Up-to-Date Evolution of Small Bowel Transplantation in Children
With Intestinal Failure
By Dominique Jan, Jean-Luc Michel, Olivier Goulet, Sabine Sarnacki, Florence Lacaitle, Diane Damotte,
Jean-Pierre Cezard, Yves Aigrain, Nicole Brousse, Michel Peuchmaur, Annick Rengeval, Virginie Colomb,
Philippe Jouvet, Claude Ricour, and Yann R~villon
Paris, France
Purpose: The aim of the authors was to report an up-to-date
review of their experience with 26 intestinal transplantations
in children since 1987.
Methods: A retrospective study was conducted of 26 patients
with a mean age of 5 years (range, 0.3 to 14 years). Three
groups were isolated. In group A (1987 to 1990), seven
patients received nine isolated intestinal transplants for short
bowel syndrome. Immunosuppression therapy consisted of
cyclosporine, aziathioprine, and corticosteroids. In group B
(1994-current), nine patients received nine isolated intestinal
transplants for short bowel syndrom (n = 2), intestinal pseu-
doobstruction (n = 2), neonatal intractable diarrhea (n = 3),
and Hirschsprung' disease (n = 1); hepatic biopsy results
showed weak cholestasis or fibrosis. In group C (1994-
current), 10 patients received 10 combined liver-small bowel
transplants for short bowel syndrome (n = 3), neonatal intrac-
table diarrhea (n = 4), and Hirschsprung' disease (n = 3);
hepatic cirrhosis related to total parenteral nutrition (TPN)
was shown in all cases. Groups B and C received immunosu-
pressive treatment consisting of tacrolimus, aziathioprine,
and corticosteroids. Posttransplant follow-up included intes-
tinal biopsies of the small bowel twice a week and more
frequently or combined with liver biopsy if rejection was
suspected.
Results: Overall patient survival (PS) and graft survival (GS)
are 61% (16 of 26) and 50% (13 of 26), respectively. In group A,
severe intestinal allograft rejection occurred in six patients
leading to graft removal (GS, 11%). Five patients died of TPN
complications after graft removal (PS, 28%). One survivor is
off TPN, and one currently is waiting for a second graft. In
group B, six patients survived (PS, 66%). Causes of death
include hepatic failure (n = 1), renal and liver failure (n = 1),
and systemic infection (n = 1). Severe intestinal allograft
rejection occurred in five patients, which neccessitated aggres-
sive immunosuppression (antilymphocyte serum) leading to
an incomplete functional recovery of the graft. Only two
patients currently are off TPN. In group C, eight patients
survived (PS, 80%) all of which are currently off TPN. One
patient died during the procedure, and one died of severe
systemic infection. Intestinal graft rejection occurred in six
patients; rejection of the liver allograft occurred in five
patients, yet all rejections were weak and successfully treated
by corticosteroids (GS, 80%).
Conclusions: Intestinal transplantation is a valid therapeutic
option for children with definitive intestinal failure and not
only for short bowel syndrome. Tacrolimus improves graft
and patient survival (group A v group B). The lower severity
of graft rejection in combined liver-small bowel transplanta-
tion improves functional results of intestinal transplantation
in children without additional mortality or morbidity (group B
vgroup C).
J Pediatr Surg 34:841-844. Copyright © 1999 by W.B. Saun-
ders Company.
INDEX WORDS: Intestinal failure, short gut, intestinal trans-
plantation, liver transplantation.
T
HE LONG-TERM OUTCOME for children who
receive parenteral nutrition is not as favorable as it
is in adults. Consequently, some patients may benefit
from small bowel transplantation. Children dependent on
total parenteral nutrition (TPN), more so than adults, are
exposed to hepatic complications. Intestinal transplanta-
tion was performed in our institution in 1987 using
cyclosporine, t With the introduction of tacrolimus we
have abruptly changed the clinical application of small
bowel transplantation. 2 This is an up-to-date review of
our experience with 28 intestinal transplantations in 26
children since 1987.
MATERIALS AND METHODS
Materials
Between January 1987 and June 1998, 26 children received 28
intestinal transplants: nine isolated small bowel (SB) using cyclosporine
from 1987 to 1991 (group A). Since 1994, nine isolated small bowel
(group B) and 10 combined liver-small bowel (LSB) transplants (group
C) using tacrolimus were performed. The liver was included as part of
the allograft when parenteral nutrition-induced end-stage liver failure
was evident. The recipients consisted of 15 boys and 11 girls, age
ranging between 0.5 to 12 years old. All patients were dependant on
definitive total parenteral nutrition. Intestinal failure was caused by
short gut syndrome (n = 14), intractable diarrhea of infancy (n = 7),
From Hopital Necker-Enfants Malades, Paris, France.
Presented at the 1998 Annual Meeting of the Section on Surgery of
the American Academy of Pediatrics, San Francisco, California,
October 16-19, 1998.
Address reprint requests to Dominique Jan, MD, Professor of
Pediatric Surgery, Hopital Necker-Enfants Malades, 149 rue de S#vres,
75015 Paris, France.
Copyright © 1999 by W.B. Saunders Company
0022-3468/99/3405-0035503.00/0
Journal of Pediatric Surgery, Vo134, No 5 (May), 1999: pp 841-844 841