SMA - spinal muscular atrophy - is a disease that until recently was incurable and fatal. In 2016, a light appeared in the tunnel, it was nusinersen, the medicinal substance that gave hope to thousands of patients around the world suffering from this rare, progressive disease.

Nusinersen is the first substance in the world that modifies the operation of the SMN1 gene, responsible for the encoding of SMN proteins. Its absence is the source of spinal muscular atrophy.

Nusinersen was approved by the US Food and Drug Administration (FDA) at the end of 2016, and in mid-2022 the therapy was approved in Europe. In Poland, the drug received reimbursement in January 2022 and wasrapidly implemented for the treatment of SMA patients , as part of the drug program "Treatment of spinal muscular atrophy". The program covered all patients with SMA without any restrictions, the only criterion being the presence of the disease confirmed by genetic tests.

"This is a breakthrough in the treatment of spinal muscular atrophy," doctors say. "The disease just stops," parents say. We asked Iga Grzybowska, mother of two sick children, and Anna Turowska, mother of a boy suffering from spinal muscular atrophy, about how the drug works and how children with SMA live.

Józia was saved by her sister

- Józio was developing properly. He started to crawl at the age of 7 months, he sat and walked around furniture - says Iga Grzybowska, mother of 6-year-old Józia with SMA, 4-year-old Tereska, who is asymptomatic with SMA, and a he althy one-year-old Tadzio. - The first symptoms appeared after a year. We have noticed that there is no progress in walking. He was a kid who never ran and that worried us a bit. We started diagnostics by physiotherapists, orthopedists and other specialists. They reassured him that it was a matter of his temperament, because he was developing very intellectually.

When the gait not only did not improve, but its regression was visible, my parents decided to see a neurologist, eventually they came to Dr. Krystyna Szymańska from the Warsaw pediatric hospital of the Medical University of Warsaw, who immediately recognized SMA. The diagnosis had to be confirmed, however, so Józio was referred for genetic blood tests. He was then a year and 5 months old.

- I am an educator, disability is nothing strange to me, but of coursein the context of their own children it is a completely different matter - says Iga Grzybowska. - First, we were incredulous about the information that our son has SMA. It was difficult for us and we went through such a period of mourning, facing the feeling of fear for what our life would be like now. In addition, at the time of diagnosis, I was already pregnant with my second child, whose he alth could also be at risk - recalls Iga Grzybowska.

And so it happened. When Tereska was born, and research showed that she also suffered from SMA. The Grzybowski family were already prepared for such an eventuality. A few months earlier, they had contacted the Italian physician Enrico Bertini, a pediatric neurologist at the Bambino Gesù Children's Hospital in Rome, who was in the process of clinical trials on a drug containing nusinersen and was looking for asymptomatic children. There are 4 types of SMA. The mild type is the so-called form 3 - during the first months of life, it is asymptomatic, children acquire certain skills, but only then, as a result of the disease, lose them. Józio and Tereska were diagnosed with this form of the disease and such patients were searched for by an Italian specialist in SMA.

- We contacted the doctor and told him our story: that we have a sick Józio and a second daughter "on the way". He assured us that he would take her for treatment - recalls Ms Iga. - As soon as Tereska was born, her tests were performed - the result of genetic tests was positive, so when she was 2 months old, we flew with her to Rome. We already knew that the new drug was very promising, so we had high hopes for this therapy.

The girl was included in the clinical trial, what's more -was also able to treat her brother . As their parents say, Terenia saved Józio at the last moment - the disease progressed, the little one was getting weaker and weaker - he hardly crawled, his muscles began to decline very quickly.

Józio later received therapy from his sister, two years ago. Today he is crawling again, he is able to take a few steps by the furniture, he walks and wears orthoses, he uses the toilet himself, he has no problems in the kindergarten, not only physical but also emotional problems related to his disability.

- He is a very open, happy boy, he rides his pram, says that he is Lightning McQueen - laughs Iga. - His independence is a great joy for us. There are no respiratory problems, no major infections. Tereska, on the other hand, does not feel the effects of the disease at all. Due to the fact that she was given the drug right after birth (and is still getting it), she is a asymptomatic child and does not require anyrehabilitation. He runs, jumps, sings, goes to kindergarten, where he dances in ballet. He's just a volcano of energy.

The parents of Józia and Tereska are full of optimism. They follow reports on the effectiveness of nusinersen therapy in the US, where the drug has been used the longest - each dose is muscle rebuilding and further improvement. It is already known that if treatment is started before the onset of SMA symptoms, it is often possible to prevent the onset of the disease altogether.

- Many parents are waiting for this "wow" effect, that is, that their children will suddenly start to walk on their own - says Iga Grzybowska. - But the longer we live with this disease, the more we enjoy the little things, trifles that affect children's independence and their mobility. Our son takes the 10th dose of the drug and each time we notice some progress, whether it is an improvement in muscle strength or a new skill.

More than a year ago, the Grzybowski family decided to have a third child. Tadzio was born he althy and is a great joy for the whole family. He has already started to crawl and wants to play with his brother, which motivates Józio to be active.

- We are believers, deciding to have a third child, we knew that she could be sick, but we trusted very much that it would not be. We were aware that even if it turned out to be so, this perspective of SMA disease is different for us - says Iga Grzybowska. - We are both therapists, we can see how strong our marriage is. Although we have a disease in our family, in retrospect we can see that it was she who gave us this strength.

Radek lost many months

For 14 months now, thanks to the reimbursement, 4-year-old Radek, who is told by his mother, Anna Turowska, has been using nusinersen.

- My son was born in asphyxia, after a transferred pregnancy, after a very difficult birth, so from the first days of his life he was under the care of a neurologist - says Anna. - Doctors, however, saw no problems, apart from the decreased muscle tone. Radek developed normally. He tried to sit up, pulled his head up, rolled, crawled. But when he was 9-10 months old and still wasn't sitting by himself, he was taking the crawling posture, but he couldn't pull himself up with his arms, we started to worry.

The parents reported the boy to a physiotherapist, then privately to a neurologist, an early development psychological and pedagogical clinic, and even to a geneticist. In the meantime, they began rehabilitation for poor muscle tone. Rehabilitation improved, but the baby was still not crawling or walking. When Radek was 12 months old, the doctor sent him to the hospital for metabolic and other tests, also for SMA, but due to the lack of places, andthen due to illness, the child did not go there until six months later.

- We waited a month for the results and finally got a diagnosis - recalls Anna Turowska. - The worst has been confirmed. All tests for genetic and metabolic diseases were negative, only SMA was positive. Additionally, I was already in the 24th week of twin pregnancy. Immediately questions arose, what about them? Doctors wanted to do amniocentesis, but they were afraid, and I also categorically refused because of the high risk of damage to the fetuses. It was a difficult time - there was no joy from pregnancy, only fear for the children, and it was necessary to take care of Radek, his rehabilitation …

Two girls were born. Already on the third day, tests were carried out on them, due to the genetic burden and the disease of the older brother. The results only came a month later, but they were good.Girls are he althy, but just like their parents, they carry the defective SMN gene.This means that they may pass it on to their children in the future.

- In a dozen or so years, we will have to have a hard conversation with them on this topic. Maybe they will ask themselves earlier, because they will see Radek, his illness. The time will come to explain it to them, maybe someday they will have to test their partners … - wonders Anna Turowska. Fortunately, there is hope for SMA treatment with nusinersen, which really gives amazing results, and there is talk of introducing newborn screening tests that would allow for early detection of the disease and introduction of immediate treatment. It is a pity that Radek did not have such a possibility -the diagnosis took too long, and we were unlucky , because if our son's test results came three days earlier, we would have a chance to include him in the clinical trial of nusinersen. The treatment results were definitely better, and yes - we've lost many months.

Radek has now taken the eighth dose of the drug for SMA - he has been receiving it since March 2022, he was the first child to receive the drug under reimbursement. The drug is administered into the cerebrospinal fluid through a lumbar puncture. The boy is much stronger after him, the disease does not progress, and he regains the skills he lost in the first years of his life. He can sit independently, he also moves in a wheelchair. He plays with his beloved cars, raises his hands high, which was impossible before. His torso is more stable, he has also regained control of holding his head, so he starts rolling on the floor, he can turn from his back to his tummy.

- Radek will have to take the drug for the rest of his life, he will also have to exercise. Rehabilitation is very muchimportant, that's why my son has it every day. A physiotherapist visits him 4 times a week, and exercises with us 3 days a week. The improvement is slow but very visible. And that's what makes us happy.

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