Hernia (2013) 17:285–288 DOI 10.1007/s10029-011-0861-6

CASE REPORT

Stenosis of the small intestine after reduction of strangulated Littre hernia in an infant S. Visˇnjic´ • A. Car • R. Kralj

Received: 5 April 2011 / Accepted: 1 July 2011 / Published online: 26 July 2011 Ó Springer-Verlag 2011

Abstract Herniation and incarceration of a Meckel’s diverticulum in a hernial sac—Littre hernia—is a relatively uncommon surgical emergency. Segmental stenosis of small intestine after hernia reduction and consecutive intestinal obstruction is a similarly rare emergency. The combination of both these disorders is extremely uncommon at any age and especially during infancy. The obvious rarity of the condition, its subtle diagnostic features, the potentially ominous course of events and the age of patient indicate early surgery as a life-saving solution. Keywords

Littre hernia
Intestinal stenosis
Infant

Introduction As originally defined by Rieke in 1841, Littre hernia is a protrusion of a Meckel’s diverticulum through an existing abdominal wall opening. It is a rare finding at any age. Due to its rare occurrence, different incidences have been reported. To our knowledge, only two papers reporting a Littre’s hernia in infancy have been published [1, 2]. After reduction of the strangulated hernia, due to temporary ischemia, stenosis of the small intestine is a well known complication. Stenosis of the small intestine in early life is uncommon. An order of events including Littre’s hernia with strangulation and progressive small bowel stenosis with obstructive symptoms is therefore extremely rare. Diagnosis is difficult, and clinical examination alone can

S. Visˇnjic´ (&)
A. Car
R. Kralj Children’s Hospital Zagreb, KBCSM, Medical School University of Zagreb, Zagreb, Croatia e-mail: [email protected]

barely differentiate Littre from other types of hernia until complications arise. Progressive symptoms of small bowel obstruction, although unspecific, should indicate an early operative approach.

Case report A 4-month-old male infant was operated due to an incarcerated left-sided inguinal hernia 6 weeks prior to hospital admittance and the onset of symptoms. He was then referred to a surgeon because of poor feeding, vomiting and abdominal distension. Physical examination revealed a firm indurated hernia in the right groin that was completely closely reduced. At that time, the patient still had normal bowel movement, regular stool emptying and no signs of inflammatory disease (normal values of white blood count and C-reactive protein). Gastric decompression with a nasogastric tube was performed to prevent emesis and gastric aspiration, but no retention was noted. In spite of hernia reduction, distension of the abdomen as well as distension of the small bowel loops progressed. The initial diagnostic approach in the case of suspected neonatal intestinal obstruction at the Children’s Hospital Zagreb is plain X-ray of the abdomen in the erect position, followed by ultrasonography of the abdomen. Serial plain X-rays showed a marked progression in number and width of the dilated loops. Sonography showed dilated bowel loops with regular peristalsis and a small amount of free fluid in the space of Douglas. A barium enema study revealed a narrow lumen of the colon, and barium contrast reached only the hepatic flexure of the colon but did not pass through coecum and the ileocoecal valve. Laboratory values of inflammatory markers were still within normal range. As there was no improvement in the overall status after hernia

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Fig. 1 Hernia sac reverted and thick band attached to hernia sac (bottom of picture)

Fig. 2 Meckel’s diverticulum congested and fragile, thick fibrous Vitelline duct band, stenotic small intestine and prestenotic dilatation

reduction during a 36-h monitoring, surgery was indicated. Operative findings revealed all the elements of a incarcerated Litrre’s hernia. Reduction of the hernia caused inversion of the whole hernia sac in the abdominal cavity, and Meckel’s diverticulum was adhered by a thick fibrous band to the bottom of the hernia sac (Fig. 1). Meckel’s diverticulum was congested and fragile but without perforation, and a thick fibrous Vitelline duct band, originating from the top of diverticulum, was attached to the peritoneal hernia sac. Severe segmental stenoses of the small intestine were noted, up to 3 cm on each side of the Meckel’s diverticulum origin. Obvious prestenotic dilatation of the proximal small bowel explains the symptoms of progressive obstruction. Poststenotioc bowel was normal in size (Fig. 2). Resection of the diverticulum as well as the stenotic and congested portion of the small bowel with an end-to-end anastomosis of the small intestine was performed. Approximately 15 cm of the small bowel was resected. Herniorraphy was also performed. Histology confirmed Meckel’s diverticulum. The immediate postoperative course was uneventful and the patient did not present any symptoms during postoperative follow up.

emergency of the utmost importance. Reduction with or without sedation is possible in one-quarter to one-third of cases, and normally leads to delayed surgery [3]. Littre’s hernia was defined as the presence of a Meckel’s diverticulum in a hernia sac. Meckel’s diverticulum is found at the antimesenteric border of the ileum, usually located up to 60 cm from the ileocecal valve, measuring up to a few centimeters in length. Embryologically, it is a persistent intestinal part of the Vitelline (omphaloenteric) duct, an early communication of the midgut and the umbilical vesicle. Littre (1700) first reported ileal diverticula, but attributed them wrongly to prolonged traction. Meckel (1809) originated a theory of their embryological origin. The nature of the herniated Meckel’s diverticulum was defined by Rieke in 1841. Treves (1897) first described partial enteral herniation. Only part of the circumference of the bowel is entrapped and strangulated in the hernia orifice-enterocele herniation or Richter’s hernia. Subsequently, Treves was the first to make a distinction between Littre’s and Richter’s hernia [4]. Usual sites of Littre hernia are: inguinal (50%), umbilical (20%), and femoral (20%), but a lumbar or ventral location have also been reported. Postincisional Littre hernia has also been described [5]. Meckel’s diverticulum may be accompanied in the sac by an ileal loop, to which it is attached. Rarely, it may undergo incarceration or strangulation, necrosis, and perforation. In children, the diverticulum is more prone to adhere to the sac [6]. During incarceration, all symptoms of strangulated hernia in infancy can be present, and there is no specific sign that advocates the presence of Meckel’s diverticulum in a hernia sac. In the preoperative examination of inguinal masses, diagnostic ultrasound is a common, but not a mandatory, procedure in the Children’s Hospital in Zagreb, bearing in mind that the diagnostic value of preoperative sonography for strangulated hernia is unreliable and may

Discussion Inguinal hernia is a frequent pathology in children due to abnormal persistence of the processus vaginalis. Incidence varies from slightly less than 1 to 4.4% for children of any age. In premature infants, the incidence reaches about 30%. The most severe complication of inguinal hernia is strangulation. Hernias are usually repaired at an early stage and the incidence of strangulated hernias with resulting complications has decreased sharply. However, although it is a rare entity (from 1.5 to 8%), it still constitutes a surgical

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be misleading [7]. Some reports advocate contrast computed tomography (CT) for hernia sac content evaluation as a highly specific imaging technique in adults [8], but the value of the method for infants who need sedation or general anesthesia for the CT scan remains unclear. Therefore, reduction of a hernia, without extensive diagnostic check up prior to reduction, is the primary option. Closed reduction-taxis is preferred to an operation. We found that immediate surgery is a better solution for refractory or nonreducible cases than reduction under sedation or general anesthesia and a delayed repair. Small intestinal stenosis due to ischaemia is a well known complication following strangulated hernia. It has been modelled with experiments on rabbits. Impairment of the arterial rather than venous circulation is responsible for the damage observed. Such experiments have shown that ischemia that persist for several hours produce a frank gangrene of the intestine; if circulation was restored before the end of a critical time interval, a number of these rabbits developed ulceration or gangrene of the mucosa, which eventually led to tubular stenosis [9]. Older populations are most affected while the incidence in infancy is rare. Disordered transit and progressive intestinal obstruction can follow immediately after hernia reduction as well as after an undefined interval and there is no predictive factor to determine it. Clinical status will determine the volume of preoperative check up and the urgency of surgery. Plain radiography of the abdomen may show a small-bowel obstruction in the form of dilated small-bowel loops with multiple air fluid levels. Preoperative diagnosis of small intestinal stenosis can be made accurately with contrast studies in up to 50% of adults, but accuracy declines with younger age. Sonographic findings can rarely be diagnostic but unspecific symptoms can sometimes contribute to the diagnosis. A Tc-99 m pertechnetate scan to rule out or detect tracer uptake in Meckel’s diverticulum gastric mucosa is still regularly used but recent studies report that Meckel’s diverticulum is currently more commonly encountered in children on CT performed for abdominal pain than in technetium pertechnetate studies [10, 11]. So, in spite of advanced imaging techniques it can be difficult to diagnose Littre’s hernia on clinical and imaging grounds alone, and many authors have concluded that the diagnosis can be made only upon explorative surgery [12]. During infancy, incompletely reducible hernias may be confused with cryptorchidism, when Meckel’s diverticulum adheres to and envelops the testicle making palpation of the gonad difficult. Rapid deterioration in infant patients usually dictates early surgery. Operative findings determine the mode and choice of the operative procedure. Conventional treatment for reduced Littre’s hernia is a wedge resection of the diverticulum and a transverse repair of the ileum. Repair of the hernia from

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within the sac should follow the initial procedure. An unresolved question is whether a simple diverticulectomy is sufficient for removing a Meckel’s diverticulum. The ectopic tissue increases the likelihood of symptomatic postoperative bleeding. Recent data reports that ectopic tissue is present in 13% of the patients with Meckel’s diverticulum. In 62% of patients with ectopic tissue, this tissue is not palpable. Palpable tissue, if found, is usually located at the base of the Meckel’s diverticulum [13]. Therefore, segmental ileal resection would be a smart option, especially for children. There is no evidence that segmental resection has any disadvantages over wedge resection or simple diverticulotomy in terms of complications or follow up results [14]. Some authors reported successful laparoscopic repair for incarcerated inguinal, or even femoral Littre’s, hernia [15, 16], and the same principles could be applied to preschool and older children but may not be applicable in infancy. Although each of these methods has their own advantages and limitations, the choice of management still depends on the patient’s condition and the surgeon’s preferences.

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