Effects of delayed presentation of intussusception in infants and children

Effects of delayed presentation of intussusception in infants and children

Mohamed Fathy Metwally, MD

Department of Surgery, Tanta University, Egypt.

Abstract

Background/purpose:  Many  children  with  intussusception   are  reported  to  present  late for definitive therapy. Attempted  nonsurgical  reduction of intussusceptions  after 48 hours is controversial because of the low probability of reduction and an increased risk of perforation. This study was conducted to determine the effect of delayed presentation on clinical parameters, management, and the outcome of childhood intussusceptions.  The author also studied the diagnostic investigations that can give criteria that may help to predict bowel viability and to choose the most suitable management.

Methods:  Comparative  analysis of  44  children with intussusception  managed from 2007 to 2011 at Tanta university and Saudi Arabia was done.

Results:  The mean time from onset of presentation  was 3 days (range  from 4 hours to 7 days). Thirty seven patients presented within 48 hours of symptoms (group 1) and    7patients presented  after 48 hours (group 2). Clinical presentations were similar in the children with the exception ofbilious vomiting, rectal bleeding, and abdominal distension that were significantly commoner in group 2 children (p<O.05).The type ofintussusceptionsfound at operation did not differ in the groups, but bowel complications  and the incidence offailed reduction and bowel resection were higher in group 2 patients (p<0.05).  Though the postoperative  complications did not differ significantly between the two groups, mortality directly related to intussusceptions occurred only in patients who presented after 48 hours.

Conclusion: Significant number of children with intussusceptions  present late for definitive treatment. These cases have a high risk of bowel complications  and intestinal resection. The outcome  of these  patients  can  be improved  through good  perioperative  care and  reducing delays in seeking health care. Also, cases of delayed presentation can safely undergo contrast enema reduction ifno bowel-wall edema ofthe intussuscipiens  or obstruction or ischemia is demonstrated.

Key words: Childhood, intussusceptions,  delayed presentation, outcome.

Introduction:

Intussusception is a common cause of gastrointestinal   obstruction   in  infants  and young children (15%).1 The peak incidence is between 4 and 6 months with a male to female ratio  of  1.3  :1.2-4  Clinical  presentations  are well known that definitive diagnosis and management in most cases of intussusception is expected  to  be  done  within  48 hours  of onset of symptoms.  The presenting  features of   intussusception    are   sudden   onset  of

intermittent   colicky   abdominal   pain   in  a previously  well  child.  Infants  and  children may  strain,  draw  their  knees  up,  act  very irritable,   and  cry  loudly.  The  child   may recover and become playful inbetween bouts of  pain,  or  become  tired  and  weak  from crying.         Vomiting usually  starts  soon  after the pain  begins. Abdominal   distention  and constipation are common, although the child may pass normal stools initially, the following stools may look  bloody  with mucus  giving

jell-like stools.  Palpable  abdominal mass and sometimes, palpable mass on rectal examination may be felt.2-7 In spite  of this, a considerable number    of   children    with   intussusception, for  ill-defined  reasons,  present  late  for treatment. This  delayed  presentation might be a significant cause for the poorer  outcome of treatment in these  cases.8

Intussusception is diagnosed by radiology, ultrasonography, Doppler, and surgery. Abdominal  radiography  may   show   dilated small bowel and absence of gas in the region of the cecum.  In some cases, a mass impression within  the colonic  gas with  an "air  crescent" sign from air trapped between the bowel walls, indicates  an intraluminal mass created  by the intussuscepting loop. Sometimes, radiographs may  appear   indeterminate  or  normal, therefore, the presence  of an unremarkable abdominal radiograph should  not be the base for excluding a diagnosis of intussusception.9

Contrast and pneumatic enemas have dual role in diagnosis and treatment of intussusception. Water - soluble  radiopaque agents as well as air are used for enemas  as they show   coiled spring appearance with arrest of barium at the end of intussusception.!

Ultrasonography has now proved its accuracy for diagnosis of intussusception. Transverse  ultrasonogram  shows      a  mass with a swirled  appearance of alternating sonolucent and  hyperechoic bowel  wall  of the     loop-within-a-loop.    On    longitudinal ultra  sonograms, the  intussuscipiens and the intussusceptum have the appearance of a submarine sandwich or pseudo  kidney sign.

There appear  to be multiple  layers, which

represent  the   walls   of   the   intussuscepted bowel        loops.   Although                        ultrasonographic examination        is                      almost always positive, overlying   loops      of   air-containing   bowel may  obscure  intussusception.9 Some authors found  that hydrostatic reduction was unlikely or  impossible  when   the   outer   rim  of  the intussusception  target    sign   on   ultrasonic was  more  than  1Omm thick  or when  a large amount  of free  peritoneal fluid  was found.l o Other authors showed no correlation between the edema, peritoneal fluid or of the occlusive syndrome  and  the  severity  of  ischemia.lO

In spite of this, clinical    and radiological findings  are unable to predict  the intensity of the vascular  injury.

The  invagination of  a segment of  bowel

induces  a mechanism of strangulation where as the  intussusceptum proceeds   distally,  its mesentery    becomes    stretched,    angulated and  compressed between  the  layers  of  the bowel   wall,  first  causing   partial  inhibition of      venous                          drainage,           venous    congestion and   edema   that   contribute  to  tighten   the intussusceptum       and         obstruct   the      small bowel.  A second  phase  of arterial  occlusion will  occur  with  hemorrhage, gangrene   and risk of  perforation. Severe  ischemia is often encountered  in  ileoileal  and  trans-valvular ileocolic  types , where the mesentery is short and the intussusceptum has a narrow  neck.ll In  intussusceptions, color   Doppler   allows an   interesting   evaluation   of   the        degree of            bowel               ischemia                      and necrosis,          can change   the   therapeutic  management                         and appears        promising          for       appreciating     the viability  of   the   occluded    bowel    loops.ll CT is generally the imaging modality of choice as  the   images   on   CT  are   pathognomonic for  intussusception.  A  CT  scan  may  have a   further    advantage    by   providing   clues to  the   etiology   of  the   intussusception as lymphadenopathy         or          malignant       lesions.l Many recent  studies  on intussusception have paid  great  attention  to children  who  present after  48  hours   of  symptoms,  in  a  trial  to develop  feasible  initiatives   for  better  early diagnosis  and   better   results   of   treatment for  the  patients.S-7,12,13  Treatment   includes air   or  hydrostatic  reduction  enema   under radiologic  or  ultrasound  guidance, surgery reduction (traditional and  laparoscopic) and in  approximately  10%   of   cases  intestinal

resection due to intestinal  ischemia.l4

Patients and methods:

This  study  was  conducted  at  Tanta University   Hospital     and    Saudi    Arabia, over   a  period   of  4  years  from   2007     to

2011.   Forty   four   patients   were   managed for intussusception. Diagnosis was based mainly   on  clinical,   radiological, ultrasound and  Doppler  evaluation. All  cases  received

fluid and electrolyte resuscitation. Definitive treatment   including   operative   intervention was done in all cases.

For the purpose of this study, delayed presentation was defined as presentation after

48 hours of onset of symptoms. The clinical features, operative treatment, complications and  outcome  were  compared   between  the cases presented after 48 hours and those presenting   earlier,   in  order   to   determine the effect of delayed presentation on these variables.

At the start of the study the author selected operative intervention for all cases (15 cases) to correlate between the clinical features, radiological investigations and operative findings.   Later,  the  author  started  to  use other methods of management  as hydrostatic reduction, in addition to the operative intervention.

Color Doppler has been attempted to determine the intensity of ischemia and the possibility   of   reducing   intussusception   in all cases.  In group  1, there  were  evidence of   venous   &  arterial   hyperemia    within the intussusceptum in 37 cases. Complete reduction by hydrostatic reduction was achieved in 7 cases (19 %), simple  reduction by   milking  after  surgical exploration in 22 cases (59 %), simple laparoscopic  reduction in 5 cases (14 %) and resection anastomosis was required in the other 3 cases (8%). The indication for resection in these 3 cases  was failure of simple reduction due to severe edema.

Considering       the  7  cases  in  group  2, there was hyperemia in 2 cases (29%)  and 5 cases (71%) without hyperemia.  No attempt for  hydrostatic or pneumatic reduction was done for  all cases and surgical  intervention was done immediately. In the 2 cases with hyperemia,    surgical   reduction   was   done with some difficulty with the aid of hot fomentations. The other 5 cases (100%) required resection anastomosis due to the marked        ischemia      and necrosis of the intestine.

Results:

The   44   children   included   37   patients (84%)   who presented within 48 hours of symptoms (group  1) and   7 patients (16 %) who presented after 48 hours of symptoms (group 2). Thirty patients  (68%) were males and 14 (32%) were females. The  median age at presentation was 7 months (range 3 months

-3 years).

All    cases    presented    with    abdominal pain. The other features varied in the two groups. In  group 1, the presenting features included bilious vomiting 14 cases (38 %), rectal bleeding 11 cases (30%), abdominal distension   14  cases  (38%),  palpable abdominal mass 17 cases (46%), and silent abdomen 3 cases (8%). The  clinical features in group 2 patients included bilious vomiting

6  cases  (86  %),   rectal   bleeding  5  cases

(71%), abdominal distension 5 cases  (71%), palpable abdominal mass 4 cases (57%), and silent abdomen 2 cases (29 %).

Table(l): Shows the comparison between the clinical presentation of intussusception in children presenting early and those presenting after 48 hours.

* Statistically significant (p< 0.05)

The average duration of symptoms before presentation to the hospital was 3 days (range

4 hours - 7 days).

At   operation,    cases   in   group   1   had ileocolic  intussusception  in  28  (76%), ileoileal  in  6 (16%),  and colocolic  in 3  (8

%). Children  in group 2, had ileocolic type in6 (86%) and colocolic one patient (14 %). Successful    reduction     of    intussusception was  done  in  34  cases  (92%)  in  group  1 (hydrostatic  reduction in 7 cases, traditional surgical       reduction          in                      22                  cases             and laparoscopic reduction in 5 cases), compared to  2  cases  (29%)  in  group  2  (p=0.03)  by traditional  surgical  reduction.  In  the  other

8  patients,  the   intussusception   was  either

irreducible  in 3 patients   in group 1, or was with  necrotic bowel in 5 patients           in group

2. The eight  cases had right hemicolectomy

(3 cases in group 1 and 5 cases in group 2).

All these  cases  were  of the  ileocolic  type. All   surgical   wounds   were   closed primarily.    None    of    the    patients    was managed in an intensive care unit. Postoperatively, the complications were superficial  wound  infection  in 3  cases, adhesive small bowel obstruction in one case and chest infection in 3 cases.

Superficial wound infection was managed by local wound care. The case of adhesive bowel obstruction responded to conservative management.

Follow  up  was  done  for  an  average  of

10 months  (range 3-29 months).  There was no recurrence of intussusception and the postoperative complications did not differ significantly between the two groups. One patient (2%) died   at the tenth day after the operation due to septicemia  in group 2.

Table (2): Shows comparison  between the postoperative complications in the 2 groups.

Figure (1a): A case of

Intu ssusception showing the passage of red currant jelly.

Figure (1b): Plain x-ray abdomen (erect) showing multiple air and fluid levels in the same case of intussusception.

Figure (lc): US of  the same case showing intu ssusception with enlarged mesenteric lymph nodes.

Figure (1d): Color Doppler ofthe same case showing marked hyperemia, indicating viable intestine.

Figure (2a): Plain x-ray of a case ofintussusception showing  absence of cecal air and an obstruction pattern are seen.

Figure (3a): US of another case showing target like lesion in a  case of ileocolic intussusception.

Figure (le): Barium enema showing coiled spring appearance with arrest

of barium at the end of intussusception.

Figure (2b): US of  the same case showing intussusception.

Figure (3b): Barium enema ofthe same case showing arrest of the barium at the hepatic flexure.

Figure (lf): Intraoperative picture showing  colocolic intussusception with viable intestine, treated by simple reduction.

Figure (2c): Laparoscopic picture of the patient with no edema or ischemia showing simple reduction of  the intussusception.

Figure (3c): Intraoperative picture ofthe same

case showing  ileocolic intussusception with viable intestine treated by simple reduction.

Figure (4a): CT scan of the patient showing the classic target sign in the sigmoid colon (arrow) . There is no evidence of bowel wall edema of the intussuscipiens  or of small bowel obstruction.

Figure (4b): x-ray showing hydrostatic reduction of

the same case of colocolic intuss.with the mass in the sigmoid colon.

Figure (4c): Reduction of

the mass into the descending colon.

Figure (4d): Reduction of the mass into the ascending colon.

Figure (4e): Complete reduction ofthe intussusception.

Figure (5a): A picture showing sunken eyes with feculent aspiration in a patient with

delayed presentation of intussusception.

Figure (5b): Plain xray abdomen (erect) ofthe same patient showing air under the Rt copula of the

diaphragm due to perforated intestine.

Figure (5c): Color Doppler ofthe same case showing sluggish arterial flow within the thickened wall of bowel. So, diagnosis ofischemic intestinal damage is highly suggested

Figure (5d): CT scan of the patient showing the classic target sign (large arrow), bowel wall edema ofthe intussuscipiens (small arrow) and dilated  small bowel consistent with  obstruction.

Discussion:

This  study   shows   that   some     children with intussusception present for definitive treatment after  48 hours  of symptoms_8,15-17

Many  reports  from  developed countries indicate that the time  of diagnosis and intervention in a majority of cases is less than

24 hours.2    This may be due to ignorance, poverty  and inadequate access to referral hospitals.

Previous  reports  have  indicated that delayed   presentation  predisposes to  bowel

complications.6,13   This    was    reflected    in

the clinical  presentation and the operative findings of the cases  that  presented  after  48 hours of symptoms in this study.

Clinical  features such  as  rectal  bleeding,

abdominal distension  and  absent  bowel sounds which may be indicative of devitalized bowel,  were commoner in children  who presented after  48 hours.  In the  same  time, a considerable number  of irreducible and devitalized  bowel     found   at  operation  in these cases, may confirm  increased bowel complications with prolonged interval  , from symptom onset  to  definitive treatment. This implies  that  one  should  have  a high  index of suspicion for the possible  presence  of devitalized  bowel   and   adequately  prepare such patients for bowel resection in case there is  need  for  it. Operative   findings  indicate that the type of intussusception does not contribute to the delay  in diagnosis.

Intussusception  is   currently   treated   by

Figure (5e): Intraoperative  picture of the same case showing a gangrenous intestine.

air  insuffiations or  hydrostatic enema.  This non  operative  reduction  of  intussusception has  been  shown   to  reduce   length   of  stay, shorten recovery, decrease  hospital costs, and decrease  the risk of complications related  to abdominal surgery_l8,19 It is known  that  the earlier  the  child  is  managed  after  the  onset of the symptoms, the higher the frequency of successful conservative treatment. The only contraindications to barium  enema  reduction is peritonitis_16,18

At the start of the study, the author did traditional  surgical   exploration to  the  first

15 cases, to correlate  the clinical,  diagnostic

investigations  and   the   operative  findings. After  that,  the  author  started   to  select   the method  of management according to the parameters determined from  the    results  of the first 15 cases.

Hydrostatic   reduction    was    attempted in 11 cases all in group 1, with no signs of ischemia, but succeeded in 7 cases only. The other 4 cases were reduced  by traditional surgical  reduction. The cause of failure  of hydrostatic reduction was due to edema of the wall  of the  intestine.   Nonsurgical reduction was not attempted in patients with delayed presentation because  of the decreased probability of success  and the increased potential  for   perforation.  However,   recent data have shown that successful hydrostatic reduction is not influenced by symptom duration.19 Many recent studies gave reports above   80%  for  non-operative reduction of

intussusception.20

Operative   intervention   is   reserved   for cases that were  complicated  at presentation or failed to respond to pressure treatment.

Laparoscopic  reduction  was attempted  in

7 cases of group 1 with no signs of massive edema  or  ischemia.  Success  was  achieved in  5  cases  only, and failed  in  2 cases  due to   severe  resistance   with   fear   of   bowel rupture.  Abdominal   exploration   was  done and reduction was done manually with some difficulty, with the aid of hot fomentations.

Patients   in group 1 were managed by traditional surgical reduction in 22 cases, and surgical resection in 3 cases, while all patients in group 2 were managed by operative reduction in 2 cases and resection in 5 cases. The high  incidence  of bowel complications in patients with delayed presentation favored routine    operative    intervention,    although, some studies reported successful pressure reduction in some cases presenting after 48 hours, and operative reduction was successful in 2   cases with delayed presentation.21,22

At operation, the procedure was chosen according   to  the  findings.   In  the  present study,  5  children  who  presented  after  48 hours of symptoms required bowel resection because of bowel complications.  The rate of bowel resection in our cases with delayed presentation was similar to many published reports_23,24

The postoperative complications in this study were those for general surgery. The complications   were   more   in   patients   in group 2 due to the complicated  intestine and the bad general condition of the patients. Though    the    postoperative     complication rates   differ  between   the  two  groups,  the absence of significant difference may be due to other factors as the operative technique, tissue handling, antibiotics used and wound management.

Recent reports from developed countries indicate that mortality from childhood Intussusception has steadily declined to under

1%.7,25,26  Most of the reported mortalities in

these settings were associated with delayed diagnosis.

In the  present  report,  mortality  directly

related  to  intussusception  occurred  only  in one patient in group 2. This indicates that early diagnosis and good patient care will improve the  outcome  of  Intussusception.  Septicemia was the main cause of mortality in our case. So, improving the perioperative care of the patients  with  aggressive  resuscitation,   use of more potent antibiotic and postoperative management  in an intensive care unit might minimize the mortality.   In the long term, improvement in time to diagnosis through maintaining  a high index of suspicion, meticulous  clinical  evaluation,  improved access  to  referral  centers,  and  provision  of basic radio diagnostic facilities may help to save these children.

Weihmiller  S  N et  al,  2011,  in  a study

of 38 patients with intussusception, they studied the predictive factors for low risk or high risk patients. Their decision  was based on the results  of abdominal  x-ray (negative or positive), age (5 or >5 months),  diarrhea (present or absent), and bilious vomiting (present or absent). They found a sensitivity of 97% for these tests.27

Kaiser AD et al, 2007, in a study for the current success rate for radiological reduction, requirements  for operative  intervention,  and the effect of delay in presentation on the outcome  of  intussusception,  they  reviewed the records for the children treated for intussusception  for  15  years.  They  studied

244 children with intussusception. Their median   age   was   8.2   months   (range,   16 days to 12.7 years). The most common presenting symptoms were emesis (81%), hematochezia (61%), and abdominal pain (59%).  Contrasted  enemas  were  performed in  190  children,  with  successful  reduction in 46%. Success in reduction was greater if symptom duration was < 24 hours compared with > 24 hours (59% vs. 36%). One hundred forty  children required  surgical  intervention for Intussusception with 50% requiring bowel resection.  Children  with  symptom  duration

> 24 hours  had  a greater  risk  of requiring

surgery (73% vs. 45%) and bowel resection (39%  vs.  17%)  than  those  with  symptoms for < 24 hours. There were 2 deaths and complications  occurring  in  19%.  Length  of

stay  after  surgical  reduction  was  3.9  days and 6.1 days if bowel resection was required. They concluded that delay in presentation decreases success in radiologic reduction and increases risk of operative intervention and bowel resection.7

Suzanne  Schuh  and  David  E.  Wesson,

1987,   studied   intussusception   in  children

2 years of age or older. They reviewed the records  of  111  children  who  were  treated for intussusception between 1974 and 1984. They found no complications of attempted barium    enema   reduction.    They   reported rate of success for this reduction of 90% regardless of the duration of illness, even for patients  who presented  up to 2 weeks after the onset of symptoms. They had 3 deaths related to the original disease and not to intussusception. They concluded that a long history is not in itself a contraindication to hydrostatic  reduction,  nor is it an automatic indication for laparotomy.28

Conclusion:

1- A considerable proportion of children with intussusception present to the mainstream medical                 practitioners    after   48   hours of   symptoms.   These   patients   have   a higher incidence of bowel complications requiring resection, and higher mortality rates. Improving the perioperative care of these cases may improve the outcome.

2- Efforts directed at improving time to diagnosis    through    enhanced    referral system and improved  diagnostic facilities may optimize the outcome in the long run.

3-     Subspecialty    and    surgeons    trammg for  the  diagnosis  and  management   of intussusception   in    children    are   very important.

4- In intussusceptions,  color Doppler allows

an  interesting  evaluation  of  the  degree of  bowel  ischemia  and  necrosis  and  it can change the therapeutic management and   appears  promising  for  appreciating the viability ofthe occluded bowel loops. This  recent  technique  seems  to  have  a place in the strategy  of the treatment  of such cases.

5-  The  ability  to  correlate  the  diagnostic

results and the surgical findings in children with intussusception, has enabled the development of imaging criteria that may indicate  which  patients  may  still safely and effectively undergo cautious attempts at barium enema or air reduction. The absence of bowel wall edema, ischemia, bowel obstruction, no peritonitis on ultrasound  and  Doppler  and  no free  air is seen on radiographic imaging, are all good indications that the bowel can be easily reduced.

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3- Huppertz HI, Soriano- Gabaro M, Grimprel

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