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13 October 2024: Articles  Japan

Retrograde Colonic Intussusception After Colonoscopy without Organic Pathology: A Case Report

Mistake in diagnosis, Diagnostic / therapeutic accidents, Rare disease

Nobuhisa Tanioka ORCID logo1AE*, Michio Kuwahara1AD, Takashi Sakai1DF, Shigeto Shimizu1BF, Shunsuke Kanazawa2DF, Kentaro Mukaida2CF, Shunsuke Uka2D, Motoki Takasaki2D, Hidekazu Abe2DF, Kensuke Munekage2DF, Toyokazu Akimori1AD

DOI: 10.12659/AJCR.945423

Am J Case Rep 2024; 25:e945423

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Abstract

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BACKGROUND: Adult colonic intussusceptions are relatively rare and are mostly caused by organic structures that serve as lead points. However, the pathogenesis of adult intussusception is not fully understood, and no cases of retrograde colonic intussusception without pathological abnormalities or associations with colonoscopy have been reported.

CASE REPORT: A 74-year-old woman presented with abdominal distension and constipation. Abdominal computed tomography (CT) revealed marked dilatation of the right and sigmoid colon, initially suggesting volvulus of the sigmoid colon. Observation of the left colon revealed no abnormal findings on the colonoscopy. Due to the persistence of abdominal symptoms from right colon dilatation, another colonoscopy was performed, and a transanal drainage tube was inserted into the transverse colon. Enterography showed a steep contrast interruption in the descending colon, which was missed at this time. The patient’s abdominal pain worsened 3 days after removal of the drainage tube. Retrograde intussusception of the sigmoid colon was discovered on abdominal CT, and a laparoscopic left hemicolectomy was performed. Pathological examination revealed multiple ulcers in the superimposed area, but no abnormal organic findings that could be considered as a lead point were found. In this case, the stretching technique and/or shear stress on the sigmoid colon by a second colonoscopy may have contributed to the development of this condition.

CONCLUSIONS: This is the first report of colonoscopy-associated retrograde colonic intussusception without organic abnormalities. Although much is unknown about the pathogenesis in this case, it may provide new insights into the pathogenesis of intussusception.

Keywords: Colonoscopy, Intussusception, Laparoscopy, Colon

Introduction

Intussusception, a condition in which a segment of the bowel is invaginated into the lumen of an adjacent segment [1], is a rare cause of intestinal obstruction that affects 1% of adults [2]. Adult intussusception occurs in the small intestine, ileocecal, and colon in 49.5%, 29.1%, and 19.9% of cases, respectively, with a higher percentage of malignancy in the colon [3]. Unlike children, who have a higher rate of idiopathic intussusception, 90% of adult intussusceptions are caused by an organic structure that serves as the lead point [3,4].

Although intussusception has had a long history since its discovery, the pathomechanisms are not fully understood due to a small number of unusual cases: adult colonic intussusception with a retrograde form [5–7], those with no definable anatomic or pathological abnormalities [8–10], and those caused by colonoscopic procedures [11–14]. Notably, no case of retrograde colonic intussusception involving colonoscopy without pathological abnormalities has ever been reported.

In this report, we present a unique case of retrograde intussusception of the sigmoid colon caused by colonoscopy to investigate fecal obstruction.

Case Report

A 74-year-old woman, who was on medication for Parkinson’s disease and constipation and had no family history of cancer, presented with abdominal distension and constipation. Abdominal computed tomography (CT) revealed marked dilatation of the right and sigmoid colon (Figure 1A, 1B). Sigmoid colon volvulus was initially suspected, and an emergency colonoscope (PCF-H290 TI, Olympus Corporation, Tokyo, Japan) was inserted. Observation up to the splenic flexure of the colon revealed no torsion or obstruction (Figure 1C, 1D). Her symptoms did not improve and she underwent another colonoscopy the following day. CT showed a dilated right colon (Figure 2A, 2B). After stretching the sigmoid colon by hooking the tip of the endoscope to the splenic flexure (Figure 2C, 2D), the trans-verse colon revealed no obstructions. Further observation was not possible because of stool retention. Because the cause of the bowel obstruction was unclear, a transanal drainage tube was placed in the transverse colon, and a water-soluble contrast medium was injected. No causative structures were found in the transverse colon (Figure 2E). A posterior view showed a steep contrast discontinuity in the descending colon at this time (Figure 2F), and the patient was admitted to the hospital.

On the first day of admission, the patient’s abdominal symptoms improved, and enterography was performed again using a transanal drainage tube. The right colon remained dilated, yet no apple core sign was observed from the transverse colon to the hepatic flexure (Figure 3A). The contrast medium did not flow into the descending colon (Figure 3B). A colonoscope was then inserted again. Dilatation of the transverse colon limited observation of the hepatic flexure, and no neoplastic lesions or ischemic changes were observed. Fecal obstruction due to decreased peristalsis involving Parkinson’s disease was suspected, and the drainage tube was removed. The patient was started on a diet and discharged home on the fourth day of admission, despite no defecation.

The patient’s abdominal pain increased that night, and she was rushed to our hospital. Her vital signs were as follows: temperature, 36.6°C; blood pressure, 112/86 mmHg; pulse rate, 114/min; SpO2, 98%; and respiratory rate, 32/min. The left abdomen was tender, and blood tests showed an elevated C-reactive protein level (9.2 mg/dL) and white blood cell count (16 500/μg). Abdominal CT showed that the sigmoid colon retrogradely invaginated the descending colon, and the contrast medium used in enterography 3d earlier accumulated from the descending to the transverse colon (Figure 4A, 4B). Emergency colonoscopy was performed, and retrograde intussusception of the sigmoid colon was diagnosed. Ischemic changes were observed in the mucosa, but there were no initial signs of necrosis (Figure 4C). After decompression of the oral colon using a transanal drainage tube (Figure 4D), laparoscopic surgery was performed.

Exploration revealed that the sigmoid colon was strongly entrapped in the descending colon, and manual repair was not possible. A laparoscopic left hemicolectomy and double-stapling technique anastomosis were performed. Macroscopic examination of the resected specimen revealed a 10-cm retrograde intussusception of the descending colon (Figure 5A, 5B). Multiple ulcers were observed in the overlapping area, but no lesions that could be a lead point for intussusception were noted (Figure 5C). Microscopic findings revealed that the ulcers were confined to the muscle layer, with mucosal necrosis, submucosal hemorrhage, edema, and neutrophilic infiltration around the ulcer (Figure 5D).

Food intake was started on the third postoperative day. Because of a coronavirus disease 2019 infection and a decrease in activities of daily living, the patient was transferred to a local doctor for rehabilitation on the 21st postoperative day.

Discussion

Over the years, several investigators have attempted to explain the pathomechanism of intussusception. The dominant theory is that peristalsis and food, along with the adjacent intestinal tract, push the lead point into the distal relaxed intestinal segment [4]. Structures that may serve as such lead points include various malignant tumors, diverticulum, postoperative adhesions, strictures, and benign neoplasms [15]. This phenomenon is especially common between the freely moving and fixed segments of the intestinal tract. In rare reports of iatrogenic intussusception involving a tube implanted in the gastrointestinal tract, the lead point is believed to be formed by negative pressure and a structure at the tip [16,17].

Retrograde intussusception is extremely rare, with limited literature describing its epidemiology. Kitahara et al cited the literature reported in Japan, stating that retrograde cases account for 0.2% of all adult intussusceptions [7]. Retrograde intussusception is thought to occur less frequently because once intussusception occurs, spontaneous remission is likely to occur owing to dilation and increased internal pressure caused by the contents of the oral intestinal tract and forward peristalsis [18]. To the best of our knowledge, retrograde intussusception of the colon is associated with a tumor larger than 2 cm [5–7,19,20], and we were unable to find a tumor-free case. This may have been caused by the oral intestinal tract sliding over the segment immobilized by a relatively large tumor. Retrograde intussusception of the colon is most commonly found in the sigmoid colon [6,7,19,20], with only 1 case reported in the transverse colon [5]. This suggests that the sigmoid colon has a free mesentery and that the antiperistaltic wave in the left colon is associated with the establishment of retrograde intussusception [20].

Several reports have described intussusception related to colonoscopy. Hashiguchi previously reviewed 14 cases of adult intussusception associated with colonoscopy, all of which were antegrade intussusceptions with onset on the right side of the colon [11]. The mechanism of intussusception after endoscopic mucosal resection or endoscopic submucosal dissection involves hematoma and mucosal edema in the treated area [14] and localized peritonitis due to thermal injury [13], which act as lead points and result in intussusception. Cases of intussusception during routine colonoscopy have also been reported, suggesting that the proximal colon retracts due to a “rapid vacuum” effect, especially when a large amount of gas that had been woven into the colonoscope during a prolonged examination is aspirated during withdrawal of the colonoscope [21–23].

A retrospective review of the timing of the occurrence of intussusception in this case suggests that it was at the time the second colonoscopy and transanal drainage tube were inserted, as enterography showed a steep contrast discontinuity image of the descending colon. Although a colonoscopy was performed in the presence of intussusception, the absence of abdominal symptoms or ischemic changes in the intestinal tract caused the intussusception to be missed. Based on a systematic review of adult intussusception, CT is considered the most accurate diagnostic method (diagnostic accuracy, 77.8%) [3]. When a colonoscopy is performed on a colon with retrograde intussusception without a lead point, stenosis is not observed in the frontal view, and its presence may be overlooked if this condition is not suggested beforehand by CT [7].

In our case, retrograde intussusception may have been caused by colonoscopy, but the mechanism remains unclear because no pathological abnormalities were found, and reports of retrograde intussusception caused by colonoscopic procedures are lacking. One possible cause is the straightening of the sigmoid colon by colonoscopy. When the sigmoid colon was stretched by hooking the tip of the camera to the splenic flexure, the elasticity of the sigmoid colon may have caused it to invaginate the dilated descending colon (Figure 6A, 6B). Another possibility is that shear stress occurs during colonoscopy insertion; in other words, friction during pushing may have pushed the sigmoid colon mucosa into the fixed descending colon (Figure 6C, 6D). However, a stenosis or organic abnormality should be present in the stacking section under this condition, but this was not evident in the specimen. Furthermore, these procedures are routinely performed during routine colonoscopy. Notably, the patient had Parkinson’s disease. The association between Parkinson’s disease and intussusception remains unclear, but is known to be associated with gastrointestinal dysfunction [24], which may have prevented spontaneous remission of retrograde intussusception, as observed in a previous report [25]. It may also have been caused by a combination of specific conditions, including the relative dilatation of the descending colon compared to the sigmoid colon.

Although traction of the transanal drainage tube or pressure with contrast injection has successfully treated retrograde intussusception of the colon in some cases [7], these methods were not viable in this case because of the long intussusception length. We performed en bloc resection of the colon because of prominent blood flow disturbances and the fact that malignant lesions are more likely to be involved in intussusception of the colon.

Conclusions

In this study, we report a unique case of retrograde colonic intussusception caused by a colonoscopy. This is the first report of retrograde colonic intussusception without evidence of organic pathology and may provide new insights into the pathogenesis of intussusception.

Figures

Abdominal computed tomography (CT) revealed marked dilatation of the right (arrowhead) and sigmoid colon (red arrow), and no obstructive origin was identified (A, B). Colonoscopy revealed no torsion or obstruction in the colon up to the splenic flexure (C, D).Figure 1.. Abdominal computed tomography (CT) revealed marked dilatation of the right (arrowhead) and sigmoid colon (red arrow), and no obstructive origin was identified (A, B). Colonoscopy revealed no torsion or obstruction in the colon up to the splenic flexure (C, D). Abdominal computed tomography (CT) revealed a collapsed sigmoid colon (red arrow) and dilatation of the right colon (arrowhead) (A, B). Colonoscopy with the tip of the endoscope hooked to the splenic flexure to stretch the sigmoid colon (C, D). Enterography revealed no causative structures in the transverse colon (E). A steep discontinuity of contrast was observed in the descending colon at this time (yellow arrow) (F).Figure 2.. Abdominal computed tomography (CT) revealed a collapsed sigmoid colon (red arrow) and dilatation of the right colon (arrowhead) (A, B). Colonoscopy with the tip of the endoscope hooked to the splenic flexure to stretch the sigmoid colon (C, D). Enterography revealed no causative structures in the transverse colon (E). A steep discontinuity of contrast was observed in the descending colon at this time (yellow arrow) (F). Enterography using a transanal drainage tube showed no apple core signs from the transverse colon to the hepatic flexure (A), and no contrast medium flowed into the descending colon (B).Figure 3.. Enterography using a transanal drainage tube showed no apple core signs from the transverse colon to the hepatic flexure (A), and no contrast medium flowed into the descending colon (B). Abdominal computed tomography (CT) revealed that the sigmoid colon retrogradely invaginated the descending colon, with contrast accumulation in the oral side (A, B). The emergency colonoscopy revealed ischemic changes in the colonic mucosa but no signs of necrosis (C). A transanal drainage tube was inserted for decompression (D).Figure 4.. Abdominal computed tomography (CT) revealed that the sigmoid colon retrogradely invaginated the descending colon, with contrast accumulation in the oral side (A, B). The emergency colonoscopy revealed ischemic changes in the colonic mucosa but no signs of necrosis (C). A transanal drainage tube was inserted for decompression (D). Macroscopic examination of the resected specimen revealed a 10-cm retrograde intussusception of the descending colon (A, B). Multiple ulcers were observed in the overlapping area (C). Microscopic findings revealed that the ulcers were confined to the muscle layer, with mucosal necrosis, submucosal hemorrhage, edema, and neutrophilic infiltration around the ulcer (arrowhead) (D).Figure 5.. Macroscopic examination of the resected specimen revealed a 10-cm retrograde intussusception of the descending colon (A, B). Multiple ulcers were observed in the overlapping area (C). Microscopic findings revealed that the ulcers were confined to the muscle layer, with mucosal necrosis, submucosal hemorrhage, edema, and neutrophilic infiltration around the ulcer (arrowhead) (D). Two possible mechanisms of retrograde intussusception resulting from colonoscopy. First, the elasticity of the sigmoid colon may have caused it to invaginate the dilated descending colon when the sigmoid colon was stretched by hooking the tip of the camera to the splenic flexure (A, B). Second, friction during pushing may have pushed the sigmoid colon mucosa into the fixed descending colon (C, D).Figure 6.. Two possible mechanisms of retrograde intussusception resulting from colonoscopy. First, the elasticity of the sigmoid colon may have caused it to invaginate the dilated descending colon when the sigmoid colon was stretched by hooking the tip of the camera to the splenic flexure (A, B). Second, friction during pushing may have pushed the sigmoid colon mucosa into the fixed descending colon (C, D).

References:

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13.. Hirasawa K, Sato C, Makazu M, Coagulation syndrome: delayed perforation after colorectal endoscopic treatments: World J Gastrointest Endosc, 2015; 7; 1055-61

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Figures

Figure 1.. Abdominal computed tomography (CT) revealed marked dilatation of the right (arrowhead) and sigmoid colon (red arrow), and no obstructive origin was identified (A, B). Colonoscopy revealed no torsion or obstruction in the colon up to the splenic flexure (C, D).Figure 2.. Abdominal computed tomography (CT) revealed a collapsed sigmoid colon (red arrow) and dilatation of the right colon (arrowhead) (A, B). Colonoscopy with the tip of the endoscope hooked to the splenic flexure to stretch the sigmoid colon (C, D). Enterography revealed no causative structures in the transverse colon (E). A steep discontinuity of contrast was observed in the descending colon at this time (yellow arrow) (F).Figure 3.. Enterography using a transanal drainage tube showed no apple core signs from the transverse colon to the hepatic flexure (A), and no contrast medium flowed into the descending colon (B).Figure 4.. Abdominal computed tomography (CT) revealed that the sigmoid colon retrogradely invaginated the descending colon, with contrast accumulation in the oral side (A, B). The emergency colonoscopy revealed ischemic changes in the colonic mucosa but no signs of necrosis (C). A transanal drainage tube was inserted for decompression (D).Figure 5.. Macroscopic examination of the resected specimen revealed a 10-cm retrograde intussusception of the descending colon (A, B). Multiple ulcers were observed in the overlapping area (C). Microscopic findings revealed that the ulcers were confined to the muscle layer, with mucosal necrosis, submucosal hemorrhage, edema, and neutrophilic infiltration around the ulcer (arrowhead) (D).Figure 6.. Two possible mechanisms of retrograde intussusception resulting from colonoscopy. First, the elasticity of the sigmoid colon may have caused it to invaginate the dilated descending colon when the sigmoid colon was stretched by hooking the tip of the camera to the splenic flexure (A, B). Second, friction during pushing may have pushed the sigmoid colon mucosa into the fixed descending colon (C, D).

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American Journal of Case Reports eISSN: 1941-5923
American Journal of Case Reports eISSN: 1941-5923