|Year : 2021 | Volume
| Issue : 3 | Page : 83-89
Complicated Littre hernias
Mariana Rita Afonso Matias1, Madara Kronberga2, Assad Aghahoseini1
1 General Surgery, York Teaching Hospital, NHS Foundation Trust, UK
2 General Surgery, University Hospitals Birmingham, NHS Foundation Trust, UK
|Date of Submission||08-Apr-2021|
|Date of Decision||12-Jul-2021|
|Date of Acceptance||05-Aug-2021|
|Date of Web Publication||30-Sep-2021|
Dr. Mariana Rita Afonso Matias
8 Forest Yard, Middleton, LS10 4WN Leeds.
Source of Support: None, Conflict of Interest: None
BACKGROUND: Meckel’s diverticulum (MD) is the most prevalent congenital anomaly of the gastrointestinal tract with an estimated prevalence ranging from 2% to 4%. The diverticulum is a remnant of the vitelline duct, which leads to the formation of a true diverticulum containing all the layers of the small intestine. The condition is usually clinically silent; however, it can present with unusual symptoms and signs and therefore can result in a delayed diagnosis, particularly in adults. The aim of this study is to review the most updated literature reporting Littre hernias (LHs) in adults, the diagnostic approach, and its subsequent management. This is a literature review, complemented by an unusual clinical case of an adult who presented with a complicated umbilical LH which required emergency surgery. MATERIALS AND METHODS: Literature reviews using PubMed, Web of Science, and other reliable sources use the possible combinations of the following keywords: Meckel’s diverticulum, Littre hernia, umbilical hernia, adults, complications, surgical treatment. RESULTS: The herniation of an MD through a potential opening in the abdominal wall is defined as an LH, representing a rare complication of the diverticula. Due to their rarity, they remain underdiagnosed, often leading to suboptimal treatment with substantial morbidity and mortality. CONCLUSION: An MD, despite being a rare clinical entity in adults, can theoretically be found in any hernia, with patients experiencing a wide range of potential complications.
Keywords: Adults, complications, Littre hernia, Meckel’s diverticulum, surgical treatment, umbilical hernia
|How to cite this article:|
Matias MA, Kronberga M, Aghahoseini A. Complicated Littre hernias. Int J Abdom Wall Hernia Surg 2021;4:83-9
| Introduction|| |
Hernia is a general term describing tissue or organ protrusion through an abnormal opening between anatomical structures. Abdominal wall hernias comprise approximately 75% of all hernias and occur when abdominal contents push through a weak point in the abdominal wall disrupting the “corset” effect of the abdominal wall muscles., There are several abdominal wall areas where there is an inherit weakness compared with others; however, hernias may occur anywhere between the chest and the pelvis. Several different conditions and aspects precipitate development of a hernia; however, the main factors leading to formation of abdominal wall hernias are increased abdominal pressure and abdominal wall weakness.,
Hernias can be classified depending on the protrusions location and/or its contents. Inguinal hernias are by far the most common of all abdominal wall hernias. Most common content found in the inguinal hernia sac is peritoneal fat or small bowel; however, hernias containing specific anatomical structures have been described by and named after several authors.
The definition of Littre hernia (LH) includes all types of hernias that contain a Meckel’s diverticulum (MD) within the hernia sac as a result of a diverticulum projection through a weakness in the abdominal wall. It was first described at the beginning of the 18th century by the French physician, anatomist, Alexis de Littre. Following the autopsy findings of two patients, he reported an “ileal diverticula in the inguinal hernia” and accredited it to traction.,,, This anatomic variant—MD—despite being initially identified by Fabricius Hildanaus in 1598—was officially named after Johann Friedrich Meckel, in 1809, after publishing a detailed description of the embryological origin of the diverticulum and its clinical features.,,
MD results from an inadequate obliteration of the proximal portion of the omphalomesenteric duct (also called the vitellointestinal duct), which links the embryonic digestive tract and yolk sac., Albeit its precise anatomic localization being quite variable, it is typically located on the antimesenteric border of the ileum, approximately 90 cm from the ileocecal valve,, and is defined as a true diverticulum as it encompasses the three intestinal layers.,,
As a congenital variant, MD is the most prevailing inherited abnormality of the gastrointestinal tract, commonly found before the age of 2 years., MD incidence in the adult population varies between 2% and 4% and is often not associated with any specific symptoms. The literature reports that only 4%-6% of the patients known to have MD experience related complications, and these occur more frequently during childhood. The leading complication in adults is bowel obstruction, often secondary to internal adhesions or hernias.
LH is a rare complication of the diverticula, and although it has been rarely witnessed in Meckel’s cases, its exact frequency remains unknown. This clinical entity must not be mistaken for a Richter’s hernia (RH) first scientifically described by August Gottlob Richter in 1778 as containing only part of the circumference of the bowel. As opposed to an LH, in an RH only the antimesenteric wall of the bowel is contained inside the hernia sac.
Bakal et al. stated that an LH can present as an inguinal, umbilical, or femoral hernia, mostly located on the right side as an inguinal hernia. However, these findings can be arguable, as they were made based on fairly small pediatric case series.
As this clinical entity is so uncommon in the adult population, it is often misdiagnosed when first assessed. However, due to the increasing number of diagnostic laparoscopies performed worldwide and the use of scintigraphy with technetium-99m pertechnetate-labeled red blood cells, the preoperative diagnostic accuracy of the condition has improved.
The emphasis of this article is on adult LHs, illustrated with a rare clinical case of a strangulated umbilical LH, to re-enforce the available literature and stimulate the scientific sharing of adult cases, clinical presentations, and treatment approaches.
| Materials and Methods|| |
We conducted a review of the recent literature using PubMed, Web of Science, and other reliable sources using the possible combinations of the following keywords: Meckel’s diverticulum, Littre hernia, umbilical hernia, adults, complications, and surgical treatment.
In this literature review, we included all original articles, written in English, with none more than 20 years old, in which LHs in adults were the key point, along with clinical assessment and surgical practice. Non-English papers, conference abstracts, and articles based on animal studies and autopsy specimens were excluded from this literature review.
The literature search yielded 52 articles, which were narrowed down after title and abstract screening. A total set of 38 full-text papers fulfilled our inclusion criteria and therefore were included in this review.
| Case Report|| |
A 75-year-old Caucasian male with a background of hypertension, chronic kidney disease, and prostate cancer presented to the hospital with a 3-day history of painful umbilical swelling. The patient described the pain as cramp-like that was relieved after vomiting. He had no history of previous abdominal surgery and no relevant family history.
On admission, he was apyrexial and hemodynamically stable. Examination of the abdomen showed marked distension with a protruding and irregular umbilical mass. The overlying skin was indurated, erythematous, and tender on palpation. There was generalized tenderness but without rebound tenderness or guarding. The umbilical mass was found to be irreducible.
Routine blood tests have shown leucocytosis (elevated neutrophils) with a mildly raised C-reactive protein and normal lactate. Due to his poor renal function, we organized a non-contrast computed tomography (CT) scan of the abdomen and pelvis that revealed an umbilical hernia with hyperdense content. Unfortunately, due to the low sensitivity of the unenhanced CT study, a strangulated bowel loop could not be excluded. The decision to therefore undergo urgent surgical intervention was made.
Intraoperatively, the umbilical defect was exposed. The hernia sac was opened and on further dissection, an MD was found firmly adherent to the strangulated hernial sac [Figure 1]. The ileal loop connected with the MD appeared discolored with a grey hue, indicating some degree of strangulation. We, therefore, proceeded with segmental resection of the ileal loop carrying the diverticulum and subsequent primary side-to-side ileoileal stapled anastomosis. The remaining bowel was carefully examined and was reduced into the abdomen. Lastly, a herniorrhaphy was performed for the fascial defect.
|Figure 1: Intraoperative specimen photograph. MD adherent to thick hernia sac strangulating diverticulum and part of the small bowel|
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The histopathology report later confirmed the operative findings, with small bowel showing an outpouching of 50 × 50 × 50 mm consistent with MD, extensive mixed inflammation, fibrosis, and hemorrhage in the wall, with mucosal ischemia.
Postoperative recovery was uneventful, the patient was deemed medically fit for discharge 72 h following surgery with the restoration of the transit and good healing of the wound.
| Discussion|| |
Different types of abdominal hernias have been reported and named based on the structures found inside the hernia sac: LH (contains an MD), RH (contains anti-mesenteric wall of bowel), and Amyand’s hernia (contains an appendix). August Gottlieb Richter (1785) first reported a hernia with partial enterocele. However, Sir Frederic Treves (1897) was the one describing the differences between RH and LH. An LH containing only an MD is labeled true LH, whereas the co-existence of small bowel and any other abdominal viscera in the hernia sac is titled a mixed LH.
An LH is an uncommon complication of MD and it is a result of its projection through a potential abdominal opening. Although its incidence is to date unknown, some studies have suggested that less than 1% of patients known to have an MD may later present with an LH.,,,
MD is a congenital malformation of the gastrointestinal tract and is routinely referred to by the “rule of 2s”: the anomaly is found in about 2% of the population, it is roughly 2 inches in length, it is normally located within 2 feet of the ileocecal valve, it is commonly diagnosed in children under 2 years, and it seems to affect males twice as often as females.,,,, The often-described anatomical location may explain Kakal et al.’s conclusion toward favoring the right-sided hernia when inguinal LH occurs. While the use of these “rules” can be helpful for clinical daily use, they are not based on accurate data.
Embryology and anatomy
MD was first reported in 1809 by Johann Friedrich Meckel as a congenital intestinal blind pouch resulting and postulated their embryologic origin as a persistent omphalomesenteric duct. The genesis of an MD can be explained by the intrauterine growth of the bowel. During the normal fetal development, the omphalomesenteric duct is responsible for the vinculum between the yolk sac and the undeveloped gastrointestinal tract and usually obliterates during the 5th to 7th week of gestation., Embryologically, an MD is formed when the duct fails to obliterate leading to numerous potential complications, such as an omphalomesenteric fistula, an umbilical sinus, an enterocyst, residual fibrous bands attaching the bowel to the umbilicus, and most commonly to an MD.,,
Anatomically, the MD is described as a real intestinal diverticulum that contains the three layers of the intestinal wall and its own blood supply from a remnant of the vitelline artery, a branch from the superior mesenteric artery., The diverticulum is typically discovered on the anti-mesenteric border of the ileum and may be located 10–150 cm (on average 90 cm) from the ileocecal junction., On average, it measures 3–6 cm in length and 2 cm in diameter. The most common sites of LH in adults are inguinal (50%), umbilical (20%), and femoral (20%)., It is also stated that from these sites, LH is generally located on the right side. In children, MD is predominantly identified inside of umbilical hernias, and it seems to be more susceptible to adhere to the hernia sac.,
Microscopically, MD is essentially coated by ileal mucosa as in the contiguous small bowel. Nevertheless, nearly 60% of MD also exhibit ectopic mucosa such as gastric, pancreatic, biliary, duodenal, or colonic, even though the real cause for the presence of heterotopic tissue in the diverticulum is unknown.,,,
Clinical findings and complications
Despite the largest part of patients found to have an MD do not present with any particular symptoms, the most recent literature states that approximately 4%-16% of these patients do experience-related complications.,, Although the diverticula can be found equally in both males and females, the incidence of complications is three to four times higher in males. About 60% of the patients diagnosed with an MD develop symptoms before the age of 10 years, but approximately 70% experience related complication before the age of 40., Along with male gender and age younger than 50, other risk factors have been identified increasing the chances of developing a symptomatic MD, such as the existence of a diverticulum with 2 cm or more in length or those that contain heterotopic mucosa. Literature has reported that when two, three, or four of these criteria were met, the percentage of symptomatic MD has increased to 25%, 42%, and 70%, respectively.
The main complication reported in adults is bowel obstruction as a result of an adhesive band or intussusception (14%-53%)., It is followed in frequency by inflammation or diverticulitis (12.7%), hemorrhage (11.8%), and ulceration (<4%). Acid secretion from ectopic mucosa found in MD results in inflammation causing diverticulitis. Other complications can result from blockage of the diverticular neck by enteroliths or foreign bodies causing stasis and infection., Meckel’s diverticulitis can often mimic the symptoms of acute appendicitis with patients presenting with right iliac fossa pain, vomiting, and low-grade fever. Recurrent intussusception can also lead to trauma, inflammation, and mucosal bleeding. MD can also be associated with rare complications including tumors (carcinoid, adenocarcinoma, sarcoma, intraductal papillary mucinous adenoma of pancreatic tissue) and vesicodiverticular fistulae.,,, The commonest tumor related to MD is a carcinoid tumor (2%). Some other uncommon complications found in the literature comprise torsion, inversion of MD, volvulus of ileum around a fibrous band, or the diverticula itself and perforation, which can happen naturally or secondary to a foreign body such as fish bones, marbles, gallstones, toothpicks, and even bullets.,,, MD perforation can lead to abscess and fistula development, and there seems to be a connection between these cases and inflammatory bowel disease.
Bleeding from the diverticulum, in particular, protracted bleeding, is unusual in adults, but can be explained by the release of acid secretions from ectopic gastric mucosa inside the diverticulum, resulting in ulceration of contiguous ileal mucosa. Symptomatic adult patients will present with ongoing crampy abdominal pain and later melena as a slow colonic transit time in adults. This ongoing slow, often unrecognized bleeding might cause hypoalbuminemia and low ferritin. Helicobacter pylori has a strong association with ulceration of gastric mucosa and bleeding. However, recent research failed to prove the same unquestionable link between H. pylori and the diagnosis of gastritis and/or bleeding from MD heterotopic mucosa., Moreover, cases of MD bleeding can initially present with iron deficiency anemia, megaloblastic anemia as a result of vitamin B12 deficiency, and bacterial overgrowth associated with the distention and immobility in surrounding obstructed ileal loops.
Literature suggests that the chances of adverse outcomes decline with increasing patients’ age, without obvious predictive factors for them to experience any of the above complications.
Incarceration into a hernia sac or MD strangulation may also arise as a potentially life-threatening complication leading to emergencies. In complicated LH, MD shows either inflammation within the hernia sac with strangulation or with ulceration of the gastric mucosa, and it cannot be reduced with the adherence of a fibrotic band or self-adherence. Although uncommon, in about one-third of incarcerated LH, the ileal loop adjacent to MD is found incarcerated within the hernia sac. Rare cases of strangulated LH have been reported. While MD is most commonly diagnosed in men, LHs are mostly found in women as a result of the substantial proportion of femoral LH., The leading symptom is usually an irreducible abdominal lump associated with tenderness, fever, and vomiting. Nevertheless, the clinical symptoms are seldom specific and commonly related to the hernia sac contents. In cases of isolated MD incarceration, patients may not develop characteristic symptoms of bowel obstruction, as a result, its diagnosis is often an intraoperative fortuitous discovery made.,
Our clinical case reports an incarcerated umbilical LH with some degree of strangulation, and a 5 cm gangrenous MD firmly adherent to the strangulated hernia sac was identified incidentally during surgery.
Quoting Charles Mayo, “MD is frequently suspected, often looked for, but seldom found.” On account of the low incidence in adults associated with an atypical presentation, the preoperative diagnosis of both MD and LH can represent an outstanding challenge. This result in less than 10% of cases of complicated LH in adults being diagnosed preoperatively., An accurate diagnosis is often difficult to institute as this clinical entity can simulate other acute abdominal conditions.
In a clinical suspicion of LH complicated by mechanical small bowel obstruction, plain abdominal radiographs may improve the preoperative diagnosis by demonstrating air-fluid levels. Nevertheless, it is important to note that in some cases thought to be incarcerated LH, intraoperatively was found that there was no true bowel obstruction, in fact, only MD was “trapped” inside the hernia sac and the remaining small bowel is free. These hernias are often misdiagnosed as RH.
Complicated LH may present with perforation following ectopic peptic ulceration due to gastric acid secretion or impaired circulation allied to strangulation. In these cases, there is a clinical indication for the use of erect chest and plain abdominal radiographs.
When the leading complication is bleeding, the diverticulum often expresses ectopic gastric mucosa. Therefore, scintigraphy with technetium-99m pertechnetate-labeled red blood cells can be used to identify gastric mucosa not less than 1.8 cm2.,, The authors report a sensitivity of 62.5%, a specificity of 9%, and an accuracy of 46% in adults that can potentially be enhanced by the co-administration of supplementary drugs, such as Pentagastrin (increases Tc uptake) and Cimetidine (prolongs Tc uptake).,, However, as this study is only specific for ectopic gastric mucosa and not exactly to MD, other clinical entities such as gut duplication cysts with ectopic gastric mucosa or vascular anomalies can be misdiagnosed. Additionally, a high number of false-negative scans have been reported in adults as a result of insufficient gastric mucosa, rapid dilution of Tc due to profuse bleeding from the mucosa, compromised vascular supply, or patients experiencing other complications rather than diverticulum bleeding.,,,, Scintigraphy with red blood cells labeled with technetium-99m is the most sensitive investigation to diagnose active gastrointestinal bleeding with bleeding rates of 0.1 ml/min. However, it remains less valuable in emergency settings.
Recent literature suggests that both abdominal ultrasonography (USS) and CT have a crucial role in the preoperative diagnosis and differentiation between hernias, but frequently they fail to provide a definitive diagnosis.,,, The appearance of MD on conventional CT is dependent on the patient’s symptoms. Studies are encouraged to use both intravenous and oral contrast material as it can assist in establishing the diagnosis of Meckel’s diverticulitis.,,
In our case, we preoperatively used a CT of the abdomen and pelvis. However, due to our patient poor renal function, the scan was performed without any contrast material. Therefore, due to the low sensitivity of the unenhanced CT study, a strangulated hernia could not be excluded; hence, we decided to proceed with emergency surgery.
According to the literature, the management of LH with symptomatic MD comprehends surgical treatment. The surgical repair of an LH includes both resection of the diverticulum and herniorrhaphy, and it can be performed either by an open (94.3%) or a laparoscopic (5.7%) approach., Some authors advocate the laparoscopic approach as it may allow for a better assessment of the bowel regarding blood supply and viability. Additionally, they believe that negative diagnostic laparoscopies can potentially avoid an unnecessary extensive approach such as an exploratory laparotomy.
The accepted treatment for symptomatic MD includes wedge diverticulectomy and division of diverticulomesenteric fibrous bands often found connected with intestinal mesentery or the abdominal wall to restore ileum transit. However, in complicated LH where there is edema, inflammation, ectopic tissue at the diverticular-intestinal connection, ischemia or perforation of the bowel, tumor, a segmental ileal resection followed by primary anastomosis should be performed.,,, This can prevent postoperative stricture of the resected segment. Literature has reported that the cumulative prevalence of early postoperative complications is 12%, which includes surgical site infection (3%), prolonged ileus (3%), and anastomotic leak (2%) with a mortality rate of 1.5%., A higher mortality rate has been associated with symptomatic patients (6%) and elderly and comorbid patients.,
Our patient underwent an emergency open umbilical hernia repair with segmental enterectomy and primary side-to-side stapled anastomosis and to date remains without complications.
Unlike the consensual management of symptomatic MD, literature seems to be divided when considering the treatment of asymptomatic diverticulum fortuitously identified during a laparotomy or laparoscopy. Although some authors defend surgical resection of MD to avoid rare but well-known complications, other authors suggest leaving the diverticulum in situ due to the unlikelihood of lifetime developing complications, decreasing with age.,, Sagar et al. reported postoperative morbidity after incidental resection up to 6%, with 33% morbidity after removal of a complicated MD, and a lifespan adverse risk predicted of about 16%.
Dumper et al. encouraged incidental diverticulectomy for patients younger in age at presentation, history of abdominal adhesions, and any palpable or visible anomalies of the MD. A length-based standard of 2 cm has also been reported to determine elective resection in asymptomatic patients. Nevertheless, none of these criteria is consistent among surgeons.
| Conclusion|| |
An MD, albeit considered a rare clinical condition in adults, may theoretically be found in any hernia, with patients experiencing a wide range of potential complications. The preoperative diagnosis of a strangulated umbilical LH such as in our clinical case is often difficult to institute. Therefore, it should warrant surgical attention and a high index of clinical suspicion. One must consider LH in the differential diagnosis in all incarcerated abdominal hernias. A thorough clinical assessment is essential and timely surgical intervention must be offered in view with ensuring the best outcome for these patients. Surgical resection of symptomatic MD followed by open or laparoscopic hernia repair is the gold standard, although the treatment of asymptomatic diverticula remains controversial. Ileal resection-anastomosis should be added to the treatment in complicated cases.
Due to the rarity of these types of hernias in adults, they remain underdiagnosed, often leading to suboptimal treatment with considerable morbidity and mortality. Therefore, it is imperative that all surgeons should not only be attentive and accurately identify them but at the same time be encouraged to routinely share similar clinical cases, thereby enhancing our community’s knowledge regarding the best clinical assessment and management of LH practices.
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Conflicts of interest
There are no conflicts of interest.
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