Evaluation of catatonia in autism and severe depression revealing Phelan-McDermid syndrome and tetrahydrobiopterin deficiency

BMJ Case Rep. 2024 Jan 4;17(1):e256155. doi: 10.1136/bcr-2023-256155.

Abstract

The authors describe a female in her late twenties, presenting with catatonia and diagnosed with epilepsy, autism spectrum disorder, mild intellectual disability, psychosis, dysthymia, anxiety and bipolar disorder, receiving weekly electroconvulsive therapy (ECT). After testing, findings indicated an interstitial deletion in the 22q13.33 region associated with Phelan-McDermid syndrome. In addition, the patient had low cerebral spinal fluid tetrahydrobiopterin (BH4) levels, suggesting dysfunction in the pterin biosynthetic pathway. As a result, the patient started on sapropterin, a BH4 replacement small molecule. After sapropterin treatment, catatonia improved, and the need for ECT decreased. There was an improvement in her cognitive ability, attention and independence. However, there has been no improvement in seizure frequency.

Keywords: Genetics; Psychiatry.

Publication types

  • Case Reports

MeSH terms

  • Adult
  • Autism Spectrum Disorder* / complications
  • Autistic Disorder* / complications
  • Autistic Disorder* / genetics
  • Catatonia* / complications
  • Catatonia* / diagnosis
  • Catatonia* / therapy
  • Chromosome Deletion
  • Chromosome Disorders* / complications
  • Chromosomes, Human, Pair 22
  • Depression
  • Depressive Disorder, Major* / complications
  • Female
  • Humans
  • Phenylketonurias* / complications

Supplementary concepts

  • Telomeric 22q13 Monosomy Syndrome