Tympanic Membrane Retractions in patients with Williams Syndrome: A Controlled Study

Oliveira, Marcelo Wierzynski; Lavinsky, Joel; Valerio, Marcel Machado; Felix, Temis Maria; Lavinsky, Luiz

doi:10.1055/s-0040-1715151

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International Archives of Otorhinolaryngology

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Original Research • iao 25 (3) • Jul-Sep 2021 • https://doi.org/10.1055/s-0040-1715151 copy

Tympanic Membrane Retractions in patients with Williams Syndrome: A Controlled Study

Authorship SCIMAGO INSTITUTIONS RANKINGS

Abstract

Introduction

The role of elastin in tympanic retractions and chronic otitis media is not well established. Williams Syndrome (WS), a pathology related to a mutation in the elastin gene, could generate tympanic retractions.

Objective

To compare the prevalence of tympanic retractions among patients with WS and controls.

Methods

WS patients (n= 43 ears) and controls (n= 130 ears) were evaluated by digital otoscopic examination and the degree of tympanic membrane retraction was classified by 2 blinded experienced otolaryngologists.

Results

The agreement rate between the evaluators was 71.1% for pars tensa and 65% for pars flaccida retraction (p< 0.001). The pars tensa and pars flaccida retractions are present in patients with WS after an adjusted residue of respectively - 2.8 and - 2.6 (p= 0.011 and p= 0.022) compared with controls.

Conclusions

Tympanic membrane retractions are not more common in the WS group when compared with controls.

Keywords
williams syndrome; elastin; chronic otitis media

Variables	Controls n = 67 n (%) or average	Williams Syndrome n = 22 n (%) or average	p-value (lower limit-upper Limit)
Female	31 (48%)	6 (27%)	0.135 (0.135-0.143)^* * Mann-Whitney.
Age	12.66	12.73	0.974 ^μ μ Student t-test.
History of oologic disorders	16 (25%)	11 (50%)	0.032 (0.028-0.037)^* * Mann-Whitney.
History of nasosinusal disorders	15 (23%)	6 (27%)	0.768 (0.758-0.779)^* * Mann-Whitney.

			Evaluator 2		Total	Kappa	p-value
			Without retraction	With retraction	Total	Kappa	p-value
Normal	Evaluator 1	Without retraction	86	8	94	0.686	< 0.001
	Evaluator 1	With Retraction	8	27	35
	Total		94	35	129
Williams Syndrome	Evaluator 1	Without retraction	41	1	42	0.788	< 0.001
	Evaluator 1	With Retraction	0	2	2
	Total		41	3	44

			Evaluator 2		Total	Kappa	p-value
			Without retraction	With retraction	Total	Kappa	p-value
Controls	Evaluator 1	Without retraction	93	13	106	0.648	< 0.001
	Evaluator 1	With retraction	3	21	24
	Total		96	34	130
Williams Syndrome	Evaluator 1	Without retraction	41	2	43	0.482	< 0.001
	Evaluator 1	With retraction	0	1	1
	Total		41	3	44

	Kappa or adjusted residual^* * Adjusted residual.	p-value
Pars tensa retractions (concordance)	0.711	< 0.001
Pars flaccida retractions (concordance)	0.65	< 0.001
Pars tensa retractions in Williams Syndrome	- 2.8^* * Adjusted residual.	0.011
Pars flaccida retractions in Williams Syndrome	- 2.6^* * Adjusted residual.	0.022

Fundação Otorrinolaringologia R. Teodoro Sampaio, 483, 05405-000 São Paulo/SP Brasil, Tel.: (55 11) 3068-9855, Fax: (55 11) 3079-6769 - São Paulo - SP - Brazil
E-mail: iaorl@iaorl.org

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[1] Address for correspondence Joel Lavinsky, MD, MSc, Departamento de Ciências Morfológicas, Universidade Federal do Rio Grande do Sul, Rua Sarmento Leite, 500, Porto Alegre, RS 90040-060, Brazil (e-mail: jlavinskybr@yahoo.com).