Clinical management and diagnosis of CLN2 disease: consensus of the Brazilian experts group.
Letícia Pereira de Brito SampaioMaria Luíza Giraldes de ManrezaAndre Luis Santos PessoaJuliana Gurgel-GiannettiAna Carolina CoanHélio van der Linden JúniorEmília Katiane E A LeãoAdélia Maria de Miranda Henriques SouzaFernando KokPublished in: Arquivos de neuro-psiquiatria (2023)
Neuronal ceroid lipofuscinosis type 2 (CLN2) is a rare neurodegenerative genetic disease that affects children in early life. Its classic form is rapidly progressive, leading to death within the first 10 years. The urge for earlier diagnosis increases with the availability of enzyme replacement therapy. A panel of nine Brazilian child neurologists combined their expertise in CLN2 with evidence from the medical literature to establish a consensus to manage this disease in Brazil. They voted 92 questions including diagnosis, clinical manifestations, and treatment of the disease, considering the access to healthcare in this country. Clinicians should suspect CLN2 disease in any child, from 2 to 4 years old, with language delay and epilepsy. Even though the classic form is the most prevalent, atypical cases with different phenotypes can be found. Electroencephalogram, magnetic resonance imaging, molecular and biochemical testing are the main tools to investigate and confirm the diagnosis. However, we have limited access to molecular testing in Brazil, and rely on the support from the pharmaceutical industry. The management of CLN2 should involve a multidisciplinary team and focus on the quality of life of patients and on family support. Enzyme replacement therapy with Cerliponase α is an innovative treatment approved in Brazil since 2018; it delays functional decline and provides quality of life. Given the difficulties for the diagnosis and treatment of rare diseases in our public health system, the early diagnosis of CLN2 needs improvement as enzyme replacement therapy is available and modifies the prognosis of patients.
Keyphrases
- replacement therapy
- healthcare
- smoking cessation
- end stage renal disease
- magnetic resonance imaging
- chronic kidney disease
- mental health
- newly diagnosed
- ejection fraction
- early life
- computed tomography
- palliative care
- systematic review
- peritoneal dialysis
- patient reported outcomes
- prognostic factors
- clinical practice
- young adults
- multiple sclerosis
- emergency department
- single molecule
- genome wide
- blood brain barrier
- magnetic resonance
- diffusion weighted imaging