Transition Zone in Total Colonic Aganglionosis and Colorectal Hirschsprung’s Disease Shows a Similar Trend of Mucosal Innervation: Image Processing and Analysis Study

Cordeiro-Rudnisky, Fernanda; Ahn, Sangtae; Sheuka, Natallia; Whyte, Christine; Boguniewicz, Ann; Fan, Rong; Ji, Lin; Jennings, Timothy A.; Lee, Hwajeong

doi:10.1177/1093526619864225

Cited by 4 publications

(11 citation statements)

References 9 publications

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“…Image processing and analysis (IPA) is an objective method for quantifying mucosal CI. 12,13 In previous studies, we have shown that there is an increasing trend in the amount of mucosal CI in the TZ of HD, from distal TZ toward normal ganglionated zone (NZ), though we were unable to define the proximal end of TZ-the boundary between TZ and NZ-due to significant variation in the amount of mucosal CI in the probable NZ and along the circumference of TZ cross sections. 12,13 This study consists of three parts.…”

Section: Introductionmentioning

confidence: 92%

“…12,13 In previous studies, we have shown that there is an increasing trend in the amount of mucosal CI in the TZ of HD, from distal TZ toward normal ganglionated zone (NZ), though we were unable to define the proximal end of TZ-the boundary between TZ and NZ-due to significant variation in the amount of mucosal CI in the probable NZ and along the circumference of TZ cross sections. 12,13 This study consists of three parts. First, we investigated whether there is a variation in the amount of mucosal CI in non-HD rectal suction biopsies along the distance from dentate line.…”

Section: Introductionmentioning

confidence: 92%

“…13 Calretinin stained pixels due to mucosal mast cells were eliminated as previously described. 13 Data Analysis…”

Section: Image Processing and Analysismentioning

confidence: 99%

“…13 Calretinin stained pixels due to mucosal mast cells were eliminated as previously described. 13 Data Analysis 1. For the rectal suction biopsies from the non-HD subjects, the average PC for the captured images was calculated for each biopsy.…”

Section: Image Processing and Analysismentioning

confidence: 99%

“…Pixel count (PC), defined as the percentage of calretinin stained pixels in the mucosa calculated by IPA, was measured for each edited image as previously described using MATLAB (MathWorks, Natick, Massachusetts, United States). 13 Calretinin stained pixels due to mucosal mast cells were eliminated as previously described. 13 Data Analysis 1.…”

Section: Image Processing and Analysismentioning

confidence: 99%

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Increasing Trend of Calretinin-Positive Mucosal Innervation from Aganglionic Zone toward Transition Zone in Hirschsprung's Disease

et al. 2021

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Introduction Diagnosis of very short-segment Hirschsprung's disease (vsHD) by rectal suction biopsy is challenging as its aganglionic zone (AZ) overlaps with physiologic hypoganglionic zone and calretinin-positive mucosal nerves may extend from the transition zone (TZ) into AZ. We studied whether an increasing trend/gradient of calretinin-positive mucosal nerves along the distance from AZ toward TZ aids in diagnosis of HD. Materials and Methods In this study, 46 rectal suction biopsies from non-HD and HD, and 15 pull-through specimens from short-segment HD were evaluated by mucosal calretinin immunostain (CI) and image processing and analysis (IPA) to measure pixel count (PC, the percentage of calretinin stained pixels in the mucosa). Consecutive longitudinal sections of proximal AZ toward distal TZ in HD pull-through specimens were utilized as a vsHD surrogate model. First, we studied variability of mucosal CI in non-HD biopsies along the distance from dentate line. Second, we determined a cutoff point of mucosal CI by IPA that separated non-HD versus HD and applied this cutoff to longitudinal sections from proximal AZ to distal TZ segments in HD pull-through specimens. Third, we studied whether an increasing trend of mucosal CI was universally observed in HD pull-through. Results Our findings included a significant variability in PC along the biopsy distance in non-HD cases. Positive mucosal CI was found in proximal AZ in 6 (43%) of 14 HD pull-through, among which 1 case lacked submucosal nerve hypertrophy in the proximal AZ. All 14 HD pull-through cases showed an increasing trend/gradient of PC from AZ toward TZ. Conclusion Based on our findings, the presence or absence of mucosal CI positivity and submucosal nerve hypertrophy may not reliably diagnose vsHD in rectal suction biopsy. While we acknowledge that the density of mucosal innervation in variable contexts and anatomical locations is unknown and yet to be explored, our study suggests that an increasing trend of positive mucosal CI from AZ toward TZ by IPA might prove to be a useful tool for the diagnosis of vsHD in the future.

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Section: Introductionmentioning

confidence: 92%

Section: Introductionmentioning

confidence: 92%

“…13 Calretinin stained pixels due to mucosal mast cells were eliminated as previously described. 13 Data Analysis…”

Section: Image Processing and Analysismentioning

confidence: 99%

Section: Image Processing and Analysismentioning

confidence: 99%

Section: Image Processing and Analysismentioning

confidence: 99%

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Increasing Trend of Calretinin-Positive Mucosal Innervation from Aganglionic Zone toward Transition Zone in Hirschsprung's Disease

et al. 2021

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Rare presentation and retrograde diagnosis of total colonic aganglionosis in a female infant: a case report

Chakhunashvili

Chakhunashvili²,

Kvirkvelia³

et al. 2023

J Med Case Reports

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Background Total colonic aganglionosis is an extremely rare variant of Hirschsprung’s disease, which is predominant in males and can be seen in 1:50,000 live births. The presented case not only depicts a rare case, but also unusual clinical, laboratory, and instrumental data. Case presentation A 2-day-old Caucasian female newborn was transferred to our hospital from maternity. The initial presentation was reverse peristalsis, abdominal distention, and inability to pass stool. Fever had started before the patient was transferred. Hirschsprung’s disease was suspected, and tests such as contrast enema and rectal suction biopsy were done. Before enterostomy, the management of the disease included fluid resuscitation, colonic irrigation, antibiotic administration, enteral feeding, and supportive therapy. During ileostomy operation, no transition zone was visualized and full-thickness biopsy samples were retrieved from the rectum and descending colon. After surgical intervention, status significantly improved—defervescence and weight gain most importantly improved. Conclusion It is well known that diagnosis of total colonic aganglionosis may be delayed for months or even years since the transition zone may not be visible and rectal suction biopsy, unlike full-thickness biopsy, is not always reliable. It might be more prudent not to be derailed because of negative radiography and rectal suction biopsy. Also, doctors should be more suspicious of the disease if signs and symptoms are starting to be consistent with Hirschsprung-associated enterocolitis, despite biopsy and radiology results.

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Inflammation and tissue remodeling contribute to fibrogenesis in stricturing Crohn’s disease: image processing and analysis study

Arslan

Brar

Goetz

et al. 2022

JPTM

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Crohn's disease (CD), a form of idiopathic inflammatory bowel disease (IBD), has an estimated annual incidence of 3.1 to 20.2 per 100,000, and a prevalence of 201 per 100,000 in North America [1,2]. Based on its clinical behavior, CD is classified as B1 (non-stricturing, non-penetrating), B2 (stricturing) and B3 (penetrating) [3][4][5]. While most CD patients initially present as B1, they can have more than one phenotype, and clinical behavior may change over time [4]; i.e., a patient with B1 phenotype may develop strictures and or fistula during the disease course and subsequently be re-classified as having a B2 or B3 phenotype [6,7]. Eventually, about 70% of CD patients develop stenosis or fistula [7]. Stricture is the most common indication for surgery in CD [8].The progression of fibrosis and eventual stricture formation is a significant disease burden requiring escalation of medical treatment or surgical intervention in CD. Although many factors, such as genetic, epigenetic, serological, clinical, environmental, and endoscopic factors are postulated to portend an increased risk for fibrosis progression, its pathogenesis is relatively poorly understood [5,9,10]. Moreover, most of the pathological studies using formalin-fixed paraffin-embedded (FFPE) tissue are limited to morphological evaluation on routine hematoxylin and eosin (H&E)

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Transition Zone in Total Colonic Aganglionosis and Colorectal Hirschsprung’s Disease Shows a Similar Trend of Mucosal Innervation: Image Processing and Analysis Study

Cited by 4 publications

References 9 publications

Increasing Trend of Calretinin-Positive Mucosal Innervation from Aganglionic Zone toward Transition Zone in Hirschsprung's Disease

Increasing Trend of Calretinin-Positive Mucosal Innervation from Aganglionic Zone toward Transition Zone in Hirschsprung's Disease

Rare presentation and retrograde diagnosis of total colonic aganglionosis in a female infant: a case report

Inflammation and tissue remodeling contribute to fibrogenesis in stricturing Crohn’s disease: image processing and analysis study

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