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Harlequin baby: a case report
S Singalavanija1, V Sangtawesin, S Horpoapan
1Dermatology Division, Children's Hospital, Bangkok, Thailand.
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.
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.
