Background. - Congential hypothyroidism is very rare compared to prima
ry hypothyroidism. Its early diagnosis may escape neonatal mass screen
ing using TSH assay. Case report. - Anthony was born at 37 weeks, weig
hing 3,060 g. He presented with hypotony, jaundice, tongue protrusion
evoking congenital hypothyroidism. Thyroid function tests favored hypo
thyroidism central in origin, while the systematic neonatal screening
was normal. Conclusion. - Clinical signs of congenital hypothyroidism
must lead to more specific tests when neonatal screening is normal.