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Harlequin baby: a case report

S Singalavanija1, V Sangtawesin, S Horpoapan

  • 1Dermatology Division, Children's Hospital, Bangkok, Thailand.

Journal of the Medical Association of Thailand = Chotmaihet Thangphaet
|June 12, 1998
PubMed
Summary

Harlequin fetus, a severe congenital ichthyosis, poses significant mortality risks. This case highlights prolonged survival in a Thai infant with intensive care and treatment, transitioning to ichthyosiform erythroderma.

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Area of Science:

  • Dermatology
  • Neonatology
  • Genetics

Background:

  • Harlequin fetus is the most severe form of congenital ichthyosis, often fatal within weeks due to complications.
  • Congenital ichthyosis encompasses a group of rare genetic skin disorders.
  • Prompt and intensive neonatal care is critical for survival in severe cases.

Observation:

  • A female infant presented with the clinical features of harlequin fetus.
  • The infant received specialized neonatal intensive care, topical emollients, and oral etretinate.
  • The patient survived the neonatal period and developed congenital non-bullous ichthyosiform erythroderma.

Findings:

  • This case represents the first reported instance of prolonged survival for a harlequin fetus in Thailand.
  • The infant demonstrated delayed growth and development despite intensive management.
  • Skin condition evolved from harlequin fetus to ichthyosiform erythroderma over three years.

Implications:

  • This case underscores the potential for improved outcomes in harlequin fetus with aggressive neonatal management.
  • Long-term follow-up is essential to monitor developmental trajectories and skin condition evolution.
  • Further research into genetic factors and therapeutic interventions for congenital ichthyosis is warranted.

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