Recurrent
Intussusception By Sigmund
in Children
H. Ein
T
HE ETIOLOGY of most intussusceptions is unknown. This makes the problem of recurrent intussusception even more puzzling. Very little has been written about this entity in children except for the recommendation that operation be performed after the first or second recurrence.‘-6 The purpose of this paper is to present, analyze, and discuss a series of recurrent intussusceptions in children. CLINICAL
MATERIAL
Six-hundred children with intussusception were treated at the Hospital for Sick Children, Toronto, over a 17-yr period, 1958 through 1974. From this group, 28 patients with 35 recurrences were collected. Twenty-three children had one recurrence each, four children had two, and one child had four recurrent intussusceptions. The overall recurrence rate was 5%. The incidence of recurrence after hydrostatic barium enema reduction was 11’~. but only 3% after operative reduction. There were no recurrences following surgical resection. After the recurrent intussusception, the risk of having a subsequent one increased. Of the 28 who suffered an initial recurrence, five demonstrated and of these five, one child (20%) went on to have a third and a fourth.
a second
recurrence
(17x),
The average age of children with recurrent intussusception was 22 mo. There were 21 boys and seven girls in the series. A viral illness preceded a recurrence in only two children. Twenty-four children (68%) had a second intussusception within 6 mo of their first, the earliest being within 12 hr and the latest 3 yr. All but one (97%) of these children had pain with the recurrent intussusception. In 21 instances (60%). there was vomiting, in 20 (57%) an abdominal mass was felt, and in ten (28%) rectal bleeding was noted. However, 13 children had fewer presenting signs and symptoms with their recurrent than with their initial intussusception. It took an average of 8 hr before the diagnosis was made as opposed to 22 hr with the first attack, and 15 of the 35 recurrent intussusceptions were diagnosed sooner than the initial intussusception. Barium enema reduction was attempted in 30 of the 35 recurrences (85%) with success in 21 of the 30 (70%). Barium enema was not attempted in five. Sixteen patients were operated on. Nine intussusceptions could be manually reduced, four were already reduced, and four resections were carried out (one child, whose intussusception was found reduced, had a bowel resection). Two leading points were found. There were 33 ileocolic, one ileoileal, and one cecocolic recurrent intussusceptions. No complications occurred from this group. DISCUSSION
In this series, 35 recurrent intussusceptions in 28 children have been analyzed. This is 59: of the total number of intussusceptions over a 17-yr period. The average age of the child in this series was 6 mo more than in the entire
From the Division of General Surgery, The Hospital for Sick Children, and Department of Surgery, Faculty of Medicine, University of Toronto, Toronto, Ontario, Canada. Presented before the 6th Annual Meeting of the American Pediatric Surgical Association, San Juan, Puerto Rico, April 10-12, 1975. Address for reprint requests: Sigmund H. Ein. M.D., F.R.C.S.(CI, F.A.C.S.. Division of General Surgery, The Hospital for Sick Children. Toronto, Ontario, Canada. 0 I975 by Grune & Stratton, Inc.
Journal of Pediatric Surgery, Vol. 10, No. 5 (October), 1975
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series. A viral illness was not more prevalent prior to the recurrent intussusception. The majority of the second intussusceptions occurred within 6 mo of the first one. No explanation has been found for this fact. When one discovers an intussusception to be present within a very few hours of hydrostatically reducing a previous one, the likelihood of reduction having been incomplete must be considered. The shortest interval between intussusceptions in this series was 12 hr. It took much less time on the average to make the diagnosis of the recurrent intussusception, and there were also fewer presenting signs and symptoms. However, pain was much more characteristic the second time around. Perhaps the onset of the typical colicky pain allerted the parents to the diagnosis much sooner. The recurrence rate after hydrostatic reduction in this series was 1 lx, similar to that reported by others. 2,4The success rate of hydrostatic reduction of the recurrent intussusceptions was higher (70%) than for the entire series (60%). A possible explanation for this improvement is the earlier presentation of the recurrent intussusception. The 3% recurrence rate after operative reduction in our series was also close to that reported by others.2*4 The previous mode of reduction (barium enema or operation) did not set a trend for future treatment of recurrent intussusceptions in the same child. For this reason, operative fixation of the ileocaecal area to the abdominal wall is not warranted. While most intussusceptions are of the ileocolic variety, this was not always the case with the recurrent intussusceptions in this series. The reason for this occasional variance is not’apparent. Although Perrin and Lindsay’s hypothesis of localized lymphoid accumulations in the terminal ileum of infants is not generally accepted, it still offers the best explanation for the high incidence of ileocolic intussusceptions.7 The incidence of leading points causing recurrent intussusceptions in children has been reported as high as 20% (about three to four times that seen in all cases of intussusception).‘*2*4 This was not the case in this series. The two leading points in this group of patients were an ileal suture line and a Meckel’s diverticulum. These were both found in children who had been operated on for previous intussusceptions requiring manual reduction, but were not caused by these leading points. These two specific recurrent intussusceptions occurred 6 mo and 3 yr, respectively, after the initial episode. In the past it has been assumed that in children who have recurrent intussusceptions, laparotomy is mandatory to rule out the possibility that there is a leading point.8 This assumption was not found to be valid, since none of 31 previously reviewed intussusceptions caused by specific pathologic lesions could be reduced by hydrostatic barium enema.g All required operation, but on the basis of failed hydrostatic reduction, not because they were recurrences. The adhesions created by the surgical manipulation of reduction of the intussusception and appendectomy may explain the reduced recurrence rate after surgical reduction of an intussusception. Indeed, ileocolic resection is claimed to virtually eliminate recurrent intussusception. Nevertheless, on the basis of this series, hydrostatic reduction, when successful, is felt to be superior treatment for recurrent intussusception.
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INTUSSUSCEPTION
SUMMARY
Twenty-eight children with 35 recurrent intussusceptions were collected and analyzed from a series of 600 intussusceptions over 17 yr. The overall recurrence rate in the entire series was 5”i, with 1 l”/, recurring after hydrostatic barium enema reduction, and 3% after operative reduction. Twenty-three children had one recurrence each, four children had two recurrences, and one child had four recurrent intussusceptions. More than two-thirds of the patients had a recurrence within 6 mo of their first intussusception, and half of these presented earlier and with fewer signs and symptoms than the previous attack had manifested. Twenty-one of 30 recurrences were reduced with barium enema. Sixteen were operated on, reducing nine manually and resecting four others. Only two leading points were found, and in both instances resection was required. All but two of the intussusceptions were ileocolic. The previous mode of reduction of each intussusception did not set a trend for future treatment of recurrent intussusceptions in the same child. While operative reduction diminishes the chances of a recurrent intussusception and ileocolic resection eliminates it, there does not seem to be any indication for surgery as long as barium enema reduction is successful. We have never observed the hydrostatic reduction of an intussusception caused by a leading point. REFERENCES I. Sarason EL, Prior JT, Prowda RL: Recurrent intussusception associated with hypertrophy of Peyer’s patches. N Engl J Med 253: 905, 1955 2. Herman BE, Becker J: Recurrent acute intussusception-A survey. Surg Clin N Am 40: 1009, 1960 3. Konigsberg K, Lee JC, Stein H: Recurrent acute intussusception. Pediatrics 53:269, 1974 4. Soper RT, Brown MJ: Recurrent acute intussusception in children. Arch Surg 89:188. 1964 5. Benson
DC.
Lloyd
JR, Fisher
H:
Intus-
susception in infants and children. Arch Surg 86~14, 1963 6. Swenson 0: Intussusception, in Swenson 0 (ed):
Pediatric
Surgery
Appleton-Century-Crofts, 7. Perrin WS, Lindsay
(ed 3). New
York,
1969, p 339 EC: Intussusception:
Monograph based on 400 cases. Br J Surg 9:46, lr‘l 8. Ravitch ***_ -’Ine MM: non-operattve treatment of intu Issusception-hydrostatic pressure reduction by hcari,am Am “YL.U,IB P~P..,Q “L.I,,BU. C..rn YUL6 f’lin _,,,1 ?.I 1. _,,I 361495-1500, 1956 9. Ein SH: Leading - noints in childhood in. tussusception.
J Pediatr
Surg (in press)
Discussion Dr. M. Ravitch (Pittsburgh): This is an extremely practical paper, because it does tell us what to do with children with recurrent intussusception. We have preached for a long time just exactly what we have just learned is wrong, namely that after a recurrence has been reduced by barium enema, the child should be studied in all the appropriate ways, and then operated on even when we haven’t found anything by barium enema, GI follow-through, etc.; even then we have never found a mechanical lesion, such as a Meckel’s or whatever, in such a child. I think that from now on we should be convinced that it is unnecessary to reoperate on those children at all. I point out a number of things presented in this good paper of Dr. Ein’s. Just as in our series, one quarter of the children operated on after presumed failure of barium enema reduction had in fact been completely reduced. This almost certainly means that they were operated on because no barium went into the small bowel. In our book, this requires operation. In fact, this failure to see barium in the small bowel was simply due to edema at the ileocecal valve with the intus-
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susception having been reduced. As far as recurrence rates are rate after barium enema reduction was 6%. Many of the papers rence rate after operative reduction. I haven’t been convinced ference in the recurrence rate of intussusception, whatever way say that if there is an advantage to operative reduction, in terms
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concerned, our own recurrence and literature report a 4% recurthat there is any significant difit is treated. One does have to of producing adhesions, which
might reduce the rate of recurrence, that would be offset by the inevitability of some cases of mechanical intestinal obstructions due to adhesions. Finally, a word of caution. I think there is a group of intussusceptions, unrelated to those discussed today, in which there are many recurrent intussusceptions and in which a tumor is always present. This is true in the Peutz-Jeghers syndrome, or in patients who have polyps, tibromas, or neurofibromas well up in the small bowel, causing a nonstrangulating intussusception that can reduce itself frequently. Many of these children have many attacks before they finally come to surgery. These are patients who would not be helped by a barium enema, since the intussusception is well up in the small bowel, and the barium enema would show the cecum filled. If you found no intussusception you would operate for a small bowel obstruction. Intussusceptions of that kind will, in fact, usually have a lead point. As far as I’m concerned, I think this has settled an important question with respect to intussusception in children. I’m grateful to Dr. Ein’s group for this. Dr. C. Minor (Wilmington): I would like to ask the author if there is ever an indication for resection of terminal ileum, cecum, and some of the ascending colon? Dr. W. Sieber (Pirrsburgh): As you know, we have been strong advocates of hydrostatic reduction as the primary treatment for simple ileocolic intussusception, and we strongly support what has been said here this morning. However, I do believe there is a place for surgical reduction of recurrent intussusception, and I point to the patients that have had not one and two recurrences, but four and five recurrences, successfully reduced over a period of 4-5 yr. Concerning Dr. Minor’s question, I believe there is a very definite indication for resection of the ileocolic area in these patients. These children inevitably have large areas of hypertrophied Peyer’s patches right on the ileocecal valve that are, in fact, the lead point, and until that is removed, there will always be recurrences. The other thing I would like to mention is that occasionally one sees a patient with agioneurotic edema that will be the source of an intussusception. I believe this must be exceedingly rare, but we have seen one child in a family with congenital angioneurotic edema with six separate intussusceptions of the transverse colon. This child was only relieved of his trouble when the entire transverse colon was resected. Dr. R. Soper (Iowa City): Actually, Dr. Sieber has said essentially what I wanted to say. About IO yr ago we reported a teenager who had had I2 documented recurrences, six hydrostatically reduced, and six confirmed and reduced at operation. In addition to that, the parents had, by saline enemata, probably reduced 20 or 30 others. When we explored this girl, we reduced the intussusception, which was ileocolic. We could not palpate anything in the ileum. We did a blind resection and were very surprised, on opening the specimen, to find a 50-cent-piece sized ectopic gastric mucosa. This then was the lead point that had been reduced hydrostatically and surgically many times. This was at a place where a Meckel’s diverticulum had, at one of the previous operations, been V-resected, without a segmental resection. I think there is an exception to the very good general principles espoused in the paper presented this morning. Dr. C. Baxter (Spokane): I disagree with the author and Dr. Ravitch because of three patients with recurrent intussusception that I have observed. Two of them had lymphosarcoma of the distal ileum, and it was with the first one of these that I resolved in my own mind that I would operate on the first recurrence even if I could reduce it successfully with hydrostatic reduction. Dr. D. Collins (San Diego): I would like to ask Dr. Ein if he has had any cases in which the first intussusception, the usual ileocolic type, required a manual reduction, and in which the second one, an ileoileal, occurred at a different place, without any special lead point, which might be construed as a postoperative intussusception? 1 have had two such cases. Dr. S. Ein (dosing): Dr. Ravitch, I have no comments, other than to thank you for your remarks. In answer to Dr. Minor’s question, about whether or not there is any indication for ileocolic resection, it’s obviously a fielder’s choice. If it were my child and I was living in Toronto with the facilities we have there and if it wasn’t too upsetting to take the child down the street four or five times, I would have this child have his intussusception reduced easily with barium, sent home. and not have him readmitted for an ileocolic resection. If it’s 12 times, in Dr. Soper’s
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area, maybe I would condescend to something like that. It becomes a fielder’s choice. I’m not trying to set up any rigid rules. I think you have to present the odds to the parents. Dr. Sieber. you pointed out that there is an indication for resection. We have not had in our series a child with angioneurotic edema. There is no question that an intussusception can be and has been idiopathic in nature with a potential lead point nearby, not involved in the intussusception. In our series in which we found 31 pathologic lead points, in the same span of time, there were five children with Meckel’s diverticula that were incidental findings. To answer Dr. Baxter, we had one lymphosarcoma. It is quite possible that the first intussusception was not caused by the lymphosarcoma and the second one was. 1 really don’t believe that you’re going to reduce an intussusception caused by a lymphosarcoma. In answer to Dr. Collins, of the recurrences there were two that occurred in a child after operative reduction. One was reduced with a barium enema. There were six recurrences after a barium enema was unsuccessful. Of those six, three were reduced with barium enemas. There were 27 after barium enema reduction. and of those 27. I7 were further reduced with barium.