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Setleis Syndrome

OMIM ID:

autosomal recesssive

Setleis Syndrome

Alternate Names

bitemporal forceps marks syndrome
facial ectodermal dysplasia
focal facial dermal dysplasia 3
FFDD3

Defective Genes

TWIST2

Clinical Characteristics

Ocular Features

The eyebrows slant upward and the lashes may be absent or appear in double rows as in distichiasis.  The periorbital skin can appear 'puckered with puffiness'.  The palpebral fissures appear short in some individuals.

Systemic Features

This condition is associated with a variety of facial skin anomalies.  The overall facial appearance has been described as 'leonine".  The facial skin often has some redundancy with a rubbery feel and may appear ‘puffy’ or wrinkled and puckered about the eyes. The lips are large.  The overall appearance has been described as 'leonine'. The nasal ridge can be flat and the nose can have a bulbous tip.  The teeth may be conical in shape.

Genetics

Inheritance

The genetic basis for this disorder seems to lie in heterozygous mutations in the TWIST2 gene (2q37.3).  Parent to child transmission has also been reported for some features but the signs in parents may be subtle.. 

The TWIST2 mutation was not present in several unrelated probands suggesting there is some genetic heterogeneity. Further, Brauer syndrome (136500) (focal facial dermal dysplasia or FFDD type I) is clinically somewhat similar to Setleis syndrome with respect to the distribution and nature of facial skin lesions and it has been suggested that the two disorders may be one and the same.  The gene responsible for Brauer syndrome has not been found but the condition is inherited in autosomal dominant fashion.  It is possible therefore that families labeled Setleis with an AD pattern of transmission actually have Brauer syndrome.  It should also be noted that the ablepharon-macrostonia syndrome (200110) and Barber-Say syndrome (209885) are also caused by mutations in TWIST2 and have some features in common with Setleis syndrome.

 

Pedigree

Autosomal recessive

In order for autosomal recessive disorders to be expressed, offspring generally must inherit two mutations, one from each carrier parent.  Carriers with only one mutation, such as the parents, do not have clinical disease.  Note that carrier parents can expect that 1 in 4 children (25%) will inherit both mutations and have the disorder, 2 in 4 children (50%) will be carriers like their parents, while 1 in 4 children (25%) inherit neither mutation.

Image
Sample pedigree of autosomal recessive inheritance

In order for autosomal recessive disorders to be expressed, offspring generally must inherit two mutations, one from each carrier parent. Carriers with only one mutation, such as the parents, do not have clinical disease. Note that carrier parents can expect that 1 in 4 children (25%) will inherit both mutations and have the disorder, 2 in 4 children (50%) will be carriers like their parents, while 1 in 4 children (25%) inherit neither mutation.

Treatment & Management

No treatment has been reported.

Selected Resources

Web Resources

Publications

Displaying 1 - 7 of 7

Autosomal dominant inheritance in Setleis syndrome

PubMedID: 764559

Autosomal Recessive Inheritance in the Setleis Bitemporal ‘Forceps Marks’ Syndrome

PubMedID: 3631024

Barber–Say syndrome and Ablepharon–Macrostomia syndrome: An overview

PubMedID: 27196381

CONGENITAL ECTODERMAL DYSPLASIA OF THE FACE

PubMedID: 14069095

Evidence for genetic homogeneity of Setleis’ syndrome and focal facial dermal dysplasia

PubMedID: 8204474

Recurrent Mutations in the Basic Domain of TWIST2 Cause Ablepharon Macrostomia and Barber-Say Syndromes

PubMedID: 26119818

Setleis syndrome: Three new cases and a review of the literature

PubMedID: 12210295