Home | About Us | Current Issue | Ahead of print | Archives | Search | Instructions | Subscription | Feedback | Editorial Board | e-Alerts | Login 
Journal of Indian Association of Pediatric Surgeons
     Journal of Indian Association of Pediatric Surgeons
Official journal of the Indian Association of Pediatric Surgeons         
 Users Online:1588 
  Print this page Email this page   Small font sizeDefault font sizeIncrease font size


 
Table of Contents   
CASE REPORT
Year : 2022  |  Volume : 27  |  Issue : 4  |  Page : 495-499
 

Spachelated intussusception a forgotten entity: Historical review and a case report


Department of Pediatric Surgery, Sawai Man Singh Medical College, Jaipur, Rajasthan, India

Date of Submission08-Jun-2021
Date of Decision30-Aug-2021
Date of Acceptance22-Nov-2021
Date of Web Publication26-Jul-2022

Correspondence Address:
Priyanka Mittal
Department of Pediatric Surgery, Sawai Man Singh Medical College and Attached Hospitals, Jaipur - 302 004, Rajasthan
India
Login to access the Email id

Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jiaps.jiaps_123_21

Rights and Permissions

 

   Abstract 


Intussusception of one or another type is frequently seen in children and is the most common cause of acute bowel obstruction. This pathology is usually promptly recognized and treated accordingly. But if untreated, this entity can have either of the two outcomes. The first one is death from peritonitis and resulting toxemia, which is the most common one. The second one is spontaneous elimination followed by auto-anastomosis and is extremely infrequent. We report one such case of this rare progression who was found to have old healed small bowel intussusception with luminal narrowing on exploration for acute bowel obstruction. We have also reviewed the world literature to understand the mechanism and circumstances under which this progression occurs.


Keywords: Auto-anastomosis, intussusception, spachelated, spontaneous elimination


How to cite this article:
Mathur P, Mittal P, Parihar P, Gupta R. Spachelated intussusception a forgotten entity: Historical review and a case report. J Indian Assoc Pediatr Surg 2022;27:495-9

How to cite this URL:
Mathur P, Mittal P, Parihar P, Gupta R. Spachelated intussusception a forgotten entity: Historical review and a case report. J Indian Assoc Pediatr Surg [serial online] 2022 [cited 2022 Aug 9];27:495-9. Available from: https://www.jiaps.com/text.asp?2022/27/4/493/352275





   Introduction Top


Intussusception is one of the most common causes of acute bowel obstruction in infants and toddlers. Timely management is of utmost importance to prevent grave outcomes. It is a well-known fact that a transient intussusception can be managed conservatively, but a fixed intussusception which is neither reduced nor resected carries fatal outcome. However, surprisingly, there are exceptions to this. Rarely, circumferential adhesions can form at the neck of intussusception, and gangrenous intussusceptum known as spachelus can slough out and may pass per rectum. Continuity of the bowel is maintained at this site of auto-anastomosis but with a luminal narrowing.[1] There have been sporadic case reports of such progression in the historic times. However, to the best of our knowledge, this is the first report of a secondary complication occurring after a spontaneously healing small bowel intussusception in the pediatric population in the past 25 years. With this intent, we report a case of spachelated intussusception and review the available literature.


   Case Report Top


A 10-month-old male child, born out of normal vaginal delivery, with no history of consanguinity and a smooth perinatal transition, reported to the emergency room (ER) with a 3-day history of severe cramping abdominal pain, bilious vomiting, and nonpassage of stools. There was a marked exacerbation of symptoms just before admission. On examination, the patient was lethargic and dehydrated. The apex beat was located in the 5th intercostal space, mid-axillary line anteriorly. There was no evident cyanosis and murmur. There was no respiratory embarrassment. On local examination, abdomen was markedly distended with visible bowel loops, with no signs of peritonitis. There was no fecal or blood staining on digital rectal examination. A plain X-ray abdomen showed multiple air-fluid levels and ultrasonography abdomen was suggestive of dilated fluid-filled small-bowel loops with sluggish peristalsis. The electrolytes were deranged. Coagulation profile and liver function tests were within normal limits. Septic screen was negative.

The patient was rehydrated, and electrolyte imbalance was corrected. The patient underwent laparotomy after necessary optimization. Exploration of the abdomen was done through supraumbilical transverse incision, and it revealed dilated small bowel loops with collapsed terminal ileum [Figure 1]a. There were no other positive findings on inspection. On bowel walking, a soft to firm mass was palpated 5cm proximal to ileocecal junction (ICJ). Unsure about the nature of mass, 5cm ileum both proximal and distal to mass and ICJ was resected and ileoascending anastomosis was performed in two layers. On opening up the resected specimen, the mass was appreciated as an intraluminal soft tethered prominence [Figure 1]b. Postoperative course was uneventful. The patient passed stool on the postoperative day (POD) 4. Nasogastric tube was removed on the same day. The patient was allowed orally on POD-5 and was discharged on POD-7.
Figure 1: (a) Intraoperative image showing dilated small bowel loops with collapsed terminal ileum. (b) An intraluminal soft tethered prominence. (c) Histopathological examination showing mucosa (black arrow) on both sides with lymphoid proliferation (red arrow) in between

Click here to view


Pathological examination of the specimen showed dense cicatrix surrounding a 2-mm lumen, with mucosa on both sides with lymphoid proliferation. Underlying stroma showed edema with dense inflammatory infiltration. No evidence of granuloma or malignancy was found [Figure 1]c.

On realizing that probably, we have removed an old strictured auto-anastomosis of a previously sloughed intussusceptum, and child's history was reviewed more thoroughly. His mother revealed that at approximately 5 months of age, the patient had an attack of severe gastroenteritis lasting for 4 weeks. After treatment with oral hydration and antibiotics at home, his diarrhea subsided. No tissue had been passed through the rectum.

At present, the child is doing well on a close follow-up of 1 year.


   Discussion Top


Intussusception develops with antegrade peristalsis when the proximal bowel (intussusceptum) drags along with its mesentery into the distal bowel (intussuscipiens). Compression of mesentery leads to edema of intussusceptum, causing impairment of lymphatic drainage and venous congestion and stasis and hence outpouring of blood and mucus. If untreated, ischemic changes amount to bowel necrosis in the intussusceptum and can be potentially fatal. Rarely, necrosed intussusceptum may get separated and expelled spontaneously, followed by auto-anastomosis of bowel ends. This entity can be defined as spachelated or stenotic old intussusception.[2] The exact incidence remains unclear. However, in 1958, Aird has estimated the incidence of this infrequent natural progression to be <2%.[3]

Although very rare even in historic times, this atypical pathophysiological progression was relatively more frequently observed about 2 centuries ago than the present times, when the operative mortality used to be high and waiting for spontaneous resolution used to be the best alternative.[4] Progression of intussusception in this manner has markedly reduced and has not been reported in pediatric population in the past 25 years. This can be ascribed to early diagnosis and intervention in the present times.

Treves is credited with the first detailed description of this atypical entity.[5] He described that if the process of vascular obstruction and gangrene is less rapid, adhesions may form between the peritoneal coats of the inner layer and outer layer or intussuscipiens at the neck of the intussusception, hereby preventing perforation at this site. This event is heralded by sloughing of intussusception, which may or may not be noticeable in stools, as in the index case, and restoration of bowel continuity by auto-anastomosis. Hence, it can be inferred that the spontaneous cure of intussusception depends on the presence of appropriate time for allowing the adhesion formation between the inner and outer layer; before the sloughing of necrosed intussusceptum [Figure 2]. This time window is more apparent in ileoileal intussusception, as in the index case, than in ileocolic intussusception where constricting effect of ileocecal valve will lead to rapid onset of vascular occlusion, gangrene, and perforation. Hence, these kinds of intussusceptions are generally ileoileal. Lichenstern has suggested that this separation occurs 11 to 21 days after intussusception.[6] Separated intussusceptum can sometimes cause a ball valve effect and can cause recurrent obstruction at the narrowed area.
Figure 2: Line diagram showing etiopathogenesis of spachelated intussusception

Click here to view


With this surgical curiosity in mind, we have attempted to review world literature as old as 145 years. For our ease of understanding, we have divided this time frame further into – Lichenstern era (1873-1877) and post-Lichenstern era (1878-present). We reviewed 2293 cases of intussusception in total. Out of these, 593 cases date back to Lichenstern era, and 1700 cases belong to post-Lichenstern era.

We have tabulated our observations in [Table 1].
Table 1: Literature review on spontaneous sloughing and auto-anastomosis in intussusception

Click here to view


Walten reported a series of intussusception in 1911, which comprised 239 cases, but only one case terminated into spontaneous elimination (which was the same as reported by Sherren).[18] In 1908, Fitzwillliams reported a series of 1000 cases of intussusception, with not even a single case undergoing spontaneous elimination.[19] Similarly, Perrin and Lindsay reported a series of 400 cases of intussusception, spanning over a period of 19 years; but spontaneous elimination was not reported in any case.[20]

Going by above data, we found that 310 cases of this entity have been reported in world literature in the past 145 years. The maximum number of such cases, i. e., 80% of cases (n = 249) were seen in Lichenstern era amounting to an incidence of 42%. This was an era when clinicians used to rely on waiting for intussusceptum to slough and auto-anastomosis to occur. Interestingly, operative interventions were also directed toward spontaneous cure in the past. Historic literature review finds the mention of the Barker's operation or Jesset's procedure where seromuscular sutures were placed at the neck of intussusception, and intussusceptum was then divided through an enterotomy in the intussuscipiens.[21],[22] Another procedure worth a mention has been described by Bayard in which the base of a sigmoid intussusceptum was banded by gaining access through the anus into the rectum to encourage intraluminal autoamputation.[23]

On pondering over post-Lichenstern era, only 20% cases (n = 61) of such progression have been reported. This infers that the incidence of this entity presently is only 3.5%. Out of these 61 cases, 70.5% of cases (n = 43) have been reported in adults, and only 29.5% of cases (n = 18) were reported in pediatric population; that too none after 1995. This brings down the incidence of spachelated intussusception in pediatric population to just 1%.

This interesting observation can be explained by earlier diagnosis and treatment of intussusception in children, in whom the clinical picture is more striking than in adults. Second, an untreated intussusception in a child will end fatally within a few days due to marked fluid and electrolyte loss which is not well tolerated by this age group; whereas, adults can tolerate fluid and electrolyte disturbances better and hence have a better chance of survival for a sufficiently long period to allow the possibility of spontaneous healing. Third, adults are more likely to have small bowel intussusception as contrary to ileocolic intussusception in pediatric population, which leads to subacute obstruction and hence more chances of spontaneous healing. This has already explained by Treves and our study validates the same.[5]

In the index case, child had a history of acute gastroenteritis lasting for 4 weeks which was most probably an overlooked small bowel intussusception. The most acceptable explanation for survival in this case was subacute intussusception leading to sloughing of intussusceptum and auto-anastomosis of the bowel ends. Five months later, the patient presented with intestinal obstruction with luminal narrowing at the site of auto-anastomosis and this has been correlated histopathologically too. Our findings are consistent with a case reported by Richard et al.[2]

Rarity of this entity coupled with the age of the patient and topography of intussusception prompted us to report the case.


   Conclusion Top


This case report intent to draw attention toward the entity of sloughed intussusceptum as a rare but possible etiology of partial bowel obstruction in childhood, especially in the cases of conservatively managed pain abdomen and altered bowel habits. It is also imperative to take a proper clinical history and meticulous examination intraoperatively and communication with the pathologist so that such interesting findings do not get missed.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient (s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initial s will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
Ravitch MM, McCune RM Jr. Intussusception in infants and children; analysis of 152 cases with a discussion of reduction by barium enema. J Pediatr 1950;37:153-73.  Back to cited text no. 1
    
2.
Royal RE, Stevenson RJ. Luminal narrowing after auto-anastomosis and intraluminal slough in intussusception: A case report and historical review. J Pediatr Surg 1995;30:752-5.  Back to cited text no. 2
    
3.
Aird I. A Companion in Surgical Studies. 2nd ed. London, England: E and S Livingstone Ltd; 1958. p. 813-6.  Back to cited text no. 3
    
4.
Robb WA, Souter W. Spontaneous sloughing and healing of intussusception. Historical review and report of a case. Br J Surg 1962;49:542-6.  Back to cited text no. 4
    
5.
Treves F. Intestinal Obstruction. 2nd ed. New York: W Wood and Co; 1899. p. 163-70.  Back to cited text no. 5
    
6.
Lichenstern O. Intussusception, invagination. In: Darmein S, Ziemssen H, editors. Ziemssen's Cyclopedia of the Practice of Medicine. Vol. 7. Leipzig, Germany: W Wood and Co; 1877. p. 610-24.  Back to cited text no. 6
    
7.
O'Connor EM. A remarkable case of intussusception. Br Med J 1894;2:123-4.  Back to cited text no. 7
    
8.
Sutcliffe A. Case of intussusception with sloughing of intestine; recovery. Br Med J 1894;2:124.  Back to cited text no. 8
    
9.
Sherren J. A clinical lecture on acute intussusception. Clin J 1906;27:185.  Back to cited text no. 9
    
10.
Marnoch J. Cases of intussusception. Scott Med Surg J 1906;19:176-84.  Back to cited text no. 10
    
11.
Thompson LD. Intussusception terminating in spontaneous elimination. Am J Dis Child 1927;34:640.  Back to cited text no. 11
    
12.
Silvermann D. Spontaneous elimination of intestine with temporary recovery. Radiology 1931;17:1208-15.  Back to cited text no. 12
    
13.
Finestone EO. Intussusception of excluded distal ileum with spontaneous expulsion per anum of sequestrated intussusception. Surgery 1947;21:34-42.  Back to cited text no. 13
    
14.
Grant RN, Bowden L. Spontaneous expulsion per anum of sequestrated transverse colon. Am J Surg 1951;82:299-302.  Back to cited text no. 14
    
15.
Benson CD, Carpenter WS, Swedenberg RD. Spontaneous expulsion of sequestrated ileum; report of recovery in two adults. Ann Surg 1953;137:261-4.  Back to cited text no. 15
    
16.
Tropea F Jr., Shepard VS. Intussusception of ileum with expulsion of intussusceptum per anum. Am J Surg 1954;87:152-4.  Back to cited text no. 16
    
17.
Peck DA, Lynn HB, DuShane JW. Intussusception in children. Surg Gynecol Obstet 1963;116:398-404.  Back to cited text no. 17
    
18.
Walton AJ. Intussusception. Practitioner 1911;87:186-222.  Back to cited text no. 18
    
19.
Fitzwilliams DC. The Pathology and Etiology of intussusception from the study of 1000 cases. Lancet 1908;1:628-709.  Back to cited text no. 19
    
20.
Perrin WS, Lindsay EC. Intussusception: A monograph based on 400 cases. Br J Surg 1921;9:46.  Back to cited text no. 20
    
21.
Treves F. A Manual of Operative Surgery. Vol. 2. Philadelphia: Lee Brothers and Co; 1901. p. 370-74.  Back to cited text no. 21
    
22.
Iason AH. Intussusception in adults. Surgery 1945;18:457-66.  Back to cited text no. 22
    
23.
Bayard HF. Intussusception of the sigmoid. Minn Med 1939;22:456-8.  Back to cited text no. 23
    


    Figures

  [Figure 1], [Figure 2]
 
 
    Tables

  [Table 1]



 

Top
Print this article  Email this article

    

 
  Search
 
  
 
    Similar in PUBMED
   Search Pubmed for
   Search in Google Scholar for
 Related articles
    Article in PDF (568 KB)
    Citation Manager
    Access Statistics
    Reader Comments
    Email Alert *
    Add to My List *
* Registration required (free)  


    Abstract
   Introduction
   Case Report
   Discussion
   Conclusion
    References
    Article Figures
    Article Tables

 Article Access Statistics
    Viewed82    
    Printed2    
    Emailed0    
    PDF Downloaded10    
    Comments [Add]    

Recommend this journal


Contact us | Sitemap | Advertise | What's New | Copyright and Disclaimer | Privacy Notice

  2005 - Journal of Indian Association of Pediatric Surgeons | Published by Wolters Kluwer - Medknow 

Online since 1st May '05