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CASE REPORT Table of Contents  
Ahead of print publication
Management of an inguinal mixed Littré hernia and incidental cryptorchidism: A case report


1 Surgery Department, Bernardino Rivadavia Hospital, Buenos Aires, Argentina
2 Early Cancer Detection Center, Max Peralta Hospital, Cartago, Costa Rica
3 Emergency Department, Bernardino Rivadavia Hospital, Buenos Aires, Argentina

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Date of Submission11-Feb-2022
Date of Decision14-Mar-2022
Date of Acceptance16-Mar-2022
Date of Web Publication14-Apr-2022
 

  Abstract 

INTRODUCTION: Littré hernia is defined as the presence of a Meckel’s diverticulum in any hernia sac. The case of an adult with Littré hernia associated with a cryptorchidic testicle in the inguinal canal has not been previously reported. Treatment of this rare case is controversial on many fronts. This report highlights the management of a case with an inguinal mixed Littré hernia and incidental cryptorchidism. CASE PRESENTATION: A 32-year-old male patient with an incarcerated right inguinal hernia presented to the emergency department. An incarcerated mixed Littré hernia was discovered associated with a cryptorchidic testicle. A Lichtenstein hernioplasty and an orchidopexy were performed without resection of Meckel’s diverticulum. DISCUSSION: There is currently no consensus on the treatment of a Littré hernia nor incidental cryptorchidism in an adult patient. Controversies arise on whether to perform diverticulectomy or not and the type of hernia repair. This case had the added unique feature of an undiagnosed cryptorchidic testicle in an adult, a pathology that also prolongs controversies on whether it is necessary to resect. CONCLUSION: Treatment of a Littré hernia, Meckel’s diverticulum, and cryptorchidism in adult patients continues to be a challenge. Given the lack of guidelines that establish appropriate treatment, it must be decided on a case by case basis; however, a conservative approach seems to be safe.

Keywords: Cryptorchidism, inguinal hernia, Meckel’s diverticulum, mixed Littré hernia


How to cite this URL:
Gamboa Miño SN, Zeledón ME, Solari AJ, Alcántara GH. Management of an inguinal mixed Littré hernia and incidental cryptorchidism: A case report. Int J Abdom Wall Hernia Surg [Epub ahead of print] [cited 2022 Jul 1]. Available from: http://www.herniasurgeryjournal.org/preprintarticle.asp?id=343316





  Key Messages: Top


Mixed Littré hernias are rare occurrences and their management is determined according to the individual case findings.

Resection of Meckel’s diverticulum should be reserved only in cases with doubts of tissue viability or the presence of ectopic tissue in the diverticulum.

Preservation of a cryptorchid testicle is a viable option if there are no doubts on the absence of neoplasia or tissue viability.


  Introduction Top


A Meckel’s diverticulum is a vestigial remnant of the omphalomesenteric duct representing the most common congenital malformation of the gastrointestinal tract. It is found in 0.3%–3.0% of the adult population.[1],[2],[3] A hernia containing Meckel’s diverticulum is called a Littré hernia and 1% of patients having a Meckel’s diverticulum will develop a Littré hernia.[1],[3] A recent systematic review identified 53 adult cases of Littré hernia in the literature.[3] It is a rare condition whose incidence is unknown. The concurrent presence of other abdominal viscera signifies a mixed Littré hernia.[1],[4]

Meckel’s diverticulum develops as a result of incomplete atrophy of the omphalomesenteric duct.[5] It is usually found 20–90 cm proximal to the ileocecal valve.[6] The treatment of Meckel’s diverticulum remains controversial. Some authors report a complication rate of 8.9% after diverticulectomy in asymptomatic cases, opting for non-resection, whereas others suggest surgical treatment, even in asymptomatic cases.[6],[7],[8],[9]

Cryptorchidism is a pathological condition defined as the failure of the testis descent to the scrotum. It is one of the most common congenital anomalies, occurring in 1%–4% of full-term male neonates.[10] The prevalence of cryptorchidism in adults is about 1% at 1 year of age. It is recognized that in children, the cryptorchid testicle should be reduced between the age of 6 and 12 months. In adults, however, there is no consensus. Orchidopexy has been the management for most patients.[11]

This report highlights the management of a 32-year-old man with a right inguinal incarcerated mixed Littré hernia and incidental cryptorchidism.


  Case History Top


A 32-year-old male patient with a 1-year history of right inguinal hernia presented to the emergency department with a 4-h history of right inguinal pain associated with vomiting. On physical examination, an irreducible and painful mass of 8 × 8 cm was observed in the right inguinal region, with no signs of inflammation. Laboratory results were normal. Symptomatic treatment was unsuccessful, so surgical treatment was elected. Informed consent was obtained from the patient.

The patient was placed in the supine position on the operating table. A right inguinal incision was performed, and dissection of the inguinal canal was performed as usual. The protrusion of 6 × 6 cm mass through the superficial inguinal ring was found. The spermatic cord was dissected, and a permeable vaginal peritoneum ductus was observed and opened. Intraoperatively, the inguinal hernia sac was opened, and the presence of an incompletely descended testis and small bowel was found. The small bowel had no signs of inflammation or necrosis [Figure 1] but presented a Meckel’s diverticulum. The diverticulum was broad based and had no signs of necrosis or inflammation. The small bowel was confirmed intact, without any evidence of compromise, and was repositioned in the abdominal cavity, along with the Meckel’s diverticulum. No resection was performed. Ectopic testis was vital, without signs of ischemia or malignancy; therefore, pexia in the scrotal pouch was decided. Spermatic cord was unscathed. Finally, a Lichtenstein hernioplasty was performed to repair the hernia. Surgery time was 1.5 h, starting at 1 am.
Figure 1: Meckel’s diverticulum without necrosis, with a broad base

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The patient progressed without complications. He was discharged from the hospital 2 days after the procedure. He was monitored for 6 months on an outpatient basis with no complications.


  Discussion Top


Management of Littré hernias poses therapeutic dilemmas as to whether diverticulectomy should routinely be performed, regardless of patient symptomatology.[1]

Some authors suggest performing diverticulectomy in addition to repairing the hernia (with or without mesh).[12] However, there is a broad consensus that hernia repair should include a mesh in patients over 30 years.[13]

Regarding the treatment of Meckel’s diverticulum, Soltero and Bill suggest that in the event of an incidental Meckel’s diverticulum, the risk of postoperative morbidity associated with resection and the low risk of non-resection justify a conservative approach.[6] Some surgeons advice against prophylactic resection arguing that morbidity rate is too high and that the reward is too low. Zani et al. found a 5.3% risk of postoperative complications after prophylactic resection and a 1.3% risk of developing symptoms (without increasing late complications) after leaving Meckel’s diverticulum in situ.[9]

On the other hand, Cullen et al. believe that resection should be performed in all cases, including incidental ones, because the incidence of postoperative complications after prophylactic resection is low.[8] Other authors describe specific criteria for resection; consequently, cases with a broad base or a short length of the diverticulum can be preserved.[9] Evola et al. concluded that resection of uncomplicated Meckel’s diverticulum should be based on identified risk factors because of possible complications following its resection. Park et al. found that patients younger than 50 years, male, diverticulum length greater than 2 cm, and ectopic or abnormal features within a diverticulum were all associated with symptomatic diverticula.[5],[14],[15],[16]

In our case, it was a broad-based Meckel’s diverticulum, which, although it had a length greater than 4 cm, showed no signs of inflammation, necrosis, or malignancy. If Meckel’s diverticulum had been resected, the possible field contamination would have made the use of a mesh difficult. This could be a risk factor resulting in the appearance of a recurrent hernia. The resection of Meckel’s diverticulum could increase possible complications such as surgical site infection, intra-abdominal abscess, anastomosis dehiscence, sepsis, and intestinal obstruction.[9],[17]

An open hernia repair was decided, considering the time of the surgery and the lack of prior experience of the surgical team with laparoscopic hernia repair. The laparoscopic approach would have taken more time.

The laparoscopic hernia repair approach has been criticized because of its technical complexity, in addition to its associated complications in the early stages of the learning curve.[18] Although the laparoscopic repair was associated with longer surgical time and a higher recurrence rate, it appeared to be a safe and effective operating technique for patients with incarcerated inguinal hernia.[19]

However, open hernia repair showed less testicular pain, shorter surgical time, and less recurrence rate. Both open and laparoscopic approaches were similar concerning chronic pain, seromas, wound infection, and neuralgia.[18]

Patterson et al. concluded that laparoscopic hernia repair may not have such a significant advantage over open hernia repair in certain situations.[20]

Few cases of cryptorchidism have been reported in adults, and its treatment remains controversial. Some authors recommend orchidectomy due to the pathological findings in the biopsy of the testis, whereas others prefer orchidopexy because they have found functional testis even in adult patients.[10] In our case, the testis did not present features of malignancy, so we opted for orchidopexy.


  Conclusion Top


Treatment of Littré hernia and cryptorchidism in adult patients continues to be a challenge. The finding of a mixed Littré Hernia, in association with a cryptorchidic testicle, has not been previously reported to the best of our knowledge. In the cases of Littré hernia with Meckel’s diverticulum with no signs of inflammation or necrosis, a broad-based diverticulum could be preserved. Lichtenstein hernioplasty would be the most suitable option. Orchidopexy is preferred over orchiectomy in testicles without signs of malignancy. Given the lack of guidelines that establish the appropriate treatment, it must be decided on a case by case basis.

Declaration of patient consent

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

Acknowledgment

The authors would like to thank Doctor and Professor Ho-Young Song (South Korean radiologist) who has been a very important person for the elaboration of this article. He taught us that you can always learn more, no matter the time.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Schizas D, Katsaros I, Koliakos N, Ntomi V, Tsilimigras DI, Moris D, et al. An alternative management of a Littré hernia case: Food for thought. G Chir 2019;40:360-3.  Back to cited text no. 1
    
2.
Hansen CC, Søreide K. Systematic review of epidemiology, presentation, and management of Meckel’s diverticulum in the 21st century. Medicine (Baltimore) 2018;97:e12154.  Back to cited text no. 2
    
3.
Schizas D, Katsaros I, Tsapralis D, Moris D, Michalinos A, Tsilimigras DI, et al. Littre’s hernia: A systematic review of the literature. Hernia 2019;23:125-30.  Back to cited text no. 3
    
4.
Johnson GGRJ, Holden J, Helewa RM, Hochman D, Hyun E. A strangulated Meckel’s diverticulum in an inguinal hernia: A case report and literature review. Int Med Case Rep J 2021;14:605-9.  Back to cited text no. 4
    
5.
Usman A, Rashid MH, Ghaffar U, Farooque U, Shabbir A. Littré’s hernia: A rare intraoperative finding. Cureus 2020;12:e11065.  Back to cited text no. 5
    
6.
Soltero MJ, Bill AH. The natural history of Meckel’s diverticulum and its relation to incidental removal. A study of 202 cases of diseased Meckel’s diverticulum found in King County, Washington, over a fifteen year period. Am J Surg 1976;132:168-73.  Back to cited text no. 6
    
7.
Karaman A, Karaman İ, Çavuşoağlu YH, Erdoağan D, Aslan MK. Management of asymptomatic or incidental Meckels diverticulum. Indian Pediatr 2010;47:1055-7.  Back to cited text no. 7
    
8.
Cullen JJ, Kelly KA, Moir CR, Hodge DO, Zinsmeister AR, Melton LJ 3rd. Surgical management of Meckel’s diverticulum. An epidemiologic, population-based study. Ann Surg 1994;220:564-8.  Back to cited text no. 8
    
9.
Zani A, Eaton S, Rees CM, Pierro A. Incidentally detected Meckel diverticulum: To resect or not to resect? Ann Surg 2008;247: 276-81.  Back to cited text no. 9
    
10.
Jeong SC, Lee S, Ku JY, Lee SD. Clinical characteristics and treatment of cryptorchidism in adults: A single-center experience. World J Mens Health 2014;32:110-5.  Back to cited text no. 10
    
11.
Avakoudjo J, Hodonou F, Sewa EV, Tandje Y, Sossa J, Yevi M, et al. Undescended testis in adult: Epidemiology and therapeutic aspects. About 23 cases in HKM University Teaching Hospital of Cotonou. Open Journal of Urology 2018;8:206-13.  Back to cited text no. 11
    
12.
Horkoff MJ, Smyth NG, Hunter JM. A large incarcerated Meckel’s diverticulum in an inguinal hernia. Int J Surg Case Rep 2014;5: 899-901.  Back to cited text no. 12
    
13.
Simons MP, Aufenacker T, Bay-Nielsen M, Bouillot JL, Campanelli G, Conze J, et al. European Hernia Society guidelines on the treatment of inguinal hernia in adult patients. Hernia 2009;13:343-403.  Back to cited text no. 13
    
14.
Evola G, Piazzese E, Bonanno S, Di Stefano C, Di Fede GF, Piazza L. Complicated Littre’s umbilical hernia with normal Meckel’s diverticulum: A case report and review of the literature. Int J Surg Case Rep 2021;84:106126.  Back to cited text no. 14
    
15.
Park J, Wolff BG, Tollefson MK, Walsh EE, Larson DR. Meckel diverticulum: The Mayo Clinic experience with 1476 patients (1950–2002). Ann Surg 2005;241:529-33.  Back to cited text no. 15
    
16.
Robijn J, Sebrechts E, Miserez M. Management of incidentally found Meckel’s diverticulum a new approach: Resection based on a risk score. Acta Chir Belg 2006;106:467-70.  Back to cited text no. 16
    
17.
Evola G, Caramma S, Caruso G, Schillaci R, Reina C, Reina GA. Intestinal obstruction and ischemia by necrotic annular Meckel’s diverticulum: Case report and review of the literature. Int J Surg Case Rep 2021;82:105897.  Back to cited text no. 17
    
18.
Bobo Z, Nan W, Qin Q, Tao W, Jianguo L, Xianli H. Meta-analysis of randomized controlled trials comparing Lichtenstein and totally extraperitoneal laparoscopic hernioplasty in treatment of inguinal hernias. J Surg Res 2014;192:409-20.  Back to cited text no. 18
    
19.
Kepičová M, Ihnát P. Laparoscopic versus open hernia repair in patients with incarcerated inguinal hernia. Rozhl Chir 2021;100:440-4.  Back to cited text no. 19
    
20.
Patterson TJ, Beck J, Currie PJ, Spence RAJ, Spence G. Meta-analysis of patient-reported outcomes after laparoscopic versus open inguinal hernia repair. Br J Surg 2019;106:824-36.  Back to cited text no. 20
    

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Correspondence Address:
Sofía N Gamboa Miño,
Surgery Department, Bernardino Rivadavia Hospital, Bulnes 1814, Suite C Floor 10, Palermo, Buenos Aires C1425DKF
Argentina
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Source of Support: None, Conflict of Interest: None



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