Hernioscopy Revealing Rare Abdominal Cocoon Syndrome in an Elderly Patient: A Novel Technique for Abdominal Pathology

Background

Incarcerated inguinal hernia (IGH) is a common clinical emergency in adults, usually requiring immediate surgery [1,2]. Intraoperative exploration can find IGH combined with bowel necrosis or even perforation, abdominal neoplasms, acute appendicitis, intestinal adhesion, and so on. Thus, anesthesia method, surgical procedures, and mesh materials for adult incarcerated or strangulated inguinal hernia are always individualized according to a patient’s case [3,4].

An abdominal cocoon (AC) is also known as “idiopathic encapsulating peritoneal sclerosis” [5]. The small intestine and colon are either totally or partially encapsulated by a fibrocollagenous sac, which mimics a cocoon [6]. To date, several reports have revealed the clinical features, etiopathogenesis, and characteristics of pathology of AC since 1978 [7–10]. However, its etiology and pathogenesis remain unclear [8], and preoperative diagnosis remains difficult [9,11]. Therefore, most cases are accidentally discovered intraoperatively. Moreover, AC is mostly observed in young adolescent women living in tropical and subtropical regions [12]. It is rarely reported in elderly men.

Several studies have reported intestinal obstruction caused by AC [9,13]. However, few studies have reported IGH combined with AC. AC is uncommon in elderly patients. Additionally, use of hernioscopy has rarely been reported in exploring intra-abdominal conditions in adults. Interestingly, IGH combined with AC was found using hernioscopy in a 90-year-old man. We present the surgical decision-making points for a case of an elderly person with IGH, enrich the diversity of AC, and propose a relatively novel approach of hernioscopy applied to elderly patients who suffer from incarcerated or strangulated groin hernias.

Case Report

A 90-year-old man was admitted with a 2-day history of progressive abdominal pain and vomiting. He had a more than 20-year history of constipation and right reducible IGH. There was no history of surgery or tuberculosis. Physical examination of the abdomen revealed apparent abdominal distention, tympanitic resonance to percussion, decreased bowel sounds, and a giant strangulated mass in the right groin. However, there were no signs of a left inguinal hernia. The skin of the scrotum was partially dark red (Figure 1A). His blood pressure was 130/66 mmHg, body temperature was 37.5°C, SpO2 was 98%, white blood cell count was 13.85×109/ml, high-sensitivity C-reactive protein (hs-CRP) level was 55.83 mg/ml, and hemoglobin concentration (Hgb) was 85 g/L. Abdominal computed tomography (CT) revealed a dilated bowel and a large mass in the right groin (Figure 1B). However, the CT scan only showed a slight broadening of the left inguinal canal and did not show a clear left hernia.

Subsequently, the patient’s condition deteriorated. Therefore, he immediately underwent surgery. We made a groin incision, resulting in less trauma and a better effect. General anesthesia was administered to meet a variety of complex surgical procedures. The mass of the ischemic intestine was densely stuck, which was very difficult to rearrange (Figure 1C). The patient’s small intestine could not be pulled out of the abdominal cavity. If we forcibly released the incarceration, it could increase the risk of small bowel damage and intestinal fistula. Thus, we slightly enlarged the right inguinal ring and returned these ischemic intestines after confirming that there was no necrosis after 15 min. However, to further investigate the type of hernia (ie, Maydl hernia) and any coexisting intra-abdominal pathology or further ischemia, hernioscopy was performed through the right open sac (Figure 1D). Surprisingly, all the bowel and mesenterium were almost completely encased in fibrous and cocoon-like membranes (Figure 1E, 1F). A greater omentum was absent (Figure 1F). Unfortunately, the fibrous membrane covering the intestines affected the observation and judgment of intestinal vitality and location. Overall, there was a small amount of purulent effusion in the lower abdomen, dilated bowel loops, and no signs of severe intestinal ischemia or intestinal necrosis. No definite signs of bowel obstruction were caused by AC syndrome. Moreover, a left inguinal hernia was accidentally identified (Figure 1G). There was only some fibrous tissue within the left inguinal hernia. Additionally, the protruding and stuck intestine was proved to be the terminal ileum. Therefore, right IGH is a common strangulated IGH rather than a specific type of hernia, such as a Richter hernia or a Maydl hernia. Finally, Lichtenstein hernia repair for the left inguinal hernia and Bassini hernia repair for the right IGH were performed. Drainage tubes were used to drain the inflammatory ascites and prevent infection (Figure 1H). Pathological tissue was obtained from the wall of the small intestine and from a fibrous membrane covering the surface of the ischemic intestine. Furthermore, histopathological results of the surface of the ischemic intestines revealed proliferation of fibrocytes and enrichment of collagen fibers with nonspecific inflammatory reactions (Figure 1I, 1J). The submucosa, muscular layer, serosal layer, and collagen fiber of the bowel and its fibrous membrane are clearly shown in Figure 1. These histopathological and macroscopic findings confirmed the diagnosis of AC. Inflammatory ascites were sent for bacterial culture and antibiotic drug sensitivity, but not for histological examination. This elderly patient was treated with conventional antibiotics after the operation. Moreover, the results of bacterial culture and antibiotic drug sensitivity tests were negative. A CT scan showed no evidence of leakage, recurrent hernia, or intestinal obstruction on the 5th day after the operation (Figure 1K). He was followed up for 6 months and reported a dramatic improvement in his quality of life (Figure 1L). Written informed consent was obtained from the patient for the publication of this report.

Discussion

When we opened the right hernia sac, a mass of the ischemic small intestine was stuck together. The surface of the small bowl was encapsulated within a thick sac (Figure 1C). We discovered that the bowel and mesenterium were almost totally wrapped by a fibrocollagenous and cocoon-like membrane. Furthermore, postoperative pathology of the small bowel wall and its fibrous membrane revealed the proliferation of fibrocytes and enrichment of collagen fibers (Figure 1I, 1J). These results are consistent with the histopathological and macroscopic manifestations of AC. Unfortunately, preoperative radiographic findings of the elderly patient were not typical AC manifestations after checking the patient’s abdominal CT with the radiologists. Overall, these histopathological and macroscopic findings confirmed the diagnosis of AC.

AC is a rare peritoneal disease. The pathogenesis and mechanisms are not fully understood [14]. Many idiopathic or secondary factors can cause AC, such as abdominal tuberculosis, long-term peritoneal dialysis, history of abdominal trauma, abdominal surgery, parasite infection, bacterial infection, and intraperitoneal chemotherapy [8,15,16]. However, this patient had no such medical history. We found that the greater omentum was absent. Additionally, the intestinal tract was encapsulated by a fibrocollagenous membrane or “cocoon”. Therefore, this was a typical case of idiopathic AC. With the popularization of laparoscopic techniques and advances in CT, an increasing number of AC cases are being diagnosed [11]. CT has been reported to be a valuable tool for making a definite diagnosis before surgery [14,17,18]. AC can be categorized into 3 types: type I involves part of the small bowel, type II involves the entire small bowel, and type III involves the small bowel along with the large bowel and other adjacent viscera [17]. The radiographic findings of AC patients’ bowel mainly include clumped loops, inter-bowel fluid, bowel wall thickening/enhancement, and so on [18]. However, not all of these manifestations are present in a single patient [17–19]. In this case, bowel wall thickening or enhancement was not confirmed on CT. In general, clinical diagnosis is difficult without specific symptoms or radiographic findings. Therefore, a part of the AC was found during the operation and then confirmed by postoperative pathology. Surgery is the most effective method of treating AC. However, the incidence of postoperative complications and mortality are high. All symptoms in this patient were mainly caused by a strangulated hernia. Therefore, we did not deal with AC. Primary AC mainly occurs in young female patients, mostly in tropical and subtropical regions [12]. Interestingly, this 90-year-old man had strangulated groin hernias and AC, and such a case has not been reported previously.

Furthermore, a new application of hernioscopy was described to explore the abdominal cavity for contralateral groin hernias in children [20]. No large case series has been published recently. This was achieved without additional skin incisions or fascial disruptions. Unfortunately, this method is not widely used for pediatric inguinal herniorrhaphy. A small transverse abdominal incision in the inguinal area was more prevalent for pediatric inguinal hernia because of dispensing with insertion into the abdominal cavity and a shorter surgery time. Moreover, the narrow abdominal space in pediatric patients is not suitable for hernioscopy.

The abdominal cavity has rarely been explored using hernios-copy in adults. According to our clinical experience, a 10-mm trocar was inserted into the opened hernia sac to establish a pneumoperitoneum of 12 mmHg. Then, a 30° laparoscopic lens was inserted into the abdomen (Figure 1D). At the induction of anesthesia, when the hernia contents retract spontaneously into the abdomen or enterocoelia, ensuring its viability can be quite difficult. This method not only allows for a good view of the entire abdomen and avoids misdiagnosis, but also reduces tissue trauma and enables quick postoperative recovery without additional skin incision. In our medical center, hernioscopy via the open hernia sac was performed in 46 patients with IGH between August 2016 and May 2020. Furthermore, mucinous neoplasm of the appendix, acute appendicitis, and AC were found by accident. Fortunately, these patients were diagnosed and underwent treatment. Therefore, this exploration method is safe and effective for investigating the abdominal cavity and organs. However, based on our clinical practice, this technique is not suitable for pediatric patients, obese patients, or patients with abdominal adhesions. The narrow space in the abdominal cavity is not conducive to performing hernioscopy and increases the possibility of injury to the abdominal organs. Generally speaking, it is a great option, especially in adults with incarcerated or strangulated groin hernias. Unfortunately, this technique has not been detected in randomized controlled trials or prospective studies. Therefore, hernioscopy used for surgical procedures needs to be further investigated in the future.

In the past, use of mesh was considered relatively contraindicated for the repair of IGH due to the possibility of blood supply disorders, bacterial migration, and infection of the surgical area. Bassini or Shouldice is usually recommended [21]. An increasing number of studies have shown that IGH is not a contraindication for mesh repair. Mesh repair of IGH does not significantly increase the incision or mesh infection rates [22–24]. Therefore, Lichtenstein hernia repair with Prolene mesh materials was performed on the left inguinal hernia. Bassini herniorrhaphy was performed on a right strangulated hernia.

Conclusions

Here, all the findings led to a diagnosis of right IGH combined with AC in a 90-year-old man, and such a case was reported for the first time. IGH, not AC, caused small bowel obstruction. IGH and AC were not precipitating factors. Surgical decision-making for the elderly patients suffering from IGH should be made according to the actual situation of the patients. Hernioscopy used in adults is a relatively novel surgical approach, and was confirmed to be safe and effective for exploring the abdominal cavity and abdominal organs. There were no intraoperative or postoperative complications in patients who underwent hernioscopy in our medical center. Hernioscopy is a great option in clinical applications, especially in adults with incarcerated or strangulated groin hernias.