Segmental filiform-polyposis in a treated case of ulcerative colitis, colon: An unusual presentation raising a doubt of primary diagnosis?

Niteeka Gurung; Surbhi Goyal; Anil Aggarwal; Amarender Singh Puri; Puja Sakhuja

doi:10.4103/ijpm.ijpm_387_21

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CASE REPORT

Year : 2023 | Volume : 66 | Issue : 2 | Page : 382-384

Segmental filiform-polyposis in a treated case of ulcerative colitis, colon: An unusual presentation raising a doubt of primary diagnosis?

Niteeka Gurung¹, Surbhi Goyal¹, Anil Aggarwal², Amarender Singh Puri³, Puja Sakhuja¹
¹ Department of Pathology, Govind Ballabh Pant Institute of Postgraduate Medical Education and Research, New Delhi, India
² Department of Gastrointestinal Surgery, Govind Ballabh Pant Institute of Postgraduate Medical Education and Research, New Delhi, India
³ Department of Gastroenterology, Medanta Hospital, Gurugram, Haryana, India

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Date of Submission	18-Apr-2021
Date of Decision	23-Dec-2021
Date of Acceptance	05-Mar-2022
Date of Web Publication	26-May-2022

Abstract

Inflammatory bowel disease is broadly classified into Crohn's disease and ulcerative colitis. The standard criteria to distinguish between the two is the manner of the involvement of the bowel, with the former showing classical skip lesions and the latter having continuous involvement of the colon, most commonly affecting the rectum. However, some cases exhibit overlapping features. Herein, we report a treated case of ulcerative colitis presenting with patchy involvement of the colon in the form of peculiar segmental filiform polyposis spanned abruptly by an intervening normal mucosa. The clinico-radiologically suspicion of carcinoma colon with Crohn's colitis was considered. The clinicians and pathologists must be aware of such atypical presentations and should not be misled to change the diagnosis from ulcerative colitis to Crohn's colitis on the post-treatment resection specimens or endoscopic biopsies solely in view of the patchy filiform polyposis (FP), which poses a drastic impact on the patient's management.

Keywords: Filiform polyposis, inflammatory bowel disease, patchy involvement

How to cite this article:
Gurung N, Goyal S, Aggarwal A, Puri AS, Sakhuja P. Segmental filiform-polyposis in a treated case of ulcerative colitis, colon: An unusual presentation raising a doubt of primary diagnosis?. Indian J Pathol Microbiol 2023;66:382-4

How to cite this URL:
Gurung N, Goyal S, Aggarwal A, Puri AS, Sakhuja P. Segmental filiform-polyposis in a treated case of ulcerative colitis, colon: An unusual presentation raising a doubt of primary diagnosis?. Indian J Pathol Microbiol [serial online] 2023 [cited 2023 Jun 3];66:382-4. Available from: https://www.ijpmonline.org/text.asp?2023/66/2/382/345869

Introduction

Filiform polyposis (FP) arising in the background of inflammatory bowel disease (IBD) is a known but rare phenomenon. This occurs because of an inflammatory insult causing mucosal ulceration which in time undergoes re-epithelialization, interrupted by the edematous mucosa that in due course of time takes the configuration of a polyp. Rarely, FP has been reported without a history of IBD with case reports in association with intestinal tuberculosis, histiocytosis X, and sigmoid diverticulitis.^[1–3] FP occurs due to repeated bouts of inflammatory insult resulting in re-epithelialization and polyp formation. Therefore, its presence does not correlate with the disease activity but is a telltale sign of a previous severe disease. We report a treated case of ulcerative colitis (UC) presenting with segmental FP spanned abruptly by an intervening normal mucosa mimicking Crohn's colitis with carcinoma clinico-radiologically.

Case

A 39-year-old male was well till January 2018 after which he started having four to five episodes/day of bloody diarrhea. He was diagnosed elsewhere as UC for which he was treated with wysolone. Later, he presented to our institute with the complaint of a non-colicky dull abdominal pain which increased after the passage of stool for the last 2 months. The colonoscopy revealed a complete loss of vascular pattern with superficial erosions in the rectum and sigmoid colon. Biopsies were taken from the rectum, transverse colon, and sigmoid colon, which were consistent with UC with moderate activity, following which the patient was continued on steroids. On computed tomography (CT) enterography, there was a presence of colonic mural thickening with enhancement extending from the mid-transverse colon up to the rectum with a maximum wall thickness of 1.9 cm. On subsequent colonoscopy, skip lesions were identified in the cecum, splenic flexure, and ascending colon with relative rectal sparing and extensive pseudopolyps in the transverse and ascending colon [Figure 1]. At the hepatic flexure and distal ascending colon, a thickening was seen which led to the suspicion of Crohn's colitis (CC) and carcinoma colon for which the patient underwent subtotal colectomy.

Figure 1: On colonoscopy, multiple pseudo polyps were seen in the transverse and descending colon

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A gross examination revealed a subtotal colectomy specimen measuring 80 cm in length. On cut open, two distinct segments of numerous tall closely packed slender worm-like polyps were seen spanning over a length of 15.5 and 12 cm each [Figure 2]a. The intervening mucosa was grossly unremarkable [Figure 2]b and the distance between the two segments was 6 cm. These polyps were thin, straight, finger-like resembling stalks without heads [Figure 2]c, more than 100, ranging from 0.5 to 4 cm in length and 0.1 to 0.5 cm in diameter. Even after meticulous examination no obvious proliferative growth or stricture was identified. The microscopic examinations revealed numerous polyps with an abrupt sharp demarcation from the adjacent uninvolved mucosa [Figure 3]a. The polyps revealed pulled-up colonic mucosa with marked crypt disorganization in the form of dilated crypts showing numerous crypt abscesses along with dense mixed acute and chronic inflammatory infiltrates in the lamina propria extending into the submucosa [Figure 3]b and [Figure 3]c. The core of the polyps revealed congested vessels [Figure 3]d. At some places, mucosal ulcerations were seen extending up to the base of the muscularis propria. However, transmural inflammation with submucosal lymphoid aggregates (Crohn's rosary), serositis, or granulomas was not seen. There was no evidence of dysplasia or malignancy in the sections examined. Dissected 25 pericolic lymph nodes showed features of reactive follicular hyperplasia. The previous biopsies were reviewed and were consistent with chronic active pancolitis (UC). Therefore, the final histological diagnosis of segmental FP in the background of treated UC was rendered. Currently, the patient is relieved of his symptoms and is on a regular follow-up.

Figure 2: A gross examination revealed two distinct segments (marked by arrow) comprising numerous tall closely packed slender worm-like polyps spanning over a length of 15.5 and 12 cm each (a). The intervening mucosa (marked by an asterisk) was completely unremarkable (b). On a closer view, the polyps were thin, straight, finger-like resembling stalks without head (c)

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Figure 3: A photomicrograph depicting filiform polyposis (a–d) Numerous polyps were identified with sharp abrupt demarcation from the surrounding uninvolved mucosae (marked up arrow) (a, h, and e, 0.5X). Tall, slender pseudopolyps were seen (b, h, and e, 40X) A higher magnification shows architectural disorganization in the form of crypt dilatation along with crypt abscesses, dense mixed acute, and chronic inflammatory infiltrate in the lamina propria (c, h, and e, 200X). Some of the polyps demonstrated a vascular core (marked by an asterisk) (d, h, and e, 100X)

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Discussion

Inflammatory pseudopolyposis in seen in around 10–20% of the cases of IBD which commonly involves the sigmoid and descending colon.^[4] FP is characteristically described as long thin slender finger- or worm-like with a diameter of 0.3–1 cm and height of 2–6 cm. Occasionally, they are larger than 15-mm diameter and are known as giant FP. FP tend to cluster, which on endoscopy and radiology may mimic a fungating proliferative malignancy.^[4] They can involve the colon focally or in a diffuse fashion resembling familial adenomatous polyposis.^[5]

The occurrence of FP in a non-IBD setting indicates that it may not always be post-inflammatory sequelae, but may also occur because of a local response to cytokines.

Odze et al.^[6] and a few other researchers have reported complete restitution of inflamed rectal mucosa resulting in rectal sparing with endoscopic and microscopic skip lesions in the UC patients who have undergone treatment.^[7],[8],[9] However, all these studies were conducted in biopsies which are prone to sampling error. In 2010, Joo et al.,^[10] for the first time, assessed the prevalence of segmental disease pattern and rectal sparing in pre-operative endoscopic biopsies and compared them with the colectomy specimens. Skip lesions in the colon were observed in 30.4% of the patients on endoscopy, which translated to 25% on biopsy examination. Nevertheless, in the resection specimens, only 10.7 and 7% of the patients exhibited relative and absolute patchy involvement, respectively.

Atypical presentation in the manner of patchy segmental involvement of a colon has been previously described, especially in the new-onset pediatric population as well as in those who have received treatment. Our case is unique because of the presence of the segmental FP with abrupt transition to unremarkable colonic mucosa in the resected specimen, which presented as a diagnostic dilemma for the clinicians as well as pathologists. The index case emphasizes that gastroenterologists, surgeons, and radiologists must be aware of such an unusual and misleading presentation in a treated case of UC, and should not be tempted to change the diagnosis from UC to CC in view of the segmental involvement of the bowel, which poses a drastic impact on the patient's medical and surgical management. The pathologists should meticulously examine the gross resection specimen and be wary of the unusual presentations of UC, especially in post-treatment colectomy specimens. To conclude, our case underscores the importance of reviewing the initial pretreatment endoscopies, biopsies, and correlation with radiology and treatment history, before changing the diagnosis to Crohn's disease (CD) especially in the post-treatment scenarios.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

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