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01-01-2018 | Original Article

Salivary glands abnormalities in oculo-auriculo-vertebral spectrum

Authors: Davide Brotto, Renzo Manara, Stefania Vio, Sara Ghiselli, Elena Cantone, Rodica Mardari, Irene Toldo, Valentina Stritoni, Alessandro Castiglione, Elisa Lovo, Patrizia Trevisi, Roberto Bovo, Alessandro Martini

Published in: Clinical Oral Investigations | Issue 1/2018

Abstract

Background

Feeding and swallowing impairment are present in up to 80% of oculo-auriculo-vertebral spectrum (OAVS) patients. Salivary gland abnormalities have been reported in OAVS patients but their rate, features, and relationship with phenotype severity have yet to be defined.

Material and methods

Parotid and submandibular salivary gland hypo/aplasia was evaluated on head MRI of 25 OAVS patients (16 with severe phenotype, Goldenhar syndrome) and 11 controls.

Results

All controls disclosed normal salivary glands. Abnormal parotid glands were found exclusively ipsilateral to facial microsomia in 21/25 OAVS patients (84%, aplasia in six patients) and showed no association with phenotype severity (14/16 patients with Goldenhar phenotype vs 7/9 patients with milder phenotype, p = 0.6). Submandibular salivary gland hypoplasia was detected in six OAVS patients, all with concomitant ipsilateral severe involvement of the parotid gland (p < 0.001). Submandibular salivary gland hypoplasia was associated to Goldenhar phenotype (p < 0.05). Parotid gland abnormalities were associated with ipsilateral fifth (p < 0.001) and seventh cranial nerve (p = 0.001) abnormalities. No association was found between parotid gland anomaly and ipsilateral internal carotid artery, inner ear, brain, eye, or spine abnormalities (p > 0.6).

Conclusions

Salivary gland abnormalities are strikingly common in OAVS. Their detection might help the management of OAVS-associated swallowing and feeding impairment.

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Title: Salivary glands abnormalities in oculo-auriculo-vertebral spectrum
Authors: Davide Brotto
Renzo Manara
Stefania Vio
Sara Ghiselli
Elena Cantone
Rodica Mardari
Irene Toldo
Valentina Stritoni
Alessandro Castiglione
Elisa Lovo
Patrizia Trevisi
Roberto Bovo
Alessandro Martini
Publication date: 01-01-2018
Publisher: Springer Berlin Heidelberg
Published in: Clinical Oral Investigations / Issue 1/2018
Print ISSN: 1432-6981
Electronic ISSN: 1436-3771
DOI: https://doi.org/10.1007/s00784-017-2125-z

Keynote webinar | Spotlight on medication adherence

Springer Medicine

Salivary glands abnormalities in oculo-auriculo-vertebral spectrum

Abstract

Background

Material and methods

Results

Conclusions

Keynote webinar | Spotlight on medication adherence

Springer Medicine

Abstract

Background

Material and methods

Results

Conclusions

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