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Serious rectal bleeding complicating suction rectal biopsy in a child.

Introduction

Rectal suction biopsy (RSB), first reported by Noblett in 1969, is considered the standard of care for the diagnosis of Hirschsprung's disease in patients under 3 years of age. (1) Although typically considered a low-risk procedure, occasionally severe adverse outcomes may occur. We report a case of a 2 year old child who developed severe bleeding requiring a transfusion after undergoing RSB.

In her initial reports, Dr. Noblett asserted that with this technique, suction biopsy specimen yielding at least 2 [mm.sup.2] of submucosa obtained above 3 cm proximal to the pectinate line and serially sectioned, and the absence of ganglion cells is diagnostic of Hirschsprung's disease. (2) As a simple, noninvasive technique, RSB has obviated the need for a full thickness rectal biopsy as a screening test for Hirschsprung's disease. (3) Modification of the suction biopsy equipment improved its safety and efficacy as a reusable tool. (4)

The procedure is considered safe and minimally invasive, often done with mild or no sedation. The most common complication as reported in many large series is minor and transient rectal bleeding. (5,7) However rare but serious complications including one fatality have been reported. (8)

Case Report

We present the case of a 2 year old Caucasian boy with a history of chronic constipation since birth. His family history is unremarkable with respect to abnormal bleeding. There was no history of any recent medication use, including non-steroidal anti-inflammatoryagents (NSAIDS). He was delivered vaginally after full term uncomplicated pregnancy and passed meconium in the first 24 hours. Immediately after discharge from the nursery, he was noted to have small caliber, hard and infrequent bowel movements. Glycerin suppositories, stool softeners and formula changes were tried without relief. His constipation worsened with the introduction of solid foods and whole milk. A single contrast barium enema revealed no transition zone, but delayed and incomplete evacuation of the barium, which was suggestive of Hirschsprung's disease. A suction rectal biopsy was performed using the rectal suction biopsy tool with negative pressure applied manually by a syringe to negative 20 cm air (Figure 1 shows suction rectal biopsy tool). Two rectal biopsies were each obtained at 2 cm and at 5 cm above the anal verge). The procedure was tolerated well with minimal blood loss noted at the time of discharge. However, shortly after discharge, his parents reported passage of progressively large amount of fresh blood followed by clots in his diapers. Twelve hours after discharge, the child returned to the clinic, where a complete blood count revealed a hemoglobin of 7.4 gm /dl, and clotting studies were normal. (PT/ INR 1.1 and PTT 32 seconds, platelet count of 347).

Following transfusion of 2 units of packed red blood cells and adequate hydration, he was taken to the operating room where, under general anesthesia, the rectal biopsy site was inspected through a speculum. Dark blood clots filled his rectum which were irrigated and suctioned. No active bleeding was noted at any of the four biopsy sites. All had fibrinous exudates covering the base of the recent biopsy sites, and none had fresh clots present. The 4 biopsy sites were then over sewn with interrupted Vicaryle sutures. The child did very well. He had no further bleeding episodes and passed non-bloody stools post operatively. On follow up, there was no recurrence of rectal bleeding 6 months after this incident and his constipation was well controlled on daily use of an osmotic laxative, lactulose.

Histological evaluation of the rectal biopsies revealed the presence of normal rectal mucosa and submucosa with mucosal lymphoid nodule identified at the two biopsy sites. Normal ganglion cells were present in the submucosa at the 5 cm specimen but not the 2 cm. No muscularis propria was noted and no vascular malformation was identified in any of the biopsies, suggesting that the technique of the biopsy was appropriate and there was no vascular predisposition to bleeding.

Discussion

The use of rectal suction biopsy to diagnose Hirschsprung's disease beyond the neonatal period has been recently evaluated. (9) With minimal complications and few insufficient samples, rectal biopsy in the older infant and toddler was shown to be a safe procedure and remains the gold standard for the diagnosis of Hirschsprung's disease. The percentage of abnormal rectal suction biopsies was significantly higher in the neonates (29%) compared to infants (15%) or toddlers above the age of 1 year (5%). Some reports in the literature question the need for a rectal biopsy in children presenting after the neonatal period, (10) however, 53% of all abnormal rectal biopsies in this series were made in children presenting after the neonatal period. (9)

Prolonged or serious rectal bleeding following suction rectal biopsy is exceptionally rare. (6,8) (Table 1 shows published series of rectal suction biopsy complications). To the best of our knowledge, this is the first reported case of serious rectal bleeding in a child older than one year of age following rectal suction biopsy. Rees and colleagues reported three neonates with rectal hemorrhages requiring transfusion in their series of 1,340 cases of rectal suction biopsies. (8) Pini-Parto et al reported two cases of persistent rectal bleeding in children less than one year of age in their series of 389 cases. (6) In our patient, an adequate sub-mucosal sample was obtained and the syringe suction limited to negative 20 cm of air. It is possible that the degree of tissue trauma and risk of serious bleeding may vary depending on the amount of suction applied during this procedure. However, Ali et al (15) reported that the use of a wall or portable machine suction when compared to syringe applied suction to the rectal biopsy forceps improves the yield of adequate sub-mucosa without increasing the risk of hemorrhage or perforation. This technique reduced the need for re-biopsy, which adds additional risk.

Conclusion

It may be prudent for pediatric surgeons and gastroenterologists using this technique to monitor patients' serial hemoglobin following rectal suction biopsy and to be familiar with this rare but real risk of serious bleeding.

Acknowledgements

The author is grateful to Stephen Sondike, MD and Mary Emmett, PhD for their assistance in vetting the manuscript.

References

(1.) Noblett HR: A rectal suction biopsy tube for use in the diagnosis of Hirschsprung's disease. J Pediatr Surg. 1969 Aug;4(4):406-9

(2.) Campbell PE, Noblett HR: Experience with rectal suction biopsy in the diagnosis of Hirschsprung's disease. J Pediatr Surg. 1969 Aug;4(4):410-5

(3.) Kurer MH, Lawson JO, Pambakian H. Suction biopsy in Hirschsprung's disease. Arch Dis Child. 1986 Jan;61(1):83-4

(4.) Freeman JK. A new instrument for suction rectal biopsy in the diagnosis of Hirschsprung's disease. Pediatr Surg Int 1997;12(1):76-7.

(5.) Meier-Ruge W, Lutterbeck PM, Herzog B, Morger R, Moser R, Scharli A. : Acetylcholinesterase activity in suction biopsies of the rectum in the diagnosis of Hirschsprung's disease. J Pediatr Surg. 1972 Feb;7(1):11-7

(6.) Pini-Prato A, Martucciello G, Jasonni V. Rectal suction biopsy in the diagnosis of intestinal dysganglionoses: 5-year experience with Solo-RBT in 389 patients. J Pediatr Surg. 2006 Jun;41(6):1043-8

(7.) Andrassy RJ, Isaacs H, Weitzman JJ: Rectal suction biopsy for the diagnosis of Hirschsprung's disease. Ann Surg. 1981 Apr;193(4):419-24.

(8.) Rees BI, Azmy A, Nigam M, Lake BD: Complications of rectal suction biopsy. J Pediatr Surg. 1983 Jun;18(3):273-5.

(9.) Rahman N, Chouhan J, Gould S, Joseph V, Grant H, Hitchcock R, Johnson P, Lakhoo K.: Rectal biopsy for Hirschsprung's disease--are we performing too many? Eur J Pediatr Surg. 2010 Mar;20(2):95-7. Epub 2010 Apr 15.

(10.) A Ghosh, D M Griffiths: Rectal biopsy in the investigation of constipation. Arch Dis Child 1998;79:266-268 doi:10.1136/adc.79.3.266.

(11.) Ali AE, Morecroft JA, Bowen JC, Bruce J, Morabito A.: Wall or machine suction rectal biopsy for Hirschsprung's disease: a simple modified technique can improve the adequacy of biopsy. Pediatr Surg Int. 2006 Aug;22(8):681-2. Epub 2006 Jul 4.

Ahmed Dahshan, MD, FAAP, FACG

Professor and Chief, Section of Pediatric

Gastroenterology, Department of Pediatrics

West Virginia University School of Medicine-Tulsa

Corresponding Author: Ahmed Dahshan, MD, Department of Pediatrics, West Virginia University School of Medicine-Tulsa, 830 Pennsylvania Ave, Suite 104, Charleston, WV 25302; ADahshan@hsc. wvu.edu

Table 1. Complications of series of rectal suction biopsies

Study                    No. of    No. of RS   Minor rectal
                        patients   Biopsies      bleeding

Andrassy 1981 (11)        444         444           0
Rees 1983 (12)           1,340       1,340          0
Freeman JK 1997 (4)        60         180           0
Gosh A 1998 (14)          141         186           0
Pini-Prato A 2006 (6)     389        1012           0
Ali AE 2006 (15)          119         127           0
Rahman N 2010 (13)        668         668           3

Study                   Major rectal   Perforation   Death
                          bleeding     and pelvic
                                         sepsis

Andrassy 1981 (11)           0              0          0
Rees 1983 (12)               3              3          1
Freeman JK 1997 (4)          0              0          0
Gosh A 1998 (14)             0              0          0
Pini-Prato A 2006 (6)        2              0          0
Ali AE 2006 (15)             0              0          0
Rahman N 2010 (13)           0              0          0
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Title Annotation:Case Report
Author:Dahshan, Ahmed
Publication:West Virginia Medical Journal
Article Type:Case study
Date:Mar 1, 2014
Words:1509
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