Letter to the Editor Open Access
Copyright ©The Author(s) 2024. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. Sep 26, 2024; 12(27): 6124-6128
Published online Sep 26, 2024. doi: 10.12998/wjcc.v12.i27.6124
When the vermiform appendix resembles a polyp: Be cautious of an intussuscepted appendix polypectomy
Raffaele Pellegrino, Antonietta Gerarda Gravina, Division of Hepatogastroenterology, Department of Precision Medicine, University of Campania Luigi Vanvitelli, Naples 80138, Italy
ORCID number: Raffaele Pellegrino (0000-0001-5074-230X); Antonietta Gerarda Gravina (0000-0001-8049-0115).
Author contributions: Pellegrino R and Gravina AG collected the literature, wrote the initial manuscript, contributed equally to this work, conceptualized the structure of the text, critically revised the manuscript for important intellectual content, and read and approved the final version of the manuscript.
Conflict-of-interest statement: The authors have no conflicts of interest to declare.
Open-Access: This article is an open-access article that was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution NonCommercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See: https://creativecommons.org/Licenses/by-nc/4.0/
Corresponding author: Antonietta Gerarda Gravina, MD, PhD, Associate Professor, Division of Hepatogastroenterology, Department of Precision Medicine, University of Campania Luigi Vanvitelli, Via L. de Crecchio, Naples 80138, Italy. antoniettagerarda.gravina@unicampania.it
Received: February 20, 2024
Revised: July 5, 2024
Accepted: July 10, 2024
Published online: September 26, 2024
Processing time: 161 Days and 8.9 Hours

Abstract

This article discusses a recently published case report on a rare instance of type IV appendiceal intussusception with a concurrent mucinous adenocarcinoma of the cecum in a young individual. The report highlights challenges in diagnosing appendiceal intussusception, emphasizing the importance of endoscopic expertise in preventing impulsive decisions such as inappropriate polypectomies. The rarity of the concurrent intussuscepted appendix and mucinous cecal cancer is underscored, prompting consideration of malignancy in appendiceal intussusception cases. Additionally, the report addresses the increasing incidence of early-onset colorectal cancer and the need for a revaluation of diagnostic paradigms in the context of evolving epidemiological trends. The awareness of potential misinterpretations and the imperative for further investigation into this rare condition are emphasized.

Key Words: Appendiceal intussusception; Colorectal cancer; Early-onset colorectal cancer; Digestive endoscopy; Abdominal pain; Bloody stools; Diarrhea

Core Tip: This article examines a recently published case report detailing a unique instance of type IV appendiceal intussusception complicated by mucinous adenocarcinoma of the cecum in a 20-year-old individual. The report emphasizes diagnostic challenges, particularly in distinguishing appendiceal intussusception from polyps, and underscores the necessity for endoscopic expertise to guide appropriate interventions. Furthermore, it discusses the rarity of concurrent occurrences of intussuscepted appendix and mucinous cecal cancer, highlighting the importance of considering malignancy in such cases. The article also addresses the increasing incidence of early-onset colorectal cancer, advocating for a reassessment of diagnostic approaches in light of evolving epidemiological trends.



TO THE EDITOR

We have perused with keen interest the report “Appendiceal intussusception complicated by adenocarcinoma of the cecum: A case report”[1], recently published in the World Journal of Clinical Cases. This noteworthy case elucidates a singular instance of a type IV[2] appendiceal intussusception involving the vermiform appendix ensconced within the cecum, juxtaposed with the presence of a mucinous adenocarcinoma of the cecum in a young individual of 20 years.

The clinical presentation of the patient was characterized by abdominal pain, bloody stool, diarrhea, nausea, and vomiting. Concurrently, notable anemia was documented, with hemoglobin values falling below 100 g/L. A precedent colonoscopy performed at an alternative medical facility yielded a diagnosis of the ileocecal lesion with high-grade adenomatous dysplasia and focal malignant transformation following biopsy sampling during endoscopic examination.

Subsequently, the patient underwent a secondary colonoscopy under the auspices of the authors, revealing a digitiform lesion within the cecal lumen. Biopsy sampling of the contiguous cecal mucosa by the authors culminated in identifying mucinous adenocarcinoma of the cecum.

The diagnosis of appendiceal intussusception was only established following the right hemicolectomy with regional lymphadenectomy, as confirmed by histological examination of the surgical specimen. This examination further identified a locally advanced cancer stage (i.e. pT3N1M0 or pIII).

WHAT INSIGHTS CAN BE DRAWN FROM THIS CASE

This intriguing report provides several points for reflection. The authors initially subjected the patient to a radiological assessment using computed tomography, which reported thickening of the ileocecal wall. However, the radiologist only discerned clear signs of intussusception through retrospective post-operative evaluation.

In the context of appendiceal intussusception, the diagnostic performance of computed tomography has been a subject of limited exploration, with only a few instances among the numerous reported cases being diagnosed prospectively[3]. Computed tomography exhibits diagnostic efficacy by identifying a target or sausage-shaped lesion encompassing the appendix. This characteristic appearance, known as the target sign, is attributed to concentric soft tissue and fat layers, delineating the involved bowel wall and the intussuscepted appendiceal fat.

As a result, this underscores how cross-sectional radiological evaluation, as rightfully emphasized by the authors, must be considered in cases of abdominal pain affecting the ileocecal region, particularly in the context of a previously identified ileocecal lesion during the index colonoscopy.

Indeed, the index colonoscopy played a crucial role, as the authors' subsequent endoscopic assessment involved a biopsy of the finger-like lesion (i.e. the intussuscepted appendix), which had initially been misinterpreted as a hyperplastic polyp. Moreover, the prior identification of declared dysplasia with focal cancerous aspects during the index colonoscopy likely dissuaded the authors from performing a polypectomy on the intussuscepted appendix, given their predisposition towards recommending a hemicolectomy for the patient. Had such a polypectomy been undertaken, it would have entailed a highly precarious "endoscopic appendectomy" with a considerable risk of intestinal perforation and iatrogenic peritonitis[4].

Regrettably, such occurrences may transpire because intussusception of the vermiform appendix can be easily misinterpreted as a polypoid lesion of the cecum[5]. This misinterpretation may lead practitioners to erroneously opt for a “polypectomy” instead of a more appropriate endoscopic intervention, such as, when feasible, per endoscopic reduction[3,5,6].

Additionally, this report stimulates discussion regarding the imperative for endoscopic expertise to guide and preclude such impulsive endoscopic decisions. This consideration is particularly pertinent when contemplating the atypical appearance of the "pseudo-polypoid" lesion associated with appendiceal intussusception, as also evident from the endoscopic image provided by the authors in their report. The atypical nature of this lesion, precluding a precise endoscopic diagnosis even with the aid of virtual chromoendoscopy techniques, underscores the importance of distinguishing it from a "typical" pedunculated polyp formation (i.e. 0-Ip according to the Paris classification[7]).

Nevertheless, the issue of polypectomies performed on intussuscepted appendices is a real concern, as several cases of inverted appendices mistaken for polyps have been documented[8].

Moreover, this case report underscores the rarity of the concurrent occurrence of intussuscepted appendix and mucinous cecal cancer. The inverse scenario, namely intussusception of the cecum with a mucinous tumor of the appendix, has also been documented[9]. Furthermore, when appendiceal intussusception is diagnosed, the potential for malignancy within its context should still be duly considered[10,11].

Additionally, there should probably have been a consideration in the discussion of this report for another aspect: the remarkably young age of the patient diagnosed with a malignant colon neoplasm, notably locally advanced with lymph node metastasis.

An alarming surge in the incidence of colorectal cancer is observed among individuals below the age of 50, delineating the phenomenon known as early-onset colorectal cancer[12]. This demographic, often excluded from widespread international screening initiatives, demonstrates a predilection for early-onset colorectal cancer, predominantly within the distal colon and rectum, frequently manifesting with obstructive symptoms[12]. Furthermore, individuals in this younger cohort tend to present with a comparatively advanced disease stage at the point of diagnosis in contrast to their counterparts aged over 50. Molecularly, these neoplasms exhibit a predilection for poor differentiation coupled with microsatellite instability[12]. Projections indicate that within the ensuing decade, 1/10 to 1/4 of colorectal cancer diagnoses will comprise early-onset colorectal cancer occurring before the age of 50[12]. Putative contributors to this trend encompass obesity, antibiotic usage, intestinal microbiota, and dietary patterns[12,13].

Conclusively, the focus is directed towards this case of cecal cancer, undeniably aligning with the classification of early-onset colorectal cancer. A thorough examination of the anamnestic details provided by the authors did not disclose a clear presence of comorbidities or a significant family history of gastrointestinal pathologies. Unfortunately, the report lacks information on whether the patient was obese, at the very least, engaged in alcohol consumption or smoking habits. Nevertheless, no molecular analyses conducted on the surgical specimen were disclosed in the report. The authors outlined an adjuvant chemotherapy plan (specific therapeutic agents unspecified).

As demonstrated in the authors' report, this evolving shift in epidemiological paradigms frequently challenges clinicians. In the case of a 25-year-old female presenting with nearly 1 mo of abdominal pain and bloody diarrhea, the initial inclination would typically be towards considering inflammatory bowel disease rather than colorectal cancer[14]. Another diagnostic hypothesis could have been infectious colitis[15]. Nevertheless, the objective finding of right lower quadrant abdominal pain, apart from radiological considerations, might have leaned more towards acute appendicitis in this age group. Notwithstanding, the intussusception of the appendix can easily be associated with appendicitis[16-19].

In other words, this report rekindles the awareness that certain epidemiological certainties guiding the exclusion of specific diagnoses must be reconsidered in these profound epidemiological shifts.

Certainly, as described by the authors, the appendix intussusception, in this case, is likely secondary to a mechanical factor associated with the growth of the cecal tumor. However, given the condition's rarity, further investigation of this aspect in future studies is also essential.

CONCLUSION

In conclusion, the recently published report on "Appendiceal intussusception complicated by adenocarcinoma of the cecum: A case report" published in the World Journal of Clinical Cases sheds light on a rare and intriguing case that intertwines appendiceal intussusception and mucinous cecal cancer. The detailed clinical presentation, diagnostic challenges, and subsequent surgical intervention with right hemicolectomy were meticulously discussed. The importance of considering appendiceal intussusception in the differential diagnosis of abdominal pain, especially in the context of a previously identified ileocecal lesion during colonoscopy, is underscored. The report highlights the limitations and strengths of radiological assessments and emphasizes the need for endoscopic expertise to discern between pseudo-polypoid lesions and typical polyp formations.

Furthermore, the case draws attention to the alarming rise in early-onset colorectal cancer, a demographic often excluded from widespread screening initiatives. The patient's young age and locally advanced disease stage prompt reflection on the evolving epidemiological paradigms surrounding colorectal cancer.

The report catalyzes broader discussions on the evolving landscape of colorectal cancer epidemiology and its rare presentations, urging clinicians to remain vigilant and reconsider traditional diagnostic approaches in the face of emerging trends.

Footnotes

Provenance and peer review: Invited article; Externally peer reviewed.

Peer-review model: Single blind

Specialty type: Medicine, research and experimental

Country of origin: Italy

Peer-review report’s classification

Scientific Quality: Grade B

Novelty: Grade B

Creativity or Innovation: Grade B

Scientific Significance: Grade B

P-Reviewer: Osera S S-Editor: Luo ML L-Editor: Filipodia P-Editor: Zhao YQ

References
1.  Long Y, Xiang YN, Huang F, Xu L, Li XY, Zhen YH. Appendiceal intussusception complicated by adenocarcinoma of the cecum: A case report. World J Clin Cases. 2024;12:1461-1466.  [PubMed]  [DOI]  [Cited in This Article: ]  [Reference Citation Analysis (0)]
2.  Mcswain B. Intussusception of the appendix: Review of the literature and report of a case. South Med J. 1941;34:263-270.  [PubMed]  [DOI]  [Cited in This Article: ]  [Cited by in Crossref: 57]  [Cited by in F6Publishing: 57]  [Article Influence: 0.7]  [Reference Citation Analysis (0)]
3.  Hines JJ, Paek GK, Lee P, Wu L, Katz DS. Beyond appendicitis; radiologic review of unusual and rare pathology of the appendix. Abdom Radiol (NY). 2016;41:568-581.  [PubMed]  [DOI]  [Cited in This Article: ]  [Cited by in Crossref: 14]  [Cited by in F6Publishing: 18]  [Article Influence: 2.3]  [Reference Citation Analysis (0)]
4.  Fazio RA, Wickremesinghe PC, Arsura EL, Rando J. Endoscopic removal of an intussuscepted appendix mimicking a polyp--an endoscopic hazard. Am J Gastroenterol. 1982;77:556-558.  [PubMed]  [DOI]  [Cited in This Article: ]
5.  Ozuner G, Davidson P, Church J. Intussusception of the vermiform appendix: preoperative colonoscopic diagnosis of two cases and review of the literature. Int J Colorectal Dis. 2000;15:185-187.  [PubMed]  [DOI]  [Cited in This Article: ]  [Cited by in Crossref: 24]  [Cited by in F6Publishing: 26]  [Article Influence: 1.1]  [Reference Citation Analysis (0)]
6.  Luzier J, Verhey P, Dobos N. Preoperative CT diagnosis of appendiceal intussusception. AJR Am J Roentgenol. 2006;187:W325-W326.  [PubMed]  [DOI]  [Cited in This Article: ]  [Cited by in Crossref: 22]  [Cited by in F6Publishing: 25]  [Article Influence: 1.4]  [Reference Citation Analysis (0)]
7.  East JE, Vleugels JL, Roelandt P, Bhandari P, Bisschops R, Dekker E, Hassan C, Horgan G, Kiesslich R, Longcroft-Wheaton G, Wilson A, Dumonceau JM. Advanced endoscopic imaging: European Society of Gastrointestinal Endoscopy (ESGE) Technology Review. Endoscopy. 2016;48:1029-1045.  [PubMed]  [DOI]  [Cited in This Article: ]  [Cited by in Crossref: 123]  [Cited by in F6Publishing: 126]  [Article Influence: 15.8]  [Reference Citation Analysis (0)]
8.  Birkness J, Lam-Himlin D, Byrnes K, Wood L, Voltaggio L. The inverted appendix - a potentially problematic diagnosis: clinicopathologic analysis of 21 cases. Histopathology. 2019;74:853-860.  [PubMed]  [DOI]  [Cited in This Article: ]  [Cited by in Crossref: 7]  [Cited by in F6Publishing: 8]  [Article Influence: 1.6]  [Reference Citation Analysis (0)]
9.  St John A, Murray R, Cooper L, Diaz J, Ghneim M. Cecal-Colon Intussusception due to Appendiceal Mucinous Adenocarcinoma. Am Surg. 2023;89:3822-3825.  [PubMed]  [DOI]  [Cited in This Article: ]  [Reference Citation Analysis (0)]
10.  Teke Z, Eray IC, Aydin E, Ortlek AB. Appendiceal intussusception caused by mucinous cystadenoma presenting as acute appendicitis. Ann R Coll Surg Engl. 2020;102:e1-e4.  [PubMed]  [DOI]  [Cited in This Article: ]  [Cited by in Crossref: 5]  [Cited by in F6Publishing: 5]  [Article Influence: 1.3]  [Reference Citation Analysis (0)]
11.  Kasetani T, Hirata H, Morita T, Furukawa H. [A Case of Appendiceal Intussusception Induced by Early Appendiceal Carcinoma]. Gan To Kagaku Ryoho. 2021;48:1728-1730.  [PubMed]  [DOI]  [Cited in This Article: ]
12.  REACCT Collaborative; Zaborowski AM, Abdile A, Adamina M, Aigner F, d'Allens L, Allmer C, Álvarez A, Anula R, Andric M, Atallah S, Bach S, Bala M, Barussaud M, Bausys A, Bebington B, Beggs A, Bellolio F, Bennett MR, Berdinskikh A, Bevan V, Biondo S, Bislenghi G, Bludau M, Boutall A, Brouwer N, Brown C, Bruns C, Buchanan DD, Buchwald P, Burger JWA, Burlov N, Campanelli M, Capdepont M, Carvello M, Chew HH, Christoforidis D, Clark D, Climent M, Cologne KG, Contreras T, Croner R, Daniels IR, Dapri G, Davies J, Delrio P, Denost Q, Deutsch M, Dias A, D'Hoore A, Drozdov E, Duek D, Dunlop M, Dziki A, Edmundson A, Efetov S, El-Hussuna A, Elliot B, Emile S, Espin E, Evans M, Faes S, Faiz O, Fleming F, Foppa C, Fowler G, Frasson M, Figueiredo N, Forgan T, Frizelle F, Gadaev S, Gellona J, Glyn T, Gong J, Goran B, Greenwood E, Guren MG, Guillon S, Gutlic I, Hahnloser D, Hampel H, Hanly A, Hasegawa H, Iversen LH, Hill A, Hill J, Hoch J, Hoffmeister M, Hompes R, Hurtado L, Iaquinandi F, Imbrasaite U, Islam R, Jafari MD, Kanemitsu Y, Karachun A, Karimuddin AA, Keller DS, Kelly J, Kennelly R, Khrykov G, Kocian P, Koh C, Kok N, Knight KA, Knol J, Kontovounisios C, Korner H, Krivokapic Z, Kronberger I, Kroon HM, Kryzauskas M, Kural S, Kusters M, Lakkis Z, Lankov T, Larson D, Lázár G, Lee KY, Lee SH, Lefèvre JH, Lepisto A, Lieu C, Loi L, Lynch C, Maillou-Martinaud H, Maroli A, Martin S, Martling A, Matzel KE, Mayol J, McDermott F, Meurette G, Millan M, Mitteregger M, Moiseenko A, Monson JRT, Morarasu S, Moritani K, Möslein G, Munini M, Nahas C, Nahas S, Negoi I, Novikova A, Ocares M, Okabayashi K, Olkina A, Oñate-Ocaña L, Otero J, Ozen C, Pace U, São Julião GP, Panaiotti L, Panis Y, Papamichael D, Park J, Patel S, Patrón Uriburu JC, Pera M, Perez RO, Petrov A, Pfeffer F, Phang PT, Poskus T, Pringle H, Proud D, Raguz I, Rama N, Rasheed S, Raval MJ, Rega D, Reissfelder C, Reyes Meneses JC, Ris F, Riss S, Rodriguez-Zentner H, Roxburgh CS, Saklani A, Salido AJ, Sammour T, Saraste D, Schneider M, Seishima R, Sekulic A, Seppala T, Sheahan K, Shine R, Shlomina A, Sica GS, Singnomklao T, Siragusa L, Smart N, Solis A, Spinelli A, Staiger RD, Stamos MJ, Steele S, Sunderland M, Tan KK, Tanis PJ, Tekkis P, Teklay B, Tengku S, Jiménez-Toscano M, Tsarkov P, Turina M, Ulrich A, Vailati BB, van Harten M, Verhoef C, Warrier S, Wexner S, de Wilt H, Weinberg BA, Wells C, Wolthuis A, Xynos E, You N, Zakharenko A, Zeballos J, Winter DC. Characteristics of Early-Onset vs Late-Onset Colorectal Cancer: A Review. JAMA Surg. 2021;156:865-874.  [PubMed]  [DOI]  [Cited in This Article: ]  [Cited by in Crossref: 99]  [Cited by in F6Publishing: 116]  [Article Influence: 38.7]  [Reference Citation Analysis (0)]
13.  Puzzono M, Mannucci A, Di Leo M, Zuppardo RA, Russo M, Ditonno I, Goni E, Notaristefano C, Azzolini F, Fanti L, Viale E, Elmore U, Pantaleo G, Cascinu S, Rosati R, Cavestro GM. Diet and Lifestyle Habits in Early-Onset Colorectal Cancer: A Pilot Case-Control Study. Dig Dis. 2022;40:710-718.  [PubMed]  [DOI]  [Cited in This Article: ]  [Cited by in Crossref: 2]  [Cited by in F6Publishing: 5]  [Article Influence: 2.5]  [Reference Citation Analysis (0)]
14.  Kucharzik T, Ellul P, Greuter T, Rahier JF, Verstockt B, Abreu C, Albuquerque A, Allocca M, Esteve M, Farraye FA, Gordon H, Karmiris K, Kopylov U, Kirchgesner J, MacMahon E, Magro F, Maaser C, de Ridder L, Taxonera C, Toruner M, Tremblay L, Scharl M, Viget N, Zabana Y, Vavricka S. ECCO Guidelines on the Prevention, Diagnosis, and Management of Infections in Inflammatory Bowel Disease. J Crohns Colitis. 2021;15:879-913.  [PubMed]  [DOI]  [Cited in This Article: ]  [Cited by in Crossref: 81]  [Cited by in F6Publishing: 202]  [Article Influence: 67.3]  [Reference Citation Analysis (32)]
15.  Iqbal T, DuPont HL. Approach to the patient with infectious colitis: clinical features, work-up and treatment. Curr Opin Gastroenterol. 2021;37:66-75.  [PubMed]  [DOI]  [Cited in This Article: ]  [Cited by in Crossref: 3]  [Cited by in F6Publishing: 2]  [Article Influence: 0.7]  [Reference Citation Analysis (0)]
16.  Phan DHL, Hong MK, Morgan MJ. Appendiceal intussusception causing appendicitis. ANZ J Surg. 2018;88:E851-E852.  [PubMed]  [DOI]  [Cited in This Article: ]  [Cited by in Crossref: 1]  [Cited by in F6Publishing: 2]  [Article Influence: 0.3]  [Reference Citation Analysis (0)]
17.  Anandan M, Kumaran P, Lau SYC, Breen D, Leong M. Appendiceal intussusception with appendiceal-caecal fistula in adults caused by acute appendicitis: case report and literature review. ANZ J Surg. 2022;92:1211-1212.  [PubMed]  [DOI]  [Cited in This Article: ]  [Reference Citation Analysis (0)]
18.  Hurtado CG, Chen L, Meckmongkol T. Concurrent Acute Appendicitis and Type III Appendiceal Intussusception: A Case Report. Cureus. 2022;14:e27310.  [PubMed]  [DOI]  [Cited in This Article: ]  [Reference Citation Analysis (0)]
19.  Narula N, Gibbs KE, Kong F, Mukherjee I. Appendiceal Intussusception, Diverticula, and Fecalith Associated With Appendicitis. Am Surg. 2023;89:6257-6259.  [PubMed]  [DOI]  [Cited in This Article: ]  [Reference Citation Analysis (0)]