Adult juvenile polyp bleeding detected by extravascular contrast leakage and treated with endoscopic clipping: A case report

Abstract

BACKGROUND

Juvenile polyps (JPs) are non-neoplastic polyps. In adults, JPs present with hematochezia in only approximately half the patients and are often found incidentally during endoscopic screening. JPs have no mucosal fascia at the tip, and spontaneous shedding and massive gastrointestinal hemorrhage may occur. Thus, the JP bleeding detected in this case by extravascular contrast leakage on computed tomography scans and treated with endoscopic clipping is rare.

CASE SUMMARY

A previously healthy 31-year-old male patient presented with a 2-day history of bloody stools. Upon hospital arrival, rectal examination revealed fresh blood, and abdominal computed tomography scans showed extravascular contrast leakage from the lower rectum’s left-side wall. His blood pressure was slightly low at 104/62 mmHg. However, his pulse rate (69 bpm) and oxygen level (99% on room air) were within normal limits. Emergency endoscopy revealed a pedunculated lesion in the rectum covered by a non-neoplastic mucosal epithelium. No neoplastic lesions were observed at the tip of the polyp; however, pulsatile bleeding was detected at the distal end. We performed endoscopic hemostasis by clipping the stem and then performed a polypectomy above the stem to examine the lesion tissue. Histopathological evaluation revealed a cystically dilated gland without neoplastic lesions. A subsequent total colonoscopy revealed two JPs with characteristic edematous, smooth, and reddish surfaces close to the hemorrhagic lesion. Subsequent histopathological evaluation indicated findings characteristic of JP, such as severe inflammatory cell infiltration of the stroma and cystic dilatation of the glandular ducts.

CONCLUSION

There are no reports of adult JPs presenting with contrast extravasation where endoscopic hemostasis was successful, as in this case.

Key Words: Contrast-enhanced computed tomography; Endoscopic clipping; Juvenile polyp; Rectal bleeding; Contrast extravasation; Case report

Core Tip: A 31-year-old male presented with hematochezia lasting for 2 days. computed tomography scans showed extravasation, and endoscopy was used to confirm and stop bleeding from the stem of a juvenile polyp (JP). Emergency endoscopy revealed persistent bleeding from the base of the polyp, and diagnostic polypectomy and hemostasis was performed. JPs are often detected with bloody stools. However, there have been no reports of JPs with active bleeding seen on computed tomography scans as extravasation. In this rare case, an endoscopic video showed persistent bleeding, and successful endoscopic hemostasis was achieved, and captured on video.

INTRODUCTION

Juvenile polyps (JPs) are categorized as non-neoplastic polyps. Approximately 80%-90% of JPs are located in the sigmoid colon and rectum and are most common in children aged 3-10 years, although many adult cases have been reported[1,2]. In terms of the trigger for detection, most childhood cases were found to present with hematochezia, whereas in adults, hematochezia is reported in only approximately half of all cases and is often found incidentally during endoscopy screening[1,3,4]. As JPs have no mucosal fascia growth at the tip of the polyp, spontaneous shedding and massive gastrointestinal hemorrhage may occur[5]. However, according to our research, no such reports were found upon searching PubMed using the keywords “juvenile polyp,” “computed tomography,” and “extravascular leakage.” Thus, the JP bleeding detected in this case by extravascular leakage of contrast medium on computed tomography (CT) scans and treated with endoscopic clipping is rare.

CASE PRESENTATION

Chief complaints

A previously healthy 31-year-old male patient presented with a 2-day history of bloody stools.

History of present illness

He stayed home but then suffered from a large volume of fresh bloody stools and was referred to our hospital after visiting his family doctor.

History of past illness

He denied any personal history of illness.

Personal and family history

There were no specific findings in his medical history or medications and no family history of JPs.

Physical examination

The patient’s vital signs were stable, with no obvious abnormalities in the physical examination; his abdomen was flat and soft, with no abdominal tenderness. Shifting dullness in the abdomen was negative. A large volume of red stools was reported.

Laboratory examinations

At the time of presentation, the patient was not anemic and had a serum hemoglobin (Hb) level of 13.7 g/dL (normal range, 13.7-16.8 g/dL).

Imaging examinations

Contrast-enhanced abdominal CT scans revealed extravascular leakage of contrast medium from the left wall of the lower rectum (Figure 1).

Figure 1 Contrast-enhanced abdominal computed tomography scan. Contrast-enhanced abdominal computed tomography scan shows extravascular leakage of the contrast medium from the left wall of the lower rectum (red arrow).

FINAL DIAGNOSIS

The final diagnosis was JPs.

TREATMENT

We performed an emergency colonoscopy and found a pedunculated polyp on the left wall of the lesion below the peritoneal reflection. The stalk was covered by non-neoplastic mucosal epithelium, and its tip was buried in a blood clot. After the clot was removed, pulsatile bleeding was observed at the dissected end of the polyp. We performed endoscopic hemostasis by clipping the stem and then performed polypectomy above the clip to examine the tissue (Figure 2, Video 1). Histopathological evaluation of the stem section revealed large blood vessels on hematoxylin and eosin staining, and some glandular ducts were dilated on the cyst, which is characteristic of JPs. We also confirmed the presence of the lamina muscularis mucosae using desmin staining. There were no findings of tearing or disruption of the lamina due to malignancy (Figure 3).

Figure 2 Endoscopic hemostasis and polypectomy. A: Pulsatile bleeding is observed from the stem of the polyp; B: Endoscopic hemostasis is performed by clipping the stem; C: Polypectomy is then performed above the clip to examine the tissue; D: The clips are added to completely stop the bleeding.

Figure 3 Histopathological evaluation. A: Hematoxylin and eosin staining showing large blood vessels in the stalk (red box) and some dilated glandular ducts on the cyst (yellow arrowheads); B: Desmin staining showing intricate lamina muscularis mucosae. There are no signs of malignancy, such as tearing or disruption of the lamina.

OUTCOME AND FOLLOW-UP

On the fourth day of hospitalization, the patient’s serum Hb level dropped to 10.4 g/dL, although no fresh bloody stools were observed. On the sixth day of hospitalization, we performed a total colonoscopy and found two semi-pedunculated polyps, approximately 15 mm in diameter, near the bleeding lesion in the rectum (Figure 3). The polyps were intensely erythematous, had smooth surfaces and erosions, and were moss-white in color. Narrow-band imaging revealed that the glands were dilated, with no irregular vessels or surface patterns, confirming diagnosis for the Japan narrow-band imaging expert team classification type 1 (Figure 4). As these polyps showed characteristic JP findings, we performed endoscopic mucosal resection. Histopathology revealed high inflammatory cell infiltrates and cystically dilated glandular formations filled with mucus. However, a lack of lamina muscularis mucosae in the head of the polyp further confirmed they were JPs (Figure 5). The patient showed favorable progress after the endoscopic procedure and was discharged on the seventh day of hospitalization. Subsequently, we performed an upper gastrointestinal endoscopy that revealed no polyps in the stomach. As the possibility of juvenile polyposis syndrome (JPS) could not be ruled out, we recommended an examination of the small intestine. However, the patient did not wish to undergo further examinations.

Figure 4 Semi-pedunculated polyps near the clipped bleeding site in the rectum. A: Two semi-pedunculated polyps, approximately 15 mm in diameter (arrow 1 and arrow 2), are visible near the clipped bleeding site in the rectum; B and C: The polyps are intensely erythematous and have smooth surfaces and erosions with moss-white colorings; D and E: Narrow band imaging reveals dilated glands.

Figure 5 Histopathological evaluation of juvenile polyps. A: Glandular ducts with dilated lumens distributed in the polyp; B: At the stem of the polyp (blue box in panel A, the lamina muscularis mucosae are present (dotted circle); C: Whereas in the head (green box in panel A), the lamina muscularis mucosae are lacking and a high inflammatory cell infiltrate is present (dotted circle).

DISCUSSION

JPs were first described by Horrilleno et al[6] in 1957. As previously mentioned, most pediatric cases (under 10 years of age) present with hematochezia, whereas in adult cases, hematochezia is mainly detected incidentally by colonoscopy. Endoscopic features are described as pedunculated, with smooth or granular surfaces and reddish-colored polyps with slight irregularities. They often show erosions, mucous membranes, and hemorrhages[3]. Histological findings are characterized by cystically dilated glandular formations filled with mucus. The differential diagnoses of JPs on endoscopy include adenomas, familial adenomatous polyposis, and inflammatory polyps[2].

Colonic polyps are a rare cause of bloody stools. According to a large Japanese study on acute hematochezia, the most common cause of hematochezia was diverticular hemorrhage (63.6%), followed by ischemic enteritis (9.1%), and only 0.4% of cases were caused by colorectal polyps. The mechanism of hematochezia of JPs with stroma is considered as follows: Stromal polyps have large blood vessels inside[7], and JPs tend to shed spontaneously and bleed profusely because the mucosal muscle plate is not embedded in the polyp head. In the case presented herein, although the head of the polyp was already detached, a large arteriovenous vein was running from the tissue of the detached transected edge and a surrounding cystic duct with dilated glandular ducts. This suggested that the head of the stemmed JP had spontaneously detached and presented with active bleeding. The rarity of this case lies in the fact that hemorrhage due to JPs was first confirmed by extravascular leakage observed on a CT scan. Considering that the frequency of extravascular leakage on contrast CT scans for hematochezia evaluations is reported to be 22% and that the aforementioned colonic polyps are the cause of 0.4% of hematochezia, one can see how unusual it is that bleeding from a colonic polyp is detected by extravascular leakage of contrast media on CT[8]. Generally, contrast-enhanced CT scans can identify the source of bleeding if the bleeding is greater than 0.5 mL/minute[9]. In this case, the patient had experienced hematochezia for 2 days, indicating that the bleeding had persisted for at least 30 hours. Furthermore, by the fourth day after hospitalization, the patient’s Hb level had decreased by 3.3 g/dL. Considering the amount of bleeding detected by CT scans[9] and given that the patient weighed 60 kg, the total volume of blood loss was estimated to be approximately 1 L.

Finally, we discuss the association between JP and cancer. Cases of JPs are usually solitary. However, cases with multiple polyps require additional examination for JPS. Patients with JPS have a high risk of developing gastrointestinal cancers, especially gastric and colorectal cancers, with an estimated cumulative risk of 9%-68%. Therefore, routine surveillance is required for patients with JPS, considering the risk of cancer[10]. JPS is diagnosed in the presence of any of the following: More than five JPs in the colorectum, multiple JPs throughout the gastrointestinal tract, or any number of JPs, and a family history of JPS. However, a single JP is believed not to increase the risk of cancer[3]. Thus, gastrointestinal surveillance is typically considered unnecessary in the absence of a family history of JPS[4,11]. However, some case reports have described adenomas or adenocarcinomas on JPs. Giardiello et al[12] reported that multiple JPs or a family history of JPs increases the risk of colon neoplasia. Thus, total colonoscopy is recommended for these patients. In the present case, at least three JPs were identified, suggesting that routine surveillance will be necessary.

A limitation of this case is that the head of the hemorrhagic pedunculated lesion had auto-amputated, and we could not establish the complete polyp form. However, the histopathology of the stalk showed dilated glandular ducts, a finding consistent with JP histology.

CONCLUSION

Although JPs are often encountered during screening endoscopies, we believe that cases of active hemorrhage observed on CT scans, as in this case, are rare. JP is often characterized by hematochezia and sometimes induces hemorrhagic shock. However, there have been no previous reports of JP in adults presenting with contrast extravasation on CT scans and where endoscopic hemostasis was successfully performed for active bleeding, as in this very rare case.