There are only a limited number of studies that assess long-term outcomes in infantile spasms and they show that, at most, one quarter of patients have a favourable long-term cognitive outcome and one third remain seizure-free. Many comorbidities are associated with infantile spasms (autism, cerebral palsy, sensory defects), and mortality is high throughout life.[77]Riikonen R. Infantile spasms: outcome in clinical studies. Pediatr Neurol. 2020 Jul;108:54-64.
https://www.pedneur.com/article/S0887-8994(20)30041-2/fulltext
http://www.ncbi.nlm.nih.gov/pubmed/32305143?tool=bestpractice.com
Several factors influence the prognosis of children with infantile spasms. Most studies investigating short- and long-term outcomes are retrospective and include different populations and treatments. Whether prognosis has changed over time is difficult to assess.
Aetiology is one of the most important prognostic indicators and improved aetiological diagnostics should allow for more accurate prognostic counselling. Where no underlying cause is found (previously called ‘idiopathic’ or ‘cryptogenic’ spasms) after first investigations, a known or presumed genetic predisposition is likely, and these patients generally have a favourable outcome. However, individuals with primarily genetic causes such as SCN2A are more likely to have poor developmental outcomes.[78]Osborne JP, Edwards SW, Dietrich Alber F, et al. The underlying etiology of infantile spasms (West syndrome): information from the International Collaborative Infantile Spasms Study (ICISS). Epilepsia. 2019 Sep;60(9):1861-9.
http://www.ncbi.nlm.nih.gov/pubmed/31418851?tool=bestpractice.com
If the aetiology remains unknown and development was normal before spasm onset, the patient can be free of spasms and have normal or nearly normal psychomotor development in about 80% cases.[79]Granström ML, Gaily E, Liukkonen E. Treatment of infantile spasms: results of a population-based study with vigabatrin as the first drug for spasms. Epilepsia. 1999 Jul;40(7):950-7.
https://onlinelibrary.wiley.com/doi/epdf/10.1111/j.1528-1157.1999.tb00802.x
http://www.ncbi.nlm.nih.gov/pubmed/10403219?tool=bestpractice.com
[80]Cohen-Sadan S, Kramer U, Ben-Zeev B, et al. Multicenter long-term follow-up of children with idiopathic West syndrome: ACTH versus vigabatrin. Eur J Neurol. 2009 Apr;16(4):482-7.
http://www.ncbi.nlm.nih.gov/pubmed/19348622?tool=bestpractice.com
[81]Kivity S, Lerman P, Ariel R, et al. Long-term cognitive outcomes of a cohort of children with cryptogenic infantile spasms treated with high-dose adrenocorticotropic hormone. Epilepsia. 2004 Mar;45(3):255-62.
https://onlinelibrary.wiley.com/doi/full/10.1111/j.0013-9580.2004.30503.x
http://www.ncbi.nlm.nih.gov/pubmed/15009227?tool=bestpractice.com
Patients with symptomatic aetiology may still have a favourable seizure outcome if the aetiology is neonatal hypoglycaemia, Down syndrome, stroke or infarct, periventricular leukomalacia, or neurofibromatosis.[82]Gaily E, Lommi M, Lapatto R, et al. Incidence and outcome of epilepsy syndromes with onset in the first year of life: a retrospective population-based study. Epilepsia. 2016 Oct;57(10):1594-601.
https://onlinelibrary.wiley.com/doi/10.1111/epi.13514
http://www.ncbi.nlm.nih.gov/pubmed/27574005?tool=bestpractice.com
The outcome is almost always poor in children with severe brain malformations, post-infectious aetiology, and tuberous sclerosis.
Several studies have found a relationship between short time (lead-time) to treatment and improved mental outcome.[39]Lombroso CT. A prospective study of infantile spasms: clinical and therapeutic correlations. Epilepsia. 1983 Apr;24(2):135-58.
http://www.ncbi.nlm.nih.gov/pubmed/6299719?tool=bestpractice.com
[79]Granström ML, Gaily E, Liukkonen E. Treatment of infantile spasms: results of a population-based study with vigabatrin as the first drug for spasms. Epilepsia. 1999 Jul;40(7):950-7.
https://onlinelibrary.wiley.com/doi/epdf/10.1111/j.1528-1157.1999.tb00802.x
http://www.ncbi.nlm.nih.gov/pubmed/10403219?tool=bestpractice.com
[80]Cohen-Sadan S, Kramer U, Ben-Zeev B, et al. Multicenter long-term follow-up of children with idiopathic West syndrome: ACTH versus vigabatrin. Eur J Neurol. 2009 Apr;16(4):482-7.
http://www.ncbi.nlm.nih.gov/pubmed/19348622?tool=bestpractice.com
[83]Jeavons PM, Bower BD, Dimitrakoudi M. Long-term prognosis of 150 cases of "West syndrome". Epilepsia. 1973 Jun;14(2):153-64.
http://www.ncbi.nlm.nih.gov/pubmed/4515986?tool=bestpractice.com
[84]O'Callaghan FJ, Lux AL, Darke K, et al. The effect of lead time to treatment and of age of onset on developmental outcome at 4 years in infantile spasms: evidence from the United Kingdom Infantile Spasms Study. Epilepsia. 2011 Jul;52(7):1359-64.
https://onlinelibrary.wiley.com/doi/10.1111/j.1528-1167.2011.03127.x
http://www.ncbi.nlm.nih.gov/pubmed/21668442?tool=bestpractice.com
[85]Auvin S, Hartman AL, Desnous B, et al. Diagnosis delay in West syndrome: misdiagnosis and consequences. Eur J Pediatr. 2012 Nov;171(11):1695-701.
http://www.ncbi.nlm.nih.gov/pubmed/22892960?tool=bestpractice.com
[86]Primec ZR, Stare J, Neubauer D. The risk of lower mental outcome in infantile spasms increases after three weeks of hypsarrhythmia duration. Epilepsia. 2006 Dec;47(12):2202-5.
https://onlinelibrary.wiley.com/doi/10.1111/j.1528-1167.2006.00888.x
http://www.ncbi.nlm.nih.gov/pubmed/17201726?tool=bestpractice.com
Adverse prognostic indicators include:[77]Riikonen R. Infantile spasms: outcome in clinical studies. Pediatr Neurol. 2020 Jul;108:54-64.
https://www.pedneur.com/article/S0887-8994(20)30041-2/fulltext
http://www.ncbi.nlm.nih.gov/pubmed/32305143?tool=bestpractice.com
Underlying aetiology (previously called ‘symptomatic’ infantile spasms): in particular, brain malformations, post-infectious aetiology, and tuberous sclerosis
Presence of developmental abnormalities at spasm onset
Longer duration of hypsarrhythmia (>3 weeks).[86]Primec ZR, Stare J, Neubauer D. The risk of lower mental outcome in infantile spasms increases after three weeks of hypsarrhythmia duration. Epilepsia. 2006 Dec;47(12):2202-5.
https://onlinelibrary.wiley.com/doi/10.1111/j.1528-1167.2006.00888.x
http://www.ncbi.nlm.nih.gov/pubmed/17201726?tool=bestpractice.com
Predictors of favorable outcome include:[77]Riikonen R. Infantile spasms: outcome in clinical studies. Pediatr Neurol. 2020 Jul;108:54-64.
https://www.pedneur.com/article/S0887-8994(20)30041-2/fulltext
http://www.ncbi.nlm.nih.gov/pubmed/32305143?tool=bestpractice.com
Unknown aetiology (previously called ‘idiopathic' or ‘cryptogenic' infantile spasms)
Normal development at spasm onset
A short duration of hypsarrhythmia (regardless of aetiology, but may be especially important in the unknown aetiology group)
Short lead-time to treatment.