Successful endoscopic decompression and detorsion of splenic flexure volvulus in a child with multimorbidity

Colonic volvulus is a disorder that can occur in the sigmoid colon, cecum, and transverse colon, which are not fixed to the retroperitoneum. Sigmoid volvulus is the most common, accounting for 60%–70% of cases, followed by cecal volvulus, accounting for 25%–40%. In contrast, volvulus of the transverse colon, including the splenic flexure, is very rare, accounting for 0.94% of all cases of colonic volvulus.1, 2 In principle, surgical treatment is recommended for volvulus of the transverse colon, including the splenic flexure, because of the high mortality rate and the high recurrence rate after detorsion.3 However, the risk of emergency surgery is high in children with severe mental disabilities who have various comorbidities. In addition, there have been no case reports of patients with a ventriculoperitoneal (VP) shunt who developed splenic flexure volvulus. Here, we report a severely mentally retarded multimorbid 17-year-old male with a VP shunt who developed splenic flexure volvulus, and illustrate that endoscopic decompression and detorsion avoided emergency surgery and led to a good prognosis. A 17-year-old boy with a VP shunt with hydrocephalus, mental retardation, chronic constipation, aerophagia, and dilated cardiomyopathy with mitral regurgitation developed acute oral feeding difficulty, defecation disturbance, and abdominal distension with ineffective enema. The patient had no other symptoms and was afebrile and hemodynamically stable. Clinical examination revealed marked, tense, diffuse abdominal distension with the percussion of drum sounds. Assessing the patient's pain was difficult because of the essential characteristics of mental retardation. Laboratory investigation results were unremarkable. Head computed tomography (CT) and X-ray evaluation revealed no exacerbation of the hydrocephalus or VP shunt rupture. However, abdominal X-ray revealed marked colonic dilatation (Figure 1a). A radiographic contrast enema revealed a beak sign at the splenic flexure, suggesting intestinal obstruction (Figure 1b). Subsequent contrast-enhanced abdominal CT revealed a splenic flexure volvulus without ischemia (Figure 1c). Endoscopic decompression and detorsion was performed using carbon dioxide (CO2) insufflation under sedation. Consistent with the CT findings, endoscopic examination showed a twisted mucosa without ischemic findings at the splenic flexure, and beyond the torsion site, a dilated lumen with a large fecal mass (Figure 1d). Endoscopic decompression and detorsion was successful (Figure 1e), and the patient's abdominal distension improved. The VP shunt likely did not function as a leading point for torsion or adhesion because its tip was mobile and away from the splenic flexure. The following day, abdominal X-ray revealed no evidence of recurrence or complications (Figure 1f), and the patient was discharged. After discussion with the patient's family, the decision was made to follow-up the patient without surgical treatment at that time, considering the high risk of surgery due to multimorbidity. However, the patient experienced a relapse of splenic flexure volvulus 3 months later. Endoscopic decompression and detorsion was repeated, and a transanal ileus tube was placed with endoscopic assistance to prevent re-volvulus. Subsequently, intestinal irrigation through the transanal ileus tube was also performed. Although a migrating transverse colon was still observed even during ileus tube placement (Figure 1g), the patient's condition remained stable. After nutritional management and preoperative evaluation in collaboration with the neurosurgery and cardiology departments, laparoscope-assisted partial resection of the transverse colon was performed. To prevent re-dilatation of the colon and further recurrence, the torsion origin was mobilized before resection, and a transanal drainage tube remained in place until anastomotic stenosis improved. Pathology revealed that the colon tissue was vasodilated and congested (Figure 1h). Postoperatively, no complications were observed, including cardiological complications and VP shunt problems. Moreover, the patient's constipation was alleviated, and the patient was able to defecate on his own. To prevent fecal impaction, an enema was continued once every 2 days, and the patient has had no recurrence of volvulus for 2 years. In a systematic review, the fatality rate of volvulus of the transverse colon, including the splenic flexure, was reported to be 14.1% (18/161) in adults. However, when limited to splenic flexure volvulus or limited to pediatric cases, the rates were only 2.2% (1/75) and 3.1% (1/63), respectively.2 This suggests that the severity rate of splenic flexure volvulus in children may not be high. In addition, volvulus of the transverse colon, including the splenic flexure, occurred more frequently in patients with constipation and psychomotor retardation.2 This suggests that children with severe mental disabilities having various comorbidities, as this patient, are at a higher risk of developing the disease. We believe that endoscopic decompression and detorsion have the great advantage of avoiding emergency surgery in these children. On the other hand, endoscopic decompression and detorsion carry a risk of intestinal perforation. The endoscopic procedure can only be performed in patients without bowel necrosis or peritonitis and with stable vital signs. During endoscopy, the endoscope should be gently advanced using intermittent low flow CO2 insufflation. There is no established technique for endoscopic decompression and detorsion of splenic flexure volvulus. In this case, the endoscope was placed in the transverse colon, the dilated colon was decompressed, and then endoscopic detorsion of the decompressed volvulus was performed by counterclockwise rotation and slowly withdrawing the endoscope, in contrast to the clockwise rotation in sigmoid volvulus. This report demonstrates that endoscopic decompression and detorsion of the splenic flexure volvulus may be a bridging treatment for operative intervention in patients with severe complications if the patient is hemodynamically compensated without evidence of vascular compromise. In addition, placement of a transanal ileus tube may be effective in preventing re-volvulus during the preoperative waiting period. Substantial contributions to the conception or design of the work: TT and AM. Acquisition, analysis, or interpretation of endoscopic findings for the work: AM and KI. Acquisition, analysis, or interpretation of operative findings for the work: KS, TJ, and KM. Acquisition, analysis, or interpretation of comprehensive data for the work: AM, KI, and HT. Drafting the work: TT and AM. Revising the work critically for important intellectual content: KM and HT. Final approval of the version to be published: TT, AM, KI, KS, TJ, KM, and HT. The authors would like to thank Dr. Ryuta Tanaka and Dr. Katsuyuki Yaita for useful discussions. We would like to thank Editage (https://www.editage.com/) for the English language editing. The authors declare no conflicts of interest. Written informed consent was obtained from the patient's parents.