A case report - iKnow

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Otolaryngology Case Reports 16 (2020) 100202

Available online 4 July 2020

Speech improvement in a child with submucous cleft palate surgery: A case report

Dini Widiarni Widodo

^a^,^*

, Tri Juda Airlangga

^b

, Gita Aryanti

^c

aDepartment of Otorhinolaryngology-Head and Neck Surgery, Facialplasty Reconstructive Division. Faculty of Medicine Universitas Indonesia, Dr. Cipto Mangunkusumo Hospital, Jakarta, Indonesia

bDepartment of Otorhinolaryngology-Head and Neck Surgery, ENT Community Division. Faculty of Medicine Universitas Indonesia, Dr. Cipto Mangunkusumo Hospital, Jakarta, Indonesia

cDepartment of Otorhinolaryngology-Head and Neck Surgery, Faculty of Medicine Universitas Indonesia, Dr. Cipto Mangunkusumo Hospital, Jakarta, Indonesia

A R T I C L E I N F O Keywords:

Speech improvement Uji gajah

Submucous cleft palate V-Y Pushback

Furlow double-opposing Z-plasty technique

A B S T R A C T

This article reports a case from Indonesia with velopharyngeal insufficiency caused by submucous cleft palate.

This 4-year-old girl, with the chief complaint of nasal sound and congenital palate defect, underwent V-Y Pushback combined with Furlow Double-Opposing Z-Plasty technique surgery. A single operation, by combining these techniques, was performed to line up the hard and soft palate of uvula. Two weeks after surgery, there was instant phonation improvement, which was evaluated by Uji Gajah (Elephant test) used in Bahasa, Indonesia. We conclude that combined palatoplasty and veloplasty surgical techniques may provide good velopharyngeal function results.

Introduction

Submucous cleft palate is a congenital defect resulting from the lack of normal fusion of the muscle within the soft palate. It occurs either alone or along with congenital anomalous syndrome. According to previous study, the incidence was 1 out of 1200 subjects with a male-to- female ratio of 1:2 [1]. This disorder affects the child’s oral growth and development and may cause speech disorder, speech delay, and diffi- culty in food intake [2,3] (see Figs. 1 and 2)

The method of surgery is determined by the location of the defect whether it is in the hard palate or the soft palate. The surgical methods for the hard palate include Veau-Wardill-Kilner V-Y pushback, von Langenbeck, two-flap, while the surgical method for soft palate is mainlyintravelar reconstruction such as Furlow Double-opposing Z- plasty [4].

Speech therapy is the most essential treatment for its management besides surgery. According to Danelz’s study, velopharyngeal insufficiency occurred in around 20–50% of patients despite palate repair operations [6]. Speech therapy can help children to learn the correct articulation and adjust the pressure in the oral cavity to increase sound production [5,6]. We used Uji Gajah (Elephant Test) in Bahasa Indonesia for the evaluation of velopharyngeal insufficiency in Indonesian patients

after surgery [7].

This report aims to describe a case of submucous cleft palate and velopharyngeal insufficiency treated with combined surgical technique of V-Y pushback and Z-plasty and its speech outcome using Uji Gajah.

Case report

The parents of a 4-year-old girl complained of unclear speech and nasal sound since the time she was able to speak. There were no other complaints. This was the first-time that the patient asked for medical help for her hypernasality. There were no face or congenital abnor- malities in the patient’s family. On investigations, there was a right perihilar and paracardial infiltrate on the chest radiograph, without any clinical complaints. On pre-operative oral examination, a thin gap defects about 3 cm �1 cm were seen both in the soft and hard palates. The patient suffered congenital submucous cleft palate. The evaluation of speech revealed hypernasal sound by Uji Gajah. Patient was diagnosed with submucous cleft palate and velopharyngeal insufficiency, and palatoplasty surgery was planned with V-Y pushback technique and Z- plasty technique. Written consent for surgery was obtained from the patient’s parents.

The surgery was conducted on April 8, 2019 with the V-Y pushback

* Corresponding author. Facialplasty Reconstructive/Otorhinolaryngology-Head and Neck Surgery. Faculty of Medicine Universitas Indonesia, Jalan Diponegoro No 71, Jakarta, 10430, Indonesia.

E-mail address: [email protected] (D.W. Widodo).

Contents lists available at ScienceDirect

Otolaryngology Case Reports

journal homepage: www.elsevier.com/locate/xocr

https://doi.org/10.1016/j.xocr.2020.100202

Received 10 May 2020; Received in revised form 30 June 2020; Accepted 1 July 2020

Downloaded for Anonymous User (n/a) at University of Indonesia from ClinicalKey.com by Elsevier on April 26, 2022. For personal use only. No other uses without permission. Copyright ©2022. Elsevier Inc. All rights reserved.

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Otolaryngology Case Reports 16 (2020) 100202

2 technique combined with Z-plasty. There was no complication related to surgery. The patient recovered in several days and asked to come for second Uji Gajah examination. The result showed that her nasal sound disappeared. She pronounced words clearer than before. The compari- son of results can be observed in video. (Vid.2) The patient’s speech further improved after several home speech therapy sessions from her mother.

Discussion

Submucous cleft palate is identical with the bifid uvula triad, the absence of posterior nasal spines, and the presence of muscle diastasis in the velum which finally gives a picture of the pellucid zone [8]. Velo- pharyngeal insufficiency occurs because the levator palatin muscle is located more anteriorly to the palate durum. Most cases of submucous cleft palate are asymptomatic. However, about 15% of them can suffer from velopharyngeal insufficiency, which is associated with limited palate mobility, shortened palate, and weakened palate [8]. In this case,

the patient was diagnosed with asymptomatic submucous cleft palate based on the defects in the soft palate and speech disorders. In this case, the indication of surgery was the presence of velopharyngeal insufficiency.

Uji Gajah (Elephant test) in Bahasa, Indonesia is a modified ‘Zoo passage’ test for nasality by Fletcher. The study was conducted in 1999 to create a measurement tool for detecting nasality in simple tests. Uji Gajah includes 22 nasal consonants/m, n, N/, and six vowels (i, e, �e, u, o, a) in Bahasa Indonesia. Those consonants were made into a short story which was read by the patients. Pronouncing Uji Gajah was aimed to enhance patient’s nasality if there was any. Along with the other studies used in Indonesia, Uji sengau and Uji Hantu, this test had been performed by physicians for more than 20 years [9].

Many surgeons performed combined techniques to shorten the length of treatment and lessen the number of surgeries [10]. The results vary based on the change in nasality of the patients. Based on the sur- geon’s experience, better treatment outcome was obtained in their patients. Neatly closed soft and hard palate may produce better Fig. 1.(a)Preoperative, (b)Post-Operative Day 1 (Image is being edited by Elsevier Illustrator).

Fig. 2.VY-Pushback and furlow double Z opposing combination technique.

D.W. Widodo et al.

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Otolaryngology Case Reports 16 (2020) 100202

3 pronunciation without any nasality.

Author statment

Dini Widiarni Widodo: Conceptualization, Writing,reviewing, and editing. Gita Aryanti: Writing- andoriginal draft preparation. Tri Juda Airlangga: Supervision

Summary

Surgical interventions can be performed to repair both hard palate and soft palate at once using V-Y Pushback combined with Z-plasty technique in submucous cleft palate patients. Soft palate reconstruction in patients with cleft palate gives better functional outcomes and reduce the incidence of velopharyngeal incompetence. Uji Gajah can be used as the measurement of nasal sound in the present case.

Declaration of competing interest

The authors declare that they have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this paper.

Appendix A. Supplementary data

Supplementary data to this article can be found online at https://doi.

org/10.1016/j.xocr.2020.100202.

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