Table 1.
Feature | Family 1 | Family 2 | Family 3 | Family 4 | Family 5 | Family 6 | |||||||
---|---|---|---|---|---|---|---|---|---|---|---|---|---|
Individuals | II.1 | II.2 | I.1 | II.2 (i) | II.4 | III.1 | II.1 | I.2 | II.1 | I.1 | II.1 (i) | II.1 (i) | II.2 |
Gender | male | female | male | female | male | female | female | female | female | male | male | male | male |
Age at diagnosis | <5 years | <5 years | forties | <5 years | <5 years | <5 years | <5 years | <5 years | <5 years | preteen | <5 years | preteen | preteen |
Hormone deficiency at diagnosis | GH, TSH | GH, TSH | GH | GH | GH | GH | GH, TSH, PRL | TSH | GH, TSH | GH | GH | GH | GH |
Height at diagnosis (SDS) | N/A | N/A | −3.7 | −5 | −5.3 | N/A | −4 | −5.42 | −3.45 | −4.2 | −4.15 | N/A | N/A |
rhGH treatment | N/A | N/A | no | yes | yes | yes | N/A | yes | yes | yes | yes | yes | N/A |
Final height (cm/SDS) | N/A | N/A | 150/−3.7 | 156.5/−0.9 | 165/−1.5 | N/A | N/A | 150.9/−2.67 | still growing | 147.9/−3.66 | still growing | N/A | N/A |
Pituitary hormone deficiencies | GH, TSH | GH, TSH | GH | GH | GH | GH | GH, TSH, PRL | GH, TSH, PRL, (ACTHa) | GH, TSH, PRL, (ACTHa) | GH | GH | GH | GH |
Pituitary MRI | disrupted stalk | disrupted stalk | normal | normal | normal | N/A | normal | normal | normal | N/A | APH | normal | N/A |
Brain MRI | N/A | N/A | N/A | N/A | N/A | N/A | N/A | 1 cm left frontal and parietal lobe abnormality | normal | normal | normal | N/A | N/A |
Dysmorphic features | none noted | none noted | none noted | large forehead | none noted | N/A | N/A | intellectual disability, strabismus, astigmatism, nystagmus, dysplastic thyroid gland | macroglossia, bilateral hearing impairment, developmental delay, dysplastic thyroid gland | none noted | short stature, frontal bossing, high pitched voice | N/A | N/A |
Molecular findings (heterozygous) | c.148T>G | c.148T>G | c.152T>G | c.152T>G | c.152T>G | c.152T>G | c.155T>G | c.157T>G | c.157T>G | c.150T>G | c.150T>G | c.153T>A | c.153T>A |
p.Ser50Ala | p.Ser50Ala | p.Ile51Ser | p.Ile51Ser | p.Ile51Ser | p.Ile51Ser | p.Leu52Trp | p.Ser53Ala | p.Ser53Ala | p.Ser50= | p.Ser50= | p.Ile51= | p.Ile51= |
Abbreviations are as follows: i, index case; N/A, not available; MRI, magnetic resonance imaging; rhGH, recombinant human growth hormone; APH, anterior pituitary hypoplasia; SDS, standard deviation score; ACTH, adrenocorticotropin.
Diagnosed in late twenties for I.2 and newborn for II.1.