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  6. Pigmentary Chorioretinopathy Due To Long-chain 3-hydroxyacyl-coa Dehydrogenase Deficiency (lchad): A Case Report With Long-term Follow-up

Pigmentary chorioretinopathy due to long-chain 3-hydroxyacyl-CoA dehydrogenase deficiency (LCHAD): a case report with long-term follow-up

N Castro Casal1, N Olivier Pascual1, R Arroyo Castillo1

  • 1Servicio de Oftalmología, Complexo Hospitalario Universitario de Ferrol, A Coruña, Spain.

Archivos De La Sociedad Espanola De Oftalmologia|June 14, 2025

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View abstract on PubMed

Summary

Long-chain 3-hydroxyacyl-CoA dehydrogenase deficiency (LCHAD) is a severe genetic disorder causing pigmentary chorioretinopathy. Early diagnosis and monitoring are crucial for managing LCHAD

Area of Science:

  • Medical Genetics
  • Ophthalmology
  • Metabolic Disorders

Background:

  • Long-chain 3-hydroxyacyl-CoA dehydrogenase deficiency (LCHAD) is a rare genetic disorder.
  • LCHAD can lead to pigmentary chorioretinopathy, affecting the retinal pigment epithelium (RPE).

Observation:

  • A 20-year-old female diagnosed with LCHAD via neonatal screening presented with RPE alterations since age 3.
  • Fundus examination revealed a salt-and-pepper pattern and peripheral chorioretinal atrophy.

Findings:

  • The patient remained visually asymptomatic and systemically stable for over 15 years.
  • Regular monitoring using retinography, OCT, FAF, and ERG was essential for disease management.

Implications:

  • This case underscores the critical role of early LCHAD diagnosis through neonatal screening.
  • Proactive management and long-term follow-up are vital for preventing decompensation and improving patient outcomes.
Keywords:
Key wordschorioretinopathycoriorretinopatíacribado neonataldéficit 3-hidroxiacil-CoA-deshidrogenasa de cadena larga (LCHAD)long-chain 3-hydroxyacyl-CoA dehydrogenase deficiency (LCHAD)neonatal screening

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