C lin ic a i a n d r a d io lo g ic a l p o s to p e r a tiv e e v a lu a tio n
o f
p o s te r io r s a g itta l a n o r e c to p la s ty in p a tie n ts w ith _ u p p e r a n d
in te r m e d ia te a n o r e c ta l m a lf o r m a tio n s
Department
of Imaging Diagnosis, Discipline of Pediatric Surgery of the Department
of Surgery
Universidade Federal de São Paulo - São Paulo, Brazil
lh e P S A R P is to d a y th e m o st-u se d su rg ica l te ch n iq u e fo r co rre ctio n o f h ig h a n d in te rm e d ia ry a n o re cta l m a lfo rm a tio n s. lh e re is m u ch co n tro ve rsy in th e lite ra tu re a b o u t th e p o st-o p e ra tive e va lu a tio n o f th e se ca se s. W e stu d ie d 2 7 ca se s o f a n o re cta l m a lfo rm a tio n s fro m clin ica i a n d ra d io lo g ica l a sp e cts, in o rd e r to a n a lyse : 1 .F e ca l co n tin e n ce 2 .R e la tio n sh ip b e tw e e n p o st-o p e ra tive fe ca l co n tin e n ce a n d th e a sso cia te d sa cra l a n o m a lie s 3 .R e la tio n sh ip b e tw e e n th e ra d io lo g ica l e va lu a tio n b y d e fe co g ra m a n d fe ca l co n tin e n ce
F ro m th e a n a lysis o f th e ca se s, w e co n clu d e d : 1 .F e ca l co n tin e n ce w a s a ch ie ve d in 4 8 .1 4 % o f th e ca se s; p a rtia l co n tin e n ce in 2 5 .9 2 % ; a n d fe ca l in co n tin e n ce in 2 5 .9 2 % o f th e ca se s. 2 .T h e p re se n ce o f fe ca l in co n tin e n ce w a s d ire ctly re la te d to th e a sso cia te d sa cra l a n o m a lie s.
U N IT E R M S : A n o re cta l m a lfo rm a tio n s. P o ste rio r sa g itta l a n o re cto p la sty.
IN T R O D U C T IO N
S
i n c e s t u d i e s d o n e b y P e n a a n d D e V r i e sI, p o s t e r i o r s a g i t t a l a n o r e c t o p l a s t y ( P S A R P ) h a s b e c o m e t h e m a i n t r e a t m e n t f o r u p p e r a n d i n t e r m e d i a t e a n o r e c t a l m a l f o r m a t i o n s ( A R M ) a r o u n d t h e w o r l d .
H o w e v e r , c a r e f u l l o n g t e r m e v a l u a t i o n m u s t b e d o n e t o a l l o w c o m p l e t e a p p r o v a l o f t h i s m e t h o d .
T h e r e f o r e , w e a n a l y z e d c l i n i c a I a n d r a d i o l o g i c a l a s s a y s o f 2 7 c h i l d r e n w i t h u p p e r a n d i n t e r m e d i a t e A R M s w h o u n d e r w e n t P S A R P .
Address for correspondence:
José Luiz M artins
R ua dos O tonis, 131. V. C lem entino
São Paulo/SP - Brasil- C EP 04025-000
P A T IE N T S
W e s t u d i e d 2 7 w h i t e c h i l d r e n w i t h 2 1 u p p e r a n d 6 i n t e r m e d i a t e A R M s , o f w h i c h 1 7 w e r e m a l e a n d 1 0 w e r e f e m a l e , w h o u n d e r w e n t c o l o s t o m i e s , P S A R P s , a n d a n a l d i l a t i o n s w i t h H e g a r 's c a n d l e , a f t e r w h i c h t h e c o l o s t o m i e s w e r e c l o s e d ; a g e s v a r i e d f r o m 4 t o 1 1 y e a r s . W e s e l e c t e d p a t i e n t s w h o w e r e o v e r 4 y e a r s o l d b e c a u s e a t t h i s a g e f e c a l c o n t i n e n c e i s w e l l - d e v e l o p e d i n n o r m a l c h i l d r e n . A l I c h i l d r e n w e r e e v a l u a t e d 3 m o n t h s o r m o r e a f t e r t h e c o l o s t o m i e s w e r e c l o s e d .
A l l c h i l d r e n w e r e e v a l u a t e d c l i n i c a l l y a n d r a d i o l o g i c a l l y b y d e f e c o g r a m s u s i n g t h e H a b e r k o r n t e c h n i q u e2 f o r t h e f o l l o w i n g p a r a m e t e r s :
1 . F e c a l c o n t i n e n c e
2 . R e l a t i o n b e t w e e n f e c a l c o n t i n e n c e a n d t h e p r e s e n c e o f s a c r a l m a l f o r m a t i o n s
M A R T IN S , J.L .; L E D E R M A N , E .M .; P IN U S , J. - C lin ica i a n d ra d io lo g ica l p o sto p e ra tive e va lu a tio n o f p o ste rio r sa g itta l a n o re cto p la sty in p a tie n ts w ith u p p e r a n d
in te rm e d ia te a n o re cta l m a lfo rm a tio n s
1567
3. R elationship betw een fecal continence and' dynam ic radiology evaluation by defecogram , analyzing the follow ing features:
a) Presence of rectal reservoir b) Presence of fecalom a
c) Im pression on posterior rectum caused by the , çontractions of the :puborectalis of the levator
ani, w hich is part of the superior sphincter m uscular com plex
d) A bsence of inferior sphincter m uscular com pIex tightening, w hich is form ed by the sphincter ani externs and is dem onstrated by constant evacuation
C hildren w ho: defecated once or tw ice a day; w ith no soiling; w hose feces and anus presented no alterations; w ith good upper and low er rectal contraction during exam ination w ere considered continent.
C hiIdren w ho: defecated three to five tim es a day; w ith norm al feces and frequent soiIing; w ho presented rectal prolapse; w ith m oderate upper or Iow er contraction during rectal exam ination w ere considered partially continent.
C hildren w ho: defecated m ore than five tim es a day; w ith liquid feces; constant and total fecalloss; anus w ith a large opening, or rectal prolapse, and loss of feces; w ith light or nil upper or low er contraction during exam ination w ere consÍdered incontinent3•
W e used chi-square test for statistical analysis of table 2 to com pare continent, partially continent, and incontinent patients according to the characteristics above. W e used Fisher's exact test4 to associate continent,
partially continent, and incontinent patients' tightening of externaI sphincter; the leveI of significance used w as
5%
( p ~0.05).
R E S U L T S
1. Fecal Continence
Thirteen of the 27 cases presented fecal continence, 7 cases presented partial continence, and 7 presented incontinence.
Eight of the thirteen continent patients had upper, A R M s and 5 had interm ediate A R M s; six of the seven partially continent-patients had upper A R M s and 1had an interm ediate A R M ; all 7 incontinent patients had upper A R M s.
D istribution according to gender w as the follow ing: C ontinent - 7 m ale and 6 fem ale.
Partial1y continent - 5 m ale and 2 fem ale. Incontinent - 5 m ale and 2 fem ale.
2. Relation Between Fecal Continence and Associated
Sacral Malformations
O ne of the thirteen continent children presented an associated sacral m alform ation; 3 of the seven partially continent children presented associated sacral anom alies; six of the seven incontinent children presented associated sacral anom alies.
V ertebral m al form ations found in these patients can be seen in TabIe
1.
T a b le 1
A s s o c ia te d V e rte b ra l M a lfo rm a tio n s
C a s e M a lfo rm a tio n
4 F u s io n o f v e rte b ra e 4 a n d 5 . A b s e n t C o c c y x
6 A b s e n c e o f 5 s a c ra l v e rte b ra e a n d c o c c y x
7 F u s io n o f s a c ra l v e rte b ra e 2 , 3 , a n d 4 .
A b s e n c e o f 5 s a c ra l v e rte b ra e a n d c o c c y x
1 0 F u s io n o f a li s a c ra l v e rte b ra e . A b s e n c e o f
c o c c y x . L o m b o s a c ra l h e m iv e rte b ra . S c o lio s is
1 1 A b s e n c e o f s a c ra l v ,e rte b ra e 5 a n d c o c c y x
1 3 F u s io n o f s a c ra l v e rte b ra e 2 , 3 , a n d 4 .
A b s e n c e o f s a c ra l v e rte b ra e 5 a n d c o c c y x
1 5 A b s e n c e o f s a c ra l v e rte b ra e 5 a n d c o c c y x
1 9 F u s io n o f s a c ra l v e rte b ra e 1 ,2 , 3 . A b s e n c e o f v e rte b ra e 4 , 5 , a n d c o c c y x . H e m ia g e n e s ia
o f le ft re m a in in g s a c ra l v e rte b ra e .
2 4 S c im ita r v e rte b ra 5 . In c re a s e o f
in te rp e d ic u la r s p a c e b e tw e e n s a c ra l
v e rte b ra e 1 a n d 2 .
2 5 A b s e n c e o f s a c ra l v e rte b ra 5 a n d c o c c y x
3. Relation Between Fecal Continence and Defecogram
R esults are show n in Table 2.
T A B L E 2
C o n tin e n c e v s . D e fe c o g ra m
N
RR
F E C A A IM P T ICC o n tin e n c e 1 3 1 3 O 1 3 1 3 1 3
P a rtia l C o n t. 7 7 1 7 7 7
In c o n tin e n c e 7 5 1 6 6 2
N = n u m b e r o f e v a lu a te d p a tie n ts A R = re c ta l re s e rv o ir
F E C = fe c a lo m a
A A
=
a n o re c ta l a n g le a lte ra tio n d u rin g e v a c u a tio nIM P
=
Im p re s s io n o f th e s u p e rio r s p h in c te r c o m p le x o n p o s te rio r re c tu mT IC = tig h te n in g o f in fe rio r s p h in c te r c o m p le x
There w as a significant association betw een incontinence and absence of tightening of externaI sphincter in the defecogram (Fig. 1and 2).,
M A R T IN S , J .L .; L E D E R M A N , E .M .; P IN U S , J . - C lin ic a i a n d ra d io lo g ic a l p o s to p e ra tiv e e v a lu a tio n o f p o s te rio r s a g itta l a n o re c to p la s ty in p a tie n ts w ith u p p e r a n d
in te rm e d ia te a n o re c ta l m a lfo rm a tio n s
1 5 6 8
F ig u r e 1 • T ig h te n in g o f e x te r n a i s p h in c te r in c o n tin e n t p a tie n t
F ig u r e 2 • A b s e n c e o f e x te r n a i s p h in c te r tig h te n in g in in c o n tin e n t
p a tie n t.
F ig u r e 3 • A b s e n c e o f e x te r n a i s p h in c te r tig h te n in g a s s o c ia te d w ith fib r o s is in in c o n tin e n t p a tie n t
D IS C U S S IO N
P o s to p e r a tiv e e v a lu a tio n o f f e c a l c o n tin e n c e a f te r c o r r e c tio n o f A R M s h a s b e e n c o n tr o v e r s ia l, a n d a c o n s e n s u s h a s n o t b e e n r e a c h e d in s p ite o f n u m e r o u s r e p o r ts ( 5 , 6 , 7 a n d 8 ) .
O u r c lin ic a I e v a lu a tio n is b a s e d o n a b le n d o f w h a t h a s b e e n p r o p o s e d r e c e n tly .
O u r r e s u lts a g r e e w ith th o s e b y o th e r a u th o r s , w ith th e e x c e p tio n o f L a n je m e ije r &M o lle n a a r - 1 9 9 1 ( 9 ) , w h o s ta te d th a t th is s u r g ic a l te c h n iq u e n e v e r a llo w s n o r m a l f e c a l c o n tin e n c e .
T h e p r e s e n c e o f s a c r a l m a lf o r m a tio n s h a v e a n e f f e c t o n f e c a l c o n tin e n c e e s p e c ia lly in u p p e r A R M . C lin ic a I m a n if e s ta tio n s a r e g r e a te r w h e n e n tir e s a c r a l s e c tio n s a r e n o t p r e s e n t, b u t e v e n h e m is a c r a l m a lf o r m a tio n s m a y b e o c c a s io n a lly r e s p o n s ib le f o r n e r v e in v o lv e m e n t; th e r e f o r e , it is im p o r ta n t to o b s e r v e th e f o llo w in g d a ta :
1 . I n n e r v a tio n o f le v a to r a n i a n d b la d d e r a r e u s u a lI y n o r m a l in m a lf o r m a tio n s o f v e r te b r a e 4 a n d 5 .
2 . M a lf o r m a tio n s o f s a c r a l v e r te b r a e 3 , 4 , a n d 5 le a d to v a r ia b le in n e r v a tio n o f b la d d e r a n d le v a to r a n i, b u t m o s t p a tie n ts b e c o m e in c o n tin e n te
3 . M a lf o r m a tio n s o f s a c r a l v e r te b r a e 1 a n d 2 a r e a s s o c ia te d w ith in c o n tin e n c e s in c e th e le v a to r a n i n o t o n ly p r e s e n ts p r o b le m s w ith its in n e r v a tio n b u t a ls o is u n d e r d e v e lo p e d .
4 . H e m is a c r a l m a lf o r m a tio n s a r e u n p r e d ic ta b le ; in v o lv e m e n t o f v e r te b r a e 4 a n d 5 in th e s e c a s e s u s u a lly le a d s to b la d d e r a n d le v a to r a n i m a lf u n c tio n s .
I n o u r s tu d y , 4 2 .8 5 % o f th e p a r tia lly c o n tin e n t a n d 8 5 .7 1 % o f th e in c o n tin e n t p a tie n ts p r e s e n te d s a c r a l m a lf o r m a tio n s . T h e s e r e s u lts a g r e e w ith s tu d ie s o f P e n a 1 0 a n d M a r tin s 1 1 , w h ic h s ta te th a t s a c r a l m a lf o r m a tio n s a r e u s u a lly a s s o c ia te d w ith n e u r o lo g ic a l p r o b le m s a n d m u s c le w e a k e n in g .
E v a lu a tio n o f p a tie n ts u s in g d e f e c o g r a m s in o u r s tu d y w a s c o n s is te n t w ith f e c a l c o n tin e n c e , a lth o u g h m a n y a u th o r s c o n s id e r p u b o r e c ta l im p r e s s io n o n th e p o s te r io r r e c tu m to b e m o r e im p o r ta n t, e g : K e lly I 2 .1 3 , C y w e s e t a l
'4.
T h e s e a u th o r s r e p o r t th a t th e p u b o r e c ta l im p r e s s io n o n th e p o s te r io r r e c tu m g iv e s a b e tte r id e a o f th e f u n c tio n o f th e s p h in c te r a n d its r e la tio n to f e c a l c o n tin e n c e . O u r s tu d y d o e s n o t a g r e e s in c e a lI p a tie n ts , in c lu d in g th e in c o n tin e n t o n e s , p r e s e n te d a p u b o r e c ta l im p r e s s io n o n th e p o s te r io r r e c tu m , w ith o n ly o n e e x c e p tio n .
O u r r e s u lts s h o w th a t th e b e s t p a r a m e te r is th e tig h te n in g o f th e e x te r n a I a n a l s p h in c te r , w h ic h w a s p r e s e n t in a ll c o n tin e n t p a tie n ts a n d n o .t p r e s e n t in 7 1 .4 2 % o f th e in c o n tin e n t p a tie n ts .
O th e r f e a tu r e s , s u c h a s th e r e c ta l r e s e r v o ir , th e p r e s e n c e o f f e c a lo m a , a n d th e a lte r a tio n o f a n o r e c ta l a n g le d id n o t s h o w a n y s ig n if ic a n t r e s u lts in th e e v a lu a tio n o f p o s to p e r a tiv e c o n tin e n c e .
Jlli:::~
M A R T IN S , J .L .; L E D E R M A N , E .M .; P IN U S , J . - C lin ic a i a n d r a d io lo g ic a l p o s to p e r a tiv e e v a lu a tio n o f p o s te r io r s a g itta l a n o r e c to p la s ty in p a tie n ts w ith u p p e r a n d
in te r m e d ia te a n o r e c ta l m a lfo r m a tio n s
C O N C L U S IO N S
1.
PSARP
was
efficient
in maintaining
fecal
continence
in 48.14%
of the cases and partial
fecal continence
in 25.92% of the cases.
1569
2.
Fecal incontinence
was associated
directly with
sacral malformations.
3.
Absence
of externaI
anal sphincter
tightening
in
patients
with
fecal
incontinence,
as
demonstrated
by defecograms,
was significantly
higher.
RESUM O
A A R P S P é a tu a lm e n te u m a d a s té c n ic a s m a is u tiliz a d a s n a c o r r e ç ã o c ir ú r g ic a d a s a n o m a lia s a n o r r e ta is a lta s e in te r m e d iá r ia s , e x is tin d o m u ita c o n tr o v é r s ia n a lite r a tu r a q u a n to a o s c r ité r io s d e a v a lia ç ã o d e s s e s p a c ie n te s n o p ó s - o p e r a tó r io . F o r a m e s tu d a d o s 2 7 p a c ie n te s d o p o n to d e v is ta c lín ic o e r a d io ló g ic o p e lo d e fe c o g r a m a , s u b m e tid o s
à
A R P S ~ v is a n d o a v a lia r o s s e g u in te s p a r â m e tr o s : 1 . C o n tin ê n c ia fe c a l; 2 . R e la ç ã o e n tr e a c o n tin ê n c ia fe c a l e a s a n o m a lia s s a c r a is a s s o c ia d a s ; 3 .R e la ç ã o e n tr e a a v a lia ç ã o r a d io ló g ic a d in â m ic a p e lo d e fe c o g r a m a e a c o n tin ê n c ia fe c a l. D a a n á lis e d a c a s u ís tic a fo r a m o b tid a s a s s e g u in te s c o n c lu s õ e s : 1 .A c o n tin ê n c ia fe c a l fo i o b tid a e m 4 8 ,1 4% d o s c a s o s ; p a r c ia lm e n te e m 2 5 ,9 2 % d o s c a s o s ; e a in c o n tin ê n c ia fe c a l fo i o b s e r v a d a e m 2 5 ,9 2 % d o s c a s o s .2 .A o c o r r ê n c ia d e in c o n tin ê n c ia fe c a l e s te v e d ir e ta m e n te r e la c io n a d a c o m a a s s o c ia ç ã o d e a n o m a lia s s a c r a is . 3 .N o s p a c ie n te s c o m in c o n tin ê n c ia fe c a l, a a u s ê n c ia d e o c lu s ã o d o e s fín c te r e x te r n o d o â n u s n o d e fe c o g r a m a fo i s ig n ific a n te m e n te m a io r .R E F E R E N C E S
1. Pena A , De Vries SPA- Posterior sagital anorectoplasty: important technical considerations and applications. J Pediatr Surg 1982; 17(6):796-811.
2. Haberkorn S, M athias A L ,Teixeira M G, et aI. -Defecograma: técnica simplificada e dados de interpretação. Rev Ass M ed Bras 1978; 24(10):361-2.
3. M artins J L - Avaliação pós-operatória de crianças portadoras de anomalias anorretais submetidas a correção cirúrgica pela anorretoplastia sagital posterior. Tese de Docência Livre. Escola Paulista de M edicina.
111p, 1993.
4. Siegel S - Estadistica parametrica. Ed.Trillas.M exico. 346p, 1975.
5. Partridge J P & Gough M H - Congenital abnormalities of the anus and rectum. Brit J Surg 1961; 49:37-50.
6. S\venson O & Donnellan W L - Preservation of the puborectalis sling in imperforate anus repair. Surg Clin North An1er 1967; 47: 173-93.
7. Stephens F D & Smith E D - Anorectal malformations in children. Year Book M edicaI Publishers. Chicago, 1971. 8. Templeton Jr J M & Diteshein J A - High imperforate anus:
quantitative results of long term fecal continence. J Pediatr Surg 1985; 20(6):645-52.
9. Langemeijeir R A T M & M ollenaar J C - Continence after posterior sagital anorectoplasty. J Pediatr Surg 1991; 26(5):587-590.
10. Pena A - Posterior sagital approach for correction of anorectal malformations. Adv Surg 1986; 19:69-100.
11. M artins J L - Complicações no tratamento das anomalias anorretais.In:M argarido N F,Saad R, Cecconello I, M artins J L & Soares L A - Complicações em Cirurgia. 1l iEd. São
Paulo: Ed.Robe, 1992:297-313.
12. Kelly
J
H - Cineradiography in anorectal malformations. J Pediatr Surg 1969; 4(5):538-46.13. Kelly J H -The clinicaI and radioIogical assesment of anal continence in childhood. Aust N Z J Surg 1972; 42(1):62-63. 14. Cywes S, Cremin B J& Louw J H - Assesment of continence after treatment for anorectal agenesis: a clinicaI and radiologic correlation.
J
Pediatr Surg 1972; 6: 132-137.M A R T IN S , J .L .; L E D E R M A N , E .M .; P IN U S , J . - C lin ic a i a n d r a d io lo g ic a l p o s to p e r a tiv e e v a lu a tio n o f p o s te r io r s a g itta l a n o r e c to p la s ty in p a tie n ts w ith u p p e r a n d
in te r m e d ia te a n o r e c ta l m a lfo r m a tio n s
