Diagnostic

l
Ureteral Triplication Radiology

Patrick J. Perkins, M.D., R. Lawrence Kroovand, M.D., and

Arthur T. Evans, M.D.

ABSTRACT-Seven new cases of ureteral triplication are added to the 53 already

reported in the literature. The embryologic development is also discussed.
This uncommon anomaly usually has a clinical presentation involving dys-
function of one or more of the multiple collecting systems, such as urinary
tract infection or urinary incontinence. I t is not found in association with
other nonurologic anomalies. The clinical significance of ureteral triplication
does not differ from that of ureteral duplication.
INDEX TERMS: Ureters, abnormalities. Urinary Tract, abnormalities
Radiology 108: 533-538, September 1973

is one of the rarest anomalies
T R I P L E URETER
of the upper urinary tract. It is reported to
be associated with an increased incidence of
congenital anomalies as well as a predisposition
to infection and calculus formation (7, 22, 28).
No recent comprehensive review of the subject
of ureteral triplication is available. To the .53
cases reported in the literature, we add 7 of our
own, and present a review and analysis of the
world's literature (TABLE I).
CASE REPORTS
CASE I. A 24-year-old woman was evaluated at the Christ
Hospital, Cincinnati, for primary amenorrhea. The patient
was short and obese, with poorly developed secondary
sexual characteristics (infantile genitalia, sparse body hair).
Excretory urography demonstrated left ureteral triplication
(Fig. 1). No additional urologic studies were performed.
Chromosomal analysis revealed an XXV mosaic pattern.
The final diagnosis was ovarian dysgenesis (Turner's syn-
drome).
CASE II. This 24-year-old man was studied at the Cin-
cinnati General Hospital for hypertension. Past history
was unremarkable. Aside from persistent elevation in
blood pressure (150/90 mm Hg) the patient was normal.
A routine hypertensive pyelogram demonstrated right
ureteral triplication (Fig. 2). Cystoscopy revealed two
ureteral orifices in the right hemitrigone; a third orifice was
located at the bladder neck. A Hutch diverticulum was
demonstrated above and medial to the right hemitrigone.
There was a single left ureteral orifice. Retrograde pyelog-
raphy on the right showed that the lower ectopic orifice
drained the middle collecting system, the upper orifice
drained the upper segment, and the middle orifice the lower
segment. A cystogram demonstrated no vesicoureteral
reflux. No therapy was given for hypertension and the
patient is currently asymptomatic.
CASE III. A 50-year-old man was admitted to the Cin-
cinnati Veterans Administration Hospital for pyuria. The
history was noncontributory and the physical examination
unremarkable. Excretory urography demonstrated left
ureteral triplication (Fig. 3). Cystoscopy showed prostatitis,
a right ureterocele, and a single left ureteral orifice. Left
retrograde pyelography revealed that the left ureters united
at the pelvic brim. No follow-up is available.
CASE IV. A six-year-old girl was evaluated for chronic
urinary tract infections at the Syracuse Memorial Hospita1.
Both history and physical examination were unremarkable.
An excretory urogram suggested right ureteral triplication
and a cystogram showed vesicoureteral reflux into the middle
collecting system on the right. Retrograde pyelography also
suggested right ureteral triplication, although the three
ureters could not be clearly demonstrated. No description is
available of other cystoscopic findings.
Surgical exploration revealed right ureteral triplication with
a hydronephrotic dysplastic upper collecting system whose
ureter was atretic. There was hydronephrosis and pyelo-
nephritis of the middle collecting system. The lower collect-
ing system was normal. A right nephrectomy was per-
formed. The postoperative course has been uneventful.
CASE V. A seven-year-old girl was admitted to the Crouse
Irving Hospital, Syracuse, New York, for evaluation of
urinary incontinence. The child had a lifelong history of a
normal voiding pattern but was continually wet between
voidings. Past history was otherwise unremarkable. Phys-
ical examination revealed a damp perineum. No ectopic
ureteral orifice could be found.
Excretory urography (Fig. 4) demonstrated left ureteral
triplication. Cystoscopy revealed two left ureteral orifices
in the trigone. Ectopic flow of urine was noted in the per-
ineum, but the orifice could not be located.
Retroperitoneal exploration revealed three left ureters.
The ectopic ureter emptied into the vagina and drained the
middle collecting system. This ureter was resected and its
stump ligated. The perineum remained dry postoperatively
and the patient has done well.
CASE VI. A 16-year-old boy was admitted to the Cin-
cinnati General Hospital with hypertension. The blood

1 From the Departments of Radiology (P. J. P.) and Urology (R. L. K., A. T. E.), University of Cincinnati Medical Center, Cincinnati,
Ohio. Accepted for publication in March 1973. ' .
Presented at the North Central Section of the American Urologic Association, Chicago, 111., Sep. 28-30, 1972, and at the American
Academy of Pediatrics, Section on Urology, New York, N. Y.. Oct. 14-19, 1972. shan
533
 Fig. 1. CASE T. Left ureteral triplication.
Fig. 2. CASE II. Routine hypertensive pyelogram demonstrates right ureteral triplication.

Fig. 3. CASE III. Excretory urogram demonstrates left ureteral triplication.

Fig. 4. CASE V. A. Excretory urogram reveals left ureteral triplication. On close inspection, a third ureter is faintly identified
between two obvious ureters just beyond the level of the kidney.
B. Composite drawing made from several films of the excretory urography sequence, none of which showed the entire collecting sys-
tem with adequate detail.
534
 Diagnostic
Vol. 108 URETERAL TRIPLICATION 535 Radiology

pressure was 140/100 mm Hg. Physical examination was
otherwise unremarkable. An excretory urogram demon-
strated left ureteral triplication (Fig. 5). The three ureters
united shortly below their origin. No additional urologic
evaluation was performed. No follow-up is available.
CASE VII: A 33-year-old woman was evaluated for
dysuria and dysmenorrhea at the University of California
Hospital. Both history and physical examination were
unremarkable.
An excretory urogram demonstrated three right ureters.
At cystoscopy, two right ureteral orifices were seen. The
patient also had acute cystitis and urethral stenosis. Retro-
grade pyelography indicated that the upper vesical orifice
drained the middle and lower collecting systems whose ureters
fused at the L-4 level (Fig. 6). The lower trigonal orifice
drained the upper collecting system. The patient under-
went a hysterectomy for endometriosis. There have been
no urinary symptoms.
DISCUSSION
Embryologic ureteral development normally
begins at five weeks with a bud from the wolffian
duct. This bud grows dorsally and then cranially
to join the metanephrogenic cap of tissue which
forms the renal parenchyma. As the embryo
develops, the wolffian duct descends and becomes
incorporated into the part of the cloaca destined to
become the bladder. When multiple ureteral
buds are present, the lowest one, which drains the
most caudal renal segment, is incorporated first
into the bladder, where it subsequently moves
cranially and laterally. On further caudal dis-
placement of the wolffian duct, the more cranial
ureter becomes incorporated medially and caudally
in the bladder or even in the more distal derivatives
of the wolffian duct below the bladder (Weigert-
Meyer law) (31). The farther apart the mul-
tiple ureteral buds are situated, the more likely
the more cranial buds will terminate extravesi-
cally in caudal wolffian duct remnants (37).
An ectopic ureteral orifice may occur anywhere
between the normal location on the trigone
and the caudal end of the wolffian duct. In
men, the wolffian duct forms the posterior urethra,
appendix epididymis, vas deferens, epididymis,
ejaculatory duct, and the seminal vesicle (7,47).
In women the wolffian duct is vestigial. Its
remnants include the duct of the epoophoron
(usually found in the broad ligament), Gartner's
duct (usually found in the lateral vaginal wall,
but sometimes at the hymen), and the supra-
sphincteric urethra (37) (Fig. 7).
Whether ureteral triplication represents a nor-
mal variant or a congenital anomaly in number
has been discussed. We agree with Day and
Nordmark (12, 36) that normal ureteral develop-
ment stops with division of the ureter into an
upper and lower major calix and that any addi-
tional division is anomalous.
'In 1946, Smith (48) reviewed the subject of

Fig. 5. CASE VI. Left ureteral triplication. The three ureters are united shortly below their origin.
Fig. 6. CASE VII. Retrograde pyelogram indicates that the upper vesical orifice drains the middle and lower collecting systems whose
ureters fuse at the L-4 level. The lower trigonal orifice drains the upper collecting system.
536 P. J. PERKINS, R. L. KROOVAND AND A. T. EVANS September 1973

BlADDER
MALE
TRIGCH

? UTERUS
TRIGONE
? CERVIX
SEMINAL VESICLE

VAGINA

VAG I HAL VEST' BULE

lI£[RAL ORIFICE ECTOPIA

Fig. 7. Composite diagram of possible locations of ectopic ureteral orifices
in the female (left half) and the male (right half).

T ABLE I: REVIE\V OF REPORTED CASES OF URETERAL triplication (Fig. 8). Spangler (49) summarized
TRIPLICATION ACCORDING TO SMITH'S CLASSIFICATION
Smith's classification thus:
Type I Type III Type I: Triple ureter (complete triplica-
Lau (25) Furstner (15)
Burt (6) Perrin (38) tion): 3 complete ureters with 3
Woodruff (56) Franck (14) ureteral orifices.
Muller (34) Schroter (44)
Wright (58) Smith (48) Type II: Double ureter with one bifid: 3
Ireland (22) Withycombe (55) ureters arise from the kidney, but 2
Demoullin (13)* Gill (16)
Ratano (23) Lorbek (28) join; 2 ureteral orifices are found.
G6tzen (19) G6tzen (18) Type III: Trifid ureter: 3 ureters arise from
Spangler (49) Stobbaerts (50)
Livaditis (27) Bauchard (4)* the kidney and one ureteral orifice
Ringer (41) Sakamoto (23) is present.
Newman (35) Nakazin (23)
Parker (37) Nagano (23) Type IV: Double ureter with "inverted V"~
Trede (52, 53) Brueziere (5) bifurcation: 2 ureters arise from
CASE II Fujii (23)
CASE V Scott (45)
the kidney; one bifurcates so that
Corney (11) there are 3 ureteral orifices.
Type II CASE III
Ureteral triplication is more common in women
Wrany (57) Type IV
(34 of 62) and more frequently left sided (35 of
Miller (33) Chwalla (9) 62). This is in concert with the studies on renal
MacLean (30) Lenke (26)
Axilrod (3) anomalies by Schmutte (42). Lorbek (28) con-
MacKelvie (29) Indeterminate curs with our findings; in addition he states that
Petrovcic (40) Alvisi (1)
Demoullin (13) * G6tzen (20) there is an hereditary tendency to the occur-
Goto (23) Cibert (10) rence of ureteral triplication as well as an associa-
Sierra Lange (46) Yelsa (59)
Reuss (21) Gogichaev (17) tion with other congenital anomalies such as
Peterson (39) CASE I syndactyly, angioma, and malformation of the
Izawa (23) CASE IV
Mikulicek (32) CASE VI sex organs for which he gave no verification. The
CASE VII ages of the 60 patients at the time of presentation
* Bilateral triplication. varied greatly (six months to seventy-four years).
 Diagnostic
Vol. 108 URETERAL TRIPLICATION 537 Radiology

I m
Fig. 8. Types of ureteral triplication according to Smith (see text).

Of the overall group of patients, 37 presented and Chute (22) offer an explanation for this lack
with symptoms referable to urinary tract infec- of absolute conformity to the Weigert-Meyer law.
tion (flank pain, hematuria, dysuria) involving Fifty-eight patients had unilateral ureteral
one or more of the collecting systems. Urinary triplication; in 2 patients (4, 13) there was bi-
calculus was found only five times; 5 patients lateral triplication. Ureteral duplication was pres-
had no urologic complaints (TABLE II) . ent in the contralateral collecting system in 19.
Ureteral ectopia involving the triplicate sys- This represents the most common anomaly as-
tem was seen 13 times. Eleven of these patients
were women, 8 of whom had some urinary in- TABLE II: CLINICAL SYMPTOMS IN 60 PATIENTS WITH
URETERAL TRIPLICATION
continence. The 2 men were asymptomatic with
reference to the ectopic ureter. The location Lumbar or flank pain 20
Urinary tract infection 18
of the ectopic ureteral orifice was always within Urinary incontinence 11
the distribution of the wolffian duct derivatives. Hematuria 6
Ureteral colic or stone 5
We have found, in agreement with others (6, 22, Asymptomatic 5
37,41,42), that complete ureteral triplication has Hypertension 3
Failure to thrive 2
a higher incidence of ectopic orifice. Postmortem or intraoperative finding 4
Smith Types I, II, and III occurred with not
disparate frequency: 26%, 23%, and 34%, re- TABLE III: ASSOCIATED FINDINGS IN PAT1ENTS WITH
URETERAL TRIPLICATION .
spectively. This probably indicates that the
degree (Smith Type I, II, or III) and extent of None noted 27
Contralateral ureteral duplication 19
ureteral triplication (as well as duplication) is Ectopic ureters 17
a temporal function of anomalous development Triplicated unit 13
Contralateral side 4
during embryogenesis. Type IV occurred in 3%. Renal dysplasia 5
The latter is difficult to explain embryologically. Stone (ureteral, renal, bladder) 5
Triplicated unit 3
Possibly it is a result of the development of two Contralateral side 2
ureteral buds from the wolffian duct which unite Hypertension 3
Contralateral triplication 2
prior to encountering the metanephrogenic cap Pyelonephritis 2
of tissue; or the third ureter may represent per- Clitoral hypospadias & V.S.D. 1
Spina bifida 1
sistence of the wolffian duct itself (24). Thirteen Ovarian dysgenesis 1
per cent of reported cases did not have sufficient Pelvic kidney 1
Hydrocele 1
information to permit accurate classification. Ureteral polyp 1
A review of the 17 examples of Type I reveals Ureterocele 1
Hutch diverticulum 1
that the Weigert-Meyer law did not hold for 5 Vesicoureteral reflux 1
of the 8 patients in whom the details were suffi- Endometriosis 1
Cystitis 1
ciently well seen to apply this rule. Ireland
538 P. J. PERKINS, R. L.
sociated with ureteral triplication. Additional
anomalies coexisting with ureteral triplication are
ureteral ectopia (17 patients) and renal dysplasia
(5 patients). Other associated findings were
unusual and rarely symptomatic (TABLE III).
ADDENDUM: Since preparation of this paper, we have seen
an additional case of ureteral triplication. I t is similar to
that recorded by MacKelvie (29) (viz., three ureters, one
of which is blind-ending; two ureteral orifices are present)
and will be reported elsewhere.
ACKNOWLEDGMENTS: We should like to thank Dr. Chapin
Hawley, Christ Hospital, Cincinnati, for permission to pub-
lish CASE I; Dr. Donald Heimbrock, Cincinnati Veterans
Administration Hospital, for CASE III; Dr. George Mitchell,
Syracuse Memorial Hospital, Syracuse, New York, for
CASES IV and V; and Dr. Hideyo Minagi, University of
California, San Francisco, for CASE VII. We also appreciate
the assistance of Dr. Benjamin Felson in the preparation
of this paper.
We greatly appreciate the translations rendered by Drs.
Alan Chambers, Ramiro Correa, Tohru Ishikawa, Vjekoslav
Mikelic and Jan Schwarz. Mr. John Barnard most gra-
ciously provided the illustrations.
Department of Radiology
Cincinnati General Hospital
Cincinnati, Ohio 45229
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