Journal List > J Korean Assoc Pediatr Surg > v.20(2) > 1071942

Kim, Kim, Kim, Namgoong, and Hwang: How Reliable Are Diagnostic Methods of Hirschsprung Disease?

Abstract

Purpose

The purpose of this study was to compare the diagnostic accuracy of the non-invasive diagnostic methods and rectal suction biopsy for the detection of Hirschsprung disease (HD).

Methods

We reviewed diagnostic methods and results retrospectively in patients who underwent anorectal manometry, barium enema and rectal suction biopsy for the diagnosis of HD at Asan Medical Center from January 2000 to December 2012.

Results

There were 97 patients (59 neonates and 38 infants) in the study period. The overall accuracy of anorectal manometry for the diagnosis of HD was 71.1% and its sensitivity was 51.4% (48.1% in neonate and 62.5% in infant, respectively) and its overall specificity was 82.3% (81.3% in neonate and 83.3% in infant, respectively). The Overall accuracy of barium enema was 66.0% (72.8% in neonate and 55.3% in infant, respectively) and specificity of barium enema was 53.2% (56.3% in neonate and 50.0% in infant, respectively). These results were lower than those of anorectal manometry. The overall sensitivity of barium enema was 88.6% (92.6% in neonate and 75.0% in infant, respectively) and it was higher than the sensitivity of anorectal manometry. Histological studies confirmed HD in 35 patients, in one of whom the suction biopsy showed negative finding.

Conclusion

Accuracy of non-invasive methods for diagnosis of HD in our study is lower than those in previous study, so we need to improve the quality of diagnostic tools in our hospital. We conclude that the rectal suction biopsy is the most accurate test for diagnosing HD, so the biopsy to confirm the diagnosis of the HD is very important.

Figures and Tables

Table 1
Test Results of Barium Enema
jkaps-20-33-i001

Values are presented as patients' number.

HD, Hirschsprung disease.

Table 2
Test Results of Anorectal Manometry
jkaps-20-33-i002

Values are presented as patients' number.

HD, Hirschsprung disease; RAIR, recto-anal inhibitory reflex.

Table 3
Test Results of Suction Biopsy
jkaps-20-33-i003

Values are presented as patients' number.

HD, Hirschsprung disease.

Table 4
Direct Comparison of Accuracy
jkaps-20-33-i004

BE, barium enema; ARM, anorectal manometry; RSB, rectal suction biopsy.

Notes

This study was presented at the 30th Annual Congress of Korean Society of Pediatric Surgeons.

No potential conflict of interest relevant to this article was reported.

References

1. de Lorijn F, Reitsma JB, Voskuijl WP, Aronson DC, Ten Kate FJ, Smets AM, et al. Diagnosis of Hirschsprung's disease: a prospective, comparative accuracy study of common tests. J Pediatr. 2005; 146:787–792.
2. de Lorijn F, Kremer LC, Reitsma JB, Benninga MA. Diagnostic tests in Hirschsprung disease: a systematic review. J Pediatr Gastroenterol Nutr. 2006; 42:496–505.
3. Osatakul S, Patrapinyokul S, Osatakul N. The diagnostic value of anorectal manometry as a screening test for Hirschsprung's disease. J Med Assoc Thai. 1999; 82:1100–1105.
4. Emir H, Akman M, Sarimurat N, Kiliç N, Erdoğan E, Söylet Y. Anorectal manometry during the neonatal period: its specificity in the diagnosis of Hirschsprung's disease. Eur J Pediatr Surg. 1999; 9:101–103.
5. Kim DY, Kim SC, Kim KM, Kim ER, Kim KS, Kim JS, et al. Diagnosis of Hirschsprung's disease of neonate and infant. J Korean Assoc Pediatr Surg. 2002; 8:1–5.
6. Kurer MH, Lawson JO, Pambakian H. Suction biopsy in Hirschsprung's disease. Arch Dis Child. 1986; 61:83–84.
7. Martucciello G, Pini Prato A, Puri P, Holschneider AM, Meier-Ruge W, Jasonni V, et al. Controversies concerning diagnostic guidelines for anomalies of the enteric nervous system: a report from the fourth International Symposium on Hirschsprung's disease and related neurocristopathies. J Pediatr Surg. 2005; 40:1527–1531.
8. Noviello C, Cobellis G, Romano M, Amici G, Martino A. Diagnosis of Hirschsprung's disease: an age-related approach in children below or above one year. Colorectal dis. 2010; 12:1044–1048.
9. Holschneider AM, Meier-Ruge W, Ure BM. Hirschsprung's disease and allied disorders--a review. Eur J Pediatr Surg. 1994; 4:260–266.
10. Choi YI, Choi SO, Park WH. Diagnostic strategy using barium enema and rectal suction biopsy with acetylcholinesterase histochemistry in neonates suspected of having Hirschsprung's disease. J Korean Assoc Pediatr Surg. 2001; 7:105–111.
11. Taxman TL, Yulish BS, Rothstein FC. How useful is the barium enema in the diagnosis of infantile Hirschsprung's disease? Am J Dis Child. 1986; 140:881–884.
12. Rosenfield NS, Ablow RC, Markowitz RI, DiPietro M, Seashore JH, Touloukian RJ, et al. Hirschsprung disease: accuracy of the barium enema examination. Radiology. 1984; 150:393–400.
13. Zhou XL, Chen FB, Ou BY, Zhang XP, Jiang MZ. Evaluation of clinical value of anorectal manometry for diagnosis of Hirschsprung's disease in neonate. Zhonghua Er Ke Za Zhi. 2004; 42:681–683.
14. Ito Y, Donahoe PK, Hendren WH. Maturation of the rectoanal response in premature and perinatal infants. J Pediatr Surg. 1977; 12:477–482.
15. Holschneider AM, Kellner E, Streibl P, Sippell WG. The development of anorectal continence and its significance in the diagnosiso of Hirschsprung's disease. J Pediatr Surg. 1976; 11:151–156.
16. Aaronson I, Nixon HH. A clinical evaluation of anorectal pressure studies in the diagnosis of Hirschsprung's disease. Gut. 1972; 13:138–146.
17. Benninga MA, Omari TI, Haslam RR, Barnett CP, Dent J, Davidson GP. Characterization of anorectal pressure and the anorectal inhibitory reflex in healthy preterm and term infants. J Pediatr. 2001; 139:233–237.
18. Kawahara H, Kubota A, Hasegawa T, Okuyama H, Ueno T, Watanabe T, et al. Anorectal sleeve micromanometry for the diagnosis of Hirschsprung's disease in newborns. J Pediatr Surg. 2007; 42:2075–2079.
19. Huang Y, Zheng S, Xiao X. Preliminary evaluation of anorectal manometry in diagnosing Hirschsprung's disease in neonates. Pediatr Surg Int. 2009; 25:41–45.
20. Andrassy RJ, Isaacs H, Weitzman JJ. Rectal suction biopsy for the diagnosis of Hirschsprung's disease. Ann Surg. 1981; 193:419–424.
21. Lake BD, Puri P, Nixon HH, Claireaux AE. Hirschsprung's disease: an appraisal of histochemically demonstrated acetylcholinesterase activity in suction rectal biopsy specimens as an aid to diagnosis. Arch Pathol Lab Med. 1978; 102:244–247.
22. Yadav L, Kini U, Das K, Mohanty S, Puttegowda D. Calretinin immunohistochemistry versus improvised rapid Acetylcholinesterase histochemistry in the evaluation of colorectal biopsies for Hirschsprung disease. Indian J Pathol Microbiol. 2014; 57:369–375.
23. Holschneider AM, Puri P. Hirschsprung's disease and allied disorders. 3rd ed. Berlin, New York: Springer;2006.
TOOLS
Similar articles