Endoscopic and laparoscopic treatment of ileocecal laterally spreading tumor with concomitant appendiceal adenoma: A case report and review of literature

Abstract

BACKGROUND

Ileocecal laterally spreading tumors (LSTs) complicated by appendiceal tubular adenoma are rare and challenging to diagnose because of the absence of typical symptoms and specific diagnostic signs. Traditionally, the primary treatment has been laparoscopic appendectomy (LA).

CASE SUMMARY

A 63-year-old female presented with changes in bowel habits. Colonoscopy revealed an ileocecal LST. The patient underwent endoscopic submucosal dissection. Postoperative follow-up colonoscopy revealed mucosal elevation at the appendiceal orifice, with pathology confirming tubular adenoma. Abdominal computed tomography indicated a suspicious appendiceal tumor, leading to LA with partial cecectomy. The postoperative recovery was uneventful. At the 1-year follow-up, colonoscopy revealed no evidence of tumor recurrence.

CONCLUSION

Ileocecal LSTs with appendiceal tubular adenomas are traditionally treated with LA. endoscopic submucosal dissection can also yield favorable outcomes.

Key Words: Appendiceal tubular adenoma; Ileocecal laterally spreading tumor; Appendiceal laterally spreading tumor; Endoscopic submucosal dissection; Laparoscopic appendectomy; Case report

Core Tip: Ileocecal laterally spreading tumors with appendiceal tubular adenoma are rare and hard to diagnose due to nonspecific symptoms. Traditionally treated by laparoscopic appendectomy, new research shows endoscopic submucosal dissection can be effective. This case innovatively applied sequential endoscopic submucosal dissection for high-grade dysplastic lesions at the appendiceal orifice, followed by appendectomy preserving the ileocecal valve. This approach improved postoperative quality of life and presents a novel strategy for deep-seated appendiceal lesions, addressing visualization challenges in deep lumen extension.

INTRODUCTION

Laterally spreading tumors (LSTs) are critical lesions in colorectal cancer, with a prevalence of 0.8% in the general population undergoing colonoscopy[1]. Appendiceal tumors are rare[2]. Collins reported that among 280000 appendectomies performed in the United States, the incidence of appendiceal tumors ranged between 0.9% and 1.4%[3], with noncancerous appendiceal tumors accounting for 0.02%-0.08%[4]. Benign appendiceal tumors are often asymptomatic and incidentally detected during clinical examinations, surgery, or pathological evaluation after appendectomy, whereas malignant tumors may cause symptoms related to regional involvement, peritoneal dissemination, or metastatic disease, with acute appendicitis being the most common initial presentation[5]. We report a case of ileocecal LST complicated by an appendiceal tubular adenoma detected during a routine physical examination and managed with endoscopic submucosal dissection (ESD) and laparoscopic appendectomy (LA).

CASE PRESENTATION

Chief complaints

A 63-year-old female presented to The Affiliated Hospital of Qingdao University with a chief complaint of “changes in bowel habits for six months”.

History of present illness

Colonoscopy revealed a 5 cm × 6 cm nodular-mixed type LST in the ileocecal region (Figure 1), with the largest nodule measuring approximately 1 cm in diameter. The appendiceal orifice was not visualized during colonoscopy, suggesting it was covered by the lesion. Pathological biopsy of the lesion revealed a tubulovillous adenoma with high-grade intraepithelial neoplasia in some areas. Given the large size of the lesion and the potential risk of invasive colon cancer, surgical resection, which typically requires right hemicolectomy, was considered. However, to avoid complications such as abdominal pain and diarrhoea associated with right hemicolectomy and maximize the preservation of colonic and ileocecal valve function, ESD of the ileocecal LST was initially performed. The pocket-creation method is used during the dissection, and we did not used any traction devices or methods to facilitate the dissection.

Figure 1 Imaging manifestations. A: Endoscopic images of the lesion before endoscopic submucosal dissection (ESD); B: Magnifying endoscopy with narrow-band imaging images of the lesion before ESD; C: Endoscopic images after ESD; D: Photographs of longitudinal sections of the gross appendix specimen; E: Endoscopic images of longitudinal sections of the gross appendix specimen; F and G: Magnifying endoscopy with narrow-band imaging images of longitudinal sections of the gross appendix specimen; H: Endoscopic images 1 year after laparoscopic appendectomy showed no signs of recurrence.

During ESD, the lesion was found to extend into the appendiceal orifice. The mucosal tissue invading the appendix was transected endoscopically, and biopsies were taken from the residual tissue at the appendiceal orifice. The final pathology of the ESD specimen revealed a tubulovillous adenoma with high-grade dysplasia and focal carcinoma, and the histologic grade is moderate differentiated (Figure 2). Surgical margins (lateral and basal) were negative. Biopsies of the appendiceal orifice mucosa revealed chronic inflammation with focal adenomatous hyperplasia. The lesion was diagnosed as early colon cancer, obviating the need for radical colectomy. Although adenomatous tissue was suspected to remain within the appendix, appendiceal ultrasound and abdominal computed tomography (CT) did not detect mucosal thickening of the appendix. After discussion with the patient, LA was planned after wound healing.

Figure 2 The final pathology of the endoscopic submucosal dissection. A: Photomicrograph ileocecal laterally spreading tumor (haematoxylin and eosin, original magnification × 20); B: Photomicrograph tubular adenoma of the appendix (haematoxylin and eosin, original magnification × 20).

Five months later, follow-up colonoscopy revealed good healing at the surgical site. An 8 mm × 8 mm mucosal elevation was noted at the appendiceal orifice, which was clearly visualized (Figure 1). Biopsy confirmed the presence of a tubular adenoma at the appendiceal opening. LA with partial cecectomy was performed. No lymph node dissection was performed, as neither preoperative contrast-enhanced CT nor intraoperative findings showed abnormal lymph nodes. Gross examination of the resected specimen revealed the appendix with a small amount of adjacent ileocecal mucosa. Upon longitudinal sectioning of the appendix (Figure 1) and magnifying observation with narrow-band imaging, adenomatous structures were found in approximately 50% of the appendiceal lumen near the orifice (consistent with endoscopic classification; Figure 1). Final pathology confirmed a tubular adenoma with negative resection margins (Figure 2). The patient was discharged on postoperative day 3. A one-year follow-up CT scan revealed no enlarged abdominal lymph nodes, and colonoscopy confirmed well-healed mucosa (Figure 1). The timeline of diagnosis and treatment is listed in Figure 3.

Figure 3  Timeline of diagnosis and treatment.

History of past illness

The patient did have past illness.

Personal and family history

The patient did have personal and family history.

Physical examination

Inspection: The abdomen is symmetric in shape, without distension or depression. Abdominal respiration is present. There is no varicose vein on the abdominal wall, and no rash, scar, gastrointestinal pattern, or peristaltic wave is observed. Palpation: The abdominal wall is soft without muscular tension. No tenderness, rebound tenderness, or abnormal mass is detected throughout the abdomen. The liver and spleen are not palpable below the costal margin. The Murphy’s sign is negative, and there is no percussion pain in the renal area. Percussion: The hepatic dullness boundary is present, located from the 5^th-6^th intercostal space at the right midclavicular line to the lower edge of the right costal arch. Shifting dullness is negative. Auscultation: Bowel sounds are normal, with a frequency of about 4-5 times per minute. No vascular murmur or friction sound is heard.

Laboratory examinations

No obvious abnormalities were found in the patient’s laboratory examinations.

Imaging examinations

March 20, 2023: Colonoscopy (flexible fiberoptic endoscopy of the colon) revealed an approximately 5 cm × 6 cm LST at the appendiceal orifice region of the cecum. August 4, 2023: Colonoscopy showed good healing of the postoperative wound. At the appendiceal orifice, there was a mucosal elevation approximately 8 mm × 8 mm in diameter, and the appendiceal orifice was visible. October 23, 2023: Colonoscopy revealed a mucosal elevation approximately 8 mm × 6 mm in diameter at the appendiceal orifice, with surrounding scarring. October 23, 2023: Dynamic enhanced CT scan of the lower abdomen: The proximal part of the appendix shows uneven thickening with mild dilation of the appendiceal cavity. Slightly enlarged lymph node shadows are seen medially in the ileocecal region. A tumor in the appendiceal area is suspected. Please make a comprehensive diagnosis combined with clinical history and colonoscopy. October 30, 2024: Colonoscopy showed no obvious abnormalities.

FINAL DIAGNOSIS

Ileocecal LST with concomitant appendiceal adenoma.

TREATMENT

March 24, 2023: ESD was performed for the ileocecal LST. October 24, 2023: LA and partial cecectomy were performed.

OUTCOME AND FOLLOW-UP

The appendiceal adenoma was completely resected. And one-year follow-up after surgery showed no obvious abnormalities on colonoscopy.

DISCUSSION

This case highlights a rare presentation of an ileocecal LST complicated by an appendiceal tubular adenoma. While ileocecal LSTs are not uncommon, their extension into the appendix is rare. Appendiceal epithelial tumors are observed in 0.2%-0.3% of appendectomy samples, most frequently in individuals aged 50-70 years[6], as in our case. Additionally, tubular adenoma is a rare histological type of nonmucinous epithelial tumor, accounting for only 30% of all appendiceal epithelial tumors[7].

Appendiceal tumors are often difficult to diagnose. Indeed, in most cases, these lesions are asymptomatic and incidentally detected during surgery or pathological examination of surgical samples. A review of appendiceal adenomas indicated that over 93% of these benign lesions are diagnosed postappendectomy or at autopsy[8], with occasional incidental diagnosis during radiological or endoscopic evaluation for other abdominal conditions[9]. In radiological imaging, ultrasound is a valuable modality for detecting appendiceal tumors but is highly operator-dependent and limited in its ability to assess tumor staging. Abdominal CT with or without intravenous and oral contrast is the most accurate method for detecting appendiceal tumors, with a sensitivity of up to 95%[10]. Endoscopic diagnosis is more feasible when a tumor involves the appendiceal orifice and extends to the caecal mucosa. In our case, the patient’s ileocecal LST with appendiceal extension was identified during endoscopy for changes in bowel habits.

After the diagnosis of an appendiceal adenoma, treatment is critical to avoid potential complications such as metastasis, appendicitis, or vomiting. Surgery is widely considered as the gold standard for managing appendiceal tumors[11-13]. However, surgical resection is limited by the challenge of ensuring negative margins without compromising the ileocecal valve. The incidence of complications increases further with more extensive procedures, such as ileocecectomy or right hemicolectomy[14].

With advancements in endoscopic technology, is being applied to regions once considered too complex for endoscopic management. ESD is widely used in many specialized centers for treating superficial colonic neoplasia owing to its lower incidence of postoperative adverse events than that of traditional surgery[15]. Lesions near the appendiceal orifice (L-PAO) were initially deemed unresectable by endoscopy because of the high risks of incomplete resection, perforation, and acute appendicitis[16,17]. However, with increasing experience, endoscopic treatment of L-PAO has been demonstrated to be safe and effective[18,19].

Endoscopic mucosal resection (EMR) has been described for managing lesions involving the appendiceal orifice. Tate et al[20] reported favorable outcomes with EMR; however, for most lesions in their series, endoscopic management was not attempted because of deep extension into the appendiceal lumen or involvement of > 50% of the orifice circumference. Traction techniques may aid in exposing lesion margins[21,22], but visualization remains challenging if the lesion extends deeply into the appendiceal lumen, even with such techniques. Some researchers have proposed underwater EMR for adenomas involving the appendiceal orifice, but its efficacy remains suboptimal in patients with deep luminal extension[22-24].

Historically, for lesions extending deeply into the appendiceal orifice (as in this case), full-thickness resection with a disc-shaped snare has been considered an ideal endoluminal approach, as retracting the lesion into a cap allows resection to deeper portions of the orifice. For lesions > 2 cm in diameter, endoscopic full-thickness resection can also be combined with prior EMR via a “hybrid” technique to ensure complete lateral excision[25]. However, the risk of appendicitis associated with full-thickness resection with a disc-shaped snare cannot be ignored: Two studies by Schmidbaur et al[26] and Ichkhanian et al[27] reported that 15%-17% of patients develop appendicitis, with 50% of patients requiring urgent appendectomy. Recent reports also suggest that this approach may lead to peritoneal seeding of adenomatous tissue in cases of incomplete resection[28].

The most recent study by Jacob et al[29] demonstrated that ESD for L-PAO achieved an overall en bloc resection rate of 94.7%, with both perforation and postoperative bleeding rates at 1.3%. Thus, ESD for L-PAO offers safety and efficacy comparable to those of ESD in extra-appendiceal orifice regions. This study included three lesions extending deep into the appendiceal orifice, all of which were resected en bloc; however, all three patients had a history of prior appendectomy. In a separate study, Oung et al[30] evaluated ESD with double clips and rubber band traction in 11 type 3 Lesions in nonappendectomized patients, achieving en bloc resection for all patients. However, there were two cases of non-R0 resection, six perforations (54.5%), one case of delayed local peritonitis resolved with antibiotics, and one patient requiring additional surgery for peritonitis. These studies concluded that caecal lesions not involving the appendiceal orifice can be resected like any other colonic lesion can, whereas deeper invasion into the appendix significantly increases the risk of incomplete resection and complications.

An alternative approach is to recommend combined endoscopic and laparoscopic surgery, which addresses challenges in identifying lesion margins, ensures en bloc resection, and improves R0 resection rates. Multiple studies[31-33] have confirmed that combined endoscopic and laparoscopic surgery is a safe procedure with favorable clinical outcomes and low recurrence rates. However, its application in nonbenign lesions is limited because of the challenge of ensuring negative margins while minimizing the resection scope.

In the present case, the physician innovatively performed ESD for the ileocecal region and appendiceal orifice first, followed by LA with preservation of the ileocecal valve for a type 3 Lesion. Preoperative endoscopic treatment of the ileocecal mass ensured negative margins and provided high-quality specimens for pathologists to accurately stage the resected lesion, guiding further management (postoperative follow-up vs surgical resection). Multiple endoscopic biopsies and ESD prior to LA confirmed adenoma at the appendiceal orifice, successfully preserving the ileocecal valve, reducing postoperative complications, and improving quality of life.

This case demonstrates that laparoscopic surgery following endoscopic ESD for appendiceal adenoma is safe and effective in achieving curative outcomes. However, a thorough histopathological examination is mandatory postoperatively. If appendectomy pathology reveals cancer, curative resection and lymph node dissection following colorectal cancer guidelines are needed. Additionally, patients should be counselled on the risks of cancer-related complications or the need for further resection after appendectomy.

CONCLUSION

An ileocecal LST with an appendiceal tubular adenoma is a clinically rare condition that is often incidentally detected during surgery. The preoperative diagnosis is limited by the frequent absence of symptoms and nonspecific diagnostic signs. With the advancement of endoscopic techniques and their lower postoperative complication rates, colorectal tumor lesions invading the appendiceal orifice should no longer be considered contraindications for endoscopic resection. Lesions should be classified according to the degree of invasion into the appendiceal orifice to guide the selection of different resection strategies.