- Email: [email protected]
Gastro colic fistula in a neonate – Case report of a rare complication of necrotizing enterocolitis
J Ped Surg Case Reports 3 (2015) 10e12
Contents lists available at ScienceDirect
Journal of Pediatric Surgery CASE REPORTS journal homepage: www.jpscasereports.com
Gastro colic fistula in a neonate e Case report of a rare complication of necrotizing enterocolitisq Shilpa Kalane*, Pradeep Suryawanshi, Umesh Vaidya, Shashank Shrotriya Division of Neonatology, Department of Pediatrics, Sahyadri Speciality Hospital, Nagar Road, Pune, Maharashtra, India
a r t i c l e i n f o
a b s t r a c t
Article history: Received 19 September 2014 Received in revised form 2 November 2014 Accepted 4 November 2014
Gastrocolic Fistula is, in the majority of cases the pathological communication between stomach and transverse colon. It occurs mostly in adults, but they can be present in infants, as well, as a result of congenital abnormalities or iatrogenic procedures (i.e. migration of naso gastric tube that placed before). We report a case of 10 days old full term, male neonate, was on naso gastric tube feeds, had clinical features of necrotizing enterocolitis (NEC), diagnosed to have gastrocolic fistula on barium enema study and confirmed on CT abdomen. Intraoperatively, baby had multiple perforations with gastrocolic fistula. Histopathological examination was suggestive of NEC. We searched literature, we could find seven case reports. Ó 2015 The Authors. Published by Elsevier Inc. All rights reserved.
Key words: Gastro colic fistula NEC Neonate
1. Case report A male neonate, born by full term emergency caesarian section to a primigravida mother for meconium stained amniotic fluid, was limp at birth, had meconium aspiration syndrome and hence was referred to us for further management. Baby was small for gestational age with weight was 1980 g, Head circumference 34 cm, and total length at birth was 49 cm. Baby was ventilated for 3 days for moderate meconium aspiration syndrome. Trophic feeds via naso gastric tube were initiated on day 1 of life, baby reached full feeds (180 ml/kg/day, expressed breast milk) on day 6 of life. Baby was lethargic and hypotonic on day 5 of life. CRP was positive, hemogram, CSF and electrolytes were normal. In view of sepsis, antibiotics were started. On day 8 of life baby had increasing yellowish greenish aspirates suggestive of feeding intolerance. X ray abdomen was done was suggestive of dilated bowel loops, Necrotizing enterocolitis was suspected. Baby was kept NPO. In view of progressively increasing aspirates, Barium enema study was done on day 10 of life showed gastro colic fistula (Fig. 1), was confirmed on CT scan abdomen (Fig. 2). On day 11, exploratory laparatomy was
q This is an open access article under the CC BY-NC-ND license (http:// creativecommons.org/licenses/by-nc-nd/3.0/). * Corresponding author. Flat no. 202, G Building, Wondercity, Katraz-S.N.76, Pune-46, Maharashtra, India. Tel.: þ91 9552024242 (mobile). E-mail address: [email protected] (S. Kalane).
done. Baby had 5 intestinal perforations (Fig. 3) e intestinal resection and diversion was done. Histopathological examination of intestinal biopsy specimen showed mucosal edema, hemorrhage, necrosis suggestive of NEC. Baby was kept NPO for 5 days postoperative. On postoperative day 6, feeds were started. On day 21 of life, baby was on exclusive breast feeding, discharged on day 24 of life with discharge weight 1990 g. 2. Discussion In children, gastro colic fistulae are very rare, especially in newborns and infants. Since 1945, only 8 cases have been reported in the literature [1]. Gastro colic fistulae result from perforation of the stomach into the colon or of the opposite, mostly among premature and immature small-for-dates infants [2]. This occurs because of stress ulcer, gastric tissue ischemia or trauma, due to complications of necrotizing enterocolitis, Hirschsprung’s disease and meconium ileus, respectively. Also, gastro colic fistulae in children can occur after migration or placement of PEG feeding tubes [2]. Strictures complicating NEC occur in 20% of patients and usually involve colon [3]. In contrast fistula formation complicating NEC is rare and may occur with or without stricture. Clinical data on our one patient and the 8 previously described in literature are summarized in Table 1. The plain film findings in the presence of fistula are nonspecific. Clinically our patient presented with increasing aspirates without
2213-5766/$ e see front matter Ó 2015 The Authors. Published by Elsevier Inc. All rights reserved. http://dx.doi.org/10.1016/j.epsc.2014.11.002
S. Kalane et al. / J Ped Surg Case Reports 3 (2015) 10e12
11
Fig. 1. Supine abdominal X ray with Barium enema study. (aed) X rays show dye slowly advancing from rectum, sigmoid (a), descending colon to stomach suggestive of gastro colic fistula (b) then to the rest of the intestine (c and d).
Fig. 2. CT scan abdomen suggestive of gastro colic fistula.
abdominal distention and previously reported cases presented with diarrhea and abdominal distention. Clinical symptoms suggestive of feeding intolerance and nonspecific bowel gas pattern on plain x ray should raise the possibility of an existing fistula. Several mechanisms may be proposed for the development of enterocolic fistula following NEC. Severe colonic ischemia and subsequent bowel necrosis may develop over time inciting an inflammatory response. Continuous inflammation may result in adherence of affected segment of colon to adjacent bowel and eventual fistulization. Alternatively, a sub acute perforation may be walled off by adjacent viscera resulting in fistula formation. In cases mentioned in literature, time gap between diagnosis of NEC and fistula formation was wide around 2 weekse10 weeks. However in our case it was early. In this case baby was IUGR, had suffered asphyxia and had MAS. This may have aggravated intestinal ischemia, leading to early postnatal fistula formation. Alternatively as mentioned earlier sub acute perforation may be walled off by adjacent viscera resulting in fistula formation. As mentioned in most of the reported cases, present case also did well postoperatively. Gastro colic fistulae, a condition with poor prognosis, can arise from a variety of pathological processes, spontaneous or iatrogenic, and the classical symptoms are increasing aspirates, abdominal distention, vomiting and diarrhea. The best diagnostic method is
12
S. Kalane et al. / J Ped Surg Case Reports 3 (2015) 10e12
Fig. 3. Small intestinal perforations (a-e) with gastro colic fistula. Table 1 Cases of enteric fistulas due to necrotizing enterocolitis. Author
Diagnosis of NEC (age) Diagnosis of fistula (age) Birth weight (g) Fistula location
Colonic stricture Clinical presentation
Follow up
Pein [4] Firor [5] Beck [6] Kosloske [7] Paley [8] Kiely [9] Levin [1] Levin [1] Case
7 days ? 5 days ? 7 days 5 days 6 days 22 days 8 days
No No Yes No Yes Yes No No No
Died post surgery Died post surgery Surgery, did well Surgery, did well Surgery, did well Surgery, did well Died pre surgery Surgery, did well Surgery, did well
35 28 18 18 63 32 25 43 10
days days weeks weeks days days days days days
? ? 2610 ? 3800 3600 820 1190 1980
the barium enema, while other radiological methods play a significant role to other parameters of the fistulae. The therapy of this condition remains surgical. After all, we must emphasize that it is a serious pathologic condition that can lead to death. Contributions Kalane S: search of the literature, partial English editing, and correction, Suryawanshi P: editing, Vaidya U: Final editing and correction, Shrotriya S: editorship of the manuscript. Conflicts of interests None. Sources of funding None.
Gastro colic Gastro jejunocolic Ileocolic Enterocolic Jejunocolic Jejunoileocolic Jejunocolic Jejunoileocolic Gastro colic
Vomiting, diarrhea Vomiting, diarrhea Distention, constipation ? Distention, vomiting Vomiting, diarrhea Distention Distention Increasing bilious aspirates
References [1] Levin TL, Brill PW, Winchester P. Enteric fistula formation secondary to necrotizing enterocolitis. Pediatr Radiol 1991;21:309e11. [2] Hager J, Gassner I. Gastrocolic fistula in a 7 week old: a rare complication after gastric perforation. J Pediatr Surg 1994;29:1597. [3] Caffey’s pediatric x-ray diagnosis. 8th ed. Chicago: Year Book Medical Publishers; 1985. p. 1860. [4] Pein NK, Witswatersrand MB. Neonatal gastrocolic fistula: report of a case. Lancet 1948;II:53. [5] Firor HV. Gastrojejunocolic fistula in an infant: a previously unrecorded etiology and reflections on management. J Pediatr Surg 1970;5:450. [6] Beck JM, Dimner M, Chappel J. Enterocolitis following exchange transfusion. S Afr J Surg 1971;9:39. [7] Kosloske AM, Martin LW. Surgical complications of necrotizing enterocolitis. Arch Surg 1973;107:223. [8] Paley RH, McCarten KM, Clevand RH. Enterocolonic fistula as a late complication of necrotizing enterocolitis. AJR Am J Roentgenol 1979;132:989. [9] Kiely E, Eckstein HB. Colonic stricture and enterocolonic fistulae following necrotizing enterocolitis. Br J Surg 1984;71:613.
Contents lists available at ScienceDirect
Journal of Pediatric Surgery CASE REPORTS journal homepage: www.jpscasereports.com
Gastro colic fistula in a neonate e Case report of a rare complication of necrotizing enterocolitisq Shilpa Kalane*, Pradeep Suryawanshi, Umesh Vaidya, Shashank Shrotriya Division of Neonatology, Department of Pediatrics, Sahyadri Speciality Hospital, Nagar Road, Pune, Maharashtra, India
a r t i c l e i n f o
a b s t r a c t
Article history: Received 19 September 2014 Received in revised form 2 November 2014 Accepted 4 November 2014
Gastrocolic Fistula is, in the majority of cases the pathological communication between stomach and transverse colon. It occurs mostly in adults, but they can be present in infants, as well, as a result of congenital abnormalities or iatrogenic procedures (i.e. migration of naso gastric tube that placed before). We report a case of 10 days old full term, male neonate, was on naso gastric tube feeds, had clinical features of necrotizing enterocolitis (NEC), diagnosed to have gastrocolic fistula on barium enema study and confirmed on CT abdomen. Intraoperatively, baby had multiple perforations with gastrocolic fistula. Histopathological examination was suggestive of NEC. We searched literature, we could find seven case reports. Ó 2015 The Authors. Published by Elsevier Inc. All rights reserved.
Key words: Gastro colic fistula NEC Neonate
1. Case report A male neonate, born by full term emergency caesarian section to a primigravida mother for meconium stained amniotic fluid, was limp at birth, had meconium aspiration syndrome and hence was referred to us for further management. Baby was small for gestational age with weight was 1980 g, Head circumference 34 cm, and total length at birth was 49 cm. Baby was ventilated for 3 days for moderate meconium aspiration syndrome. Trophic feeds via naso gastric tube were initiated on day 1 of life, baby reached full feeds (180 ml/kg/day, expressed breast milk) on day 6 of life. Baby was lethargic and hypotonic on day 5 of life. CRP was positive, hemogram, CSF and electrolytes were normal. In view of sepsis, antibiotics were started. On day 8 of life baby had increasing yellowish greenish aspirates suggestive of feeding intolerance. X ray abdomen was done was suggestive of dilated bowel loops, Necrotizing enterocolitis was suspected. Baby was kept NPO. In view of progressively increasing aspirates, Barium enema study was done on day 10 of life showed gastro colic fistula (Fig. 1), was confirmed on CT scan abdomen (Fig. 2). On day 11, exploratory laparatomy was
q This is an open access article under the CC BY-NC-ND license (http:// creativecommons.org/licenses/by-nc-nd/3.0/). * Corresponding author. Flat no. 202, G Building, Wondercity, Katraz-S.N.76, Pune-46, Maharashtra, India. Tel.: þ91 9552024242 (mobile). E-mail address: [email protected] (S. Kalane).
done. Baby had 5 intestinal perforations (Fig. 3) e intestinal resection and diversion was done. Histopathological examination of intestinal biopsy specimen showed mucosal edema, hemorrhage, necrosis suggestive of NEC. Baby was kept NPO for 5 days postoperative. On postoperative day 6, feeds were started. On day 21 of life, baby was on exclusive breast feeding, discharged on day 24 of life with discharge weight 1990 g. 2. Discussion In children, gastro colic fistulae are very rare, especially in newborns and infants. Since 1945, only 8 cases have been reported in the literature [1]. Gastro colic fistulae result from perforation of the stomach into the colon or of the opposite, mostly among premature and immature small-for-dates infants [2]. This occurs because of stress ulcer, gastric tissue ischemia or trauma, due to complications of necrotizing enterocolitis, Hirschsprung’s disease and meconium ileus, respectively. Also, gastro colic fistulae in children can occur after migration or placement of PEG feeding tubes [2]. Strictures complicating NEC occur in 20% of patients and usually involve colon [3]. In contrast fistula formation complicating NEC is rare and may occur with or without stricture. Clinical data on our one patient and the 8 previously described in literature are summarized in Table 1. The plain film findings in the presence of fistula are nonspecific. Clinically our patient presented with increasing aspirates without
2213-5766/$ e see front matter Ó 2015 The Authors. Published by Elsevier Inc. All rights reserved. http://dx.doi.org/10.1016/j.epsc.2014.11.002
S. Kalane et al. / J Ped Surg Case Reports 3 (2015) 10e12
11
Fig. 1. Supine abdominal X ray with Barium enema study. (aed) X rays show dye slowly advancing from rectum, sigmoid (a), descending colon to stomach suggestive of gastro colic fistula (b) then to the rest of the intestine (c and d).
Fig. 2. CT scan abdomen suggestive of gastro colic fistula.
abdominal distention and previously reported cases presented with diarrhea and abdominal distention. Clinical symptoms suggestive of feeding intolerance and nonspecific bowel gas pattern on plain x ray should raise the possibility of an existing fistula. Several mechanisms may be proposed for the development of enterocolic fistula following NEC. Severe colonic ischemia and subsequent bowel necrosis may develop over time inciting an inflammatory response. Continuous inflammation may result in adherence of affected segment of colon to adjacent bowel and eventual fistulization. Alternatively, a sub acute perforation may be walled off by adjacent viscera resulting in fistula formation. In cases mentioned in literature, time gap between diagnosis of NEC and fistula formation was wide around 2 weekse10 weeks. However in our case it was early. In this case baby was IUGR, had suffered asphyxia and had MAS. This may have aggravated intestinal ischemia, leading to early postnatal fistula formation. Alternatively as mentioned earlier sub acute perforation may be walled off by adjacent viscera resulting in fistula formation. As mentioned in most of the reported cases, present case also did well postoperatively. Gastro colic fistulae, a condition with poor prognosis, can arise from a variety of pathological processes, spontaneous or iatrogenic, and the classical symptoms are increasing aspirates, abdominal distention, vomiting and diarrhea. The best diagnostic method is
12
S. Kalane et al. / J Ped Surg Case Reports 3 (2015) 10e12
Fig. 3. Small intestinal perforations (a-e) with gastro colic fistula. Table 1 Cases of enteric fistulas due to necrotizing enterocolitis. Author
Diagnosis of NEC (age) Diagnosis of fistula (age) Birth weight (g) Fistula location
Colonic stricture Clinical presentation
Follow up
Pein [4] Firor [5] Beck [6] Kosloske [7] Paley [8] Kiely [9] Levin [1] Levin [1] Case
7 days ? 5 days ? 7 days 5 days 6 days 22 days 8 days
No No Yes No Yes Yes No No No
Died post surgery Died post surgery Surgery, did well Surgery, did well Surgery, did well Surgery, did well Died pre surgery Surgery, did well Surgery, did well
35 28 18 18 63 32 25 43 10
days days weeks weeks days days days days days
? ? 2610 ? 3800 3600 820 1190 1980
the barium enema, while other radiological methods play a significant role to other parameters of the fistulae. The therapy of this condition remains surgical. After all, we must emphasize that it is a serious pathologic condition that can lead to death. Contributions Kalane S: search of the literature, partial English editing, and correction, Suryawanshi P: editing, Vaidya U: Final editing and correction, Shrotriya S: editorship of the manuscript. Conflicts of interests None. Sources of funding None.
Gastro colic Gastro jejunocolic Ileocolic Enterocolic Jejunocolic Jejunoileocolic Jejunocolic Jejunoileocolic Gastro colic
Vomiting, diarrhea Vomiting, diarrhea Distention, constipation ? Distention, vomiting Vomiting, diarrhea Distention Distention Increasing bilious aspirates
References [1] Levin TL, Brill PW, Winchester P. Enteric fistula formation secondary to necrotizing enterocolitis. Pediatr Radiol 1991;21:309e11. [2] Hager J, Gassner I. Gastrocolic fistula in a 7 week old: a rare complication after gastric perforation. J Pediatr Surg 1994;29:1597. [3] Caffey’s pediatric x-ray diagnosis. 8th ed. Chicago: Year Book Medical Publishers; 1985. p. 1860. [4] Pein NK, Witswatersrand MB. Neonatal gastrocolic fistula: report of a case. Lancet 1948;II:53. [5] Firor HV. Gastrojejunocolic fistula in an infant: a previously unrecorded etiology and reflections on management. J Pediatr Surg 1970;5:450. [6] Beck JM, Dimner M, Chappel J. Enterocolitis following exchange transfusion. S Afr J Surg 1971;9:39. [7] Kosloske AM, Martin LW. Surgical complications of necrotizing enterocolitis. Arch Surg 1973;107:223. [8] Paley RH, McCarten KM, Clevand RH. Enterocolonic fistula as a late complication of necrotizing enterocolitis. AJR Am J Roentgenol 1979;132:989. [9] Kiely E, Eckstein HB. Colonic stricture and enterocolonic fistulae following necrotizing enterocolitis. Br J Surg 1984;71:613.