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Neonatal sigmoid volvulus: A complication of anal stenosis
Neonatal Sigmoid Volvulus: A Complication of Anal Stenosis By D. De Caluwe´, J. Kelleher, and M.T. Corbally Dublin, Ireland
Sigmoid volvulus is an exceptionally rare cause of intestinal obstruction in neonates. Only 7 cases have been reported in the English and French literature. The authors report a recent case of sigmoid volvulus in a neonate secondary to anal stenosis and review the diagnosis and management of this serious condition. The authors believe that carefully per-
formed radiologic reduction is the preferable alternative to surgical intervention. J Pediatr Surg 36:1079-1081. Copyright © 2001 by W.B. Saunders Company. INDEX WORDS: Sigmoid volvulus, anal stenosis, neonatal.
S
IGMOID VOLVULUS is a diagnosis rarely made in the neonatal population. Most articles are limited to single case reports.1-4 The first report of a sigmoid volvulus in a 14-day-old boy was published by Carter and Hinshaw in 1961.5 Previously, management of neonatal sigmoid volvulus has always been surgical. Hydrostatic reduction by barium enema was first described in 1980 in a 5-week-old infant.1 We report the first case of volvulus of the sigmoid colon in a 2-day-old boy secondary to anal stenosis treated solely by omnipaque contrast enema. CASE REPORT A 2-day-old full-term boy presented with poor feeding, nonbilious vomiting, and increasing abdominal distension. Meconium had been passed, and physical examination showed a well-hydrated baby with obvious abdominal distension, audible bowel sounds, but no tenderness or palpable masses. The buttocks were well formed, but the anus was tight not admitting a small finger. Plain abdominal radiographs showed largely distended intestinal loops but no free air (Fig 1). The baby was started on intravenous fluids and antibiotics, and a nasogastric tube was left on free drainage. A contrast enema with omnipaque showed a sigmoid volvulus that was reduced successfully, and a rectal tube was left in place for 24 hours (Fig 2). Rectal washouts were then started. Suction rectal biopsy results were normal, and anal dilatation was performed under general anaesthesia. Subsequently, the baby ate well and tolerated twice daily dilatations. At the age of 10 months, he is thriving well and has regular bowel motions with twice-daily dilatations.
DISCUSSION Fig 1.
The plain film suggests presence of a sigmoid volvulus.
Sigmoid volvulus is an extremely rare cause of large bowel obstruction in the neonate. Only 7 cases have been reported in the English and French literature (Table 1).1-4,6 There is a strong male predominance.4 The predisposing factor in all age groups appears to be a redundant sigmoid colon associated with an absent, shortened, or abnormally attached mesenteric root.1-4,6,7 The redundant sigmoid may twist around its narrow point of
From Our Lady’s Hospital for Sick Children, Dublin, Ireland. Address reprint requests to M.T. Corbally, Our Lady’s Hospital for Sick Children, Crumlin, Dublin 12, Ireland. Copyright © 2001 by W.B. Saunders Company 0022-3468/01/3607-0027$35.00/0 doi:10.1053/jpsu.2001.24759
Journal of Pediatric Surgery, Vol 36, No 7 (July), 2001: pp 1079-1081
1079
1080
DE CALUWE´, KELLEHER, AND CORBALLY
Fig 2. (A) Contrast injected into the rectum only passed proximally by rotating the patient lying on the left side to a supine position and from there to a slightly right side down position. At this stage, contrast passed across the area of volvulus into the very dilated sigmoid and subsequently into undilated proximal colon. (B) The contrast is shown throughout the colon with residual abnormal distension of the sigmoid. The proximal small bowel is normal.
attachment with luminal obstruction as a result. Occlusion of the mesenteric vessels with ischemia of the sigmoid loop occurs and results in infarction and gangrene. Bacterial translocation appears and results in peritonitis.5 In all age groups, it is associated with constipation.6,7 Hirschsprung’s disease always should be considered and excluded in neonates presenting with a sigmoid volvulus because 3 of the 7 neonates were found to have Hirschsprung’s disease (Table 1). Failure to pass meconium or to have spontaneous bowel motions, bilious or nonbilious vomiting, and increasing abdominal distension are the most common presenting symptoms.6,8-10 In 1 reported case, the baby had an imperforate anus.6 In our case, the baby was noticed to have anal stenosis. Plain x-rays, although occasionally dramatic, are not specific for sigmoid volvulus but should prompt further evaluation. The neonatal age group does not tend to exhibit the early findings of a “coffee bean” abdominal gas pattern, which shows the dilated sigmoid loop rising from the pelvis into the upper part of the abdomen and
suggests a closed loop obstruction.1,2,4,11 Barium enema usually confirms the diagnosis.2,7 All but one of the reported neonatal patients in the literature underwent laparotomy and detorsion or resection with or without colostomy of the twisted sigmoid.2 A contrast enema can derotate the volvulus and relief the obstruction successfully.2,4 Recurrence after derotation by contrast enema is rare.9 Nadalo et al1 reported the successful hydrostatic reduction of a sigmoid volvulus in a 5-week-old infant. Diagnosis and reduction of a sigmoid volvulus by means of a contrast enema in a 1-day-old baby with Hirschsprung’s disease has been reported by Venugopal et al.2 We have reported here on a 2-day-old neonate with sigmoid volvulus secondary to anal stenosis successfully corrected by an omnipaque enema. We believe this procedure is the preferable alternative to surgery in the neonate presenting with a sigmoid volvulus. Surgical intervention only should be necessary in case of unsuccessful derotation by contrast enema or recurrent volvulus.
NEONATAL SIGMOID VOLVULUS
1081
Table 1. Reported Cases of Neonatal Sigmoid Volvulus Study
Age/Sex
Plain Film Findings
Diagnosis
Carter and Hinshaw,5 1961
14 d/M
Abdominal distension, Bile-stained vomiting
Severe colonic distension
Ba enema, Exploratory laparatomy
None, Spontaneous detorsion
Srouji et al,10 1974
2 d/M
Moderate distension, Airfluid levels, No air in rectum
Ba enema, Exploratory laparatomy
Valla et al,4 1982
5 d/M
Abdominal distension, Bile-stained vomiting, No meconium passage Acute distal bowel obstruction Acute distal-bowel obstruction Normal gas pattern, No meconium passage, Imperforate anus Abdominal distension, Vomiting, No meconium passage Abdominal distension, Non-bilious vomiting
Air-fluid levels
Laparatomy
Sigmoid resection, Colostomy, Closure, Colostomy at 3 months Sigmoid resection
Air-fluid levels
Ba enema
Laparatomy, Detorsion
Increasing distension, No air in rectum, Pneumo peritoneum Dilated small and large bowel
Exploratory laparatomy
Loop colostomy
Exploratory laparatomy
Dilated bowel loops
Ba enema
Detorsion, Sigmoid colostomy at 14 days Detorsion
Dilated bowel loops
Omnipaque enema
Detorsion
6 d/M Janik et al,6 1983
1 d/M
McCalla et al,9 1985
2 d/M
Venugopal et al,2 1997
1 d/M
Current report, 2000
2 d/M
Symptoms
Abdominal distension, Non-bilious vomiting, Fight anus
Treatment
Outcome Postoperative peritonitis, Death after 2 days Enterocolic fistula, TPN sepsis, Death at 8 months Hirschsprung’s disease, Well Persistent constipation, Encopresis Abdomino-perineal pull-through Hirschsprung’s disease, Soave procedure at 11 months, Well Hirschsprung’s disease, Soave procedure as neonate, Well Anal dilatation at day 4, Well
REFERENCES 1. Nadalo MAJ LA, Ramirez CTP H: The successful hydrostatic reduction of sigmoid volvulus in an infant: Case report and literature review. Milit Med 2:132-134, 1980 2. Venugopal KS, Wilcox DT, Bruce J: Case report: Hirschsprung’s disease presenting as sigmoid volvulus in a newborn. Eur J Pediatr Surg 7:172-173, 1997 3. Leeba JM, Boas RN: Simultaneous intussusception and sigmoid volvulus in a child. Pediatr Radiol 16:248-249, 1986 4. Valla JS, Louis D, Berard J, et al: Volvulus du sigmoide chez l’enfant. A propos de 6 observations. Chir Pe´diatr 23:93-96, 1982 5. Carter R, Hinshaw DB: Acute sigmoid volvulus in children. Am J Dis Child 101:631-634, 1961 6. Janik JS, Humphrey R, Nagaraj HS: Sigmoid volvulus in a neonate with imperforate anus. J Pediatr Surg 18:636-638, 1983
7. Ismail A: Recurrent colonic volvulus in children. J Pediatr Surg 32:1739-1742, 1997 8. Sarioglu A, Tanyel FC, Bu¨yu¨kpamukc¸u N, et al: Colonic volvulus: A rare presentation of Hirschsprung’s disease. J Pediatr Surg 321:117-118, 1997 9. McCalla TH, Arensman RM, Falterman KW: Sigmoid volvulus in children. Am Surg 51:514-519, 1985 10. Srouji MN, Finnegan LP, Boas RN: Case reports: Neonatal sigmoid volvulus with absence of mesocolon. J Pediatr Surg 9:779-781, 1974 11. Mellor MFA, Drake DG: Colonic volvulus in children: Value of barium enema for diagnosis and treatment in 14 children. AJR 162: 1157-1159, 1994
Sigmoid volvulus is an exceptionally rare cause of intestinal obstruction in neonates. Only 7 cases have been reported in the English and French literature. The authors report a recent case of sigmoid volvulus in a neonate secondary to anal stenosis and review the diagnosis and management of this serious condition. The authors believe that carefully per-
formed radiologic reduction is the preferable alternative to surgical intervention. J Pediatr Surg 36:1079-1081. Copyright © 2001 by W.B. Saunders Company. INDEX WORDS: Sigmoid volvulus, anal stenosis, neonatal.
S
IGMOID VOLVULUS is a diagnosis rarely made in the neonatal population. Most articles are limited to single case reports.1-4 The first report of a sigmoid volvulus in a 14-day-old boy was published by Carter and Hinshaw in 1961.5 Previously, management of neonatal sigmoid volvulus has always been surgical. Hydrostatic reduction by barium enema was first described in 1980 in a 5-week-old infant.1 We report the first case of volvulus of the sigmoid colon in a 2-day-old boy secondary to anal stenosis treated solely by omnipaque contrast enema. CASE REPORT A 2-day-old full-term boy presented with poor feeding, nonbilious vomiting, and increasing abdominal distension. Meconium had been passed, and physical examination showed a well-hydrated baby with obvious abdominal distension, audible bowel sounds, but no tenderness or palpable masses. The buttocks were well formed, but the anus was tight not admitting a small finger. Plain abdominal radiographs showed largely distended intestinal loops but no free air (Fig 1). The baby was started on intravenous fluids and antibiotics, and a nasogastric tube was left on free drainage. A contrast enema with omnipaque showed a sigmoid volvulus that was reduced successfully, and a rectal tube was left in place for 24 hours (Fig 2). Rectal washouts were then started. Suction rectal biopsy results were normal, and anal dilatation was performed under general anaesthesia. Subsequently, the baby ate well and tolerated twice daily dilatations. At the age of 10 months, he is thriving well and has regular bowel motions with twice-daily dilatations.
DISCUSSION Fig 1.
The plain film suggests presence of a sigmoid volvulus.
Sigmoid volvulus is an extremely rare cause of large bowel obstruction in the neonate. Only 7 cases have been reported in the English and French literature (Table 1).1-4,6 There is a strong male predominance.4 The predisposing factor in all age groups appears to be a redundant sigmoid colon associated with an absent, shortened, or abnormally attached mesenteric root.1-4,6,7 The redundant sigmoid may twist around its narrow point of
From Our Lady’s Hospital for Sick Children, Dublin, Ireland. Address reprint requests to M.T. Corbally, Our Lady’s Hospital for Sick Children, Crumlin, Dublin 12, Ireland. Copyright © 2001 by W.B. Saunders Company 0022-3468/01/3607-0027$35.00/0 doi:10.1053/jpsu.2001.24759
Journal of Pediatric Surgery, Vol 36, No 7 (July), 2001: pp 1079-1081
1079
1080
DE CALUWE´, KELLEHER, AND CORBALLY
Fig 2. (A) Contrast injected into the rectum only passed proximally by rotating the patient lying on the left side to a supine position and from there to a slightly right side down position. At this stage, contrast passed across the area of volvulus into the very dilated sigmoid and subsequently into undilated proximal colon. (B) The contrast is shown throughout the colon with residual abnormal distension of the sigmoid. The proximal small bowel is normal.
attachment with luminal obstruction as a result. Occlusion of the mesenteric vessels with ischemia of the sigmoid loop occurs and results in infarction and gangrene. Bacterial translocation appears and results in peritonitis.5 In all age groups, it is associated with constipation.6,7 Hirschsprung’s disease always should be considered and excluded in neonates presenting with a sigmoid volvulus because 3 of the 7 neonates were found to have Hirschsprung’s disease (Table 1). Failure to pass meconium or to have spontaneous bowel motions, bilious or nonbilious vomiting, and increasing abdominal distension are the most common presenting symptoms.6,8-10 In 1 reported case, the baby had an imperforate anus.6 In our case, the baby was noticed to have anal stenosis. Plain x-rays, although occasionally dramatic, are not specific for sigmoid volvulus but should prompt further evaluation. The neonatal age group does not tend to exhibit the early findings of a “coffee bean” abdominal gas pattern, which shows the dilated sigmoid loop rising from the pelvis into the upper part of the abdomen and
suggests a closed loop obstruction.1,2,4,11 Barium enema usually confirms the diagnosis.2,7 All but one of the reported neonatal patients in the literature underwent laparotomy and detorsion or resection with or without colostomy of the twisted sigmoid.2 A contrast enema can derotate the volvulus and relief the obstruction successfully.2,4 Recurrence after derotation by contrast enema is rare.9 Nadalo et al1 reported the successful hydrostatic reduction of a sigmoid volvulus in a 5-week-old infant. Diagnosis and reduction of a sigmoid volvulus by means of a contrast enema in a 1-day-old baby with Hirschsprung’s disease has been reported by Venugopal et al.2 We have reported here on a 2-day-old neonate with sigmoid volvulus secondary to anal stenosis successfully corrected by an omnipaque enema. We believe this procedure is the preferable alternative to surgery in the neonate presenting with a sigmoid volvulus. Surgical intervention only should be necessary in case of unsuccessful derotation by contrast enema or recurrent volvulus.
NEONATAL SIGMOID VOLVULUS
1081
Table 1. Reported Cases of Neonatal Sigmoid Volvulus Study
Age/Sex
Plain Film Findings
Diagnosis
Carter and Hinshaw,5 1961
14 d/M
Abdominal distension, Bile-stained vomiting
Severe colonic distension
Ba enema, Exploratory laparatomy
None, Spontaneous detorsion
Srouji et al,10 1974
2 d/M
Moderate distension, Airfluid levels, No air in rectum
Ba enema, Exploratory laparatomy
Valla et al,4 1982
5 d/M
Abdominal distension, Bile-stained vomiting, No meconium passage Acute distal bowel obstruction Acute distal-bowel obstruction Normal gas pattern, No meconium passage, Imperforate anus Abdominal distension, Vomiting, No meconium passage Abdominal distension, Non-bilious vomiting
Air-fluid levels
Laparatomy
Sigmoid resection, Colostomy, Closure, Colostomy at 3 months Sigmoid resection
Air-fluid levels
Ba enema
Laparatomy, Detorsion
Increasing distension, No air in rectum, Pneumo peritoneum Dilated small and large bowel
Exploratory laparatomy
Loop colostomy
Exploratory laparatomy
Dilated bowel loops
Ba enema
Detorsion, Sigmoid colostomy at 14 days Detorsion
Dilated bowel loops
Omnipaque enema
Detorsion
6 d/M Janik et al,6 1983
1 d/M
McCalla et al,9 1985
2 d/M
Venugopal et al,2 1997
1 d/M
Current report, 2000
2 d/M
Symptoms
Abdominal distension, Non-bilious vomiting, Fight anus
Treatment
Outcome Postoperative peritonitis, Death after 2 days Enterocolic fistula, TPN sepsis, Death at 8 months Hirschsprung’s disease, Well Persistent constipation, Encopresis Abdomino-perineal pull-through Hirschsprung’s disease, Soave procedure at 11 months, Well Hirschsprung’s disease, Soave procedure as neonate, Well Anal dilatation at day 4, Well
REFERENCES 1. Nadalo MAJ LA, Ramirez CTP H: The successful hydrostatic reduction of sigmoid volvulus in an infant: Case report and literature review. Milit Med 2:132-134, 1980 2. Venugopal KS, Wilcox DT, Bruce J: Case report: Hirschsprung’s disease presenting as sigmoid volvulus in a newborn. Eur J Pediatr Surg 7:172-173, 1997 3. Leeba JM, Boas RN: Simultaneous intussusception and sigmoid volvulus in a child. Pediatr Radiol 16:248-249, 1986 4. Valla JS, Louis D, Berard J, et al: Volvulus du sigmoide chez l’enfant. A propos de 6 observations. Chir Pe´diatr 23:93-96, 1982 5. Carter R, Hinshaw DB: Acute sigmoid volvulus in children. Am J Dis Child 101:631-634, 1961 6. Janik JS, Humphrey R, Nagaraj HS: Sigmoid volvulus in a neonate with imperforate anus. J Pediatr Surg 18:636-638, 1983
7. Ismail A: Recurrent colonic volvulus in children. J Pediatr Surg 32:1739-1742, 1997 8. Sarioglu A, Tanyel FC, Bu¨yu¨kpamukc¸u N, et al: Colonic volvulus: A rare presentation of Hirschsprung’s disease. J Pediatr Surg 321:117-118, 1997 9. McCalla TH, Arensman RM, Falterman KW: Sigmoid volvulus in children. Am Surg 51:514-519, 1985 10. Srouji MN, Finnegan LP, Boas RN: Case reports: Neonatal sigmoid volvulus with absence of mesocolon. J Pediatr Surg 9:779-781, 1974 11. Mellor MFA, Drake DG: Colonic volvulus in children: Value of barium enema for diagnosis and treatment in 14 children. AJR 162: 1157-1159, 1994