Description of a multidisciplinary model of care in a French cohort of adult patients with tuberous sclerosis complex

J Med Genet. 2021 Jan;58(1):25-31. doi: 10.1136/jmedgenet-2019-106607. Epub 2020 May 14.

Abstract

Background: Tuberous sclerosis complex (TSC) is a rare autosomal dominant genetic disorder. Due to the various manifestations of TSC and their potential complications, a multidisciplinary care approach is recommended by consensus guidelines.

Objectives: Our study aimed to give a complete description of our TSC adult cohort and to evaluate the multidisciplinary and interdisciplinary management model.

Methods: Data on each adult patient diagnosed with TSC, including disease manifestations, interventions and outcomes, were collected at baseline and updated annually. A multidisciplinary TSC approach with all the recommended explorations was carried out annually.

Results: 90 patients were enrolled in Centre Hospitalier Universitaire de Bordeaux, between January 2000 and September 2018. Median age of patients at inclusion was 37 years (range, 27-47) and 20 years old at diagnosis of TSC. Regarding the occurrence of TSC manifestations, 97% of the patients had cutaneous lesions, 89% had neurological manifestations, 83% had renal manifestations and 100% had dental lesions with pits. More than half the patients had sclerotic bone lesions (68%), TSC-associated neuropsychiatric disorders (64%) and lymphangioleiomyomatosis (59%). A TSC multidisciplinary approach was developed including a global follow-up and an evaluation of TSC targeting organs, according to the recommendations. A satisfaction survey revealed global and entire satisfaction of patients with TSC.

Conclusion: We obtained an accurate description of a cohort of adult patients with TSC. Our multidisciplinary approach model allowed us to provide optimal management of patients with TSC with a high level of patient satisfaction.

Keywords: mTOR inhibitor; quality of life; registry; renal angiomyolipoma; tuberous sclerosis complex.

MeSH terms

  • Adult
  • Cohort Studies
  • Disease Management*
  • Female
  • France / epidemiology
  • Guidelines as Topic
  • Humans
  • Lymphangioleiomyomatosis / complications
  • Lymphangioleiomyomatosis / epidemiology*
  • Lymphangioleiomyomatosis / pathology
  • Lymphangioleiomyomatosis / therapy
  • Male
  • Mental Disorders / complications
  • Mental Disorders / epidemiology*
  • Mental Disorders / pathology
  • Mental Disorders / therapy
  • Middle Aged
  • Surveys and Questionnaires
  • Tuberous Sclerosis / complications
  • Tuberous Sclerosis / epidemiology*
  • Tuberous Sclerosis / pathology
  • Tuberous Sclerosis / therapy