Combined mutations of NKX2-1 and surfactant protein C genes for refractory low oxyhemoglobin saturation and interstitial pneumonia: A case report

Medicine (Baltimore). 2020 Mar;99(12):e19650. doi: 10.1097/MD.0000000000019650.

Abstract

Rationale: Mutations of the NKX2-1 gene are associated with brain-lung-thyroid syndrome, which is characterized by benign hereditary chorea, hypothyroidism, and pulmonary disease with variable presentation. Surfactant protein C (SFTPC) gene mutations result in chronic interstitial lung disease in adults or severe neonatal respiratory distress syndrome.

Patient concerns: Recurrent hypoxemia was observed shortly after birth in a baby at a gestational age of 40 weeks and birth weight of 3150 g. The need for respiratory support gradually increased. He had hypothyroidism and experienced feeding difficulties and irritability.

Diagnosis: Genetic examination of the peripheral blood revealed combined mutations of the NKX2-1 and SFTPC genes.

Interventions: The patient was administered respiratory support, antibiotics, low-dose dexamethasone, supplementary thyroxine, venous nutrition, and other supportive measures.

Outcomes: The patient's guardian stopped treatment 3 months after commencement of treatment, due to the seriousness of his condition and the patient died.

Lessons: Combined mutations of NKX2-1 and SFTPC genes are very rare. Thus, idiopathic interstitial pneumonia with hypothyroidism and neurological disorders require special attention.

Publication types

  • Case Reports

MeSH terms

  • Athetosis / blood
  • Athetosis / diagnosis
  • Athetosis / genetics*
  • Athetosis / therapy
  • Chorea / blood
  • Chorea / diagnosis
  • Chorea / genetics*
  • Chorea / therapy
  • Congenital Hypothyroidism / blood
  • Congenital Hypothyroidism / diagnosis
  • Congenital Hypothyroidism / genetics*
  • Congenital Hypothyroidism / therapy
  • Fatal Outcome
  • Feeding and Eating Disorders / diagnosis
  • Feeding and Eating Disorders / etiology
  • Humans
  • Hypothyroidism / diagnosis
  • Hypothyroidism / etiology
  • Hypoxia / diagnosis
  • Hypoxia / etiology
  • Infant, Newborn
  • Karyotyping
  • Lung Diseases, Interstitial / diagnosis
  • Lung Diseases, Interstitial / etiology
  • Male
  • Mutation
  • Palliative Care / methods
  • Protein C / metabolism*
  • Pulmonary Surfactants / metabolism*
  • Recurrence
  • Respiratory Distress Syndrome, Newborn / blood
  • Respiratory Distress Syndrome, Newborn / diagnosis
  • Respiratory Distress Syndrome, Newborn / etiology
  • Respiratory Distress Syndrome, Newborn / genetics*
  • Respiratory Distress Syndrome, Newborn / therapy
  • Thyroid Nuclear Factor 1 / genetics*

Substances

  • NKX2-1 protein, human
  • Protein C
  • Pulmonary Surfactants
  • Thyroid Nuclear Factor 1

Supplementary concepts

  • Choreoathetosis, Hypothyroidism, And Neonatal Respiratory Distress