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Diagnosis of Hirschsprung's Disease by Immunostaining Rectal Suction Biopsies for Calretinin, S100 Protein and Protein Gene Product 9.5
Published on: April 26, 2019
Increased Utilization of Abdominal Surgical Procedures, Endoscopy and Imaging After Negative Rectal Biopsies for
Niels Bjørn1,2,3, Gunvor Madsen4, Rasmus Gaardskær Nielsen5
1Research Unit for Surgery, Odense University Hospital, University of Southern Denmark, 5230 Odense, Denmark.
Insights
Children undergoing rectal biopsy for Hirschsprung's disease (HD) but not diagnosed with HD still experience significantly higher gastrointestinal healthcare utilization. This highlights the need for further investigation into the long-term outcomes for these pediatric patients.
Area of Science:
- Pediatric Gastroenterology
- Colorectal Surgery
- Medical Diagnostics
Background:
- Functional constipation is common in children, often leading to rectal biopsy to rule out Hirschsprung's disease (HD).
- A significant majority of pediatric patients biopsied for suspected HD receive a negative diagnosis, with limited understanding of their subsequent healthcare needs.
- Post-biopsy healthcare utilization in children without HD remains understudied.
Purpose of the Study:
- To assess and compare gastrointestinal (GI)-related healthcare utilization in children with negative rectal biopsies for HD versus those diagnosed with HD and healthy controls.
- To quantify the frequency of GI surgeries, endoscopies, and imaging procedures in these pediatric cohorts.
- To provide evidence for improved management strategies and resource allocation for children with functional constipation.
Main Methods:
- A nationwide Danish registry-based cohort study (1998-2018) included children under 18 undergoing rectal biopsy for suspected HD.
- The cohort comprised 1105 children: 128 diagnosed with HD and 977 without HD.
- A control group of healthy children was matched 1:10 to the non-HD cohort, with GI-related healthcare utilization analyzed pre- and post-biopsy.
Main Results:
- Children without HD showed substantially higher rates of post-biopsy GI surgeries (11.2% vs. 1.6%), endoscopies (9.4% vs. 0.5%), and imaging (37.1% vs. 7.8%) compared to controls.
- These increased utilization rates were statistically significant (p < 0.001 for all).
- The non-HD group experienced 5-10 fold increased GI-related procedures compared to the general population.
Conclusions:
- Pediatric patients with negative rectal biopsies for Hirschsprung's disease exhibit significantly elevated gastrointestinal healthcare utilization.
- This finding underscores the potential for undiagnosed conditions or long-term sequelae following negative HD biopsies.
- Further research is warranted to elucidate the reasons for increased GI healthcare utilization and optimize care pathways for these children.
Abstract:
Objective: Functional constipation affects up to one third of children. When standard treatments fail, the child may be referred to rectal biopsy on the suspicion of Hirschsprung's disease (HD), but only 7-28% will have a confirmed HD. For those with a negative biopsy for HD, there is limited evidence of the post-biopsy utilization of healthcare services. This study aimed to investigate gastrointestinal-related healthcare utilization after rectal biopsy in non-HD children compared to matched healthy controls and patients diagnosed with HD. Methods: This nationwide registry-based cohort study included all Danish children <18 years who underwent rectal biopsy for HD during the period from 1998 to 2018. The cohort was matched 1:10 to form a control cohort. Outcomes included gastrointestinal surgeries, endoscopies, and imaging procedures pre- and post-biopsy. Results: Among the 1105 children included in the cohort, 128 were diagnosed with Hirschsprung's disease (HD), while 977 were non-HD. Compared to the control group, the non-HD showed significantly higher rates of post-biopsy surgery (11.2% vs. 1.6%, p < 0.001), endoscopy (9.4% vs. 0.5%, p < 0.001), and imaging (37.1% vs. 7.8%, p < 0.001) related to the gastrointestinal tract. Conclusions: Children with a HD-negative rectal biopsy had 5-10 fold increased frequency of gastrointestinal-related surgeries, endoscopies or imaging during the follow-up period compared to the background population.
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