E d w a r d E s te v e s , J a q u e s P in u s , R e n a to F r o ta d e A lb u q u e r q u e M a r a n h a o , S im o n e d e C a m p o s V ie ir a A b ib , J o s e P in u s
C rossed T esticular E ctopia
Pediatric Surgery Division of the Department
of Surgery,
Escola Paulista de Medicina - Silo Paulo, Brazil.
C ro s s e d te s tic u la r e c to p ia (C T E ) is a ra re a n o m a ly , c h a ra c te riz e d b y m ig ra tio n o f o n e te s tis to w a rd s th e o p p o s ite in g u in a l c a n a l. P re s e n te d h e re is a c a s e o f c ro s s e d e c to p ia o f th e rig h t te s tis , tre a te d b y e x tra p e rito n e a l tra n s p o s itio n o f th e g o n a d a n d rig h t o rc h io p e x y . E m b rio lo g y a n d s u rg ic a l fin d in g s s u g g e s t th a t C T E is a c o m m o n c o n s e q u e n c e o f m a n y u n c le a r e th io lo g ic fa c to rs , s p e c ia lly m e c h a n i-c a l o n e s , a n d i-c a n b e a s s o i-c ia te d w ith M u lle r d u i-c t p e rs is te n i-c e . R e v ie w o f lite ra tu re s u g g e s ts a i-c la s s ifii-c a tio n o f C T E in to 3 ty p e s : I -a s s o c i-a te d w ith in g u in -a l h e rn i-a -a lo n e ; II - -a s s o c i-a te d w ith p e rs is te n t m u lle ri-a n re m n -a n ts ; III - -a s s o c i-a te d w ith o th e r -a n o m -a lie s w ith o u t m u lle ria n re m n a n ts . T re a tm e n t in c lu d e s tra n s e p ta l o rc h io p e x y o r e x tra p e rito n e a l tra n s p o s itio n o f th e te s tis , re s e a rc h fo r m u lle ria n re m n a n ts a n d o th e r a n o m a lie s , a n d lo n g te rm p o s to p e ra tiv e fo llo w -u p , d u e to th e ris k o f b e c o m in g m a lig n a n t.
U n ite rm s : C ry p to rc h id is m . E c to p ic te s tis . O rc h io p e x y . T e s tic u la r e c to p ia .
T
esticu larch aracterizedecto p iab y lo calizatio nis an an o m alyo f th e testis o u t o f itso f testicu lar d escen t n o rm al m ig ratio n p ath w ay to w ard s th e scro tu m .T h ere are k n o w n fiv e ty p es o f testicu lar ecto p ia: su p erficial
in g u in al (in terstitial), fem o ral (cru ral), p erin eal,
p u b o p en ile, an d cro ssed .
In cro ssed testicu lar ecto p ia (C T E ), th e ecto p ic testis
is fo u n d in th e o p p o site g ro in o r h em iscro tu m , b esid e th e
o th er testis. It is also called tran sv erse testicu lar ecto p ia,
u n ilateral d o u b le testis, testicu lar p seu d o d u p licatio n o r
A d d r e s s fo r c o r r e s p o n d e n c e : E d w a r d E s te v e s P e r e ir a
A v . D r . A ltin o A r a n te s , 1 1 3 2 -A p . 5 2
V ila C le m e n tin o - S a o P a u lo /S P - B r a s il - C E P 0 4 0 4 2 - 0 0 5
tran sv erse ab erran t testis. C T E is a v ery rare co n g en ital
an o m aly , as th ere are ab o u t 1 4 7 rep o rted cases sin ce th e
first d escrip tio n b y V o n L en h o ssek , 1 8 8 6 .
P resen ted h ere is a case o f C T E treated b y th e
P ed iatric S u rg ery D iv isio n o fE sco la P au lista d e M ed icin a,
an d co n sid eratio n s are d iscu ssed ab o u t n ew em b rio lo g ical
co n cep ts, a m o rp h o lo g ical classificatio n as w ell as clin ical,
p h y sio p ath o lo g ical an d th erap eu tic featu res.
C A S E R E P O R T
A 2 -y ear o ld w h ite b o y p resen ted w ith a sw ellin g in
th e left g ro in an d em p ty scro tu m at th e rig h t sid e. P h y sical
sig n s sh o w ed left in g u in o -scro tal h ern ia w ith easily
red u cib le co n ten t, p alp ab le retractile left testis, an d b o th
E S T E V E S , E .; P IN U S , J.;M A R A N H A O , R .F A ; A S IS , S .C .V . & P IN U S , J. -C ro s s e d T e s tic u la r E c to p ia
F ig u re 1 - L e ft in g u in o to m y sh o w in g le ft re tra ctile te stis (th in a rro w ) a n d e cto p ic rig h t te stis in sid e th e h e rn ia l sa c (th ick a rro w ).
F ig u re 2 - S ch e m e sh o w in g o rig in a l p o sitio n s o f th e te ste s (fu ll tra ce ), a n d fin a l p o sitio n s a fte r su rg e ry (cu t tra ce ).
m e s o rc h ia w a s fu s e d b y p e rito n e u m
ju s t a b o v e th e in te rn a l in g u in a l rin g .
T h e le ft te s tis w a s lo c a te d in th e
s c ro tu m b u t w a s re tra c tile , a n d th e
e c to p ic rig h t te s ts la y in th e g ro in ,
n e a r th e le ft in g u in a l rin g . A fte r
is o la tio n a n d h ig h lig a tu re o f th e
s a c , re tro p e rito n e a l d is s e c tio n d e
-m o n s tra te d th a t th e L E F T g o n a d
h a d its v a s d e fe re n s a n d b lo o d
v e s s e ls c o m in g fro m th e le ft s id e .
T h e v e s s e ls a n d v a s o f th e R IG H T
g o n a d o rig in a te d o n th e rig h t s id e ,
c ro s s in g th e b la d d e r a n te rio rly u n til
th e le ft in g u in a l rin g , a n d n o g u b e
r-n a c u lu m w a s s e e n a tta c h e d to th e e c to p ic te s tis (fig . 1 ).
T h e re tra c tile L E F T te s tis
w a s fix e d in th e le ft s c ro tu m
p o u c h . H o w e v e r, a fte r d is s e c tio n ,
th e s p e rm a tic c o rd o f th e R IG H T
te s tis w a s n o t lo n g e n o u g h to a llo w
tra n s e p ta l o rc h io p e x y w ith o u t
te n s io n . T h e re fo re , it w a s d e c id e d
to p e rfo rm a re tro p e rito n e a l tra n s p o s itio n o f th e g o n a d to
th e rig h t g ro in . R IG H T in g u in o to m y re v e a le d a s m a ll
e x te rn a l in g u in a l rin g a ro u n d th e ilio in g u in a l n e rv e , a s w e ll
a s a n e m p ty in g u in a l c a n a l a n d a n a b s e n t in te rn a l rin g .
T h e fa s c ia tra n s v e rs a lis w a s o p e n e d a n d
re tro p e rito n e a l d is s e c tio n p e rm itte d b o th tra n s p o s itio n o f
th e e c to p ic te s tis to th e rig h t g ro in a n d s tra ig h te n in g o f
th e c o rd (fig . 2 ). O rc h ip e x y to th e rig h t s c ro ta l p o u c h w a s
p e rfo rm e d w ith o u t te n s io n , in c is io n s w e re c lo s e d a n d
c o n v a le s c e n c e w a s u n e v e n tfu l. T h e c a rio ty p e a n a ly s e d
p o s te rio rly d e m o n s tra te d g e n o ty p e 4 6 X Y a n d b o th
a b d o m in a l u ltra s o u n d a n d in tra v e n o u s p ie lo g ra p h y w e re
n o rm a l. A fte r th re e y e a rs b o th te s te s w e re p a lp a b le in th e s c ro tu m .
D IS C U S S IO N
T h e re a re a b o u t 1 4 7 c a s e s o f C T E d e s c rib e d s in c e
1 8 8 6 , w h e n V o n L e n h o s s e k re p o rte d a n e c ro p s y fin d in g
(2 8 ). T h e la rg e s t in c id e n c e is fo u n d in E u ro p e a n d J a p a n . S ix ty c a s e s h a v in g b e e n p u b lis h e d in J a p a n e s e lite ra tu re
s in c e th e firs t re p o rt in 1 9 1 2 , b y I w a s a k i(3 7 ).
D u rin g th e la s t 2 0 y e a rs o u r s e rv ic e h a s a tte n d e d 7 5 0
c a s e s o f a n o m a lo u s p o s itio n o f te s tis , a n d o n ly o n e o fC T E 0 • •
I
fixa tio n o f th e
le ft te stis
(re tra ctile ) n o n -p a lp a b le rig h t te s tis a n d s p e rm a tic c o rd . S u s p e c te d o f
h a v in g a le ft in g u in a l h e rn ia a n d rig h t c ry p to rc h id is m , h e
w a s s u b m itte d to s u rg e ry . T h e L E F T in g u in o to m y re v e a le d
tw o te s te s o f e q u a l a p p e a ra n c e w ith in th e h e rn ia l s a c ,
s e p a ra te d e p id id y m id e s a n d v a s a d e fe re n tia , a n d th e
tra n sp o sitio n
o f th e e cto p ic rig h t
te stis
S a o P a u lo M e d ica l Jo u rn a l/R P M 1 1 3 (4 ): 9 3 5 -9 4 0 , 1 9 9 5 E S T E V E S , E .; P IN U S , J .;M A R A N H A o , R .F A ; A S IS , S .C .V . & P IN U S , J .
(1 :750). A fter an extensive review of the literature,
com paring this index w ith the data from C am pbell (1959)
(9), B enson and L ofti (1967) (6), W ooley (1979) (50) and
dem ographic statistics of the U nited N ations (15), the
estim ation of global incidence of C T E is about 1:4 m illion.
T here is no statistical difference in regard to the
affected side, and over the last 20 years the m ean age at
diagnosis has been 9.3 years. O ne case of fam ily incidence
w as reporded by Stauber, in tw o brothers w ith C T E and
persistent m ullerian rem nants (45).
O ur child presented a clinical picture analogous to
the m ost frequently found in C T E . T he patient usually
presents an inguinal hernia w ith ipsilateral palpable testis and an im palpable testis on the other side. T he differential
diagnosis includes unilateral anorchia or criptorchidism ,
or C T E . O ccasionally tw o globular structures are palpable
on the hernia side, and preoperative diagnosis m ay be: a)
crossed testicular ectopi a; b) cord hydrocele,
polyorchidism , sperm atocele, epididym itis or testis tum or,
splenogonadal fusion or acessory adrenal, all associated
w ith contralateral anorchia or abdom inal cryptorchidism .
A s surgery is indicated for the hernia, and due to the rarity of C T E and the other associations above, diagnosis
is generally m ade at operation. Preoperative localization
of the im palpable testis m ay be attem pted w ith ultrasound,
C T , M N R , and m ore accurately, w ith laparoscopy,
herniography (46), arteriography or venography. H ow ever,
if palpation or exam s don't identify the testis, surgical
exploration w ill allow diagnosis and treatm ent.
In all cases w here cariotype w as analysed, the genotype w as 46 X Y . T he m ost frequent anom alies
associated w ith C T E are inguinal hernia, persistent
m ullerian rem nants and incom plete descent of the
non-ectopic testis (tab. 1). A lthough there are only tw o reported cases (19) of high urinary tract abnorm alities (1,2 % ), m ost
authors suggest urinary tract evaluation of patients w ith
C T E .
T here are som e differences am ong the various cases
of C T E , w hich have produced several theories to explain the gene'sis of this rare entity (18,26,28,29,37.). M any
authors propose that abnorm al or absent gubernaculum
could be im portant factors, although has been
dem onstraded norm al testes m igration after gubernaculum
ablation in anim al fetuses (14).
M ost authors agree that each testis is form ed on
different sides, and som ehow one crosses tow ard the
opposite side in the m ajor part of the m igration trajectory.
W e believe that m echanical causes of C T E , like internal inguinal obstruction (14), absent perotoneum -vaginalis
process, absent gubernaculum , m esorchia aderences, and
duct or gonads fusion, are certainly relevant factors
Table 1
Associated anomalies in 148 cases of CTE
(see references)
A n o m a ly n %
In g u in a l h e rn ia 1 4 5 9 8 ,0
P e rs is te n t M O ile r d u c t re m a n e n t 5 6 3 7 ,8
In c o m p le te d e s c e n t o f te s tis 1 2 8 ,1
H id ro c e le 7 4 ,7
T e s tis tu m o r 6 4 ,0
H y p o s p a d ia 6 4 ,0
A b s e n t s e m in a l v e s ic le 3 2 ,0
E c to p ic s e m in a l v e s ic le 1 0 ,6
E c to p ic s c o tu m 1 0 ,6
S u p ra n u m e ra ry e c to p ic e p id id y m is 1 0 ,6
B ila te ra l d u p lic a tio n o f v a s a d e fe re n tia 1 0 ,6
P ie lo u re te ra l ju n c tio n s te n o s is +
re n a l d y s g e n e s is + 0 ,6
s e m in a l v e s ic le c y s t
P ie lic d u p lic a tio n 0 ,6
(20,24,27,47). O ur patient, like others, had neither internal
inguinal ring nor vaginalis process at the right, and no
gubernaculum w as attached to the right testis.
Persistent param enosoneph ic (M U lIer' s) duct
rem anent - tubes, rudim entary uterus, hem iuterus
-occurred in 38% of C T E cases (T ab. 1). Persistence of
m ullerian rem anent in fenotipically norm al m ales represents a recessi ve trait w ith m ale sex restriction (11,21),
in chrom ossom e 19, resulting in abnorm al m ullerian
inhibiting substance (M IS) receptors, or inactive form s of
M IS or even inadequate shintesis of M IS, by the fetal testis
(21,38). In these cases, the testis w ould initially aggregate
the m ullerian rem anent and w ould be carried to the opposite side. Inversely, it is likely that C T E could be the
cause, not the consequence, of som e cases of M uller's duct
rem anent. C onsidering that the ectopic testis has already m igrated to the other side before 8_9th w eek, the period of
the M U ller duct's sensivity to M IS (25). T he concentration
of M IS w ould be insufficient on the original side, leading
to non-degeneration of pisilateral m ullerian structures.
A ctually, in m ost cases the rem anent are hem iuterus, tubes
or m ixed structures associated w ith an abnorm al ectopic
testis.
T hevathasan postulated a classification of C T E
considering the eventual etiology (47). W e suggest a sim ple
classification into three types, based upon the objective
presence of associated anom alies, w hich w ould im ply distinct therapeutic approaches (tab. 2). M any authors
E S T E V E S , E .; P IN U S , J.;M A R A N H A O , R .F .A .; A B IB , S .C .V . & P IN U S , J. -C ro s s e d T e s ti9 u la r E c to p ia
Table
2
Classification of crossed testicular ectopia (CTE)
T y p e I: S im p le e T E , a s s o c ia te d to in g u in a l h e rn ia a lo n e
T y p e II: e T E a s s o c ia te d to p e rs is te n t m u lle ria n re m n a n ts
T y p e III: e T E a s s o c ia te d to o th e r a n o m a lie s
consider that only cases w ithout persistent m tillerian
rem anent m ust be term ed authentic crossed testicular
ectopia(6, 17), w hich w ould resum e the ocurrence to only
about 92 cases in the literature.
It's im portant to assign that thestis tum or in C T E w as
reported in six cases, including five sem inom as in adults
w ith both testes inside the scrotum (19,31,37.). T herefore
long term follow -up and w ise orientation should be
accom plished to all patients.
TREATMENT
In the evidence of C T E at the operation, the approach
depends on the operatory findings:
T ype I (C T E w ith inguinal hernia only):
a) . D issection and high ligature of the hernia sac.
b) D issection and isolation of both cords and vas
deferens m ay be perform ed, allow ing m esorchia
separation and appraisal of both testes origin, in order
to exclude polyorchidism .
c) O rchiopexy. If sperm atic cord lenght is good, as in
m ost cases, it's recom m ended to fix the ectopic testis
in the opposed scrotal pouch by T R A N S E P T A L
technique (O m bredanne-M iller) (36). If the sperm atic
cord is short, as in our case, one can perform testis
T R A N S P O S IT IO N to the other groin through
contralateral inguinotom y, w ith or w ithout
laparotom y, by intra or extraperotoneal approach, or
by staged orchiopexy.
d) C ontralateral inguinal exploration is im portant to rule
out polyorchidism and m ay provide ectopic testis
transposition in cases of short sperm atic cord.
e) Intraoperative vasography is suggested by-F ujita (19)
and P eterson (41), in order to evaluate anatom y,
exclude polyorchidism , and eventually avoid
unnecessary laparotom y or contralateral
inguinotom y. W e consider vasography difficult and
dangerous to the fine vas of a child.
f) It the testis is noted to be atrophic of if C T E occurs
after puberty, orchiectom y is indicated due the high
risk of m alignization (4% ).
T ype II (C T E associated w ith m ullerian rem nants):
besides the steps quited above, in the presence of m ullerian
rem nants, their ablation is not obligatory, and just a
segm entary ressection m ay be done to provide pathological
study. T he inferior uterine segm ent is often adhered to the
vas deferens and m ust be preserved to avoid iatrogenic
infertility (18,21,33).
T ype III (C T E associated w ith different other
abnom alities): In the evidence of other anom alies,
treatm ent should be appropriate to each case.
G enetic evaluation shall be perform ed m ethodically.
D espite association of 1,2% of high urinary tract
anom alies, it's recom m ended urographic investigation in
all cases of crossed testicular ectopia.
RESUMO
In tro d u ~ a o : A e c to p ia te s tic u la r c ru z a d a (E T C ) e u m a a n o m a lia ra ra , c a ra c te riz a d a p e la d e s c id a d e u m te s tftu lo n o c a n a l !n g u in a l d o la d e o p o s to . A p re s e n ta m o s u m c a s o d e e c to p ia c ru z a d a d o te s tfc u lo d ire ito , tra ta d o p o r tra n s p o s iy 8 .o e x tra p e rito n e a l d a g 6 n a d a e o rq u ip e x ia d ire ita . C o n c lu s a o : a s c o n h e c im e n to s e m b rio l6 g ic o s e o s a c h a d o s c iru rg ic o s s u g e re m q u e a E T C s e ja u m a c o n s e q O e n c ia c o m u m d e v a rio s fa to re s e tio l6 g ic o s , s o b re tu d o fa to re s m e c a n ic o s , e p o d e c a u s a r p e rs is te n c ia d o d u c to d e M O ile r. A p 6 s e x te n s a re v is 8 .o d a lite ra tu ra ,s u g e rim o s u m a c la s s ific a c ;8 .o d a E T C e m 3 tip o s : 1 - a s s o c ia d a s o m e n te
a
h e rn ia in g u in a l; II a s s o c ia d a a re m a n e s c e n te s m u lle ria n o s ; III - a s s o c ia d a a o u tra s a n o m a lia s , s e m re m a n e s c e n te s m u lle ria n o s . a tra ta m e n to in c lu i o rq u ip e x ia tra n s -s e p ta l o u tra n s p o s iy 8 .o tra n s -a b d o m in a l d o te s tfc u lo , p e s q u is a d e re m a n e s c e n te s m u lle ria n o s e o u tra s a n o m a lia s , e s e g u im e n to p 6 s -o p e ra t6 rio a la n g e p ra z o , d e v id o a ris c o d e m a lig n iz a y 8 .o .s a o P a u lo M e d ic a l J o u rn a l/R P M 1 1 3 (4 ): 9 3 5 -9 4 0 , 1 9 9 5 E S T E V E S , E .; P IN U S , J.; M A R A N H A O , R .E A .; A S IS , S .C .V . & P IN U S , J.
939
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