Interview with Prof. Gisele Martins, University of Brasilia (UnB), Brazil (main part) and Dr. Argerie Tsimicalis, McGill University, Canada

Who are you & what is your relationship to OI?

My name is Gisele Martins and I’m a professor of nursing at the University of Brasilia (UnB), Brazil. My involvement with the OI community started seeing OI kids with bladder and bowel issues in my advanced nurse-led clinic in the field of Pediatric Urology, located in our teaching hospital. Given my nurse-scientist background, I decided to conduct a preliminary study to estimate the prevalence and presentation of bladder, bowel, and combined bladder and bowel symptoms experienced by Brazilian children with OI. This research was published as follows: 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.

How did the project start?

This first preliminary Brazilian study was led by my research team at the University of Brasilia, Department of Nursing along with a collaboration between McGill University and Shriners Hospital for Children, Montreal, Canada. At that time (2015), Dr. Argerie Tsimicalis and I conducted a descriptive study with a convenience sample of 31 Brazilian parent-child pairs of toilet-trained children aged from 3 to 18 years. There was no funding.

In 2018-2019, I spent a bit of my sabbatical year under Dr. Tsimicalis’ supervision to conduct a mixed-method study which allowed for a comprehensive and rich account of the state of bladder and bowel issues in children living with chronic musculoskeletal conditions such as arthrogryposis multiplex congenita (AMC), Osteogenesis imperfecta (OI), and spastic Cerebral Palsy (CP). The study was grounded in the perspective of key stakeholders, including children, their family, and healthcare providers. It was funded by Shriners Hospital for Children and the findings were not published yet.

What was your research project about?

It was about bladder and bowel issues in children living with impaired physical mobility. The first preliminary study was conducted with a Brazilian sample of 31 children with OI and the second was composed by 45 Canadian children living with impaired physical mobility, including children with OI. A detailed understanding of the continence issues, the challenges associated with toileting practices and barriers related to public access will help individuals, families, and healthcare providers to advocate for treatment, services, devices and environmental modifications.

What were your most interesting findings?

Speaking of children with OI, constipation was the prevalent symptom in both samples (Brazilian & Canadian). Moreover, 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) was also reported, while isolated bladder issues were not reported by our samples.

What is the most important take home message for clinical work?

This research is an important step toward effective screening, detection, and access to care and treatment, especially for individuals living with impaired physical mobility.

Do you think the findings are relevant for adults with OI as well?

The OI Foundation has identified underemphasized health conditions in adults with OI ‘‘urinary tract’’ or ‘‘gastrointestinal system’’ as the organ systems that had the most significant negative impact on their quality of life. Additionally, early identification of gastrointestinal problems and a timely referral to a specialist is crucial to avoid these symptoms throughout the life course.

Argerie (Tsimicalis), can you tell us about the pooping video?

Gisele‘s work in Brazil, followed by our work at Shriners together (unpublished) found that some of the children are constipated. Now, the risk of constipation is greater after surgery. Being constipated is not very pleasant, not very pleasant to talk about and can even have some embarrassing moments.

Now, how does one talk about poop? As part of our effort to increase to educational resources available, our team has created a colouring book for children with OI called OI Colour OI Learn. The book is supplemented with colouring sheets, greeting cards and animated videos. Learning how to have a successful poop after surgery, is one of the animated videos. Why animation? Makes it fun, adds humor, help destigmatize, and welcomes children to talk about poop and farts, and learn what is needed to make those Bowls move.

Should more research be done on this topic?

Conservative interventions, including lifestyle changes and behavioral therapies, have not been examined specifically in children with OI experiencing bladder and bowel issues, and represent an optimal opportunity to provide relief and improve quality of life.

Were patients/patient organizations involved in this project? How?

Absolutely! Our team is composed by scientists, healthcare providers and patients to facilitate and mobilize research evidence in the hopes of increasing awareness of bladder and bowel issues, particularly in children living with impaired physical mobility.

Any messages for the readers of OIFE Magazine?

Advocate for bladder and bowel assessment across lifespan. It is also important that healthcare providers, parents, and when appropriate the child, can identify the red flags of incontinence, such as delayed toileting training, urgency associated with ‘accidents’, soiling, and continually monitor bladder and bowel control with a low threshold for referral.