Long-term outcome of neonates with suspected Hirschsprung's disease, but normal rectal biopsy.
OBJECTIVE
Hirschsprung's disease (HD) must always be considered in very early-onset constipation. Although HD has a well-described clinical course, little is known about those neonates in whom HD was excluded. We aimed to describe the long-term clinical outcomes of neonates with a clinical suspicion of HD that was excluded by rectal suction biopsy.


METHODS
This is a single-center double-cohort comparative study. Neonates who underwent rectal mucosa biopsy for suspected HD were age and sex matched with healthy controls. A survey on clinical outcomes, stooling patterns, and other gastrointestinal (GI)-related conditions was sent to parents. Pathology slides were re-reported by an experienced histopathologist blinded to the clinical data.


RESULTS
A total of 51 neonates were included [25 cases, 26 controls; 41% males, median time of follow-up 4.25 years (interquartile range 2.7-6.9)]. Nine (36%) of patients in the case group required prolonged laxative use for constipation during the first year of life compared with 0 (0%) controls (P<0.001). This difference was maintained at the end of follow-up, with 5 (20%) versus 0 (0%), respectively (P=0.02). Case neonates were significantly more likely to be hospitalized or to be diagnosed with a chronic GI-related condition than the controls (33 vs. 12%, P=0.01; and 19 vs. 8%, P=0.04, respectively).


CONCLUSIONS
Neonatal constipation is associated with long-term GI-related disorders and should be considered clinically significant even when the diagnosis of HD is excluded. Neonates with early-onset abnormal stooling patterns should be monitored with adequate pediatrician or pediatric gastroenterologist follow-up.
aShaare Zedek Medical Center bThe Hebrew University of Jerusalem, Jerusalem, Israel.
Daniel
D
Harlev
Oleg
O
Kharenko
Jacob
J
Waxman
Tanya
T
Frankel
Dan
D
Turner
Oren
O
Ledder
D001706:Biopsy; D016022:Case-Control Studies; D002648:Child; D002675:Child, Preschool; D003248:Constipation / Q000175:diagnosis* / Q000188:drug therapy / Q000209:etiology / Q000503:physiopathology; D003672:Defecation / Q000187:drug effects; D005260:Female; D006627:Hirschsprung Disease / Q000150:complications / Q000175:diagnosis* / Q000473:pathology / Q000503:physiopathology; D006801:Humans; D007223:Infant; D007231:Infant, Newborn; D007413:Intestinal Mucosa / Q000473:pathology*; D007557:Israel; D054368:Laxatives / Q000627:therapeutic use; D008297:Male; D011237:Predictive Value of Tests; D011379:Prognosis; D012007:Rectum / Q000473:pathology* / Q000503:physiopathology; D012307:Risk Factors; D013997:Time Factors
D054368:Laxatives