Massive surgical emphysema after perineal proctosigmoidectomy

Abstract

An 83-year-old woman underwent an elective perineal proctosigmoidectomy (Altemeier procedure) for a rectal prolapse. On postoperative day 1, the patient presented with impressive subcutaneous emphysema involving the chest, neck and face without any other symptoms. A CT scan showed free air in the retroperitoneum, the intraperitoneal cavity, the mediastinum and a subcutaneous emphysema of the neck and the face. Air was also found around the coloanal anastomosis and an anastomotic leak was proven by rectal contrast agent. In this situation, a rectoscopy followed by a laparoscopy were immediately performed. The leak could not be visualised. Peritoneal lavage and drainage, followed by protective sigmoidostomy were carried out. After surgery, the follow-up was uneventful except a persistent but asymptomatic leak with a presacral cavity. The coloanal dehiscence was later proven in rectoscopy. Although sutured, it is still present and colostomy closure will eventually be possible in a few months.

Background

Rectal prolapse is a clinical entity where the rectal wall protrudes through the anal orifice.¹ It is generally more common in elderly women. Further risk factors include pregnancy (especially multiparous), previous surgery such as hysterectomy or rectocele repair, pelvic neuropathies and neurological disease, high gastrointestinal helminthic loads (Trichuris dysentery syndrome), chronic obstructive pulmonary disease and cystic fibrosis.^1–3 Progressive loss of function of the pelvic muscles and ligaments can lead to faecal incontinence, pelvic pain, defecation disorders, rectal bleeding and ulcers. Definitive treatment of a rectal prolapse can only be accomplished by surgery. There are more than 50 types of procedures described in the literature and a consensus has not yet been reached on the preferred method to be used.⁴ Basically, the procedure can be performed through an abdominal or perineal approach, or a combination of the two. Rectopexy is carried out with or without rectosigmoid resection. The Altemeier procedure, a perineal full-thickness rectosigmoidectomy, is considered as a possible option for elderly patients because it obviates the need for abdominal surgery.⁴ Potential complications referred to the Altemeier procedure are anastomotic leakage, postoperative haemorrhage, postoperative faecal impaction, anastomotic stricture and recurrence of the prolapse or persisting functional symptoms.⁵ The presented case describes classic risk factors of rectal prolapse while the postoperative complication is quite unique and has never been published in this context.

Case presentation

An 83-year-old woman was referred for evaluation of a rectal prolapse. The case history showed a progress of the prolapse with several episodes of difficult reposition. The examination confirmed a full-thickness rectal prolapse 8 cm in length along with third-degree haemorrhoids. The digital rectal examination revealed a rough mucosa as a sign of a persistent prolapse as well as a defect of the pelvic floor. The medical history included vaginal hysterectomy with rectocele repair 9 years earlier. Furthermore, the patient had sustained a pulmonary embolism 12 months before with persistent third-degree dyspnoea, and suffered from chronic renal failure and incipient dementia. We decided to perform an Altemeier procedure as would be less invasive than an abdominal approach in this elderly and multimorbid patient. The haemorrhoids were treated simultaneously by haemorrhoidectomy (Ferguson procedure). Twelve hours after surgery the patient presented with progressive subcutaneous emphysema (SCE) of the chest. The patient was haemodynamically stable and showed no signs of sepsis or dyspnoea. In the following hours the emphysema progressed up to the neck and face.

Investigations

Laboratory findings were unremarkable with a white cell count of 10.6 G/L and a C reactive protein of 39 mg/L. In favour of further investigation a craniothoracoabdominal CT scan was administered (figure 1). Free air was found in the retroperitoneum and in the abdominal cavity, and extended up through the mediastinum (pneumomediastinum) into the neck and the face (SCE). Air was also present in the greater omentum and the prevesical space. Rectal contrast agent was applied and was found perirectal, proving an anastomotic leak.

Figure 1.

Origin of leak and schematic pathway of the emphysema. Colour-enhanced CT slices (emphysema in blue); axial (1), sagittal (2) and coronal (3) view. Air is visible in the retroperitoneum (A), along the vessels (B), intraperitoneal (C), preperitoneal (D), in the mediastinum (E), in the subcutaneous tissue of the neck (F) and face (G).

Differential diagnosis

The differential diagnosis of an SCE is broad and asks for a detailed evaluation. Basically, gas may enter the subcutaneous space from another compartment (eg, trauma) or is produced by bacteria (infection).⁶ A detailed patient history can often narrow the aetiology of an SCE. In the present case, an infection with gas-producing bacteria was very unlikely, because the patient was in a quite good condition. Our first thought was a pneumothorax caused by a barotrauma after intubation. Because of the extent and the progression of the emphysema, a CT scan was performed showing the air leak around the anastomosis. However, an SCE could be caused by an air leak at any point in between the oesophagus and the anus. The anatomical site of the perforation usually determines the location of any SCE. Extra abdominal perforations are more likely to lead to an SCE.^{7 8}

Treatment

As there were clear CT findings of an anastomotic leak we decided to perform an emergency rigid rectoscopy. However, it was inconclusive and the leak could not be visualised. The following laparoscopy showed evidence of free fluid in the pelvis and visible air in the retroperitoneum, predominantly left of the descending and sigmoid colon as well as in the lesser omentum (figure 2). The leak itself could not be visualised by the air insufflation test. We decided to perform a peritoneal lavage and a Jackson-Pratt drain was placed near the anastomosis. In addition, a diverting sigmoidostomy was successfully carried out and a perianal drainage was placed into the presacral space.

Figure 2.

Intraoperative findings: air in ligamentum teres (1), lesser omentum (2), greater omentum (3), retroperitoneum (4) and near the anastomosis (5).

Outcome and follow-up

After surgery, broad-spectrum antibiotic treatment with piperacillin was administered and the patient was monitored on the intensive care unit for several days. She stayed haemodynamically stable and the use of vasopressors was not necessary at any point. After 3 weeks the patient was discharged in good condition to a rehabilitation facility. At follow-up 6 weeks postoperatively, the patient was asymptomatic. A CT scan with contrast agent through the colostomy showed a persistent leak ventral of the anastomosis with a small presacral collection. In a rectoscopy we were able to visualise a 5 mm dehiscence of the ventral part of the anastomosis. We decided to close the dehiscence by sutures. So far, the patient has tolerated the procedure very well and has no symptoms. We plan another contrast-enhanced CT scan in a few months to check the status of the leak. Only after the local situation has healed will we discuss the closure of the colostomy.

Discussion

To the best of our knowledge, this is the first reported case of an air leak of this extent after an Altemeier procedure. There are two similar cases of pneumoperitoneum, pneumoretroperitoneum, pneumomediastinum and pneumopericardium after Longo haemorrhoidopexy, one of them also with SCE of the neck.^{9 10} Furthermore, we found several reports of cervical SCE after perforation including an occult perforation of sigmoid diverticulum⁷ and a spontaneous rectal perforation.¹¹

There is no reported case of emphysema of the neck and face after a perineal proctosigmoidectomy, as described in the presented case. As mentioned above, in absence of any signs of sepsis our first thought was that the massive emphysema had to be related to a barotrauma after intubation. It was only the CT scan that proved otherwise and led to the diagnosis of an anastomotic leak. To our surprise, other published cases described a lack of signs of sepsis as well, although there is continuity from within the septic colon through the retroperitoneum, the mediastinum to the subcutaneous tissue. An SCE of the upper part of the body without any other symptoms can be caused by a perforation of the colon or the rectum. Therefore, after colorectal surgery, an extraperitoneal anastomotic leak or perforation must be excluded.^{7 10 11}

The Altemeier technique starts with a circumferential incision of the prolapsed rectal wall. The rectum can then be gently pulled out until no additional prolapse is possible. Further, a levator repair can be performed anterior or posterior to the rectum. The bowel is divided and the anastomosis is accomplished.⁴ In the described case, surgery was performed in lithotomy position and an anterior levator repair was carried out. It is our assumption that these sutures led to a ventral lesion and finally to the anastomotic leak. Importantly, in lithotomy position the view of the ventral bowel part is always limited and at risk for a lesion.

The study of the anatomic layers and spaces can be illustrated very well in this case. Through just a small dehiscence of the ventral anastomosis air leaked around the rectum and extended into the rectum fascia. From there it made its way to the retroperitoneum and prevesical space, further around the kidneys and probably along the mesenteric root into the peritoneum. The connection to the mediastinum can be found paraoesophageally. Just above the sternum in the jugulum the gas gets its access to the subcutaneous layers.⁹ In addition, it is progressing through the neck (along the vessels and subcutaneous) into the retropharyngeal space, all the way up to the face.

With the help of this case, the expansion of an infectious disease can easily be remembered and it becomes clear why an infection at the cervical site can lead to a fatal mediastinitis.¹²

In conclusion, an anastomotic leak must be excluded by CT scan and rectoscopy if an SCE occurs after a perineal proctosigmoidectomy, even if the patient is asymptomatic and shows no signs of sepsis.

Learning points.

• A subcutaneous emphysema of the upper part of the body may be the only sign of an anastomotic leak after perineal proctosigmoidectomy.

• Therefore, an anastomotic leak must be excluded by CT scan/rectoscopy in such cases.

• Gas and infections spread along fascial layers; the retroperitoneum, the prevesical space, the mediastinum and the subcutaneous spaces are all connected, and further cavities can be reached along vessels.

• The ventral part of the anastomosis in the Altemeier procedure is a critical step due to the limited visibility in lithotomy position.