Monday, April 3, 2017

Syngap

Having one child with a serious form of epilepsy and autism would be difficult. Having two is almost unimaginable. 

But that is reality for Melbourne couple Danielle and Danny Williams. Their daughters, seven-year-old Jaeli and five-year-old Dali were two of the first people in Australia to be diagnosed with a rare genetic condition called Syngap.



The sisters can have multiple seizures every hour (sometimes experiencing up to 50 seizures in an hour). They hardly sleep, are still in nappies, have impaired speech, autism, motor difficulties and behavioural issues.

When they are outside they have to be monitored constantly, as one of their behavioural issues is pica — which means they will pick anything up off the ground and try and eat it. If they are experiencing extreme seizures they have to wear helmets for their own safety.

Jaeli has seizures throughout the evening and as such suffers from insomnia. The longest stretch she can sleep is four hours. She wakes her mother up each time she has a seizure, which means Danielle is often working four days a week on four hours sleep. And this has been going on for six years.

For the first year of Jaeli’s life there didn’t seem to be anything wrong.

“She missed a few developmental milestones, but nothing alarming,” recalls Danielle.

“She was sitting up eating and babbling — she wasn’t that far behind other kids.

“The first time we realised something was wrong with her was when we were already pregnant with our second child. When Jaeli was about 18-months old Danny (who is an osteopath) noticed these little eyelid flutters. The pediatrician made us feel a bit like paranoid parents but we took her to the hospital for tests and that’s when my husband’s suspicions were confirmed ... that they were little seizures”.

By the time Dali was born Jaeli’s seizures were severe. At the worst she was being flung back and dropping to the ground multiple times throughout the day.

“For the first three months of the new baby’s life I was in hospital with Jaeli, having to express milk for my newborn who was at home with my mother,” recalls Danielle.

For the first year of her new daughter’s life, Danielle held onto the hope that Dali wouldn’t be affected like her sister.

Dali “hit all the milestones” and they thought she was in the clear, until Danielle noticed the eyelid flutters at the 12-month mark.

It was a devastating moment.

“I didn’t tell anyone initially,” she says.

“I just wanted to make sure ... and then I remember telling my close girlfriend ... that was bad. I’m tearing up just remembering it.

“The first time (with Jaeli) we had no idea what we were in for ... we didn’t realise the worst was yet to come. But with Dali we knew exactly. It was hard.”

Specialists had trouble diagnosing the girls’ condition. Jaeli went through 22 different diagnoses and 27 different treatments. She endured countless invasive medical tests including lumbar punctures, spinal taps and blood tests. At one point Jaeli was poisoned by one of the anti-convulsive medications she was taking.

But last year there was a breakthrough. After undergoing genome sequencing they were finally able to identify that the girls suffered from a rare neurological condition where the SYNGAP1 gene is mutated. They were the first people in Australia to be diagnosed with the Syngap condition.
It was a huge moment for the family.

“We had been looking for a cause for five years — we had invested in so many tests to try and find what was wrong and to just be told, it was an end to all the poking and prodding and we felt like we had a bit of control, a bit of power over this thing that had held us for so long,” explains Danielle.

There is currently no cure and the life expectancy for syngap sufferers is yet to be determined.

“But now that we have a diagnosis, we are focused on research, specifically into finding a way to help this mutated gene,” said Danielle.

The family is hoping to raise $150k a year for the next three years to fund research to find a cure.
They’ve launched the Secret4Syngap campaign to help raise awareness of the condition. They’re asking people to simply share a secret online by using the #secret4syngap hashtag…

Despite their challenges as a family, both Danielle and Danny work hard to remain positive.

But it hasn’t always been easy and there have been times when they have felt overwhelmed.

“The girls get a bus now, but when I used to drive the kids to the special school before they were in prep I used to get into my car in the morning and I used to just cry. It was all just so overwhelming,” recalls Danielle.

“But one day I remember another mum pulled up in front of me and proceeded to go to the boot of her car to take out a walking frame. When she turned to the side, I got a glimpse of a very pretty woman who was heavy with a similar pain to mine. I got a glimpse of what her original self was, what she would have otherwise been.

“Then I saw a little fist knocking on the inside of the back seat. As she went around to open the door, I saw her chronically saddened face light up. She opened the door and stood there for a minute or so while she and her ghostlike son embraced. Both clearly smitten with each other.

“People without special needs kids ... they just don’t get to appreciate rare moments like that.”

http://www.news.com.au/lifestyle/health/health-problems/they-hardly-talk-theyre-still-in-nappies-they-have-seizures-every-hour/news-story/bc77bdf0a8b11f5a1bac07ccf5531dab

Courtesy of:  http://www.foxnews.com/health/2017/03/31/rare-condition-causes-familys-2-daughters-to-suffer-multiple-seizures-every-hour.html

2 comments:

  1. Parker MJ, Fryer AE, Shears DJ, Lachlan KL, McKee SA, Magee AC, Mohammed S, Vasudevan PC, Park SM, Benoit V, Lederer D, Maystadt I, Study D, FitzPatrick DR. De novo, heterozygous, loss-of-function mutations in SYNGAP1 cause a syndromic form of intellectual disability. Am J Med Genet A. 2015 Oct;167A(10):2231-7.

    Abstract
    De novo mutations (DNM) in SYNGAP1, encoding Ras/Rap GTPase-activating protein SynGAP, have been reported in individuals with nonsyndromic intellectual disability (ID). We identified 10 previously unreported individuals with SYNGAP1 DNM; seven via the Deciphering Developmental Disorders (DDD) Study, one through clinical analysis for copy number variation and the remaining two (monozygotic twins) via a research multi-gene panel analysis. Seven of the nine heterozygous mutations are likely to result in loss-of-function (3 nonsense; 3 frameshift; 1 whole gene deletion). The remaining two mutations, one of which affected the monozygotic twins, were missense variants. Each individual carrying a DNM in SYNGAP1 had moderate-to-severe ID and 7/10 had epilepsy; typically myoclonic seizures, absences or drop attacks. 8/10 had hypotonia, 5/10 had significant constipation, 7/10 had wide-based/unsteady gait, 3/10 had strabismus, and 2/10 had significant hip dysplasia. A proportion of the affected individuals had a similar, myopathic facial appearance, with broad nasal bridge, relatively long nose and full lower lip vermilion. A distinctive behavioral phenotype was also observed with aggressive/challenging behavior and significant sleep problems being common. 7/10 individuals had MR imaging of the brain each of which was reported as normal. The clinical features of the individuals reported here show significant overlap with those associated with 6p21.3 microdeletions, confirming that haploinsufficiency for SYNGAP1 is responsible for both disorders.

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  2. Jeyabalan N, Clement JP. SYNGAP1: Mind the Gap. Front Cell Neurosci. 2016 Feb 15;10:32.

    Abstract
    A cardinal feature of early stages of human brain development centers on the sensory, cognitive, and emotional experiences that shape neuronal-circuit formation and refinement. Consequently, alterations in these processes account for many psychiatric and neurodevelopmental disorders. Neurodevelopment disorders affect 3-4% of the world population. The impact of these disorders presents a major challenge to clinicians, geneticists, and neuroscientists. Mutations that cause neurodevelopmental disorders are commonly found in genes encoding proteins that regulate synaptic function. Investigation of the underlying mechanisms using gain or loss of function approaches has revealed alterations in dendritic spine structure, function, and plasticity, consequently modulating the neuronal circuit formation and thereby raising the possibility of neurodevelopmental disorders resulting from synaptopathies. One such gene, SYNGAP1 (Synaptic Ras-GTPase-activating protein) has been shown to cause Intellectual Disability (ID) with comorbid Autism Spectrum Disorder (ASD) and epilepsy in children. SYNGAP1 is a negative regulator of Ras, Rap and of AMPA receptor trafficking to the postsynaptic membrane, thereby regulating not only synaptic plasticity, but also neuronal homeostasis. Recent studies on the neurophysiology of SYNGAP1, using Syngap1 mouse models, have provided deeper insights into how downstream signaling proteins and synaptic plasticity are regulated by SYNGAP1. This knowledge has led to a better understanding of the function of SYNGAP1 and suggests a potential target during critical period of development when the brain is more susceptible to therapeutic intervention.

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