INTESTINAL STRICTURES IN THE NEONATE* A COMPLICATION OF JSCHEMIC BOWEL DISEASE

Transcription

1 APRIL, 1975 INTESTINAL STRICTURES IN THE NEONATE* A COMPLICATION OF JSCHEMIC BOWEL DISEASE By HOWARD COCKRILL, M.D., MICHAEL OLIPHANT, M.D., and HERMAN GROSSMAN, M.D. DURHAM, NORTH CAROLINA O VER the past decade ischemic bowel disease (IBD) has become an incneasingly well recognized clinical entity in the adult patient However, until recently very little had been written about the subject in the neonate. Although there is a prior knowledge that vascular accidents can occur in the pediatric age group, all too often the possibility fails to be considered in acute problems of the gastrointestinal tract of the infant. Concomitantly, stricture formation is a well documented late complication of IBD in the adult, but the radiologic literature concerning this problem in the neonate is sparce. It is the purpose of this paper to discuss 4 patients with IBD due to varying etiologies, who later developed gastrointestinal strictures. Classically, the underlying factors responsible for IBD in the adult have been divided into occlusive and nonocclusive categories.27 Although the etiologic mechanisms may be quite different in the neonate, this classification of occlusive and nonocclusive ischemia is still applicable (Table i). OCCLUSIVE ISCHEMIC BOWEL DISEASE- MECHANICAL OCCLUSION Intussusception of the newborn is a relatively rare phenomenon,29 but it is capable of creating an ischemic bowel segment by a mechanical occlusion of the vessels. A 3 day old infant presented with bilious vomiting and blood per rectum. An iliocolonic intussusception was diagnosed by barium enema (Fig. IA) and then surgically reduced. Five months later, because of failure to thrive, recurrent vomiting, and abdominal distention, another barium enema study was performed (Fig. i B); it demonstrated a localized narrowing of the proximal left colon. Re-exploration proved that the narrowed segment was due to ischemic stricture. This narrowing had not been present at the original surgery. Apparently, the degree of ischemia to the bowel wall from the intussusception was not sufficient to have caused a full thickness infarction with subsequent perforation. However, restoration of the blood supply following reduction of the intussusception did allow healing of this segment, but with associated fibrosis and stricture. The stenotic area in the colon was resected, and an end-to-end anastomosis was performed. Trauma represents another etiologic mechanism in the pathogenesis of IBD. In some instances the precipitating event may be iatrogenic. A newborn male was found to have a high imperforate anus for which a pull through operation was performed on the first day of life. It is unusual to perform this type of surgery in a neonate; considerable manipulation of the distal colon, and its blood supply, is required. At 3 weeks of age the patient was referred to Duke Hospital with a partial intestinal obstruction. A contrast study of the colon (Fig. 2A) demonstrated a stricture in the sigmoid colon. A carefully selected dietary regimen permitted the child to grow, but at i year of age severe constipation led to a repeat barium enema examination. This study demonstrated a persistence of the narrowing in the sigmoid colon; this lesion was felt to represent an ischemic stricture secondary to inadvertent * Presented at the Seventy-fifth Annual Meeting of the American Roentgen Ray Society, San Francisco, California, September 24-27, From the Departments of Radiology and Pediatrics, Duke University Medical Center, Durham, North Carolina. 764

2 VOL. 123, No. Intestinal Strictures in the Neonate 765 TABLE I CAUSE5 OF ISCHEMIC BOWEL DISEASE IN THE NEONATE I. Occlusive A. Primarily arterial involvement I. Mechanical arterial occlusion a. intussusception b. trauma-postoperative c. mid-gut volvulus 2. Thromboembolic phenomena a. emboli originating in placental or umbilical veins9 b. postcardiac surgery mural thrombi7 c. umbilical artery catheterization #{176} Primarily venous involvement I. perforation following exchange transfusionl4,16u 2. dehydration7 2. sepsis7 4. postoperative-trauma5 8 II. Nonocclusive A. Necrotizing enterocolitis 4 25 interruption of the blood supply of the colon at the time of surgery. The patient is currently being given a trial of medical management. OCCLUSIVE ISCHEMIC BOWEL DISEASE- THROMBOEMBOLIC PHENOMENA Embolization from thrombi in the venous channels of the placenta or from thrombi in the umbilical vein has been cited as a cause of gastrointestinal ischemia.9 An embolus originating in either of these locations may travel through the ductus venosus and ductus arteniosus (or foramen ovale) to reach the splanchnic circulation. Such an embolus could either be spon- FIG. i. (A) A 3 day old infant presented with bilious vomiting and blood per rectum. Top arrow points to filling defect caused by an ileocolic intussusception. Bottom arrow marks the wall of the distal transverse colon. (B) Five months later, symptoms of a partial intestinal obstruction prompted another barium enema examination. A stricture of the proximal left colon was demonstrated. I

3 766 H. Cockrill, M. Oliphant and H. Grossman APRIL, 1975 per.orrned on t.. first day... weeks later the patient was referred v... symptoms of a large bowel obstruction. Barium enema examination revealed an ischemic stricture in the sigmoid colon. (B) A repeat barium enema study I year later shows persistence of the stenosis. taneous, or it could be iatrogenic, following umbilical vein catheterization. Occlusion of a mesentenic vessel by the embolus would lead to varying degrees of bowel colonic stenosis in this 2 a neonate. - in the umbilical cord caused venous congestion and thrombus formation in the umbilical vein. Emboli from this source may well have caused a mesenteric vascular accident in utero. ischemia. With only modenate ischemia and good collateral flow, total necrosis of the bowel wall might not occur, but subsequent healing of the wall by cicatnix would lead to stricture formation. A full term infant developed abdominal distention after birth, and on the second day of life a barium enema study revealed an area of narrowing of the splenic flexure (Fig. 3). Erskine9 has suggested that congenital intestinal stenoses, such as seen in this patient, may in fact be the result of thromboembolic vascular accidents in utero or in the immediate neonatal period. At the time of delivery, a complete knot was noted in the umbilical cord of this patient. Microscopic sections of the portion of the cord between the knot and the placenta revealed evidence of venous congestion, focal hemorrhage, and umbilical vein fibrin thrombi. Via the fetal circulatory pathways, these umbilical venous thrombi could have easily embolized to the mesentenic vessels. It has been documented in experimental animals that occlusion on partial occlusion of the mesentenic vasculatune of the fetus leads to bowel stenosis or atnesia when the animal is delivered a few weeks laten.l,15 It seems reasonable to

4 VOL. 123, No. Intestinal Strictures in the Neonate 767 postulate that the colonic stenosis in this neonate was the result of such an intrauterine vascular accident. NONOCCLUSIVE ISCHEMIC BOWEL DISEASE In neonates it is also possible for intestinal ischemia to develop even when the mesentenic vasculatune remains patent. This nonocclusive form of bowel ischemia revolves around the concept of a selective shunting of blood flow away from the splanchnic circulation toward the heart and central nervous system during periods of hypoxia and/or shock. This selective redistribution of blood flow has been linked to the diving reflex seen in aquatic mammals.14 Experimental studies have shown that during these periods of hypoxic stress there is a marked decrease in the perfusion of the stomach, ileum, and colon. 4 2 Necnotizing entenocolitis represents an example of nonocclusive IBD. This is to be differentiated from pneumatosis intestinalis (intramural bowel gas) due to umbilical artery catheters or to mechanical bowel obstruction.2#{176} The syndrome is characterized by abdominal distention, bilious vomiting and bloody stools; it is most often found in prematures.2#{176} The early roentgenograms demonstrate multiple distended bowel loops and intramural gas (pneumatosis intestinalis). Complications are portal venous gas and/or bowel perforation. No single mechanism satisfactorily explains all patients with this entity. Most recent investigatons agree that infection and hypoxia both play important roles in the pathogenesis of necrotizing enterocoli- 9,20,22,25 Regardless of the initiating event, in most cases of necrotizing enterocolitis there is an element of hypoxia which contributes to the pathologic lesion in the intestine. Thus, in the stressed newborn there is a selective shunting of blood away from the splanchnic bed, giving rise to the intestinal ischemia. 4 With progression of the process, the combination of tissue ischemia and bacterial proliferation results in perforation FIG. 4. This patient had necrotizing enterocolitis and was treated medically. Subsequently, he developed this area of stenosis in the transverse colon. and peritonitis. With less serious ischemia the bowel wall may be able to repair itself, but often with considerable fibrosis and resultant stenosis. A premature infant that was treated medically for necrotizing enterocolitis survived the acute phase, but 3 weeks later developed a long segment of colonic stenosis (Fig. 4). This complication of an ischemic stricture has also been reported by others ,17,21 As seen in Table i, there are also other causes of IBD. In the well known neonatal entity of mid gut volvulus, the twisting nature of the volvulus effectively decreases perfusion to the bowel. When this occurs in utero or in the peninatal period, the sequela may be stenosis or even atresia of the small bowel. The incidence of thrombus formation associated with the use of an umbilical arterial catheter is quite high.26 Mesenteric vascular accidents result from thromboembolic phenomena secondary to these umbilical arterial catheters. #{176} 2#{176} Perforation of the nonobstructed bowel occurs following exchange transfusions via the umbilical vein Clinical information

5 768 H. Cocknill, M. Oliphant and H. Grossman APRIL, 1975 and experimental data have implicated the umbilical venous catheter as the source of portal hemodynamic on embolic complications which have eventually resulted in an ischemic lesion.2 Colonic stenosis following repair of this ischemic injury has been reported. CONCLUSION With minor degrees of localized ischemia, the bowel may be able to fully recover and return to normal. On the other hand, severe ischemia may nesult in full thickness necrosis of the bowel wall. Thus, perforation may be an early complication of severe IBD. Between these two opposite ends of the spectrum there will be instances when the degree of ischemia is sufficient to impart considerable damage to the bowel wall, but not enough to cause perforation. Such a damaged bowel can heal by fibrosis with concomitant stricture formation. Thus, ischemic stenosis represents a late complication of IBD. The radiologist should be aware that a contrast enema examination should not be performed during the acute phase of ischemic entenitis for fear of perforating the already friable bowel wall. Following the acute episode, however, if there are problems with alimentation, a barium enema study and small bowel series should be done to rule out ischemic stricture formation. Likewise, when the radiologist demonstrates an idiopathic or congenital stricture in an infant, a careful review of the patient s peninatal history may well uncover a cause for the stenosis (Table i). Herman Grossman, M.D. Box 3834 Duke University Medical Center Durham, North Carolina REFERENCES i. BARNARD, C. N. Genesis of intestinal atresia. S. Forum, 1956, 7, BECK, J. M., DINNER, M., and CHAPPEL, J. Enterocoli tis following exchange transfusion. South African 7. Surg., 1971, 9, BELL, M. J., KosLosKE, A. M., BENTON, C., and MARTIN, L. W. Neonatal necrotizing enterocolitis: prevention of perforation. 7. Pediat. Surg., 1973, 8, 6oi-6o. 4. CORKERY, J. J., DUBOWITZ, V., LI5TER, J., and MoosA, A. Colonic perforation after exchange transfusion. Brit. M. 7., 1968, t, DAVIES, R. H. Mesenteric vein thrombosis in child who had undergone operative repair of imperforate anus and meningomyelocele. 7. Pediat. Surg., 1973, 8, DESA, D. J., MUCLOW, E. S., and GOUGH, M. H. Neonatal gut infarction. 7. Pediat. Surg., 1970, 5, DE MUTH, W. E. Mesenteric vascular occlusion in children. 7.A.M.A., 1962, 179, DUDGEON, D. L., CORAN, A. G., LAUPPE, F. A., HODGMAN, J. E., and ROSENKRANTZ, J. G. Surgical management of acute necrotizing enterocolitis. 7. Pediat. Surg., 1973, 8, ERSKINE, J. M. Colonic stenosis in newborn: possible thromboembolic etiology of intestinal stenosis and atresia. 7. Pediat. Surg., 3970, 5, KITTERMAN, J. A., PHIBB5, R. H., and TOOLEY, W. H. Catheterization of umbilical vessels in newborn infants. Pediat. Clin. North America, 1970, 17, II. KRASNA, I. H., BECKER, J. M., SCHNEIDER, K. M., and BECK, A. R. Colonic stenosis following necrotizing enterocolitis of newborn. 7. Pediat. Surg., 1970, 5, KRASNA, I. H., Fox, H. A., SCHNEIDER, K. M., and BECKER, J. M. Low molecular weight dextran in treatment of necrotizing enterocolitis and midgut volvulus in infants. 7. Pediat. Surg., 1973, 8, LLOYD, D. A., and CYWES, S. Intestinal stenosis and enterocyst formation as late complication of neonatal necrotizing enterocolitis. 7. Pediat. Surg., 1973, 8, LLOYD, J. R. Etiology of gastrointestinal perforations in newborn. 7. Pediat. Surg., 3969, 4, Louw, J. H., and BARNARD, C. N. Congenital intestinal atresia: observations on its origin. Lancet, 1955, 2, i6. ORME, R. L., and EADES, S. M. Perforation of bowel in newborn as complication of exchange transfusion. Brit. M. 7., 1968, 4, I. 17. RABINOWITZ, J. G., WOLF, B. S., FELLER, M. R., and KRA5NA, I. Colonic changes following necrotizing enterocolitis. in newborn. AM. J. ROENTGENOL., RAD. THERAPY & NUCLEAR MED.,,I 968, 103, i8. RATNER, I. A., and SWENSON, 0. Mesenteric vascular occlusion in infancy and childhood. New England 7. Med., 1960, 263, 1I22-I ROBACK, S. A., FOKER, J., FRANTZ, I. F., HUNT, C. E., ENGEL, R. R., and LEONARD, A. S.

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7 This article has been cited by: 1. Michael Born, Leif O. Holgersen, Farrokh Shahrivar, Edward Stanley-Brown, Carol Hilfer Routine contrast enemas for diagnosing and managing strictures following nonoperative treatment of necrotizing enterocolitis. Journal of Pediatric Surgery 20:4, [CrossRef] 2. Marshall Z. Schwartz, C. Joan Richardson, C. Keith Hayden, Leonard E. Swischuk, Kenneth R.T. Tyson Intestinal stenosis following successful medical management of necrotizing enterocolitis. Journal of Pediatric Surgery 15:6, [CrossRef]