Ogilvie`s syndrome after pediatric spinal deformity surgery

J Neurosurg Pediatrics 14:255–258, 2014
©AANS, 2014
Ogilvie’s syndrome after pediatric spinal deformity surgery:
successful treatment with neostigmine
Case report
Kristopher G. Hooten, M.D.,1 Seth F. Oliveria, M.D., Ph.D.,1 Shawn D. Larson, M.D., 2
and David W. Pincus, M.D., Ph.D.1
Department of Neurological Surgery and 2Division of Pediatric Surgery, University of Florida, Gainesville,
Florida
1
Ogilvie’s syndrome is a rare and potentially fatal disease that can easily be mistaken for postoperative ileus.
Also known as acute colonic pseudo-obstruction, early recognition and diagnosis of the syndrome allows for treatment prior to bowel perforation and requisite abdominal surgery. The authors report a case of Ogilvie’s syndrome
following spinal deformity correction and tethered cord release in an adolescent who presented with acute abdominal
distension, nausea, and vomiting on postoperative Day 0. The patient was initially diagnosed with adynamic ileus
and treated conservatively with bowel rest, reduction in narcotic dosage, and a regimen of stool softeners, laxatives,
and enemas. Despite this treatment, her clinical course failed to improve, and she demonstrated significant colonic
distension radiographically. Intravenous neostigmine was administered as a bolus with a rapid and dramatic response.
This case is the first reported instance of neostigmine use for Ogilvie’s syndrome treatment following a pediatric
neurosurgical operation.
(http://thejns.org/doi/abs/10.3171/2014.6.PEDS13636)
Key Words • Ogilvie’s syndrome • neostigmine • pediatric • adolescent spinal deformity • neurosurgery • spine
O
gilvie’s
syndrome, or acute colonic pseudoobstruction, is a rare condition characterized by
decreased gastrointestinal motility and colonic
dilation that, if untreated, can result in cecal perforation
and death.26 In the postoperative period following adolescent scoliosis surgery, gastrointestinal complications are
common; the differential diagnosis includes adynamic
ileus, superior mesenteric artery syndrome, and pancreatitis.7,16,24 Paralytic ileus is more common and initially
has similar clinical symptoms that can mask an underlying bowel emergency. In the case of Ogilvie’s syndrome,
which can carry a mortality rate of 25%–60%, early recognition and appropriate treatment can prevent bowel ischemia and perforation.2,26 Common invasive treatments
focus on decompression and include colonoscopy and
laparotomy with tube cecostomy. We present a case occurring after scoliosis surgery in an adolescent in whom
medical management with neostigmine was used as a safe
and effective alternative to these invasive measures.
Case Report
History and Examination. A 15-year-old girl presented with 1 year of worsening back pain and progresJ Neurosurg: Pediatrics / Volume 14 / September 2014
sive scoliosis. On physical examination she was noted to
have an elevated left shoulder and scapula and a right rib
prominence. She was neurologically intact with no bowel
or bladder symptoms. Further workup demonstrated the
presence of a tethered cord with a filum lipoma that was
believed to be a factor potentially contributing to her back
pain and the development of her scoliosis. The scoliosis
was classified as a progressive main thoracic curve of 43°
and a thoracolumbar curve of 24°, Lenke Class 1AN.
Operation. The patient underwent an L-5 laminectomy and tethered cord release combined with selective
thoracic fusion from T-4 to L-1 using segmental pedicle
screw and rod instrumentation and autograft obtained
from the same incision. Surgery was uneventful; electrophysiological monitoring of somatosensory and motor
evoked potentials remained stable throughout the procedure. Total operative time was approximately 6 hours.
Postoperative Course. The patient was intact on
postoperative neurological examination. That evening,
approximately 4 hours after surgery was completed, the
patient developed worsening abdominal pain and nausea.
Examination at this time revealed a distended abdomen
with diminished bowel sounds. The immediately postoperative spine radiographs and subsequent abdominal films
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demonstrated marked colonic dilation and a gaseous pattern (Fig. 1).
Postoperative paralytic ileus was initially diagnosed,
and treatment included bowel rest; a combination of stool
softeners, laxatives, and enemas; and limitation of narcotic dosage. While the patient remained clinically stable,
disease progression was noted radiographically (Fig. 2).
On postoperative Day 6, her symptoms of abdominal
distension and nausea progressively worsened, and plain
abdominal radiographs demonstrated an increase in colon diameter from 11 to 15 cm (Fig. 3). A nasogastric
tube was placed, and a pediatric surgery consultation
was obtained. The patient was transferred to the pediatric intensive care unit for treatment with neostigmine.
Within 10 minutes of neostigmine administration, the
patient noted a dramatic improvement in abdominal pain
and distension, which was followed by flatus and bowel
movement shortly thereafter. Abdominal radiography 12
hours after neostigmine administration revealed dramatically reduced colonic distension (Fig. 4). The patient was
observed on bowel rest until the return of function. She
was discharged home in good condition, and she reported
continued normal bowel function at her follow-up appointment 1 week later.
Discussion
Sir William Ogilvie first described the syndrome
of colonic pseudo-obstruction in 1948 in a series of two
patients with tumor invasion of the celiac plexus. At that
time he theorized the cause to be sympathetic denervation
Fig. 2. Postoperative Day 4 abdominal radiograph. Patient remained
clinically stable, but increased air was noted in the colon, most notably
in the cecum measuring 11 cm.
Fig. 1. Postoperative Day 0 abdominal radiograph demonstrating
gastric air in addition to colon distension.
of the colon.18 There have since been numerous theories
regarding the etiology of the syndrome, including loss of
sympathetic stimulation, prostaglandin abnormalities,
transient ischemia, mechanical air-fluid lock of the bowel,
diminished parasympathetic stimulation, and/or autonomic imbalance.2,3,9,19,23 While the underlying pathophysiology remains unclear, the syndrome is characterized by
decreased gastrointestinal motility with massive colonic
dilation in the absence of mechanical obstruction and
with limited small bowel involvement. This distinguishes
the syndrome from severe postoperative adynamic ileus,
which affects the small bowel and does not respond well
to neostigmine.17,26 Treatment of Ogilvie’s syndrome must
be undertaken to prevent bowel ischemia and perforation,
which carries a high mortality rate as noted above.15
Numerous conditions have been associated with
Ogilvie’s syndrome, including advanced age, postpartum
state, alcohol abuse, narcotic use, irregular bowel habits, laxative abuse, bed rest, diabetes mellitus, congestive
heart failure, uremia, hypokalemia, critical illness, sepsis,
and trauma.2,10,13 Several studies have suggested endogenous opioid release following surgical stress as a potential
cause.8 In neurosurgical cases, colonic pseudo-obstruction has been associated with complex spine surgery and
craniotomies but has also been reported in basic lumbar
and cervical discectomies. Ogilvie’s syndrome has also
been associated with other neurological diseases, including Parkinson’s disease, brain injury, multiple sclerosis,
stroke, meningitis, and Guillain-Barré syndrome.2–5,9
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Ogilvie’s syndrome
Fig. 3. Postoperative Day 6 abdominal radiograph. Patient worsened
clinically and cecum increased to 15 cm in its largest diameter.
Ogilvie’s syndrome is rare in adults and occurs even
less frequently in the pediatric population. Description
of Ogilvie’s syndrome within the pediatric literature is
limited to a small number of case reports. These reports
describe disease risk factors similar to those in adults,
including malignancy, sepsis, systemic illness, electrolyte
imbalances, narcotic usage, and surgery.11,14,22 Though the
explanation for a higher incidence in the adult population
remains unclear, this discrepancy suggests a difference
in pediatric physiology and/or age-related changes in colonic motility.
Early diagnosis is essential for the successful treatment
of Ogilvie’s syndrome. The cecum is the thinnest-walled
portion of the colon with the greatest diameter. Rapid cecal dilation or a cecal diameter larger than 11–13 cm has
been associated with an increased risk of cecal ischemia,
necrosis, and perforation.12 As a result, treatment in adults
should be initiated at a cecal diameter greater than 9 cm.
However, the relative rarity of the disease and age-related
variations in colonic size make it difficult to establish an
equivalent threshold in the pediatric population.11,21 Once a
bowel obstruction has been ruled out with appropriate imaging, early treatment of Ogilvie’s may include bowel decompression via nasogastric suction, cessation of any oral
intake, laxatives, enemas, promotility agents, limitation
of narcotic medication, and rectal tube placement. Many
cases will resolve spontaneously over a few days with this
conservative treatment regimen.8 When required, more aggressive treatments consist of colonoscopic decompression,
cecostomy, or right colon resection. The latter are typically
reserved for cases of bowel perforation.
J Neurosurg: Pediatrics / Volume 14 / September 2014
Fig. 4. Postoperative Day 7 abdominal radiograph obtained 12 hours
after neostigmine administration, demonstrating a dramatic reduction
in colonic air.
Neostigmine is a parasympathomimetic that acts as a
reversible acetylcholinesterase inhibitor that can be used
if bowel ischemia and/or perforation have been excluded.20 It was first used in 1969 for the treatment of ileus
and has been demonstrated to enhance colonic propulsion and accelerate gastrointestinal transit time.6 It has
been extensively tested with good results in critically ill
adults with acute colonic pseudo-obstruction; however,
its use in children for this purpose has been limited to
case reports.1,11,20,25 Potential side effects of the medication include cholinergic bronchospasm and bradycardia.
In the featured case we treated our patient with 0.1 mg
glycopyrronium bromide prior to the 0.04-mg/kg neostigmine bolus; however, one report suggests infusing at
0.025–0.1 mg/kg cautiously with the potential use of atropine for symptomatic bradycardia.11 Regardless of the
specific treatment regimen, we recommend monitoring in
an intensive care setting prior to neostigmine administration because of the potential side effects of bradycardia,
hypotension, and bronchospasm. The presence of any of
these factors prior to treatment represents a relative contraindication to neostigmine use, while underlying bowel
perforation is an absolute contraindication.
Ogilvie’s syndrome has been described in only one
other case following adolescent scoliosis surgery; however, diagnosis in that case was delayed, leading to bowel perforation.4 Given our experience, we recommend
heightened suspicion and close clinical evaluation when
signs of bowel function do not return after the average
postoperative ileus duration of 48–72 hours.17 Bowel
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K. G. Hooten et al.
habits do vary greatly between individuals, and thus a
thorough preoperative bowl habit history should be obtained to grade an individual’s postoperative motility
issues. Although it was unlikely to change the patient’s
clinical course significantly, we believe, in retrospect, that
we should have consulted the pediatric surgery service
24 hours earlier. Since there is no clearly defined time
threshold for postoperative ileus to resolve in pediatrics,
pediatric surgery consultation is recommended when cecal dilation is present and clinical suspicion is high. Lastly, this case demonstrates that this rare syndrome can be
medically treated even in the setting of a 15-cm cecum
when perforation has been ruled out.
Conclusions
Ogilvie’s syndrome is a rare complication following
adolescent spinal surgery. Patients with prolonged symptoms of postoperative ileus should be closely monitored
with a high index of suspicion. Neostigmine can be safely
used with close monitoring and is associated with successful outcomes in pediatric patients.
Disclosure
The authors report no conflict of interest concerning the materials or methods used in this study or the findings specified in this
paper.
Author contributions to the study and manuscript preparation
include the following. Conception and design: Pincus. Acquisition
of data: Hooten, Oliveria. Analysis and interpretation of data: all
authors. Drafting the article: Hooten. Critically revising the article:
all authors. Reviewed submitted version of manuscript: all authors.
Approved the final version of the manuscript on behalf of all authors:
Hooten.
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Manuscript submitted December 2, 2013.
Accepted June 19, 2014.
Please include this information when citing this paper: published online July 18, 2014; DOI: 10.3171/2014.6.PEDS13636.
Address correspondence to: Kristopher G. Hooten, M.D., University of Florida, Box 100265, Gainesville, FL 32610-0261. email:
[email protected].
J Neurosurg: Pediatrics / Volume 14 / September 2014