Professional Documents
Culture Documents
www.elsevier.com/locate/jpedsurg
Key words:
Large bowel volvulus;
Cecal volvulus;
Neurodevelopmental delay;
Chronic constipation
Abstract
Background: Proximal large bowel volvulus is considered as an extremely rare surgical emergency in
children. Approximately 40 cases have been reported, and because of its rarity, the diagnosis is often
missed or delayed. The purpose of this study was to review the presentation, treatment, and clinical
outcome of proximal large bowel volvulus.
Methods: A systematic review and analysis of the data relating to 6 patients from the author's practice
and cases published in the English literature from 1965 to 2010 was performed. Detailed information
regarding demographics, clinical presentation and methods of diagnosis, surgical procedure,
complications, and outcome were recorded.
Results: Thirty-six cases of proximal large bowel volvulus were retrieved from the English literature,
and 6 cases, from the author's practice. The male-female ratio was 1:1, with a median age of 10 years.
There were 29 (69%) cases with neurodevelopmental delay. Clinical presentation included 29 (69%)
cases with constipation, 41 (98%) with colicky abdominal pain, 42 (100%) with abdominal distension,
and 35 (83%) with vomiting. Plain radiography was specific in 64% (27/42) of cases, barium enema in
100% (15/15), and computed tomography in 100% (2/2). All patients underwent surgery, with resection
and primary anastomosis in 24 (57%) cases, stoma formation in 11 (26%), and detorsion of volvulus
without resection in 7 (17%) cases. Six patients (14%) died postoperatively.
Conclusion: A child with neurodevelopmental delay and a history of constipation presenting with an
acute onset of colicky abdominal pain and progressive abdominal distension with vomiting should be
suspected of having a cecal and proximal large bowel volvulus.
2012 Elsevier Inc. All rights reserved.
1. Methods
We retrospectively analyzed the case notes of patients
who had a diagnosis of PLBV in 3 institutions: Royal
Manchester Children's Hospital, United Kingdom; Temple
Street University Children's Hospital Dublin, Ireland; and
Medical Health Science Centre University of Debrecen,
Hungary. We looked at patient demographics, presenting
features, investigations, definitive management, and mortality.
A PubMed search was performed for all case reports and
reviews of large bowel volvulus in children and cecal
volvulus in children. We analyzed the literature and included
cases of cecal and PLBV only. We gathered information under
the same headings of demographics, presenting features, investigations, definitive management, and mortality.
2. Results
There were 6 cases from our institutions and 36 cases
identified in the English literature of cecal and PLBV.
The median age was 10 years with a range of 0 to 18
years. There was no sex predominance with a male-female
ratio of 1:1. Twenty-nine (69%) patients had neurodevelopmental delay (ND), 29 (69%) presented with constipation, 41
(98%) with colicky abdominal pain, 42 (100%) with
abdominal distension, and 35 (83%) with vomiting (see
Table 1). All of the patients with constipation with the
exception of 1 had ND. All patients were investigated with a
plain abdominal radiograph, but this was only diagnostic in
27 (64%) cases. The main finding on plain radiography was
grossly distended bowel (Fig. 1). Fifteen patients underwent
a barium enema, and 2 patients had an abdominal/pelvis
computed tomographic (CT) scan, with both investigation
modalities having 100% specificity. The barium enema
findings were typically that of a bird's beak deformity
representing the cutoff point in the large bowel (Fig. 2).
Computed tomography was able to demonstrate the dilated
segment of large bowel with an air-fluid level (Fig. 3). All
1573
Table 1 Summary of demographics, clinical features,
investigations, management, and mortality
Clinical features
Our
series
Published Summary
data
No. of patients
Age (y), median (range)
Male-female
Neurodevelopmental delay
History of constipation
Colicky abdominal pain
Abdominal distension
Vomiting
Plain abdominal radiograph
Barium enema
Surgery
Mortality
Cecum involved
Terminal ileum involved
Ascending colon involved
Transverse colon involved
6
12 (3-14)
2:4
5/6
6/6
6/6
6/6
5/6
6/6
1/6
6/6
1/6
6/6
1/6
4/6
3/6
36
9.5 (0-18)
19:17
24/36
23/36
35/36
36/36
30/36
36/36
14/36
36/36
5/36
26/36
4/36
12/36
14/36
42
10 (0-18)
21:21
29/42
29/42
41/42
42/42
35/42
42/42
15/42
42/42
6/42
32/42
5/42
16/42
17/42
3. Discussion
Colonic volvulus was first described by Rokitansky in
1836. The sigmoid, cecal, and transverse colon volvulus are
1574
Fig. 1 Figs. 1 and 2 are findings in a patient with neurodevelomental delay and chronic constipation. The extremely dilated large
bowel (not only cecum but ascending, transverse, and descending
colon as well) was loaded with feces. The volvulus resulted in an
extremely dilated and necrotic cecum. The ascending colon was
involved half way up to the hepatic flexure. The rest of the colon was
dilated as well, but fully viable. Because of the chronic constipation
and fecal loading, the twisted colon was more dilated than the
intestine proximal of the obstruction. Fig. 1. is a plain abdominal
radiograph showing large bowel distension. One arrow points to the
distended bowel loop, whereas the other points to the fecal impaction.
Fine gas pattern can be seen in the transverse colon and rectum. This
is consistent with massive fecal impaction and dilatation.
1575
The overall mortality rate of 14% in this study is relatively
high compared with the mortality rate of 9% in patients with
intestinal malrotation [15]. This may be caused by the fact
that patients who develop PLBV tend to have comorbid
conditions such as neurodevelopmental impairment that predisposes them to a more poor postoperative recovery.
Despite PLBV being more common in neurologically
impaired children, it should be considered in any chronically
constipated child with fecal loading, who presents with acute
onset of colicky abdominal pain and vomiting.
References
[1] Gonil AH, Shah NN, Trivedi PS. Volvulus of the caecum in children
a report of two cases. Indian Pediatr 1978;15:361-2.
[2] McGraw JP, Kremen AJ, Rigler LG. The Roentgen diagnosis of
volvulus of the cecum. Surgery 1948;24:793-804.
[3] Anderson JR, Lee D. Acute caecal volvulus. Br J Surg 1980;67:39-41.
[4] Samuel M, Boddy SA, Nicholls E, et al. Large bowel volvulus in
childhood. Aust N Z J Surg 2000;70:258-62.
[5] Vo NJ, O'Hara SM, Alonso MH. Cecal volvulus: a rare cause of bowel
obstruction in a pediatric patient diagnosed pre-operatively by
conventional imaging studies. Pediatr Radiol 2005;35:1128-31.
[6] Takada K, Hamada Y, Sato M, et al. Cecal volvulus in children with
mental disability. Pediatr Surg Int 2007;23:1011-4.
[7] Mellor MF, Drake DG. Colonic volvulus in children: value of barium
enema for diagnosis and treatment in 14 children. AJR Am J
Roentgenol 1994;162:1157-9.
[8] Mulas C, Bruna M, Garca-Armengol J, et al. Management of colonic
volvulus. Experience in 75 patients. Rev Esp Enferm Dig 2010;102:
239-48.
[9] Ghazi A, Shinya H, Wolfe WI. Treatment of volvulus of the colon by
colonoscopy. Ann Surg 1976;183:263-5.
[10] Geer DA, Arnaud G, Beitler A, et al. Colonic volvulus. The Army
Medical Center experience 1983-1987. Am Surg 1991;57:295-300.
[11] Black RE, Cox JA. Volvulus of the transverse colon in children
report of a case and review of the literature. Z Kinderchir 1984;39:
69-71.
[12] Eisenstat TE, Raneri AJ, Mason GR. Volvulus of the transverse colon.
Am J Surg 1977;134:396-9.
[13] Patton D. Of gastrointestinal radiology, bread and butter; or, the
flowering of barium sulfate. Invest Radiol 1994;29:472.
[14] Spitz L. Malrotation in Puri P. Newborn Surgery. 2nd Edition.
London: Arnold; 2003. p. 435.
[15] Rescorla FJ, Shedd FJ, Grosfeld JL, et al. Anomalies of intestinal
rotation in childhood: analysis of 447 cases. Surgery 1990;108:710-5
[discussion 715-6].
All in-text references underlined in blue are linked to publications on ResearchGate, letting you access and read them immediately.