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Pleurisy with Effusion Associated with Pseudomucinous Cystadenoma (Meig's Syndrome)
DISEASES of the CHEST VOLUME
XIII
SEPTEMBER-OCTOBER,
1947
NUMBER
5
Pleurisy with Effusion Associated with Pseudomucinous Cystadenoma (Meig's Syndrome) ERNEST D. NORA, M.D. RICHARD M. DAVISON, M.D., F.C.C.P. Chicago, Illinois
_.
The general paucity of information, relative to a syndrome of the hydrothorax with benign ovarian tumors and ascites, has too frequently led to a diagnosis of malignancy with chest metastasis. It is the object of this paper to review the literature, analyze thirty-seven cases, and to present another case which is unusual from the standpoint of tumor patho.1ogy. There is only one similar case presented in the literature. The association of ovarian tumors with ascites has not been an uncommon thing. Boldt' in 1910 reported a case of an ovarian fibroid tumor with ascites, and Titus 2 in 1913, in a discussion of a fibroma of the ovary, stated that ascites was an accompaniment in about half of the cases. In 1914 Fullerton 3 commented on the frequent early occurrence of ascites in cases of a fibroid tumor of the ovary. In 1914 Hellman:' reviewed the literature and found that ascites was associated with at least five per cent or all of the cases of ovarian fibroma, which is considerably less than Titus and Fullerton would lead us to believe. Reel,5 Baint," Richardson,7 Macdonald,8 and Owen? all presented similar cases between the years of 1917 and 1923. Although Owen mentioned the fact that hydrothorax might occur with ascites and ovarian tumor, he presented no evidence to substantiate his statement. Hoon 1 0 in 1923, in review of all of the cases of ovarian fibroma of the Mayo Clinic from 1910 to 1921 mentioned two with a hydrothorax and ascites which disappeared after surgical removal of the tumor. Salmon 1 1 presented two cases in 1934 and stressed the association of a -From the Service of Dr. Frederick Tice, aided by the Cuneo Research Foundation. Copyright, 19.7. by the American Colleee ot Chest Physicians.
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NORA AND DAVISON
Sept.-OCt., 19."
hydrothorax with benign pelvic tumors with ascites and pleural effusions; however, it was not until 1937 that this associatton .was recognized as a syndrome. At this time, Meigs and Cass12 reported Hoon's two cases, a case of Leo's, and four additional cases from the records of the Massachusetts General Hospital. They emphasized the fact that the hydrothorax and ascites cleared up quickly after surgical removal of the tumor. This work inspired such an interest in the syndrome that a number of cases, old and new, literally sprang into the literature. The earliest recorded case was that of Cullingworth's13 in 1879. Apparently, his patient died from non-treatment. The necropsy revealed a left hydrothorax with a collapsed left lung,congestion and edema of the right lung, marked ascites, bilateral large solid tumors of the ovary, and a thickened pleura and peritoneum. Tait14 presented a second case in 1890 of a left hydrothorax, marked ascites, and a large round solid tumor. Since malignancy was suspected, an operation was postponed. Thirty paracenteses were done with no relief to the patient. Several thoracenteses had been performed previously with apparent cure of the condition. After the tumor was removed, the patient made a slow recovery, which was interrupted only by the formation of a retrouterine abscess. This abscess cleared up after opening and drainage. In 1937 Rhoads and Terrel 16 presented a case; Weld,15 in 1938, presented two cases. In 1940 Bomze and Kirshbaum17 presented two cases. In the next two years Harris and Meyer,18 Henderson,19 Lock and Collins,20 Jones,21 Glass and Goldsmith,22 and Ritv0 23 added six cases to .the list. In 1943 and 1944 an additional six cases, all of benign ovarian fibroma associated with ascites and hydrothorax, were presented by Herrick, Tyson, and Watson,25 Keleman,26, Clay, Johnston, and Samson,27 Mendel and Tyrone,28 and Gardiner and Lloyd-Hart. 29 A case similar to our case was presented by Macfee 24 in 1941. Although the tumor was a multilocular cystadenoma, a variation from the usual fibroma, it, with ours, fits into the symptom complex emphasized by Meigs and Cass. A case was presented by Mlllett and She1l30 early in 1945 that is almost identical to our case. The patient, 39 years old, com. plained of dyspnea on mUd exertion. The examination, confirmed by fluoroscopy, revealed a right hydrothorax and an enlarged abdomen. She had no gastric distress, no peripheral edema, and no urinary symptoms. Her menses were regular until three months previous to the examination. During that month she menstruated twice. A thoracentesis was done on the day of admittance, twice before surgery, and once on the twelfth postoperative day. Cultures and guinea pig examinations of the fluid were negative. The
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425
laparotomy revealed a moderate amount of fiuid, a large ovarian cyst, later found to be a pseudomucinous cystadenoma, and several mucinous implants on the peritoneum. The patient made an uneventful recovery. CASE HISTORY
A white female, A. F., forty-two years old, was admitted to the hospital on January 4, 1945, complaining of anorexia and pressure in the epigastric region. The patient's family history was negative. She had been married ten years and never able to become pregnant. Menstruation began at the age of eighteen with no irregularity until the early part of 1944, at which time her periods varied from two to ten and twelve days, and sometimes she did not menstruate at all . She also noticed distention of her abdomen. In August 1944, she was told that she had a pleural effusion. Several thoracenteses were done which partially relieved her dyspn~ . Six weeks prior to admission to the hospital, the doctor told her that she had . an abdominal tumor and hydrothorax. Four thoracenteses were done. The only abdominal symptom the patient had was the "pressure feeling." Just before her first aspiration, her left lower extremity became edematous from the foot to the knee. The edema receded completely , after the aspiration. She was aspirated at two to three week intervals, and at each of the four aspirations approximately two liters of fluid were removed. Cultures of the fluid were sterile; guinea pig tnnoculations were negative for tuberculosis, and the fluid had the appearance of a transudate. A physical examination revealed that the thoracic respiratory expan-
Figure 1 Figure 2 Figure 1: X-ray of the line of pleural effusion on August 13, 1944. Figure 2: X-ray of the line of pleural effusion on september 12, 1944.
426
NORA AND DAVISON
8ept.-oct., 10'.,
sion was the same bilaterally even though a thoracentesis had been performed just before the examination. Prior to the thoracentesis, the right intercostal spaces were larger. On palpation, an increased vocal phremitus was elicited on the left and upper part of the right side. Percussion revealed a normal pulmonary resonance at the right upper lobe. There was a dullness and even a flatness at the base of the right lung up to the third intercostal space. Auscultation revealed bronchovesicular respiration in the left side. Clinically. there was no evidence of any parenchymal consolidation. No murmurs or extrasystoles were present in the heart. and there was no arrhythmia. There was a collateral venous circulation in the anterior wall of the chest. Examination of the abdomen revealed an engorgement of the superficial veins. A large tumor mass was palpated. It extended five centimeters above the umbilicus. The surface appeared smooth to the touch, and it was painless to palpation. The mass was situated in the hypogastric region and resembled a uterus almost at full term. The liver and spleen wer't. not palpable. The Blood Count, Urinalysis, and Kahn were negative. The impression was that it was either a malignancy of the internal genitalia with metastasis to the lung or a Meig's Syndro.me. The patient was operated on January 5, 1945, and a large ovarian cyst, weighing 4,200 grams, was removed . The peritoneum was adherent and studded. There was m1l1ary studding on the surface of the liver, and ascites. A tube was inserted in the right chest cavity. The Pathological Examination revealed a multilocular, grayish red, mucinous cyst. It was filled with a stringy, gray, mucinous fluid which was not precipitated by Acetic Acid. The daughter and granddaughter cysts varied in size from a split pea to a small orange. There were many gray, necrotic areas in the cyst wall of the parent cyst, which extended into the daughter and granddaughter cysts.
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Figure 3 Figure 4 Figure 3: X-ray of the line of pleural effusion on october 30, 1944. Figure 4: Note clearness of chest on December 10, 1945, one year after surgery.
Volume XItt
PLEulUsY W1TR J!:FPUSION
The Microscopic Examination revealed the typical palllsadlng of tall columnar cells with large quantities of degenerated, colloidal material having a rather reddish hue. No definite evidence of anaplasia was encountered. Diagnosis : Pseudomucinous cystadenoma of the ovary. On the first postoperative day 175 cc. of fluid were aspirated from the chest. By the third postoperative day, no further tendency to fluid development was observed, and the tube was removed; however, the wound did not heal satisfactorily, and some omental tissue was protruding from the wound. On the fourteenth postoperative. day, a secondary closure was done after removal of some of the omental tissue . A microscopic examination of the omental tissue revealed an acute peritonitis with some pseudomyxomatous peritoneal implants. An x-ray of the patient's chest on January 19, 1945, showed a circular area of infiltration at the right base at the level of the dome of the diaphragm with a corresponding fluid level. The patient was discharged from the hospital in a good condition February 2, 1945. X-ray of chest in January, 1946 revealed no fluid. In analyzing the thirty-eight cases listed, we find that the tumors most frequently occur during the menopause or just before. It is especially significant to chest men that of the thirty-eight cases of ovarian tumor and ascites, seventeen were in the abdomen only. In all of the cases, cultures made of the fluid were sterile, and the fluid was a transudate devoid of malignant cells. There seems to be no relationship between the amount of hydroperitoneum and hydrothorax. Fifteen cases had marked ascites; thirteen cases had relatively small amounts of fluid . In five of the cases the amount of fluid was relatively large, and the pleural effusion was relatively small. In three cases the amount of fluid was small in both the chest and abdominal cavities.
F1gure 5
Microscopic section revealing the l1ning of the cyst wall which reveals pallisading tall columnar epithelium. .
30 2 Considerable On 2nd one amount
18pts. Itpts. 15 pts. 18pts. 280oa. None
Repeated
80ozs. 62ozs.
5 2-4qts. 8ozs. 8ou. 80Z1. 8-10ozs. 2qts.
1
Bllateral Right
RIght RIght
RIght
RIght
1901
1902 1908
1917 1920
1128
M.GB.
M.GB.
M.GB.
Hoon
Hoon
Leo
3.
4.
5.
6.
7.
8.
Left
Repeated. 1000ccs. every few clays.
1600ecs.
4Oozs.
6Oozs.
58ozs. 48 ozs.
88.5 ozs.
None
'~CC8.
None
overS pta.
Large amount
8everalllters
6-8qts.
several quarts
6-8 qts.
Large amount
Died before surgery
1891
None
Taft
None'
2.
Bllateral
1879
CUlllngworth
1.
Preoperative Paracenteses
Year
Operative Abdominal Effusion
Author
No. of Thoracenteses and Amount
Oase No.
Site of Pleural Effusion
Benign
Fibroma
Fibroma
Fibroma
Fibromyoma
Plbroma
Fibroma
Fibroma
of Tumor
Type
Left ovary
Right ovary
Right ovary
Left Broad Ligament
RIght ovary
Not reported
Right ovary
Both ovaries
Location of Tumor
Dyspnea. Pain in chest.
Bloating. Pain between scapula.
Cough. Bloating of abdomen.
Pain in shoulder, especially in the left one.
Pleurisy.
Pain in right chest.
Abdominal swelling.
Metrorrhagia Dyspnea. Ascites.
Ohlef Complaint
~
~
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j...
~
·fr
I
t:1
~
~ ~
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Fibromyoma uteri
300 ecs,
None None
Repeated 3 None None 1 Repeated None
Right Not Reported Right Right Right Right Left
1934 1936 1936 1936 1937 1937 1937
M.G.B.
Miller
Weld
Weld
Macomber
Rhoads and Terrell
Bomzeand Kirshbaum
13.
14.
15.
16.
17.
18.
19.
2qts.
Some
750ccs.
Fibroma
Some
None 1
Fibroma
3500 ccs.
None
Fibroma
Fibroma
Fibroma
Fibroma
Not reported
None
Fibroma
Large amount
1
None
600c08. 700 ccs,
Right
1932
Salmon
12.
Fibroma
500ccs.
None
1500 c08. 1500 c08. 2000 ccs ..
Right
1932
Salmon
11.
1930
Fibroma
Present
None
Fibroma
None
1000 ccs,
Bilateral
Bomzeand Kirshbaum
10.
Left 9
1928
1
de Rouville, etal
9.
Left ovary
Right ovary
Left
Right
Bilateral
Right
Left ovary
uterus
Right ovary
Left ovary
Right ovary
Lower abdominal mass Slight dyspnea. Bearing down sensation. Edematous feet and ankles.
Dyspnea.
Abdominal tumor. Dyspnea.
Abdominal enlargement.
Swelling of abdomen.
Pressure. Weakness. Disability.
Dyspnea. Change in bowel habits.
Menorrhagia for two years.
Mass in lower abdomen with irregular cramps.
Pain in abdomen with lower abdominal mass.
Cough. Emaciation.
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1941 1941
Borg
Henderson
Lock and Collins
MacFee
Jones
Glass and Goldsmith
Meigs
22.
23.
24.
25.
26.
27.
28.
1941
1941
1940
1939
1939
1938
Harris and Meyer
21.
1937
Year
Schenk and Eis
Author
20.
Case No.
5 times in thirty days None
1
Right Left Right
4
None
None
Right
Right
None
4
Right
1
None
None
None
2 1
None
ccs,
3000
None
Preoperative Paracenteses
1000ccs,
No. of Thoracenteses and Amount
Bilateral
Right
Right
Site of Pleural Effusion
Fibroma
Fibroma
400 ecs,
ccs, 500
Fibroma
Left ovary
Right ovary
Left ovary
Left ovary
Cystadenoma
Pleurisy. Backache. Asthma. Dyspnea.
Shortness of breath. Abdom1nal distention.
Loss of weight. Dyspnea. Abdominal tumor.
Swelling of abdomen. Dyspnea.
~0
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.,
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Abdominal pain. Distention. Mass. Left ovary
Fibroma
Fibroma
~
~
0
~
Abdominal pain. Dyspnea.
Dyspnea. Pain in right chest.
Heartburn with dull epigastric pain.
Chief Complaint
0
Pain in right chest. Breathlessness.
Both ovaries
Left ovary
Left ovary
Location of Tumor
.,.w
Both ovaries
Fibroma
Fibroma
Papillary adenocarcinoma
?:,ype of Tumor
9qts.
Smallamo~t
411ters
Large amount
Moderate amount of ascites at autopsy
200 ccs.
Not reported
Operative Abdominal Effusion
47
52
60
55
50
33
57
45
12
13
14
15
16
17
18
19 Married
•
Good
8
:Married
•
No Good
Good
Single
Good
Good
Good
Good
•
•
No
No
SoB.
Good
Death
Good
Good
Good
Good
Good
Good
Married
•
Single
Single
Married
Married
•
52
SB.
11
Married
37
10
8
1
Yes
7
Married
2
Married
53
6
Married
Yes
58
36
5
Married
•
38
4
Good
No
Single
•
55
3
Good
•
Single
9
42
2
Death
BndResult
Yes
Married
64
36
1
status
No
36
Oase
No. of Ohildren
Married
Age
16 months
7 weeks
Not reported
7 months
3 months
Not reported
16 months
17 months
24 months
Not reported
3 months
7 months
3 years
27 years
11 years
15 months
Not reported
Pollow-Up
Not reported Not reported
Not reported Not reported . Negative Not reported
Not reported Sterne Not reported
Not reported
Not reported
Not reported
2 plus
Not reported
Not reported
Negative
Negative
Not reported
Not reported
Negative
Negative
Negative
Not reported
Not reported
~
CD
•-:a
r...
!
~
i
~ Not reported Negative
tj
~ Negative
Not reported
~
0
~
Negative
Not reported
Not reported
Not reported
Not reported
Not reported
Not reported
Wassermann
.... w
Not reported
Not reported
Negative
Not reported
Negative
Negative
Not reported
Guinea Pig Innoculation
Not reported
Not reported
sterne
sterne
sterne
Not reported
sterne
sterne
sterne
Not reported .
sterne
Not reported
Sterne
Not reported
Not reported
Pleural Fluid CUltures
Good Good Good Good
Good Good Good Good Good
3
• Yes No
• 5
No 1
'. No
2 No
Married
• Married Married
• Married Married Married Married
• Married Married Married
50
73
51
66
57
49
75
59
47
44
38
39
42
26
27
28
29
30
31-
32
33
34
35
36
37
38
• Not reported.
S.B. Stlllbirth.
Good
2 abor.
Married
54
25
Yes
Good
1
Married
31
24
,
Not reported
2montbs
Good
•
•
42
23
Good
Good
Good
Good
sterne
11 months
Death
Yes
Married
44
Not reported Negative Not reported Negative
Not reported Negative Negative
Not reported Sterne Sterne
Not reported 3 months Not reported
1 month
Not reported
Not reported
10 weeks
Not reported
Not reported
Not reported Not reported
3 months
Not reported
Not reported
Negative Sterne
2 months
Not reported
Negative
Negative
Sterne
11 months
Not reported Not reported
Not reported
Not reported Not reported
Not reported
Not reported
Not reported
Not reported
4 plus
Not reported
Not reported
Not reported
Not reported
Not reported
Negative
Not reported
Not reported
Not reported
Not reported
Not reported
Negative
Not reported
Not reported
Not reported
Negative
Negative
Not reported
Not reported
Not reported
Not reported
Not reported
6 months
Not reported
Not reported
sterne
22
6 months
Good
2
Married
sterne
67
21
7 months
Good
1
Married
50
20
e.
<:
w
.,.w
s
m
!....
~
~
i
a
5 c.
434
NORA AND DAVISON
. 8ept.-oct., 1M.,
Neither does there seem to by any connection between the location of the tumor and the side of the hydrothorax. All combinations are present, such as: Right ovarian tumor with left hydrothorax, left ovarian tumor with right hydrothorax, bllateral tumors with right, left, and bllateral hydrothorax. Up to the present time, no cause for the syndrome has been agreed upon. It has been suggested that there may be a lack of right lung drainage by the Azygos vein. Inasmuch as a left hydrothorax has been found, this hypothesis does not seem plausible. At the necropsy of Case No. 22 very large Azygos veins were found. Selye,31 who used data obtained from experimental work on animals, stated that repeated minor trauma to the peritoneum by the tumor causes a resistance of the tissue which .later returns to normal; however, after a long period of continued trauma, the resistance disappears, and a histamine toxicosis or anaphylactic shock appears with accumulation of peritoneal and pleural transudates. This reaction can neither be considered proved nor contradicted. Meigs'32 study of two cases. showed that the hydrothorax and ascites were almost identical in protein composition and established some form of communication betwen the two by showing that particulate carbon, after being Injected into the abdomen, almost immediately appeared in the pleural fluid but not in the blood stream. SUMMARY
1. The second case of Meig's Syndrome due to pseudomucinous cystadenoma of the ovary is presented. 2. The case was presented to make people more conscious of a condition that responds very well to a simple operation or second operation as in this case. 3. The literature is reviewed, brought up-to-date, with the addition of the leg portion of the syn<:lrome. 4. The fact that the fluid in the peritoneal cavity differed from that in the thoracic cavity would mean that the cells found in the peritoneum did not get beyond the diaphragmatic barriers.
RESUMEN 1. Be presenta el segundo caso del S1nd~ome de Meig causado por un cistoadenoma seudomucinoso del ovario. 2. Se ha presentado este caso con el objeto de llamar la atencton de los medicos hacia un estado que responde satisfactorlamente a una operaci6n senctlla, 0 a una segunda operaeton, como este caso.
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PLEURISY WITH EPPUSION
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. 3. Be revtsa la literatura y se la pone al dla con la adtcton de la parte del sindrome refertble a la pterna. 4. El hecho de que el llquldo en la cavidad peritoneal fue diferente al que se hallO en la cavidad toractca parece lndicar que las celulas que se encontraron en el peritoneo no penetraron mas alIa de la barrera dtarragmatlca. 1 2 3 4 5 6 7 8 9 10 11 12 13 14 15 16 17
REFERENCES Boldt, H. J.: J. ou«, 61:794, 1910. Titus, R. S.: Boston M. and S. J., 169:381, 1913. Fullerton, W. D.: Surg., Gynec. and Obst., 18:451, 1914. Hellman, A. M.: Surg., GYMC. ana os«; 20:269, 1915. Reel, P. J.: Am. J. Of os«; 75:400, 1917. 8a1nt, 8. F. M.: Edinburgh Med. J., 22:383, 1919. Richardson, A.: Proc. ROt/J. Soc. ue«, 13:210, 1919-20. Macdonald, A. W.: J.A.M.A., 78:106, 1922. Owen, A. W.: Lancet, 1:1211, 1923. Hoon, M. R.: Surg., Gynec. and Obst., 36:247, 1923. salmon, U. J.: J. Mt. Sinai Hosp., N. J., 1:169, 1934. Meigs, J. V., and Cass, J. W.: Am. J. Obst. and Gynec., 33:249, 1937. Culllngworth, C. J.: Tr. Obst. soc; London, 21:276,1879. . Tait, Lawson: Medico-Chirurgical Tram., 75:109, 1892. Weld, S. B.: New Eng. J. uea; 218:262, 1938. Rhoads, J. E., and Terrell, A. W.: J.A.M.A., 109:1684, 1937. Bomze, E. J., and Kirshbaum, J. D.: Am. J. Obst. and Gynec., 40:281,
1940. 18 Harris, F. I., and Meyer, M. A.: Surgery, 9:87, 1941. 19 Henderson, H. T.: Personal Communication to J. V. Meigs, Am. J. Obst'.and Gynec., 46:19, 1943. . 20 Lock, F. R., and Colllns, C. G.: Am. J. Obst. and Gynec., 41:517, 1941. 21 Jones, W. N.: J. M. A. State of Alabama, 12:199, 1943. 22 Glass, M., and Goldsmith, J. W. Jr.: Am. J. Obst. and Gynec., 43: 1048, 1944. 23 Rltvo, Max: Am. J. Roent., 48:152, 1942. 24 MacFee, W. F.: Annals of Surg., 113:549, 1941. 25 Herrick, W. W., Watson, T. L., and Watson, B. P.: Arch. Int. Med., 71 :370, 1943. 26 Kelemen, E.: Am. J. Obst. and Gynec., 47:275, 1944. 27 Clay, A. C., Johnston, R. N., and Bamsonl L.: Brit. Mea. J., !uly, 1944. 28 Mendel, E. B., and Tyrone, C.: Am. J. Oost. and Gynec., 48:211, 1944. 29 Gardiner, R. H., and Lloyd-Hart, V.: Lancet, 2:500, 1944. 30 Millett, J., and Shell, J.: Am. J. Med. s«, 209:327, March 1945. 31 Karady, st., Browne, S. L., and Selye, H.: Quart. J. Exper. PhY8., 28: 23, 1936. 32 Meigs, J. V., Armstrong, S. H., and Hamllton, H. H.: 46:19,1943.
XIII
SEPTEMBER-OCTOBER,
1947
NUMBER
5
Pleurisy with Effusion Associated with Pseudomucinous Cystadenoma (Meig's Syndrome) ERNEST D. NORA, M.D. RICHARD M. DAVISON, M.D., F.C.C.P. Chicago, Illinois
_.
The general paucity of information, relative to a syndrome of the hydrothorax with benign ovarian tumors and ascites, has too frequently led to a diagnosis of malignancy with chest metastasis. It is the object of this paper to review the literature, analyze thirty-seven cases, and to present another case which is unusual from the standpoint of tumor patho.1ogy. There is only one similar case presented in the literature. The association of ovarian tumors with ascites has not been an uncommon thing. Boldt' in 1910 reported a case of an ovarian fibroid tumor with ascites, and Titus 2 in 1913, in a discussion of a fibroma of the ovary, stated that ascites was an accompaniment in about half of the cases. In 1914 Fullerton 3 commented on the frequent early occurrence of ascites in cases of a fibroid tumor of the ovary. In 1914 Hellman:' reviewed the literature and found that ascites was associated with at least five per cent or all of the cases of ovarian fibroma, which is considerably less than Titus and Fullerton would lead us to believe. Reel,5 Baint," Richardson,7 Macdonald,8 and Owen? all presented similar cases between the years of 1917 and 1923. Although Owen mentioned the fact that hydrothorax might occur with ascites and ovarian tumor, he presented no evidence to substantiate his statement. Hoon 1 0 in 1923, in review of all of the cases of ovarian fibroma of the Mayo Clinic from 1910 to 1921 mentioned two with a hydrothorax and ascites which disappeared after surgical removal of the tumor. Salmon 1 1 presented two cases in 1934 and stressed the association of a -From the Service of Dr. Frederick Tice, aided by the Cuneo Research Foundation. Copyright, 19.7. by the American Colleee ot Chest Physicians.
423 Th1.s
One
~5TTS-A58-XLXY I I I I I I I I I~
11111111111
424
NORA AND DAVISON
Sept.-OCt., 19."
hydrothorax with benign pelvic tumors with ascites and pleural effusions; however, it was not until 1937 that this associatton .was recognized as a syndrome. At this time, Meigs and Cass12 reported Hoon's two cases, a case of Leo's, and four additional cases from the records of the Massachusetts General Hospital. They emphasized the fact that the hydrothorax and ascites cleared up quickly after surgical removal of the tumor. This work inspired such an interest in the syndrome that a number of cases, old and new, literally sprang into the literature. The earliest recorded case was that of Cullingworth's13 in 1879. Apparently, his patient died from non-treatment. The necropsy revealed a left hydrothorax with a collapsed left lung,congestion and edema of the right lung, marked ascites, bilateral large solid tumors of the ovary, and a thickened pleura and peritoneum. Tait14 presented a second case in 1890 of a left hydrothorax, marked ascites, and a large round solid tumor. Since malignancy was suspected, an operation was postponed. Thirty paracenteses were done with no relief to the patient. Several thoracenteses had been performed previously with apparent cure of the condition. After the tumor was removed, the patient made a slow recovery, which was interrupted only by the formation of a retrouterine abscess. This abscess cleared up after opening and drainage. In 1937 Rhoads and Terrel 16 presented a case; Weld,15 in 1938, presented two cases. In 1940 Bomze and Kirshbaum17 presented two cases. In the next two years Harris and Meyer,18 Henderson,19 Lock and Collins,20 Jones,21 Glass and Goldsmith,22 and Ritv0 23 added six cases to .the list. In 1943 and 1944 an additional six cases, all of benign ovarian fibroma associated with ascites and hydrothorax, were presented by Herrick, Tyson, and Watson,25 Keleman,26, Clay, Johnston, and Samson,27 Mendel and Tyrone,28 and Gardiner and Lloyd-Hart. 29 A case similar to our case was presented by Macfee 24 in 1941. Although the tumor was a multilocular cystadenoma, a variation from the usual fibroma, it, with ours, fits into the symptom complex emphasized by Meigs and Cass. A case was presented by Mlllett and She1l30 early in 1945 that is almost identical to our case. The patient, 39 years old, com. plained of dyspnea on mUd exertion. The examination, confirmed by fluoroscopy, revealed a right hydrothorax and an enlarged abdomen. She had no gastric distress, no peripheral edema, and no urinary symptoms. Her menses were regular until three months previous to the examination. During that month she menstruated twice. A thoracentesis was done on the day of admittance, twice before surgery, and once on the twelfth postoperative day. Cultures and guinea pig examinations of the fluid were negative. The
VolumeXItt
PLEU1US~
WITH EFFUSION
425
laparotomy revealed a moderate amount of fiuid, a large ovarian cyst, later found to be a pseudomucinous cystadenoma, and several mucinous implants on the peritoneum. The patient made an uneventful recovery. CASE HISTORY
A white female, A. F., forty-two years old, was admitted to the hospital on January 4, 1945, complaining of anorexia and pressure in the epigastric region. The patient's family history was negative. She had been married ten years and never able to become pregnant. Menstruation began at the age of eighteen with no irregularity until the early part of 1944, at which time her periods varied from two to ten and twelve days, and sometimes she did not menstruate at all . She also noticed distention of her abdomen. In August 1944, she was told that she had a pleural effusion. Several thoracenteses were done which partially relieved her dyspn~ . Six weeks prior to admission to the hospital, the doctor told her that she had . an abdominal tumor and hydrothorax. Four thoracenteses were done. The only abdominal symptom the patient had was the "pressure feeling." Just before her first aspiration, her left lower extremity became edematous from the foot to the knee. The edema receded completely , after the aspiration. She was aspirated at two to three week intervals, and at each of the four aspirations approximately two liters of fluid were removed. Cultures of the fluid were sterile; guinea pig tnnoculations were negative for tuberculosis, and the fluid had the appearance of a transudate. A physical examination revealed that the thoracic respiratory expan-
Figure 1 Figure 2 Figure 1: X-ray of the line of pleural effusion on August 13, 1944. Figure 2: X-ray of the line of pleural effusion on september 12, 1944.
426
NORA AND DAVISON
8ept.-oct., 10'.,
sion was the same bilaterally even though a thoracentesis had been performed just before the examination. Prior to the thoracentesis, the right intercostal spaces were larger. On palpation, an increased vocal phremitus was elicited on the left and upper part of the right side. Percussion revealed a normal pulmonary resonance at the right upper lobe. There was a dullness and even a flatness at the base of the right lung up to the third intercostal space. Auscultation revealed bronchovesicular respiration in the left side. Clinically. there was no evidence of any parenchymal consolidation. No murmurs or extrasystoles were present in the heart. and there was no arrhythmia. There was a collateral venous circulation in the anterior wall of the chest. Examination of the abdomen revealed an engorgement of the superficial veins. A large tumor mass was palpated. It extended five centimeters above the umbilicus. The surface appeared smooth to the touch, and it was painless to palpation. The mass was situated in the hypogastric region and resembled a uterus almost at full term. The liver and spleen wer't. not palpable. The Blood Count, Urinalysis, and Kahn were negative. The impression was that it was either a malignancy of the internal genitalia with metastasis to the lung or a Meig's Syndro.me. The patient was operated on January 5, 1945, and a large ovarian cyst, weighing 4,200 grams, was removed . The peritoneum was adherent and studded. There was m1l1ary studding on the surface of the liver, and ascites. A tube was inserted in the right chest cavity. The Pathological Examination revealed a multilocular, grayish red, mucinous cyst. It was filled with a stringy, gray, mucinous fluid which was not precipitated by Acetic Acid. The daughter and granddaughter cysts varied in size from a split pea to a small orange. There were many gray, necrotic areas in the cyst wall of the parent cyst, which extended into the daughter and granddaughter cysts.
.
'.,
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e,
.
~.~ .
•
0
Figure 3 Figure 4 Figure 3: X-ray of the line of pleural effusion on october 30, 1944. Figure 4: Note clearness of chest on December 10, 1945, one year after surgery.
Volume XItt
PLEulUsY W1TR J!:FPUSION
The Microscopic Examination revealed the typical palllsadlng of tall columnar cells with large quantities of degenerated, colloidal material having a rather reddish hue. No definite evidence of anaplasia was encountered. Diagnosis : Pseudomucinous cystadenoma of the ovary. On the first postoperative day 175 cc. of fluid were aspirated from the chest. By the third postoperative day, no further tendency to fluid development was observed, and the tube was removed; however, the wound did not heal satisfactorily, and some omental tissue was protruding from the wound. On the fourteenth postoperative. day, a secondary closure was done after removal of some of the omental tissue . A microscopic examination of the omental tissue revealed an acute peritonitis with some pseudomyxomatous peritoneal implants. An x-ray of the patient's chest on January 19, 1945, showed a circular area of infiltration at the right base at the level of the dome of the diaphragm with a corresponding fluid level. The patient was discharged from the hospital in a good condition February 2, 1945. X-ray of chest in January, 1946 revealed no fluid. In analyzing the thirty-eight cases listed, we find that the tumors most frequently occur during the menopause or just before. It is especially significant to chest men that of the thirty-eight cases of ovarian tumor and ascites, seventeen were in the abdomen only. In all of the cases, cultures made of the fluid were sterile, and the fluid was a transudate devoid of malignant cells. There seems to be no relationship between the amount of hydroperitoneum and hydrothorax. Fifteen cases had marked ascites; thirteen cases had relatively small amounts of fluid . In five of the cases the amount of fluid was relatively large, and the pleural effusion was relatively small. In three cases the amount of fluid was small in both the chest and abdominal cavities.
F1gure 5
Microscopic section revealing the l1ning of the cyst wall which reveals pallisading tall columnar epithelium. .
30 2 Considerable On 2nd one amount
18pts. Itpts. 15 pts. 18pts. 280oa. None
Repeated
80ozs. 62ozs.
5 2-4qts. 8ozs. 8ou. 80Z1. 8-10ozs. 2qts.
1
Bllateral Right
RIght RIght
RIght
RIght
1901
1902 1908
1917 1920
1128
M.GB.
M.GB.
M.GB.
Hoon
Hoon
Leo
3.
4.
5.
6.
7.
8.
Left
Repeated. 1000ccs. every few clays.
1600ecs.
4Oozs.
6Oozs.
58ozs. 48 ozs.
88.5 ozs.
None
'~CC8.
None
overS pta.
Large amount
8everalllters
6-8qts.
several quarts
6-8 qts.
Large amount
Died before surgery
1891
None
Taft
None'
2.
Bllateral
1879
CUlllngworth
1.
Preoperative Paracenteses
Year
Operative Abdominal Effusion
Author
No. of Thoracenteses and Amount
Oase No.
Site of Pleural Effusion
Benign
Fibroma
Fibroma
Fibroma
Fibromyoma
Plbroma
Fibroma
Fibroma
of Tumor
Type
Left ovary
Right ovary
Right ovary
Left Broad Ligament
RIght ovary
Not reported
Right ovary
Both ovaries
Location of Tumor
Dyspnea. Pain in chest.
Bloating. Pain between scapula.
Cough. Bloating of abdomen.
Pain in shoulder, especially in the left one.
Pleurisy.
Pain in right chest.
Abdominal swelling.
Metrorrhagia Dyspnea. Ascites.
Ohlef Complaint
~
~
G
•
j...
~
·fr
I
t:1
~
~ ~
()C)
Fibromyoma uteri
300 ecs,
None None
Repeated 3 None None 1 Repeated None
Right Not Reported Right Right Right Right Left
1934 1936 1936 1936 1937 1937 1937
M.G.B.
Miller
Weld
Weld
Macomber
Rhoads and Terrell
Bomzeand Kirshbaum
13.
14.
15.
16.
17.
18.
19.
2qts.
Some
750ccs.
Fibroma
Some
None 1
Fibroma
3500 ccs.
None
Fibroma
Fibroma
Fibroma
Fibroma
Not reported
None
Fibroma
Large amount
1
None
600c08. 700 ccs,
Right
1932
Salmon
12.
Fibroma
500ccs.
None
1500 c08. 1500 c08. 2000 ccs ..
Right
1932
Salmon
11.
1930
Fibroma
Present
None
Fibroma
None
1000 ccs,
Bilateral
Bomzeand Kirshbaum
10.
Left 9
1928
1
de Rouville, etal
9.
Left ovary
Right ovary
Left
Right
Bilateral
Right
Left ovary
uterus
Right ovary
Left ovary
Right ovary
Lower abdominal mass Slight dyspnea. Bearing down sensation. Edematous feet and ankles.
Dyspnea.
Abdominal tumor. Dyspnea.
Abdominal enlargement.
Swelling of abdomen.
Pressure. Weakness. Disability.
Dyspnea. Change in bowel habits.
Menorrhagia for two years.
Mass in lower abdomen with irregular cramps.
Pain in abdomen with lower abdominal mass.
Cough. Emaciation.
~
fD
~
~
....rn 0
~
~
~ rn t<
~
~
a
<: S. c:: S CD
1941 1941
Borg
Henderson
Lock and Collins
MacFee
Jones
Glass and Goldsmith
Meigs
22.
23.
24.
25.
26.
27.
28.
1941
1941
1940
1939
1939
1938
Harris and Meyer
21.
1937
Year
Schenk and Eis
Author
20.
Case No.
5 times in thirty days None
1
Right Left Right
4
None
None
Right
Right
None
4
Right
1
None
None
None
2 1
None
ccs,
3000
None
Preoperative Paracenteses
1000ccs,
No. of Thoracenteses and Amount
Bilateral
Right
Right
Site of Pleural Effusion
Fibroma
Fibroma
400 ecs,
ccs, 500
Fibroma
Left ovary
Right ovary
Left ovary
Left ovary
Cystadenoma
Pleurisy. Backache. Asthma. Dyspnea.
Shortness of breath. Abdom1nal distention.
Loss of weight. Dyspnea. Abdominal tumor.
Swelling of abdomen. Dyspnea.
~0
~
~
CD
r...~
.,
W 'a c-.
Z
tj
Abdominal pain. Distention. Mass. Left ovary
Fibroma
Fibroma
~
~
0
~
Abdominal pain. Dyspnea.
Dyspnea. Pain in right chest.
Heartburn with dull epigastric pain.
Chief Complaint
0
Pain in right chest. Breathlessness.
Both ovaries
Left ovary
Left ovary
Location of Tumor
.,.w
Both ovaries
Fibroma
Fibroma
Papillary adenocarcinoma
?:,ype of Tumor
9qts.
Smallamo~t
411ters
Large amount
Moderate amount of ascites at autopsy
200 ccs.
Not reported
Operative Abdominal Effusion
47
52
60
55
50
33
57
45
12
13
14
15
16
17
18
19 Married
•
Good
8
:Married
•
No Good
Good
Single
Good
Good
Good
Good
•
•
No
No
SoB.
Good
Death
Good
Good
Good
Good
Good
Good
Married
•
Single
Single
Married
Married
•
52
SB.
11
Married
37
10
8
1
Yes
7
Married
2
Married
53
6
Married
Yes
58
36
5
Married
•
38
4
Good
No
Single
•
55
3
Good
•
Single
9
42
2
Death
BndResult
Yes
Married
64
36
1
status
No
36
Oase
No. of Ohildren
Married
Age
16 months
7 weeks
Not reported
7 months
3 months
Not reported
16 months
17 months
24 months
Not reported
3 months
7 months
3 years
27 years
11 years
15 months
Not reported
Pollow-Up
Not reported Not reported
Not reported Not reported . Negative Not reported
Not reported Sterne Not reported
Not reported
Not reported
Not reported
2 plus
Not reported
Not reported
Negative
Negative
Not reported
Not reported
Negative
Negative
Negative
Not reported
Not reported
~
CD
•-:a
r...
!
~
i
~ Not reported Negative
tj
~ Negative
Not reported
~
0
~
Negative
Not reported
Not reported
Not reported
Not reported
Not reported
Not reported
Wassermann
.... w
Not reported
Not reported
Negative
Not reported
Negative
Negative
Not reported
Guinea Pig Innoculation
Not reported
Not reported
sterne
sterne
sterne
Not reported
sterne
sterne
sterne
Not reported .
sterne
Not reported
Sterne
Not reported
Not reported
Pleural Fluid CUltures
Good Good Good Good
Good Good Good Good Good
3
• Yes No
• 5
No 1
'. No
2 No
Married
• Married Married
• Married Married Married Married
• Married Married Married
50
73
51
66
57
49
75
59
47
44
38
39
42
26
27
28
29
30
31-
32
33
34
35
36
37
38
• Not reported.
S.B. Stlllbirth.
Good
2 abor.
Married
54
25
Yes
Good
1
Married
31
24
,
Not reported
2montbs
Good
•
•
42
23
Good
Good
Good
Good
sterne
11 months
Death
Yes
Married
44
Not reported Negative Not reported Negative
Not reported Negative Negative
Not reported Sterne Sterne
Not reported 3 months Not reported
1 month
Not reported
Not reported
10 weeks
Not reported
Not reported
Not reported Not reported
3 months
Not reported
Not reported
Negative Sterne
2 months
Not reported
Negative
Negative
Sterne
11 months
Not reported Not reported
Not reported
Not reported Not reported
Not reported
Not reported
Not reported
Not reported
4 plus
Not reported
Not reported
Not reported
Not reported
Not reported
Negative
Not reported
Not reported
Not reported
Not reported
Not reported
Negative
Not reported
Not reported
Not reported
Negative
Negative
Not reported
Not reported
Not reported
Not reported
Not reported
6 months
Not reported
Not reported
sterne
22
6 months
Good
2
Married
sterne
67
21
7 months
Good
1
Married
50
20
e.
<:
w
.,.w
s
m
!....
~
~
i
a
5 c.
434
NORA AND DAVISON
. 8ept.-oct., 1M.,
Neither does there seem to by any connection between the location of the tumor and the side of the hydrothorax. All combinations are present, such as: Right ovarian tumor with left hydrothorax, left ovarian tumor with right hydrothorax, bllateral tumors with right, left, and bllateral hydrothorax. Up to the present time, no cause for the syndrome has been agreed upon. It has been suggested that there may be a lack of right lung drainage by the Azygos vein. Inasmuch as a left hydrothorax has been found, this hypothesis does not seem plausible. At the necropsy of Case No. 22 very large Azygos veins were found. Selye,31 who used data obtained from experimental work on animals, stated that repeated minor trauma to the peritoneum by the tumor causes a resistance of the tissue which .later returns to normal; however, after a long period of continued trauma, the resistance disappears, and a histamine toxicosis or anaphylactic shock appears with accumulation of peritoneal and pleural transudates. This reaction can neither be considered proved nor contradicted. Meigs'32 study of two cases. showed that the hydrothorax and ascites were almost identical in protein composition and established some form of communication betwen the two by showing that particulate carbon, after being Injected into the abdomen, almost immediately appeared in the pleural fluid but not in the blood stream. SUMMARY
1. The second case of Meig's Syndrome due to pseudomucinous cystadenoma of the ovary is presented. 2. The case was presented to make people more conscious of a condition that responds very well to a simple operation or second operation as in this case. 3. The literature is reviewed, brought up-to-date, with the addition of the leg portion of the syn<:lrome. 4. The fact that the fluid in the peritoneal cavity differed from that in the thoracic cavity would mean that the cells found in the peritoneum did not get beyond the diaphragmatic barriers.
RESUMEN 1. Be presenta el segundo caso del S1nd~ome de Meig causado por un cistoadenoma seudomucinoso del ovario. 2. Se ha presentado este caso con el objeto de llamar la atencton de los medicos hacia un estado que responde satisfactorlamente a una operaci6n senctlla, 0 a una segunda operaeton, como este caso.
VolumeXDI
PLEURISY WITH EPPUSION
435
. 3. Be revtsa la literatura y se la pone al dla con la adtcton de la parte del sindrome refertble a la pterna. 4. El hecho de que el llquldo en la cavidad peritoneal fue diferente al que se hallO en la cavidad toractca parece lndicar que las celulas que se encontraron en el peritoneo no penetraron mas alIa de la barrera dtarragmatlca. 1 2 3 4 5 6 7 8 9 10 11 12 13 14 15 16 17
REFERENCES Boldt, H. J.: J. ou«, 61:794, 1910. Titus, R. S.: Boston M. and S. J., 169:381, 1913. Fullerton, W. D.: Surg., Gynec. and Obst., 18:451, 1914. Hellman, A. M.: Surg., GYMC. ana os«; 20:269, 1915. Reel, P. J.: Am. J. Of os«; 75:400, 1917. 8a1nt, 8. F. M.: Edinburgh Med. J., 22:383, 1919. Richardson, A.: Proc. ROt/J. Soc. ue«, 13:210, 1919-20. Macdonald, A. W.: J.A.M.A., 78:106, 1922. Owen, A. W.: Lancet, 1:1211, 1923. Hoon, M. R.: Surg., Gynec. and Obst., 36:247, 1923. salmon, U. J.: J. Mt. Sinai Hosp., N. J., 1:169, 1934. Meigs, J. V., and Cass, J. W.: Am. J. Obst. and Gynec., 33:249, 1937. Culllngworth, C. J.: Tr. Obst. soc; London, 21:276,1879. . Tait, Lawson: Medico-Chirurgical Tram., 75:109, 1892. Weld, S. B.: New Eng. J. uea; 218:262, 1938. Rhoads, J. E., and Terrell, A. W.: J.A.M.A., 109:1684, 1937. Bomze, E. J., and Kirshbaum, J. D.: Am. J. Obst. and Gynec., 40:281,
1940. 18 Harris, F. I., and Meyer, M. A.: Surgery, 9:87, 1941. 19 Henderson, H. T.: Personal Communication to J. V. Meigs, Am. J. Obst'.and Gynec., 46:19, 1943. . 20 Lock, F. R., and Colllns, C. G.: Am. J. Obst. and Gynec., 41:517, 1941. 21 Jones, W. N.: J. M. A. State of Alabama, 12:199, 1943. 22 Glass, M., and Goldsmith, J. W. Jr.: Am. J. Obst. and Gynec., 43: 1048, 1944. 23 Rltvo, Max: Am. J. Roent., 48:152, 1942. 24 MacFee, W. F.: Annals of Surg., 113:549, 1941. 25 Herrick, W. W., Watson, T. L., and Watson, B. P.: Arch. Int. Med., 71 :370, 1943. 26 Kelemen, E.: Am. J. Obst. and Gynec., 47:275, 1944. 27 Clay, A. C., Johnston, R. N., and Bamsonl L.: Brit. Mea. J., !uly, 1944. 28 Mendel, E. B., and Tyrone, C.: Am. J. Oost. and Gynec., 48:211, 1944. 29 Gardiner, R. H., and Lloyd-Hart, V.: Lancet, 2:500, 1944. 30 Millett, J., and Shell, J.: Am. J. Med. s«, 209:327, March 1945. 31 Karady, st., Browne, S. L., and Selye, H.: Quart. J. Exper. PhY8., 28: 23, 1936. 32 Meigs, J. V., Armstrong, S. H., and Hamllton, H. H.: 46:19,1943.