Childhood tuberous sclerosis complex in southern Sweden: a paradigm shift in diagnosis and treatment

BMC Pediatr. 2023 Jun 29;23(1):329. doi: 10.1186/s12887-023-04137-4.

Abstract

Aim: To investigate the complete clinical spectrum of individuals with paediatric tuberous sclerosis complex in southern Sweden and explore changes over time.

Methods: In this retrospective observational study, 52 individuals aged up to 18 years at the study start were followed-up at regional hospitals and centres for habilitation from 2000 to 2020.

Results: Cardiac rhabdomyoma was detected prenatally/neonatally in 69.2% of the subjects born during the latest ten years of the study period. Epilepsy was diagnosed in 82.7% of subjects, and 10 (19%) were treated with everolimus, mainly (80%) for a neurological indication. Renal cysts were detected in 53%, angiomyolipomas in 47%, astrocytic hamartomas in 28% of the individuals. There was a paucity of standardized follow-up of cardiac, renal, and ophthalmological manifestations and no structured transition to adult care.

Conclusion: Our in-depth analysis shows a clear shift towards an earlier diagnosis of tuberous sclerosis complex in the latter part of the study period, where more than 60% of cases showed evidence of this condition already in utero due to the presence of a cardiac rhabdomyoma. This allows for preventive treatment of epilepsy with vigabatrin and early intervention with everolimus for potential mitigation of other symptoms of tuberous sclerosis complex.

Keywords: Angiomyolipoma; Astrocytic hamartoma; Cardiac rhabdomyoma; Epilepsy; Everolimus.

Publication types

  • Observational Study
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adult
  • Aged
  • Child
  • Early Intervention, Educational
  • Everolimus / therapeutic use
  • Heart Neoplasms* / diagnosis
  • Heart Neoplasms* / therapy
  • Humans
  • Rhabdomyoma* / diagnosis
  • Rhabdomyoma* / therapy
  • Sweden / epidemiology
  • Tuberous Sclerosis* / complications
  • Tuberous Sclerosis* / diagnosis
  • Tuberous Sclerosis* / therapy

Substances

  • Everolimus