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Branchiootic syndrome 1(BOS1)

MedGen UID:
351307
Concept ID:
C1865143
Disease or Syndrome
Synonyms: BO syndrome 1; BOS1; Branchiootic dysplasia
 
Gene (location): EYA1 (8q13.3)
 
Monarch Initiative: MONDO:0011258
OMIM®: 602588

Disease characteristics

Excerpted from the GeneReview: Branchiootorenal Spectrum Disorder
Branchiootorenal spectrum disorder (BORSD) is characterized by malformations of the outer, middle, and inner ear associated with conductive, sensorineural, or mixed hearing impairment, branchial fistulae and cysts, and renal malformations ranging from mild renal hypoplasia to bilateral renal agenesis. Some individuals progress to end-stage renal disease (ESRD) later in life. Extreme variability can be observed in the presence, severity, and type of branchial arch, otologic, audiologic, and renal abnormality from right side to left side in an affected individual and also among individuals in the same family. [from GeneReviews]
Authors:
Richard JH Smith   view full author information

Additional descriptions

From OMIM
Individuals with the branchiootic syndrome (BOS) are affected by the same branchial and otic anomalies as those seen in individuals with the branchiootorenal syndrome (see BOR1, 113650), but lack renal anomalies (Vincent et al., 1997). Although Melnick et al. (1978) maintained that the BO syndrome is distinct from the BOR syndrome because of the lack of renal anomalies and variable presence of deafness in the former, Cremers and Fikkers-van Noord (1980) suggested that the 2 syndromes represent a single entity. See 113600 for a discussion of branchial cleft anomalies, which may be related. Genetic Heterogeneity of Branchiootic Syndrome See also BOS2 (120502), mapped to chromosome 1, and BOS3 (608389), caused by mutation in the SIX1 gene (601205) on chromosome 14q23.  http://www.omim.org/entry/602588
From MedlinePlus Genetics
"Renal" refers to the kidneys; BOR syndrome (but not BO syndrome) causes abnormalities of kidney structure and function. These abnormalities range from mild to severe and can affect one or both kidneys. In some cases, end-stage renal disease (ESRD) develops later in life. This serious condition occurs when the kidneys become unable to filter fluids and waste products from the body effectively.

"Oto-" and "-otic" refer to the ear; most people with BOR/BO syndrome have hearing loss and other ear abnormalities. The hearing loss can be sensorineural, meaning it is caused by abnormalities in the inner ear; conductive, meaning it results from changes in the small bones in the middle ear; or mixed, meaning it is caused by a combination of inner ear and middle ear abnormalities. Some affected people have tiny holes in the skin or extra bits of tissue just in front of the ear. These are called preauricular pits and preauricular tags, respectively.

"Branchio-" refers to the second branchial arch, which is a structure in the developing embryo that gives rise to tissues in the front and side of the neck. In people with BOR/BO syndrome, abnormal development of the second branchial arch can result in the formation of masses in the neck called branchial cleft cysts. Some affected people have abnormal holes or pits called fistulae in the side of the neck just above the collarbone. Fistulae can form tunnels into the neck, exiting in the mouth near the tonsil. Branchial cleft cysts and fistulae can cause health problems if they become infected, so they are often removed surgically.

Branchiootorenal (BOR) syndrome is a condition that disrupts the development of tissues in the neck and causes malformations of the ears and kidneys. The signs and symptoms of this condition vary widely, even among members of the same family. Branchiootic (BO) syndrome includes many of the same features as BOR syndrome, but affected individuals do not have kidney abnormalities. The two conditions are otherwise so similar that researchers often consider them together (BOR/BO syndrome or branchiootorenal spectrum disorders).  https://medlineplus.gov/genetics/condition/branchiootorenal-branchiootic-syndrome

Clinical features

From HPO
Sensorineural hearing loss disorder
MedGen UID:
9164
Concept ID:
C0018784
Disease or Syndrome
A type of hearing impairment in one or both ears related to an abnormal functionality of the cochlear nerve.
Microtia
MedGen UID:
57535
Concept ID:
C0152423
Congenital Abnormality
Underdevelopment of the external ear.
Mixed hearing impairment
MedGen UID:
102336
Concept ID:
C0155552
Disease or Syndrome
A type of hearing loss resulting from a combination of conductive hearing impairment and sensorineural hearing impairment.
Low-set ears
MedGen UID:
65980
Concept ID:
C0239234
Congenital Abnormality
Upper insertion of the ear to the scalp below an imaginary horizontal line drawn between the inner canthi of the eye and extending posteriorly to the ear.
Cupped ear
MedGen UID:
335186
Concept ID:
C1845447
Congenital Abnormality
Laterally protruding ear that lacks antihelical folding (including absence of inferior and superior crura).
Cochlear malformation
MedGen UID:
396278
Concept ID:
C1862050
Finding
The presence of a malformed cochlea.
Dilatated internal auditory canal
MedGen UID:
382996
Concept ID:
C2676973
Finding
The presence of a dilated inner part of external acoustic meatus.
Hypoplasia of the cochlea
MedGen UID:
436824
Concept ID:
C2676974
Finding
Developmental hypoplasia of the cochlea.
Retrognathia
MedGen UID:
19766
Concept ID:
C0035353
Congenital Abnormality
An abnormality in which the mandible is mislocalised posteriorly.
Branchial fistula
MedGen UID:
107802
Concept ID:
C0546968
Congenital Abnormality
A congenital fistula in the neck resulting from incomplete closure of a branchial cleft.
Preauricular pit
MedGen UID:
120587
Concept ID:
C0266610
Congenital Abnormality
Small indentation anterior to the insertion of the ear.

Term Hierarchy

Professional guidelines

PubMed

Jovic TH, Saldanha F, Kuo R, Ahmad T
J Plast Reconstr Aesthet Surg 2014 Sep;67(9):1288-90. Epub 2014 May 14 doi: 10.1016/j.bjps.2014.04.012. PMID: 24933236

Recent clinical studies

Diagnosis

Lin XF, Luo JW, Liu G, Zhu YB, Jin Z, Lin X
Mol Med Rep 2018 Nov;18(5):4271-4280. Epub 2018 Sep 5 doi: 10.3892/mmr.2018.9455. PMID: 30221713Free PMC Article

Clinical prediction guides

Lin XF, Luo JW, Liu G, Zhu YB, Jin Z, Lin X
Mol Med Rep 2018 Nov;18(5):4271-4280. Epub 2018 Sep 5 doi: 10.3892/mmr.2018.9455. PMID: 30221713Free PMC Article

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