African Journal of Paediatric Surgery About APSON | PAPSA  
Home About us Editorial Board Current issue Search Archives Ahead Of Print Subscribe Instructions Submission Contact Login 
Users Online: 1359Print this page  Email this page Bookmark this page Small font size Default font size Increase font size 
 
 


 
CASE REPORT Table of Contents   
Year : 2011  |  Volume : 8  |  Issue : 2  |  Page : 221-224
Littre hernia in childhood: A case report with a brief review of the literature


1 Department of Pediatric Surgery, Ufuk University, Faculty of Medicine, Ankara, Turkey
2 Department of Pathology, Ataturk University, Faculty of Medicine, Erzurum, Turkey

Click here for correspondence address and email

Date of Web Publication14-Oct-2011
 

   Abstract 

A 3-year-old boy with a right-sided and painful inguinal swelling for the last 2 h was admitted to the emergency department. As there were no apparent peritoneal irritation findings, right-sided incarcerated inguinal hernia was reduced and the patient was scheduled for an elective herniorrhaphy. Perioperatively even though the sac seemed empty, it was opened in order to inspect its content. The adherence of Meckel's diverticulum (MD) to the base of hernial sac was realized and Littre hernia (LH) was diagnosed. The hernia was highly ligated after the wedge resection of the diverticulum and anastomosis. The patient was fed on the second postoperative day and discharged on the third postoperative day. Despite numerous presentations of LH in the adult age group in the literature, there are limited data about the disease in the childhood period. Even though the scarcity of the data, there are some so-called rules for LH in childhood like protruding more common through umbilical hernias, containing heterotopic tissues more frequent than adult age and incarcerating/strangulating more often. The aim of this study is to review the reported LH cases, present a new case and discuss the features of LH in childhood period.

Keywords: Children, littre hernia, meckel′s diverticulum in hernial sac

How to cite this article:
Pampal A, Aksakal ED. Littre hernia in childhood: A case report with a brief review of the literature. Afr J Paediatr Surg 2011;8:221-4

How to cite this URL:
Pampal A, Aksakal ED. Littre hernia in childhood: A case report with a brief review of the literature. Afr J Paediatr Surg [serial online] 2011 [cited 2019 Sep 19];8:221-4. Available from: http://www.afrjpaedsurg.org/text.asp?2011/8/2/221/86068

   Introduction Top


Littre hernia (LH) is the protrusion of a Meckel diverticulum (MD) through a potential abdominal opening with a container sac. This clinical entity has been described after the autopsy findings of two patients by Alexis de Littre for the first time as 'ileal diverticula in the inguinal hernia' in 1700. But the concept of "Littre hernia" has only been improved after a century, as J Frederich Meckel defined the true congenital diverticulum of the gut which is now entitled with his name. [1]

MD commonly complicates before the age of 2 and because of this reason, it is accepted as a childhood disease. In 75% of the patients, as the tip of MD is free in the abdomen it is actually more prone to protrude through any abdominal opening. [1] But this presentation is quite rare in childhood and the true incidence of LH is unknown. Despite numerous presentations of LH in the adult age group in the literature, there are limited data about the disease in the childhood period. Albeit the lack of informative data, there are some so-called rules for LH in childhood like protruding more common through umbilical hernias, containing heterotopic tissues more frequent than adult age and incarcerating/strangulating more often. [1]

The review of English literature of LH in the childhood period from the beginning of the 20 th century revealed 69 cases in case reports [Table 1] and case series [Table 2]. [2],[3],[4],[5],[6],[7],[8],[9],[10],[11],[12],[13],[14],[15],[16],[17],[18],[19],[20],[21],[22],[23],[24],[25],[26],[27],[28],[29],[30],[31],[32],[33],[34] The aim of this study is to review these cases, present a new case and discuss the features of LH in the childhood period.


   Case Report Top


A 3-year-old boy with a right-sided and painful inguinal swelling for the last 2 h was admitted to the emergency department. He was neither complaining of nausea/vomiting nor did have either episode of gastrointestinal bleeding or intestinal obstruction. The physical examination revealed a right-sided incarcerated inguinal hernia with no apparent peritoneal irritation findings. After the manual reduction, the patient was scheduled for an elective open herniorrhaphy. Perioperatively the widened hernial sac was dissected through the deep inguinal ring. As the sac was opened in order to inspect its content, the MD was seen. It was found adherent to the base of the hernial sac circumferentially [Figure 1]. The diverticulum was dissected from the hernial sac and high ligation of the hernia sac was completed after the wedge resection of the diverticulum and anastomosis. The intestinal contents were addressed to the intraperitoneal cavity. The patient was fed on the second postoperative day and discharged on the third postoperative day. The histopathological findings revealed MD with an ectopic pancreatic mucosa.


   Discussion Top


LH is the protrusion of a MD through any potential abdominal opening. It is a rare perioperative finding in adults with an incidence of 4-10% and even rarer in the childhood period. [1]

This literature review containing the presented case divulged 70 cases of LH from early 1900s to these days and by the help of these cases we aimed to discuss the features of LH in childhood. LH seems to be a problem of infancy generally in male gender. Fifty one of these patients presented with LH in the infancy period and the vast majority of these case reports and case series are composed of male gender. Even though LH can be seen at any age, the infancy period seems to be the most risky period. It is probably attributed to the tendency of incarceration at this age group.

Even though the half of the LH is located in the inguinal region in the adult age, the most common site is accepted as 'umbilical hernia' for children with a reported incidence of 85%. [1] This literature review revealed that not solely umbilical hernias but any kind of persisting umbilical openings (including umbilical hernias, umbilical cord hernias, exomphalos minor and major) are the most common sites for LH. This common location is due to the foetal umbilicus being a component of omphalomesenteric duct. [1] Apart from the previous reports, the prevalence of this presentation is found lower in this series like 64.7%. (44/68; as total number of the cases with known location of LH is 68). The second common site is the inguinal region with a prevalence of 33.8% in all age groups (23/68). Diverse from adulthood, the protrusion from the femoral region seems negligible in childhood. That is probably due to the rarity of femoral hernias in the childhood period. Most interesting site for protrusion of MD in this series is through a diaphragmatic hernia in a baby. [17]

Despite the difficulty of preoperative diagnosis of a LH, findings like incomplete manual reduction of an incarcerated hernia, hernial faecal fistulas and previous history of rectal bleeding were accepted to alert the clinician about a LH. [14] Even though the manual reduction was the procedure of choice in most of the patients in this series, it was found to be not completed in four patients and failed in three. Only three patients of this series presented faecal fistulas. The history of rectal bleeding or repetitive abdominal pain was not present in any of the cases in this review.

The complication rate of LH in this series is about 38.5%. (27/70). Twenty four of these patients incarcerated and one of the incarcerated patient experienced intra-abdominal perforation. Three of the patients in the complication group presented with faecal fistula, one patient through an umbilical hernia and two babies through exomphalos sacs. No other sides for faecal fistulas were reported even in cases who admitted hospital lately. In terms of fistula formation, umbilical region seems to be at risk for when compared to the other abdominal openings. It is not only due to the absence of muscular layers but also due to the lesser amount of subcutaneous tissue at the umbilicus in children.

The presence of heterotopic mucosa of LH is a rare finding in this series and 66.7% of the cases with heterotopic mucosa (4/6) were found to be complicated either by adhering to the adjacent tissues or by perforating. Six cases presented with adhesions to the adjacent tissues and pathological findings of half of these patients revealed heterotopic mucosa. The formation of adhesions is said to be due to either inflammatory processes related to the heterotopic mucosa or congestion and exudation due to closed loop obstruction in heterotrophy free MDs. [7],[14]

In conclusion, the anamnesis of a LH is no longer different from any hernia whether complicated or not. While evaluating a newborn with a congenital abdominal wall defect, a MD through any umbilical opening should be kept in mind especially for the ones with small defects (exomphalos minor and umbilical cord hernia). Quick decision and early intervention in such cases could preclude perforation and related faecal fistula formation. Meanwhile while evaluating a child beyond newborn period with an incarcerated hernia, an incomplete or failed manual reduction should bring forth the possibility of a LH. Furthermore, querying the history of rectal bleeding or repetitive abdominal pain in such cases should increase the suspicion of a LH.

 
   References Top

1.Skandalakis PN, Zoras O, Skandalakis JE, Mirilas P. Littre hernia: Surgical anatomy, embryology, and technique of repair. Am Surg 2006;72:238-43.  Back to cited text no. 1
    
2.Bunts FE. Meckel's Diverticulum: With Report of Strangulated Inguinal Hernia of Same. Ann Surg 1904;40:536-54.   Back to cited text no. 2
    
3.Bird WE. Littre's umbilical hernia. Case report. Am J Surg 1943;60:81-7.  Back to cited text no. 3
    
4.Rendle-Short J, Havard C. Incarcerated and strangulated inguinal hernia in the first year of life; report on 45 cases. Br Med J 1954;1:680-2.  Back to cited text no. 4
    
5.Davis CE Jr. Littre's hernia; report of two cases. Ann Surg 1954;139:370-3.  Back to cited text no. 5
    
6.Kline AH. Incarceration of Meckel's diverticulum in an inguinal hernia. J Pediatr 1958;53:479-80.   Back to cited text no. 6
    
7.Baillie RC. Incarceration of a Meckel's inguinal hernia in an infant. Br J Surg 1959;46:459-61.  Back to cited text no. 7
    
8.Wollgast GF, Hilz JM. Littre's hernia: Strangulation of Meckel's diverticulum in a femoral hernia and an inguinal hernia. Am Surg 1962;28:741-4.  Back to cited text no. 8
    
9.Gracey LR, Williams JA. Meckel's diverticulum in children. Br J Clin Pract 1963;17:315-8.  Back to cited text no. 9
    
10.Rutherford RB, Akers DR. Meckel's diverticulum: A review of 148 pediatric patients, with special reference to the pattern of bleeding and to mesodiverticular vascular bands. Surgery 1966;59:618-26.  Back to cited text no. 10
    
11.Kabbani SS, Lewis JE Jr. Strangulated hernia of Meckel's diverticulum in an infant. J Pediatr Surg 1966;1:579-82.  Back to cited text no. 11
    
12.Kremer RM, Morton JH. Strangulated Littre's umbilical hernia. Am Surg 1968;34:432-5.  Back to cited text no. 12
    
13.Krausz M, Rubin S, Schiller M. Infantile strangulated Littré's hernia with a gangrenous homolateral testis: Report of a case. Aust N Z J Surg 1974;44:45-7.  Back to cited text no. 13
    
14.Mishalany HG, Pereyra R, Longerbean JK. Littre's hernia in infancy presenting as undescended testicle. J Pediatr Surg 1982;17:67-9.   Back to cited text no. 14
    
15.Ravikumar K, Khope S, Ganapathi BP. Littre's hernia in a child--an operative surprise (a case report). J Postgrad Med 1989;35:112-3.  Back to cited text no. 15
[PUBMED]  Medknow Journal  
16.Das PC, Rao PL, Radhakrishna K. Meckel's diverticulum in children. J Postgrad Med 1992;38:19-20.  Back to cited text no. 16
[PUBMED]  Medknow Journal  
17.Kang IS, Ahn SM, Han A, Oh JT, Han SJ, Choi SH, et al. Giant Meckel's diverticulum associated with a congenital diaphragmatic hernia. Yonsei Med J 2004;45:177-9.  Back to cited text no. 17
    
18.Messina M, Ferrucci E, Meucci D, Di Maggio G, Molinaro F, Buonocore G. Littre's hernia in newborn infants: Report of two cases. Pediatr Surg Int 2005;21:485-7.  Back to cited text no. 18
    
19.Chirdan LB, Uba AF, Kidmas AT. Incarcerated umbilical hernia in children. Eur J Pediatr Surg 2006;16:45-8.   Back to cited text no. 19
    
20.Vaos G. Perforation of an inguinoscrotal Littre's hernia due to Meckel's diverticulitis: An unusual cause of acute hemiscrotum. Acta Paediatr 2006;95:251-2.  Back to cited text no. 20
    
21.Ein SH, Njere I, Ein A. Six thousand three hundred sixty-one pediatric inguinal hernias: A 35-year review. J Pediatr Surg 2006;41:980-6.  Back to cited text no. 21
    
22.Chan KW, Lee KH, Mou JW, Cheung ST, Tam YH. The use of laparoscopy in the management of Littre's hernia in children. Pediatr Surg Int 2008;24:855-8.  Back to cited text no. 22
    
23.Al-Mandil M, Khoury AE, El-Hout Y, Kogon M, Dave S, Farhat WA. Potential complications with the prescrotal approach for the palpable undescended testis? A comparison of single prescrotal incision to the traditional inguinal approach. J Urol 2008;180:686-9.  Back to cited text no. 23
    
24.Akin M, Kurukahvecioglu O, Tezcaner T, Anadol AZ, Onur K. Acute hemiscrotum due to inguinoscrotal Littre hernia: A case report. Am J Emerg Med 2008;26:973.e3-4.  Back to cited text no. 24
    
25.Komlatsè AN, Komla G, Komla A, Azanledji BM, Abossisso SK, Hubert T. Meckel's diverticulum strangulated in an umbilical hernia. Afr J Paediatr Surg 2009;6:118-9.  Back to cited text no. 25
    
26.Williams C. Unusual Surgical Lesions of the Umbilicus: Report of Cases of Congenital Origin. Ann Surg 1946;124:1108-24.  Back to cited text no. 26
    
27.Aitken J. Exomphalos. Analysis of a 10-year series of 32 cases. Arch Dis Child 1963;38:126-9.  Back to cited text no. 27
    
28.Nicol JW, MacKinlay GA. Meckel's diverticulum in exomphalos minor. J R Coll Surg Edinb 1994;39:6-7.  Back to cited text no. 28
    
29.Wakhlu A, Wakhlu AK. The management of exomphalos. J Pediatr Surg 2000;35:73-6.  Back to cited text no. 29
    
30.Hale PC, Agrawal M. Congenital fistulation of a Meckel's diverticulum to the surface of an exomphalos sac. Br J Clin Pract 1993;47:273.  Back to cited text no. 30
    
31.Tourne G, Chauleur C, Varlet MN, Tardieu D, Varlet F, Seffert P. Prenatal discovery of an omphalocele associated with an inner umbilical cord Meckel's diverticulum. J Matern Fetal Neonatal Med 2007;20:427-30.   Back to cited text no. 31
    
32.Kumar HR, Jester AL, Ladd AP. Impact of omphalocele size on associated conditions. J Pediatr Surg 2008;43:2216-9.   Back to cited text no. 32
    
33.Mavridis G, Livaditi E, Vassiliadou E, Christopoulos-Geroulanos G. Intrauterine fistulation of perforated Meckel's diverticulum to the surface of the sac of an intact exomphalos minor. Minerva Pediatr 2008;60:253-4.  Back to cited text no. 33
    
34.Pal K, Ashri H, Al Wabari A. Congenital hernia of the cord. Indian J Pediatr 2009;76:319-21.  Back to cited text no. 34
    

Top
Correspondence Address:
Arzu Pampal
Ufuk Universitesi Tip Fakultesi, Cocuk Cerrahi Bolumu, Konya Yolu No:86-88, Balgat 06520 Ankara
Turkey
Login to access the Email id

Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0189-6725.86068

Rights and Permissions


    Figures

  [Figure 1]
 
 
    Tables

  [Table 1], [Table 2]

This article has been cited by
1 Diverse Presentations in Pediatric Meckelęs Diverticulum: A Review of 100 Cases
Chen-Chuan Huang,Ming-Wei Lai,Fang-Ming Hwang,Yu-Chen Yeh,Shih-Yen Chen,Man-Shan Kong,Jin-Yao Lai,Jeng-Chang Chen,Yung-Ching Ming
Pediatrics & Neonatology. 2014;
[Pubmed] | [DOI]



 

Top
 
 
  Search
 
    Similar in PUBMED
   Search Pubmed for
   Search in Google Scholar for
 Related articles
    Email Alert *
    Add to My List *
* Registration required (free)  
 


    Abstract
   Introduction
   Case Report
   Discussion
    References
    Article Figures
    Article Tables

 Article Access Statistics
    Viewed3220    
    Printed150    
    Emailed0    
    PDF Downloaded171    
    Comments [Add]    
    Cited by others 1    

Recommend this journal