Bladder and bowel symptoms experienced by children with osteogenesis imperfecta

Martins, Gisele; Siedlikowski, Maia; Coelho, Anna Kristina Silva; Rauch, Frank; Tsimicalis, Argerie

doi:10.1016/j.jped.2018.12.008

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Original articles • J. Pediatr. (Rio J.) 96 (4) • Jul-Aug 2020 • https://doi.org/10.1016/j.jped.2018.12.008 copy

Bladder and bowel symptoms experienced by children with osteogenesis imperfecta^☆ ☆ Please cite this article as: Martins G, Siedlikowski M, Coelho AK, Rauch F, Tsimicalis A. Bladder and bowel symptoms experienced by children with osteogenesis imperfecta. J Pediatr (Rio J). 2020;96:472-8.

Authorship SCIMAGO INSTITUTIONS RANKINGS

Abstract

Objective:

To estimate the prevalence and presentation of bladder, bowel, and combined bladder and bowel symptoms experienced by children with osteogenesis imperfecta and to describe the socio-demographic and clinical profile of these children.

Method:

A descriptive study was conducted with a convenience sample of parent-child pairs of toilet-trained children aged from 3 to 18 years. Pairs were interviewed using three tools: (1) Socio-Demographic and Clinical Questionnaire; (2) Dysfunctional Voiding Scoring System; (3) Rome III Criteria along with the Bristol Stool Scale. Data were stratified by socio-demographic and clinical variables and analyzed using descriptive statistics.

Results:

Thirty-one parent-child pairs participated in the study; 38.7% (n = 12) children reported bowel symptoms, 19.4% (n = 6) reported a combination of bladder issues (such as holding maneuvers and urgency) and bowel symptoms (such as hard or painful bowel movements and large diameter stools). There were no reports of isolated bladder issues. Among the child participants, 16 (51.7%) identified as female and 20 (64.5%) were 5-14 years old. The most prevalent type of osteogenesis imperfecta was type III (n = 12; 38.7%) and eight (25.8%) children reported using a wheelchair.

Conclusion:

This is the first study to examine the prevalence and presentation of bladder, bowel, and combined bladder and bowel symptoms in children with osteogenesis imperfecta, offering a preliminary socio-demographic and clinical profile of these children. This research is an important step toward effective screening, detection, and access to care and treatment, especially for clinicians working with this group of very fragile patients.

KEYWORDS
Children; Osteogenesis imperfecta; Lower urinary tract symptoms; Constipation; Bowel and bladder dysfunction

	n ^a a n = 31.	%
Child gender
Female	16	51.7
Male	15	48.3
Primary caregiver
Mother	21	67.7
Father	6	19.4
Female guardian	4	12.9
Child's age when toilet training was started
Before 18 months	11	35.5
Between 18 up to 30 months	11	35.5
More than 30 months	8	25.8
Unsure/can't tell	1	3.2
Toilet training duration
<3 months	12	38.7
Between 3 and 6 months	7	22.6
>6 months	5	16.1
Unsure/can't tell	7	22.6
Report of toilet training difficulties
Yes	8	25.8
No	20	64.5
Unsure/can't tell	3	9.7

Prevalence of bladder/bowel symptoms assessed by the DVSS	n^a a n = 31. (%)
Urinary incontinence	1 (3.2)
Decreased frequency of bowel movements	9 (29.0)
Have to push to evacuate	9 (29.0)
Holding maneuvers	4 (12.9)
Urgency	6 (19.4)
Have to push to urinate	2 (6.4)
Pain to urinate	2 (6.4)
Presence of stressful events	15 (48.4)
Presence of suggestive symptoms of abnormal voiding, measured by the DVSS^b b Total DVSS score (female score >6 points; male score >9 points).	6 (19.4)

Prevalence of bowel symptoms assessed by the Rome IIIc	n (%)
Two or fewer evacuations per week	3 (9.7)
At least one episode of fecal incontinence per week	3 (9.7)
History of retentive posture or voluntary retention	4 (12.9)
History of hard or painful evacuation	16 (51.6)
Presence of large fecal mass in rectum	5 (16.1)
Feces of large diameter that obstruct the toilet	8 (25.8)
Presence of suggestive symptoms of functional constipation, measured by the Rome III criteria^c c The presence of at least two out of six intestinal symptoms.	12 (38.7)

Prevalence of combined bladder & bowel symptoms (DVSSb + Rome IIIc )	n (%)
Presence of suggestive symptoms of bladder and bowel dysfunction (BBD)	6 (19.4)

Variables	Categories	Prevalence rates
		Bladder symptoms (DVSS)^a a Total DVSS score (female score >6 points; male score >9 points).		Bowel symptoms (Rome III)^b b The presence of at least two out of six intestinal symptoms.
		Yes	No	Yes	No
Child gender	Female	3	13	7	9
Child gender	Male	3	12	5	10
Age range	2 < 8 years	2	9	7	4
Age range	8 < 18 years	4	16	5	15
Child's birth order	1st child	4	9	8	5
	2nd child and younger	2	16	4	14
Number of children in the family	1-2	5	16	10	11
	>3	1	9	2	8
Distance to hospital	Same city	3	11	3	2
Distance to hospital	Different city	3	14	9	17
Family income/year ($)	Up to 7900	5	16	8	13
	$ 7920-13,200	0	5	3	2
	$ 13,200-39,600	1	4	1	4
OI type	I	1	7	2	6
	III	4	8	5	7
	IV	0	5	3	2
	Unknown	1	5	2	4
Ambulatory status	Ambulatory	4	20	10	14
Ambulatory status	Non-ambulatory	2	5	2	5
Mobility assistive devices	Yes	3	13	4	8
Mobility assistive devices	No	3	12	8	11

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E-mail: jped@jped.com.br

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[1] * Corresponding author. E-mail: martinsgise@gmail.com (G. Martins).

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Bladder and bowel symptoms experienced by children with osteogenesis imperfecta☆ ☆ Please cite this article as: Martins G, Siedlikowski M, Coelho AK, Rauch F, Tsimicalis A. Bladder and bowel symptoms experienced by children with osteogenesis imperfecta. J Pediatr (Rio J). 2020;96:472-8.

Abstract

Objective:

Method:

Results:

Conclusion:

Bladder and bowel symptoms experienced by children with osteogenesis imperfecta^☆ ☆ Please cite this article as: Martins G, Siedlikowski M, Coelho AK, Rauch F, Tsimicalis A. Bladder and bowel symptoms experienced by children with osteogenesis imperfecta. J Pediatr (Rio J). 2020;96:472-8.