Entry - 264475 - PSEUDOPAPILLEDEMA, OCULAR HYPOTELORISM, BLEPHAROPHIMOSIS, AND HAND ANOMALIES - OMIM
264475

PSEUDOPAPILLEDEMA, OCULAR HYPOTELORISM, BLEPHAROPHIMOSIS, AND HAND ANOMALIES


Alternative titles; symbols

ACROOTOOCULAR SYNDROME
AOO SYNDROME


Clinical Synopsis
 

INHERITANCE
- Autosomal recessive
GROWTH
Height
- Short stature
Weight
- Birthweight <10th percentile
Other
- Failure to thrive
HEAD & NECK
Face
- Micrognathia
Ears
- Low-set ears
- External auditory meatus atresia
- Mixed deafness
Eyes
- Pseudopapilledema
- Hypotelorism
- Epicanthal folds
- Blepharophimosis
- Downslanting palpebral fissures
- Absence of sulcus orbitopalpebralis superior (upper eyelid crease)
Nose
- Broad nasal base
Mouth
- High-arched palate
- Narrow palate
Teeth
- Malocclusion
- Darkly colored teeth (reddish-brown)
- Anodontia
- Supernumerary teeth
- Delayed dental eruption
CHEST
Ribs Sternum Clavicles & Scapulae
- Pectus excavatum
ABDOMEN
Gastrointestinal
- Multiple episodes of choking (1st year of life)
SKELETAL
- Delayed bone age
Spine
- Kyphoscoliosis
Hands
- Single flexion crease fingers 2, 3, +/-5
- Hypoplastic thenar, hypothenar, and interdigital eminences
- Palmar keratosis
- Short fingers
- Partial syndactyly
- Short metacarpals (4th-5th)
Feet
- Small feet
- Widely spaced 1st-2nd toes
- Zygodactyly
- Short 3rd-4th toes
- Protruding calcaneus
SKIN, NAILS, & HAIR
Skin
- Palmar keratosis
- Poorly developed palmar creases
- Multiple pigmented nevi
ENDOCRINE FEATURES
- Partial growth hormone deficiency

TEXT

Clinical Features

In 3 individuals from 2 inbred sibships of a Brazilian kindred, Paes-Alves et al. (1991) described a syndrome characterized by malformation of the face, ears, hands and feet, associated with mixed deafness and pseudopapilledema. Facial features included ocular hypotelorism, epicanthal folds, and blepharophimosis. The ears were low set and malformed. The thenar, hypothenar, and interdigital areas of the hands were flat and the fingers mildly webbed. A broad space separated the first and second toes. A fourth affected individual in 1 of the sibships was not described in detail. It was suggested that heterozygotes could be identified on the basis of the number of related anomalies they demonstrated.

Bertola et al. (1997) described the case of a 23-year-old man born to consanguineous parents and presenting with pseudopapilledema, mixed hearing loss, and minor facial and limb anomalies. They suggested the designation acrootoocular syndrome.


REFERENCES

  1. Bertola, D. R., Wolf, L. M., Toriello, H. V., Netzloff, M. L. Acro-oto-ocular syndrome: further evidence for a new autosomal recessive disorder. Am. J. Med. Genet. 73: 442-446, 1997. [PubMed: 9415472, related citations]

  2. Paes-Alves, A. F., Azevedo, E. S., Sousa, M. G. F., Almeida-Melo, N., Oliveira-Filho, O. J. Autosomal recessive malformation syndrome with minor manifestation in the heterozygotes: a preliminary report of a possible new syndrome. Am. J. Med. Genet. 41: 141-152, 1991. [PubMed: 1785624, related citations] [Full Text]


Contributors:
Victor A. McKusick - updated : 1/13/1998
Creation Date:
Victor A. McKusick : 11/12/1991
carol : 07/25/2012
mark : 1/16/1998
terry : 1/13/1998
carol : 4/28/1994
mimadm : 3/12/1994
supermim : 3/17/1992
carol : 11/14/1991
carol : 11/12/1991

264475

PSEUDOPAPILLEDEMA, OCULAR HYPOTELORISM, BLEPHAROPHIMOSIS, AND HAND ANOMALIES


Alternative titles; symbols

ACROOTOOCULAR SYNDROME
AOO SYNDROME


SNOMEDCT: 720410001;   ORPHA: 2980;  



TEXT

Clinical Features

In 3 individuals from 2 inbred sibships of a Brazilian kindred, Paes-Alves et al. (1991) described a syndrome characterized by malformation of the face, ears, hands and feet, associated with mixed deafness and pseudopapilledema. Facial features included ocular hypotelorism, epicanthal folds, and blepharophimosis. The ears were low set and malformed. The thenar, hypothenar, and interdigital areas of the hands were flat and the fingers mildly webbed. A broad space separated the first and second toes. A fourth affected individual in 1 of the sibships was not described in detail. It was suggested that heterozygotes could be identified on the basis of the number of related anomalies they demonstrated.

Bertola et al. (1997) described the case of a 23-year-old man born to consanguineous parents and presenting with pseudopapilledema, mixed hearing loss, and minor facial and limb anomalies. They suggested the designation acrootoocular syndrome.


REFERENCES

  1. Bertola, D. R., Wolf, L. M., Toriello, H. V., Netzloff, M. L. Acro-oto-ocular syndrome: further evidence for a new autosomal recessive disorder. Am. J. Med. Genet. 73: 442-446, 1997. [PubMed: 9415472]

  2. Paes-Alves, A. F., Azevedo, E. S., Sousa, M. G. F., Almeida-Melo, N., Oliveira-Filho, O. J. Autosomal recessive malformation syndrome with minor manifestation in the heterozygotes: a preliminary report of a possible new syndrome. Am. J. Med. Genet. 41: 141-152, 1991. [PubMed: 1785624] [Full Text: https://doi.org/10.1002/ajmg.1320410202]


Contributors:
Victor A. McKusick - updated : 1/13/1998

Creation Date:
Victor A. McKusick : 11/12/1991

Edit History:
carol : 07/25/2012
mark : 1/16/1998
terry : 1/13/1998
carol : 4/28/1994
mimadm : 3/12/1994
supermim : 3/17/1992
carol : 11/14/1991
carol : 11/12/1991