,0((1-
lion
of Complaints
tumor !
of hid1 otho0 peratwn
Jax
1944 1947
·11
M
?
Marked mcrease lil Right Right Total hysterecto my including S!ZC of abdomen right adnPxa as well as left
2
Baskin and 1950 1951 ( ~ounst>ller 0
46
M
2
Cough, dyspnea. Pnlargement of abdomen
Left
3
E"slcy"
1954
1955
•}8
M
2
.\scites and hydrothorax
Right Right
4
Schuldcs 7
1955 1956
·!9
M
0
Enlargement of ab- Left domen and pain Ill right sidP of chest
5
N
1959
52
M
8
Enlargement of abdomen and dyspnea
Right Bilat- Bilateral salpingooophorectomy era!
6
Zellner;'
1961
ti:)
M
4
Enlargement of abdomen
Left
7
Kawahara
M
2
Cough and dyspnea Left
Calmenson r•t ~]!
1961
Wl
----·----------·----·- --------------
Fig. l. Right hydrothorax.
-----
------------- ··-----
Right Same
Right Bilateral oophorectomy
Right Same Right Left salpinguoophorectomy ----
------------·--·-
carried out. A large lobular, solid and 'ystic tumor, about 1::l em. in diameter, was found in the left
Volume 85 Number 1
Struma ovar11 with ascites and hydrothorax
87
Fig. 2. The left ovarian tumor is lobular and cystic. On section, lobulated cysts are encountered which contained mucus and somewhat gelatinoid material.
,..
Fig. 3. Closely packed, small glandular structures contained little colloid.
Fig. 4. An occasional large acinus contained colloid.
88
Jauuar; 1, 1963 Am, J. Obst. & Gvnee.
Kawahara
Table II. Laboratory findings m chest and abdominal fluid -----------c---------.-----Specific gravity Rivalta reaction Protein Sugar Pseudo-
Abdominal fluid
Chest fluid
1_020
1.022
+
+ 4.4 Gm./100 mi. 92 mg./100 mi.
4.8 Gm./100 mi. 100 mg./100 mL
illllClll
Comment
Meigs 11 • 15 has redefined and limited the syndrome to include cases showing ( 1) a fibroma-like tumor of the ovary (fibroma, thecoma, granulosa cell tumor, and Brenner tumor); (2) ascites; (3) hydrothorax; and ( ·l! cure following removal of the tumor. Yet there is some confusion in this definition. Since a certain number of the theca cell and granulosa cell tumors are malignant, as Kimbrough 16 said, their inclusion seems illogicaL On the other hand, various ovarian tumors associated with ascites and hydrothorax, even malignant ovarian tumors without peritoneal and pleural metastasis, have been reported. Some authors 17 - 20 have included such malignant ovarian tumors with ascites and hydrothorax in the Meigs' syndrome group. Kimbrough 16 has offered two alternatives in the definition of Meigs' syndrome with which I am in agreement--first, that the syndrome might include only fibroma of the ovary with associated ascites and hydrothorax as originally defined; second, perhaps more logically, the syndrome should include all ovarian tumors both solid and cystic
REFERENCES
1. Meigs, J. V., and Cass, J. W.: AM. J. OssT. & GYNEC. 33: 249, 1937. 2. Rhoads, J. E., and Terrell, A. W.: J. A. M. A. 109: 1684, 1937. 3. Higuchi, K., and Kato, T.: Sanfuginkanosekai (Japanese) 12: 729, 1960. t. Calmenson, M., Dockerty, M. B., and Bianco, J. J.: Surg. Gynec. & Obst. 84: 181, 1947.
with associated ascites and hydrothorax, provided there is no evidence of metastasis in the peritoneal and pleural cavities Lemon and Higgins 21 have demonstrated experimentally the pathway hv ,vhich lymphatic absorption of particulate matter proceeds through the diaphragm. Meigs, Armstrong, and Hamilton 22 were able to test this tht'ory on 2 patients with Meigs' syndrome. This experiment is suggestiYc evidence that the abdominal fluid arriws in the chest by the same pathway as thf' India ink particles There are also many theories and speculations as to why the fluid occurs in the abdomen with benign ovarian tumors. Some have attempted to explain the origin of ascites on the basis of Selye's 23 alarm reaction. Schenck and Eis 24 offered an interesting hypothesis derived from Selye's alarm reaction with the added factor of lymphatic obstruction by the compressing effect of the tumor. One of the more recent and more nearly adequate explanations of the etiologic basis for the ascitic fluid is that offered by Dockerty and Masson, 25 who believed the significant factor in the production of ascites was edema in the tumor itself or in its pedicle. Summary
1. A case of struma ovarii associated with ascites and hydrothorax is reported. 2. Since there is some inconsistency in the definitions of Meigs' syndrome it is proposed that either ( 1) only fibromas or {2) all ovarian tumors both benign and malignant should be included, provided there is no evidence of metastasis in the peritoneal or pleural cavities.
5. Baskin, R. H., and Counseller, \T. S.: Proc. Staff. Meet. Mayo Clin. 20: 60, 1951. 6. Easley, C. M.: Obst. & Gynec. 6: 630, 1955. 7. Schuldes, H.: Geburtsh. u. Frauenh. 16: 450, 1956. 8. Nakasato, T.: Sanfuginkanosekai (]apanesei 11: 1620, 1959. 9. Zellner, K.: Acta obst. et gynec. scandinav. 40: 40, 1961.
Volume 85 Number 1
Struma ovarii with ascites and hydrothorax 89
10. Anderson, W. A. D.: Pathology, ed. 3, St. Louis, 1957, The C. V. Mosby Company, p. 1081. 11. Moench, G. L.: Surg. Gynec. & Obst. 49: 150, 1929. 12. Kleine, H.: Arch. Gynak. 158: 62, 1934. 13. Kovacs, F.: Arch. Gynak. 122: 766, 1924. 14. Meigs, J. V.: AM. J. OssT. & GYNEC. 67: 962, 1954. 15. Meigs, J. V.: Obst. & Gynec. 3: 471, 1954. 16. Kimbrough, R. A.: Discussion of Meigs.14 17. Beresford, 0. D., and Aidin, R.: Lancet 2: 211, 1950.
18. Neilson, K., and Dennison, C. W.: Ann. Int. Med. 34: 1055, 1951. 19. Deacon, A. L.: Brit. M. J. 1: 317, 1954. 20. Mokrohisky, J. F.: Radiology 70: 578, 1958. 21. Lemon, W. S., and Higgins, G. M.: Am. J. M. Sc. 181: 697, 1931. 22. Meigs, J. V., Armstrong, S. H., and Hamilton, H. H.: AM. J. OssT. & GYNEC. 46: 19, 1943. 23. Selye, H.: Nature 138: 32, 1936. 24. Schenck, S. B., and Eis, B. M.: AM. J. OssT. & GYNEC. 38: 327, 1939. 25. Dockerty, M. B., and Masson, J. C.: AM. J. OssT. & GYNEC. 47: 741, 1944.