Multilocular Cyst of the Kidney
Report of Three Cases with Review of the Literature
MARY C. B A L D A U F , M.D.,
AND D A L E M. S C H U L Z ,
M.D.
Department of Pathology, Methodist Hospital, Indianapolis, Indiana 46202
ABSTRACT
Baldauf, Mary C , and Schulz, Dale M.: Multilocular cyst of the kidney.
Report of three cases with review of the literature. Am J Clin Pathol 65:
93-102, 1976. Multilocular cyst of the kidney is an uncommon lesion.
Approximately 70 cases are reported in the literature, with more than
half occurring in children. T h e etiology and pathogenesis are not known,
although the microscopic appearance in certain cases resembles that of
Wilms' tumor. Three additional cases are presented and the literature is
reviewed. (Key words: Kidneys, cystic; Multilocular cyst; Wilms' tumor;
Benign multilocular cystic nephroma.)
MULTILOCULAR CYST of the kidney is an
uncommon lesion, with approximately 70
cases having been reported. More than
half of these cases have been in children
less than 4 years of age, with the remainder
occurring in adults. Three additional
cases are presented and previously reported cases are summarized.
Report of Three Cases
Case 1. An 11-month-old boy was discovered to have a swelling of the left
flank. No significant past or family history
was obtained. T h e blood pressure was
130/90 mm. Hg. Physical examination disclosed a bulging abdominal mass on the
left. Intravenous pyelography demonstrated a few blunted calyces at the inferior
margin of the mass, and the lesion was
found to be cystic with ultrasound. T h e
child was taken to surgery with a preoperative diagnosis of Wilms' tumor, and a left
nephrectomy was performed. T h e child
Received April 9, 1975; accepted for publication
April 22, 1975.
Address reprint requests to Dr. Schulz.
93
became normotensive after operation and
was discharged after an uncomplicated
postoperative course.
Pathologic Findings
T h e dimensions of the kidney were 10
X 8 X 6 cm. T h e cortical surface was
smooth, with several patches of hemorrhagic discoloration as much as 2 cm. in
maximum dimension. T h e cut surface
of the kidney showed a well-encapsulated,
ovoid, cystic structure measuring 9 x 5
x 6 cm, occupying the central portion
(Fig. 1). T h e mass was composed of numerous thin-walled pale cysts as large as 3 cm.
in diameter filled with clear fluid. At the
poles of the kidney, compressed remnants
of firm, tan, renal parenchyma were seen
(Fig. 2). A portion of the cyst wall extended
into the renal pelvis; however, no communication between cyst and pelvis was
found. Microscopically, the cysts comprising the mass were lined by flattened
and cuboidal epithelium surrounded by
loose stroma. Occasional cysts contained a
jagged epithelium composed of large
94
BALDAUF AND SCHULZ
4
A.J.C.P.—Vol.
65
5
DATE_
FIG. 1 (left). Case 1. A multiloculated, cystic mass replaces most of the kidney. A thin rim of compressed
renal tissue can be seen at both poles.
FIG. 2 (right). Case 1. Detail demonstrating the central, cystic mass, the compressed,
slit-like renal pelvis, and an outer rim of renal parenchyma.
eosinophilic cells forming irregular projec- raphy demonstrated a normal left kidney
tions (Fig. 3). The main portion of the wall and a low-lying right kidney. The right
of the mass was fibrous and blended into kidney was rotated downward with angulaatrophic renal tissue, with scattered dilated tion at the uretero-pelvic junction, with
tubules and glomeruli being present in the the mid-portion of the renal pelvis lying
fibrous tissue. T h e remaining renal tissue opposite L2. The diagnoses of hydroneappeared normal histologically.
phrosis and ptosis of the right kidney
Case 2. A 60-year-old woman was ad- with probable neoplasm were made. A
mitted to the hospital with many com- right nephrectomy was performed and a
plaints, including long-standing, intermit- hard, nodular, fixed, hydronephrotic
tent, mild, right-sided abdominal pain. kidney was removed.
Physical findings included a blood pressure
of 164/90 mm. Hg. A hard, smooth, fixed
Pathologic Findings
mass was palpated in the right upper
quadrant of the abdomen, extending from
The weight of the kidney was 282 Gm.
the costal margin to the umbilicus. Labora- The capsular surface was granular and
tory data were noncontributory. Plain dotted by numerous petechiae. The cut
roentgenograms of the abdomen failed to surface revealed a large cystic area approxioutline the kidneys. Intravenous pyelog- mately 9 cm. in diameter. The mass had a
January 1976
MULTILOCULAR CYST OF T H E KIDNEY
95
* *• fits' 4-T£'>£r i
FlG. 3. Case 1. Large, atypical epithelial cells lining a small cyst. Hematoxylin and eosin. X210.
FIG. 4. Case 2. Sharply delineated, 9 cm., multiloculated cyst occupying the central portion of the
kidney with remaining, uninvolved kidney at both poles.
96
BALDAUF AND SCHULZ
A.J.C.P.—Vol.
65
gram showed a large mass occupying the
lower pole of the left kidney, displacing
the calyceal system. A left nephrectomy
was performed. The child remained well
until the age of 10 years, when he experienced an acute episode of rectal
bleeding. The source was found to be a
Meckel's diverticulum. T h e patient's
condition responded well to conservative
management.
Pathologic Findings
FlG. 5. Case 3. T h e lower pole of the kidney is
replaced by a 7.5-cm., multiloculated cyst, a pedunculated portion of which has prolapsed into the
renal pelvis (lower right).
T h e weight of the kidney was 215 Gm.
and it measured 1 2 x 8 x 6 cm. The cut
surface disclosed a multiloculated cystic
lesion 7.5 cm. in diameter occupying the
lower pole. The individual cysts ranged in
size from 1 to 2.5 cm. A portion of the cyst
had prolapsed into the renal pelvis (Fig.
5). The upper pole of the kidney was
composed of tissue which appeared normal
grossly. The cysts were lined by cuboidal
epithelium predominantly. A variety of
patterns was seen in the cystic structures
lying within the septa. Small cysts were
lined by plump, cuboidal cells obliterating
the lumen, while large cysts were lined
by a layer of flattened cells. Most striking
were cysts lined by irregular epithelium,
which was heaped up and papillary (Fig. 6).
The cells were large with an eosinophilic
cytoplasm and large, often basally placed,
nuclei. Also found were cysts of an intermediate size lined by a uniform columnar
epithelium. The stroma was variable, with
portions being loose and edematous and
containing small spindle cells, while other
areas were relatively more dense. A
scattered, often focal, lymphocytic infiltrate was seen. Glomerular structures
were not recognized in the septa of the
mass. In the adjacent kidney, compressed
and atrophic tubules and glomeruli were
seen.
honeycomb pattern, being composed of
numerous extremely thick-walled and
fibrous, fluid-filled locules measuring less
than 1 cm. to 2.5 cm. (Fig. 4). The mass
did not communicate with the renal pelvis,
which was dilated. A rim of compressed
renal parenchyma surrounded the cystic
mass. Microscopically the cysts were lined
by flattened cells with ovoid, elongated
nuclei. The stroma was fibrous, dense and
relatively acellular. The remaining renal
parenchyma showed degeneration, thickening of the arterioles, and increased
connective tissue.
Case 3. During a routine pediatric examination, a 17-month-old boy was discovered
to have an abdominal mass. The left
side of the abdomen was protuberant, and
Discussion
a firm, non-tender, movable mass extending from the eleventh rib to the iliac
Powell and co-workers, 36 in 1951, sugcrest was palpated. An intravenous pyelo- gested eight criteria characteristic of
January 1976
MULTILOCULAR CYST OF T H E KIDNEY
97
FIG. 6. Case 3. Loosely arranged, cellular stroma surrounds small cysts, in some of which the
epithelium forms short, papillary projections. Hematoxylin and eosin. x 8 5 .
multilocular cyst of the kidney: (1) the cyst
should be unilateral, (2) it should be solitary, (3) it should be multilocular, (4) the
cysts should not communicate with the
renal pelvis, (5) the loculi should not communicate with one another, (6) the loculi
should be lined by epithelium, (7) no renal
elements should be present within the main
cyst, and (8) remaining kidney tissue, if
present, should be normal. These criteria
were modified by Boggs and Kimmelstiel,6 who stated that fully developed
nephrons should be absent from the septa
of the cyst.
T h e first reported case is credited to
Edmunds, 14 in 1892, who described the
case of an 18-year-old girl, using the term
"cystic adenoma of the kidney." In 1932,
Meland and Braasch 31 collected ten cases
and concluded that multilocular cyst was
a rare butdefinite entity. By 1951, Frazier 18
cited additional cases in American, Canadian, French and Italian literature, in-
cluding two cases of his own, bringing
the total reported cases to 31. In the same
year, however, Powell36 found only 13
examples conforming to his criteria, and
excluded many previously accepted cases.
Aterman and colleagues 1 reviewed the
literature in 1973 and considered 40 cases
to be acceptable. Eighteen of these occurred in children. Included in this group
were two examples termed "lymphangioma" that were felt to be indistinguishable from multilocular cyst.
Uson and co-workers 43 found one multilocular cyst in 35 cases of cystic renal
disease and 53 cases of Wilms' tumor in
children. Lattimer 20 states that of 650
children with abdominal masses, six had
multilocular cysts of the kidney. The true
incidence of multilocular cyst, however,
is difficult to determine. One problem is
the confused terminology. Various names
have been used, including "polycystic
nephroblastoma," "benign multilocular
98
BALDAUF AND SCHULZ
A.J.C.P.—Vol. 65
Table 1. Pediatric Cases
Case
1-18
Author
Year
Patient's
Age, Sex
Side
Presenting
Symptom
Comment
Aterman and
associates'
1940-72
19
Wakeley*44
1930
20 mo., M
R
Mass
Unilateral polycystic kidney; no normal renal
tissue
20
Lynch and
Thompson* 29
1951
14 mo., F
L
Mass
Unilateral multicystic
kidney; no normal
renal tissue
21-22
DeLaPenaand
co-workers 13
1957
14 mo., F
2'/2 yr., M
L
L
Mass
Fever, hematuria,
mass
"Benign nephroblastomas of infancy"
Landing 27
1958
10 mo., F
L
Fever, vomiting,
diarrhea
"Well-differentiated and
polycystic Wilms'
tumor"
Marion and
colleagues 30
1964
7Vi mo., F
14 mo., F
18 mo., F
R
R
L
Mass, anorexia
Hematuria, mass
Mass
Mass
23
24-26
Eighteen pediatric cases
summarized
27
Caron and
associates9
1965
15 mo., M
L
28
Osathanondh
and Potter 33
1968
40 min., F
L
29
Kawamura and
Miyakawa25
1969
14 mo., M
Diarrhea, mass
30
Steiner and
co-workers 40
1970
14 mo., M
Mass
Johnson and
colleagues 24
1973
28 mo., F
11 mo., F
13 mo., M
R
L
R
Mass
Mass
Mass
31-33
Multiple congenital
anomalies (picture and
description not included)
34
Felman and
associates15
1973
9'/z mo., F
R
Mass
35
Austin and
Castellino 4
1973
18 mo., M
R
Mass
36
Belloli and
colleagues 5
1974
12 mo., F
R
Mass
37-38
Current cases
1975
11 mo., M
L
Mass
17 mo., M
L
Mass
* Powell's original cases.
Pedunculation of cysts
into pelvis
Actinomycin D given
preoperatively (cases
in Japanese literature
summarized: 2 pediatric, 4 adult)
Vincristine, actinomycin
D and preoperative
irradiation
Hypertension, 130/90
mm. Hg
Prolapse of cysts into
pelvis
Table 2. Adult Cases
Case
Author
Year
Patient's
Age (Yr.),
Sex
1
Edmunds* 14
1892
18, F
L
Mass
Cystic adenoma of kidney
2
Haslinger*
21
1926
53, F
R
Pain
Not accepted by Aterman 1
3
Ravitch*37
1931
33, F
R
Pain
Multilocular tuberculous cyst
1933
71,M
64, M
44, F
66, M
R
R
R
R
Pain
Frequency
4-7
Meland and
Braasch* 31
Side
Presenting
Symptom
Comment
Incidental finding at autopsy
Incidental finding at autopsy
Blood pressure 165/105 mm. Hg
8
Lieberthal*"
1939
50, F
L
Pain
Multilocular solitary cyst of renal
hilus
9
Heckel and
Gould* 22
1940
45, F
R
Mass
Papillary cystadenoma
10
Reinecke 39
1925
54, F
L
Pain
Solitary multichambered cyst
1928
55, F
L
Pain
Cystic lymphangioma
34
11
Perlmann
12
Wynn-Williams
and Morgan 45
1949
33, F
R
Hematuria
Lymphangioma, patient pregnant
Powell and
associates36
1951
—, F
L
Mass
Cyst removal with reconstructed
kidney functional, patient
pregnant
37, F
L
Mass
R
Hematuria,
pain
13-14
15
O'Flynn 32
1953
62, F
16
Attwood and
Greive 3
1958
19, M
R
Pain,
hematuria
17
Gibson20
1961
64, F
R
Mass
Pedunculation into renal pelvis
Uson and
Melicow42
1963
48, F
L
55, F
L
Pain,
hematuria
Hematuria
45, F
R
Mass
Intrapelvic herniation of cyst;
blood pressure 134/88 mm. Hg
Herniation of cyst with ureteropelvic obstruction; blood
pressure 180/90 mm. Hg
Herniation of cyst, hydronephrosis,
chronic pyelonephritis
18-20
21
Fine 18
1963
44, F
R
Hematuria
22
Piper and
Beswick35
1970
46, F
L
Mass, pain,
frequency
23
Brown and
associates7
1970
65, M
R
Hematuria
24
Javadpour and
colleagues 23
1973
57, M
R
Pain
Austin and
Castellino 4
1973
55, M
55, M
L
L
Mass
Trauma
Hypertension
27
Friday and
co-workers 18
1974
48, F
R
Mass
Preoperative diagnosis with cyst
removal
28
Present case
1975
60, F
R
Mass
25-26
* Powell's original cases.
Herniation of cyst into renal pelvis
causing ureteral obstruction;
blood pressure 210/110 mm. Hg
Calcification simulating carcinoma
seen on pyelogram; preoperative
irradiation
100
BALDAUF AND SCHULZ
cystic nephroma," and "focal" or "partial"
polycystic kidney, in addition to those
terms already mentioned. In the French
literature cases reported as "kystes multiloculaires" include examples of both
multilocular cyst and unilateral multicystic
(dysplastic) kidney. Although most authors
apply Powell's criteria, there are many
exceptions, and some reported cases do
not meet all of these criteria, while others
are incompletely documented. Of the 13
cases originally accepted by Powell, two
were devoid of remaining renal tissue.29,44
He concluded that these were a late stage
of multilocular cyst. A more recent case38
of unilateral renal cystic disease in a newborn infant has been reported as congenital multilocular cyst and, similarly,
contained no remaining renal tissue.
Another lesion difficult to categorize is
exemplified by a case reported by Carver 10
in which scattered cysts occurred in the
renal parenchyma in addition to the main
multilocular cyst.
Austin and Castellino 4 found that of nine
multilocular cysts studied by angiography, four demonstrated neovascularity
suggestive of neoplasm. A recent report 7
presented a case in which history, physical
examination, and radiographic studies
demonstrating diffuse renal calcification
gave results indistinguishable from those
caused by renal-cell carcinoma. Friday
and associates19 reported the successful
preoperative diagnosis of multilocular
cyst by discordant angiographic and ultrasonic findings, confirmed by cyst puncture
and injection, allowing cyst removal rather
than nephrectomy.
Tables 1 and 2 summarize the current
and previously r e p o r t e d cases. T h e
pediatric patients ranged in age from birth
to 414 years, with a mean age of 17 months.
The distribution between male and female
is approximately equal. The most common
presenting symptom is an abdominal mass.
There is not a family history of renal cystic
disease. One child had multiple congenital
AJ.C.P.—Vol.
65
anomalies, 33 another had Meckel's diverticulum, and a third had intussusception. 6
A preoperative diagnosis of Wilms' tumor
was usually made in the pediatric age
group. On several occasions, preoperative
treatment has been given; it consisted of
radiotherapy in three cases 7,17 ' 41 and
chemotherapy in combination with irradiation in two others. 11,24 Chemotherapy alone
was administered in one case. 25
Adult patients ranged in age from 18
to 71 years. Most cases occurred between
the fifth and seventh decades of life, with
the greatest number in the sixth decade.
In contrast to the distribution in children,
twice as many adult cases have occurred
in women as in men. The presenting symptoms are usually an abdominal mass, pain,
and hematuria. Two cases have occurred
in association with pregnancy. 36,45 Hypertension has been found in both adult and
pediatric patients. 1,4,16,31,41,42
Seven cases in the Japanese literature
have been summarized by Kawamura and
Miyakawa.25 This series is noteworthy in
that one of the three pediatric cases was
that of a 5-year-old girl, the oldest child
thus far reported. The adult patients
ranged in age from 49 to 64 years and
included a man who had renal-cell carcinoma and pulmonary metastases. Multilocular cyst of the kidney has, on occasion,
been confused with cystic renal carcinoma.
The etiology and pathogenesis of multilocular cyst are not known. Meland and
Braasch 31 found incomplete septa in solitary renal cysts and felt that multilocular
cysts were similar in origin, resulting from
the failure of fusion of the septa. Arey 2
considered multilocular cyst to be a
hemartomatous lesion. Powell36 suggested
that the lesion arose primarily within the
kidney as a group of sequestered tubules,
or Connheim rests, the number of locules
corresponding to the number of sequestered tubules, with distention being the
chief factor in the development and enlargement of the lesion. Similarly, Bur-
January 1976
MULTILOCULAR CYST OF T H E KIDNEY
rell 8 thought that embryonic tubules
might lie dormant until later development would cause cyst formation due to
their missed connection with the nephron.
Osathanondh and Potter 33 included this
lesion under "Type 2" polycystic kidneys
(caused by a developmental error in which
portions of tubules are converted into
cysts), called "multicystic kidney" when
unilateral and involving the entire kidney, and "multilocular cyst" when only a
portion of the kidney is affected.
The microscopic picture is variable.
Kissane 26 found plates of dysontogenic
cartilage in the wall of a multilocular
cyst. Other cases6,11 •12.17.18.20,27,43 n a v e <j em _
onstrated organoid structures within the
septa. Boggs and Kimmelstiel 6 first noted
frustrate tubules, glomerular anlage,
and embryonic connective tissue reminiscent of Wilms' tumor. They concluded
that these tissues represent neoplastic
growths derived from metanephric blastoma, and called the lesion "benign multilocular cystic nephroma."
Uson and co-workers, 43 when confronted
with the gross appearance of multilocular
cyst but the microscopic pattern of Wilms'
tumor, postulated that the lesion represents separate developmental aberrations of
the mesonephric blastema, and that both
errors occurred simultaneously. Christ 11
also considered multilocular cyst to be
neoplastic, but as an intermediate lesion
with combined features of nephroblastoma
and polycystic kidney, hence the designation "polycystic nephroblastoma." Fowler 17 felt that multilocular cyst was a differentiated nephroblastoma arising from the
same embryonic matrix as Wilms' tumor,
yet differing in the maturity and the proportion of neoplastic elements present.
Available evidence does not yet establish
any of these hypotheses. That the lesions
increase in size is indicated by compression
of renal parenchyma at the periphery of
the mass and the trapping of atrophic
tubules and glomeruli in the capsule. Of
101
interest in this connection is a case in which
a multilocular cyst was discovered in a
patient who four years previously had had
a normal urogram. 42 Progressive increase
in size does not, of course, necessarily
imply neoplastic growth. T h e presence of
atypical tubules that do not look like
trapped, atrophic renal tubules and cysts
lined by a variety of epithelia, as well as
the cellular stroma in some cases, all suggest a neoplastic origin. The epithelium
illustrated in Figure 3 is similar enough to
hyperplastic or neoplastic endothelium to
explain why some investigators have found
the lesions to be indistinguishable from
vascular neoplasms. The fact remains, however, that many authors have not found
such structures microscopically. Boggs
and Kimmelstiel 6 suggest that because
such foci are small they may easily be
overlooked. An alternative explanation
is that although multilocular cysts of the
kidney may all have certain gross characteristics in common, they may not all
have a common cause. This concept would
explain why some appear to be malformations while others strongly suggest a neoplastic origin.
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