Jc. Langer et E. Birnbaum, PRELIMINARY EXPERIENCE WITH INTRASPHINCTERIC BOTULINUM TOXIN FOR PERSISTENT CONSTIPATION AFTER PULL-THROUGH FOR HIRSCHSPRUNGS-DISEASE, Journal of pediatric surgery, 32(7), 1997, pp. 1059-1061
Although most children who have Hirschsprung's disease have an excelle
nt result after pull-through surgery, some experience persistent const
ipation caused by ''internal sphincter achalasia.'' Anal myectomy has
been advocated for this problem, but it results in permanent injury to
the sphincter and is not universally effective. Botulinum toxin has b
een safely used to selectively and reversibly weaken a variety of volu
ntary muscles and sphincters in both adults and children. Injection of
botulinum toxin into the internal anal sphincter (IAS) should theoret
ically produce the same functional result as anal myectomy without per
manent sphincter injury. Four children aged 4 to 8 years presented wit
h persistent constipation after a pull-through procedure for Hirschspr
ung's disease. Two had associated encopresis, both of whom had previou
s myectomies. The authors performed four-quadrant intrasphincteric bot
ulinum toxin injection (total dose, 15 U). Resting IAS pressure decrea
sed in all children 4 to 8 weeks after injection. Patients have been f
ollowed up for 7 to 9 months. One child (with Down's syndrome) remaine
d symptomatically unchanged. The other three families reported signifi
cant improvement in bowel function in their children. In two of these,
there was a return of symptoms 6 months after injection; one child un
derwent reinjection with good results. Postinjection incontinence occu
rred in three children, but resolved after several weeks in the one wh
o did not have encopresis before botulinum toxin injection. These prel
iminary results suggest that botulinum toxin may represent a less inva
sive alternative to anal myectomy for children who have severe constip
ation after surgery for Hirschsprung's disease. If myectomy is contemp
lated, botulinum toxin may be useful as a means of predicting which ch
ildren may benefit.