- Email: [email protected]
Assessment of chronic childhood dysphonia
European Annals of Otorhinolaryngology, Head and Neck diseases 131 (2014) 309–312
Available online at
ScienceDirect www.sciencedirect.com
Review
Assessment of chronic childhood dysphonia E. Mornet a , B. Coulombeau b , P. Fayoux c , J.-P. Marie d , R. Nicollas e , D. Robert-Rochet f , R. Marianowski a,∗ a
Service ORL et chirurgie cervico-faciale, hôpital Morvan, 2, avenue Foch, 29609 Brest cedex, France Service ORL et chirurgie cervico-faciale, hôpital Femme-Mère-Enfant, 59, boulevard Pinel, 69677 Bron cedex, France Service ORL et chirurgie cervico-faciale, hôpital Jeanne-de-Flandre, avenue Eugène-Avinée, 59037 Lille, France d Service ORL et chirurgie cervico-faciale, hôpital Charles-Nicolle, 1, rue de Germont, 76031 Rouen cedex, France e Service ORL et chirurgie cervico-faciale, hôpital d’enfant de la Timone, 264, rue Saint-Pierre, 13385 Marseille cedex 05, France f Service ORL et chirurgie cervico-faciale, hôpital de la Timone Adulte, 264, rue Saint-Pierre, 13385 Marseille cedex 5, France b c
a r t i c l e
i n f o
Keywords: Voice Dysphonia Children Endoscopic examination
a b s t r a c t Chronic childhood dysphonia is a common condition in the school-age period. Perceived functional disorder is subjective and the alert is usually given by a person not belonging to the child’s immediate environment. History-taking often suggests a malformation or acquired lesion. Functional assessment helps measure and diagnose the vocal impairment. Physical and endoscopic assessment in consultation is the key examination: it is only rarely impossible in children and can often found diagnosis. Additional examinations are sometimes necessary. © 2014 Elsevier Masson SAS. All rights reserved.
1. Introduction Chronic childhood voice disorder is a frequent condition, especially in the school-age period [1–4]. When there are no associated symptoms, consultation is usually triggered by the child’s school or music teachers rather than by family members. It impacts on school, family, and social life [4,5]. Impaired communication may impair social integration (difficulty in making oneself heard, remarks about vocal timbre or intensity, etc.) [6]; it may be a source of anxiety for parents and/or cause physical discomfort that may in some cases be painful. The present article describes the epidemiology of chronic childhood voice disorder and the diagnostic approach, which must be multidisciplinary [6–9]. We shall deal only with laryngeal conditions liable to induce isolated chronic childhood voice disorder, excluding acute dysphonia of infectious onset and vocal disorder induced by endocrine pathology. 2. Epidemiology The few studies of the frequency of childhood dysphonia report prevalence of 6% to 38% in school-age children [1,10,11].
∗ Corresponding author. E-mail addresses: [email protected], [email protected] (R. Marianowski). http://dx.doi.org/10.1016/j.anorl.2013.02.001 1879-7296/© 2014 Elsevier Masson SAS. All rights reserved.
Two large-scale systematic studies found a peak incidence between 8 and 10 years (43–44%), although about 30% of patients were aged 7 or under [12,13]. Age at onset is often difficult to determine precisely. In the absence of associated symptoms, the interval between onset and consultation tends to be more than 1 year. There is a strong male predominance of about 60% in dysphonia [1,2,11–13]; this is less marked before 7 years of age, followed by a male predominance which equalizes by 11 years and then turns into a female predominance as of 13 years [5,12,13]. Nodular pathology is the most frequent (55–68%), followed by congenital lesions (27–41%).
3. Clinical presentation 3.1. Functional impairment Assessment criteria for dysphonia vary greatly [14,15]. A recent transverse study of a large cohort of 8 years old from the UK [1] reported a prevalence of 6% when assessment was made by a clinician and of almost 12% when made by the child’s parents. Almost 5% of children were assessed by clinicians as being dysphonic despite their parents having noticed no signs of dysphonia. It is unusual (1%) for consultation to be the child’s own request; it is more often (about 60%) that of an outside party (family physician, speech therapist, music teacher, etc.) and in about 40% of cases that of a close friend or family member [4,16].
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3.2. Anamnesis and history 3.2.1. Anamnesis The first consultation includes history-taking with the child and parents [6,12]. A fundamental point, which might at first sight seem unimportant, concerns the person who reported or noticed the dysphonia and triggered consultation. This is an important indication when the time comes to suggest the optimal form of treatment. The interview determines the history of the dysphonia, and notably the age at onset of vocal disturbance. Dysphonia may be reported by friends or relatives as early as the first years or even months of life, without the parents themselves being at all worried by what they take to be simply the child’s own particular voice. Vocal disorder may be noticed when the child begins school, at around 3 or 4 years of age, after a more or less clear period in which the child’s voice had been considered normal. Schematically, longstanding dysphonia going back to the child’s first vocalizations, although not pathognomic, is suggestive of congenital pathology. Onset later than the beginning of schooling is suggestive of acquired pathology. A correlation with onset of ENT inflammation may also be a trigger. Evolution, and especially variation in dysphonia according to varying vocal demand, provides useful information for both diagnosis and treatment. Improvement at weekends or school holidays suggest that the child’s vocal behaviour is an important factor. Dysphonia, however severe, that is stable over the year or presenting variations unrelated to any vocal effort is suggestive of congenital malformation of vocal fold structure. 3.2.2. History Prematurity, neonatal intensive care, a particularity about the first cry or familial history of dysphonia are suggestive of a congenital lesion, especially in case of associated respiratory symptoms. Any psychomotor developmental abnormalities and the chronology of language acquisition are to be noted. Previous or current swallowing impairment and associated ENT (infection, auditory disorder, allergy), pneumologic (asthma, allergy) or digestive pathology (gastro-oesophageal reflux) should be screened for [1,8]. Finally, the interview should cover corticosteroid inhalation therapy [17] and history of tracheal intubation or ENT surgery. 3.2.3. General presentation It is essential to obtain details of the child’s personality and environment. It needs to be established whether the child is introverted or extraverted, carefree or anxious, sociable or solitary and how easily he or she communicates with adults and other children in the family and school environment. The existence of siblings and the patient’s position among them, a noisy home environment and a family habit of talking loudly are factors for functional dysphonia. School level, school-related and sports activities (individual or team sports) and musical or arts activities shed light on the child’s personality and help assess the degree of personal trouble. Finally, very young children may be mimicking a dysphonic parent [18]. 4. Voice function assessment Laryngeal examination, although central, is not the only clinical examination to be performed in consultation for childhood
dysphonia. Vocal characteristics should be noted, observing and assessing the attitudes the child adopts when using the voice [4]. Such assessment may be made, in whole or in part, during each of the examinations performed and is especially important at the first examination, setting an initial benchmark from which to assess the impact of the vocal disorder and the potential investment and motivation of child and family in any intended therapy; later, it will serve to assess treatment benefit and guide subsequent indications. Direct listening to the voice, by ear [19], is fundamental and is one of the most precise and reliable means of perceptual assessment when performed by an experienced examiner. It begins during the interview with the child and the parents, with a general assessment of the impact of the dysphonia on the child’s communication. Subsequently, more systematic assessment is required [20], including if at all possible recordings that can be kept for reference; these may be digital (computerized or not) or analogic. They should allow easy comparison during successive consultations. Various samples of voice use should be recorded [21]: reading a simple, fairly short text, if the child can read; singing voice (with a well-known children’s song); increased-volume speaking voice (e.g., counting as loudly as possible) and possibly prolonged vowelsounds (/a/ /e/ /u/). Ideally, collecting and recording samples should be associated to measuring the mean intensity of each on a sonometer. These recordings allow the main acoustic features of the voice to be assessed: tone, volume and pitch. Pitch is assessed as the mean fundamental frequency for the sample [22]. It will vary greatly according to age and type of expression [8,12,21,23–25] but, for the conversational voice or the reading voice after 6 years of age, averages 320 Hz around 3 years, 280–300 Hz around 4–5 years and 250–270 Hz around 6–7 years; it also depends on gender, averaging 240–260 Hz in boys and 250–270 Hz in girls around 11 years. With puberty-induced voice changes, tone varies radically in boys (120–140 Hz) but little in girls (220–250 Hz). It can be assessed by ear, with the help of a keyboard, with a little practice, or by asking the child to prolong the final syllable for a count of three [12]. It can also by measured on a frequency analysis system [26–28]. Volume is measured in dBSPL , using a sonometer. It generally approximates 65–70 dB for the speaking voice and 90 dB for the increased-volume speaking voice [12]. It can be useful to note the minimum sustainable intensity for a continuous vowel-sound; this is one of the parameters used in calculating the DSI (dysphonia severity index) [29]. Tone is “characteristic” of each individual, and largely results from transformation of the sound emitted from the laryngeal vestibule by crossing resonance cavities. It is the hardest aspect of voice quality to assess. The human ear is able to discern voice pitch and its variations with great finesse, but the difficulty lies in describing these observations. The most widely used scale in phoniatrics is Hirano’s [4,20], rating 5 voice pathology parameters on 4 levels (0 = normal to 3 = severe): G (grade), R (roughness), B (breathiness), A (asthenicity), S (strain). It was usefully supplemented by a 6th parameter (I: instability) by Dejonckere et al. [30]. Finally, although it is not always feasible to obtain a voice range profile [31] in due form in consultation, a rough assessment of vocal ambit (range between the lowest and highest sound the child can emit) can easily be made. The highest producible frequency is also a parameter in the DSI [29]. Objective instrumental voice assessment has developed spectacularly over the last two decades and numerous parameters have been reported. Their interest for diagnosis and follow-up in everyday clinical practice is debatable, but three are worth assessing and are relatively accessible.
E. Mornet et al. / European Annals of Otorhinolaryngology, Head and Neck diseases 131 (2014) 309–312
Maximum phonation time [20] for a given vowel-sound, usually /a/ or /e/, may if possible be associated to phonation flow rate, which approximates 60–100 mL/s for a comfortable intensity [19]. Jitter is the mean frequency variation on a given periodic sound, measured cycle to cycle; it is easy to determine for a vowel-sound on a frequency analysis system [19,26–28]. Both maximum phonation time and jitter are used in the DSI [29]. The s/z ratio between the longest /s/ and /z/ sounds the child can sustain is easy to determine: it usually approximates 1 and increases in case of poor laryngeal vibratory function. During the interview and the above examinations, the child’s vocal behaviour should also be assessed. Posture, verticality, seated and standing balance, face and neck muscle attitudes, respiratory gestures and the child’s commitment help complete the general assessment of vocal dysfunction, adapt treatment and subsequently assess benefit. The patient’s own subjective assessment [20] is becoming a reference in clinical practice, quantifying the disability in everyday, social, school and family life induced by the dysphonia. The Voice Handicap Index [32] has now been validated in several European languages [33] and is widely used in clinical practice. It has been suggested that it should be adapted for use with children [34,35].
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breathing) and getting used to the contact of the endoscope on the tongue. Local anesthesia will then be superfluous [37] (although this has been disputed [38]). After these preliminaries, the examination itself is quite quick. Unlike the recommended procedure in adults [37], the examiner holds the child’s tongue without pulling. The choice between a 70◦ and a 90◦ endoscope basically depends on the examiner’s habits: a 70◦ lens provides better visualization of the anterior commissure and avoids the uvula, whereas a 90◦ model enables better assessment of the laryngopharynx as a whole and leaves the head in a more “physiological” position. As soon as possible, stroboscopic examination of laryngeal vibration should be associated, providing precious diagnostic information [20,39,40]. In young children, the first examination may often provide images that are too brief for stroboscopy to be associated [13,37]. In older children, over successive examinations, proper and interpretable stroboscopy can be performed. Exceptionally, laryngeal examination proves impossible [13]; the child may then be examined often much more easily after a fairly short delay (of a few weeks, if possible) [21]. Systematic video-tape or computerized recording is especially interesting when images are short, allowing finer analysis of the sequences obtained and helping in explaining matters to the child and family. As with voice recording, comparison with subsequent examinations is often very useful.
5. Physical examination Physical examination is often quite easy to perform during consultation if one takes time to explain things to the child, especially when he or she is young. In some 20% of cases, there can be real difficulties, although only in 2% of cases does it prove impossible to obtain sufficient imaging for precise diagnosis. Examination is often informative, as it is very unusual (0.7–1.5% [12,13]) to find normal vocal folds in a dysphonic child, unlike in adults. 5.1. Rigid endoscopy Although often intuitively thought to be difficult [36] or impossible in children [2], rigid endoscopy is in fact usually feasible as of 4½ to 5 years of age, or even younger [4,37]. It is essential for the child to be prepared so as to ensure cooperation, briefly trying out each step of the examination (sonorization, tonality, mouth
5.2. Flexible endoscopy Flexible endoscopy can use adult caliber fibers if the child is big enough, but pediatric fibers are often preferable [4], with a diameter of about 2 mm, being better tolerated, although the reduced image definition and the impossibility of associating stroboscopy may impair the precision of diagnosis. The development and marketing of digital 3 mm flexible endoscopes are making it possible to get round these limitations and obtain better quality images and analyzable stroboscopic examinations [38]. It is not rare for flexible endoscopy to be poorly tolerated, especially by younger children, and it should be then replaced by rigid endoscopy whenever possible. Flexible endoscopy, however, is of unquestioned interest for assessing pharyngolaryngeal behaviour, especially in child singer [12], or when blowing in a musical instrument.
Chronic childhood dysphonia
syndromic
functional
Isolated lesion congenital
-Anterior commissure synechia / glottic stenosis -Epidermoid cyst – sulcus / open cyst – mucosal bridge
-Impaired laryngeal mobility
acquired -Papillomatosis -Nodular lesion
-Psychogenic or psychological dysphonia (and dysphonia)
- Adolescent voice disorder
-Polyp -Mucus retention cyst or glandular cyst
-Sulcus / vergeture
-Glottic stenosis
- Diastema
- Tumor
Fig. 1. Etiologies of chronic childhood dysphonia.
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5.3. “Classic” ENT examination Systematic oro-pharyngoscopy, otoscopy and anterior rhinoscopy complete assessment, seeking risk factors and associated factors. 6. Endoscopy under general anesthesia Endoscopy under general anesthesia is seldom required for diagnosis. It can, however, determine the topography of stenoses and tumours. Diagnostic biopsy of papillomatous and tumoral lesions can be performed during endoscopy. 7. Imaging Etiologic diagnosis is mainly clinical and endoscopic. Ultrasound, CT and MRI are indicated for tumoral syndrome assessment. Stenosis assessment requires CT. Virtual endoscopy has been suggested and should be assessed as a replacement for classical endoscopy during follow-up. 8. Etiologies In isolated dysphonia, at the end of the clinical examination, a laryngeal malformation or acquired lesion is usually diagnosed [12,13]. Functional disorder is less often suggested (Fig. 1). 9. Conclusion Assessment of childhood dysphonia should determine the cause and also the severity and the impact on the child’s life. The interview and clinical examination are usually enough to allow diagnosis. Sometimes complementary examinations and endoscopy under general anesthesia allow more precise diagnosis, enabling the dysphonia to be treated. Disclosure of interest The authors declare that they have no conflicts of interest concerning this article. References [1] Carding PN, Roulstone S, Northstone K, et al. The prevalence of childhood dysphonia: a cross-sectional study. J Voice 2006;20(4):623–30. [2] Connelly A, Clement WA, Kubba H. Management of dysphonia in children. J Laryngol Otol 2009;123(6):642–7. [3] Dejonckere PH. Voice problems in children: pathogenesis and diagnosis. Int J Pediatr Otorhinolaryngol 1999;49(Suppl. 1):S311–4. [4] Kaufmann I, Lina-Granade G, Truy E, et al. La dysphonie chronique de l’enfant. Mise au point à la lumière d’une série personnelle de 54 cas. Pediatrie 1992;47:313–9. [5] Connor NP, Cohen SB, Theis SM, et al. Attitudes of children with dysphonia. J Voice 2008;22(2):197–209. [6] McMurray JS. Disorders of phonation in children. Pediatr Clin North Am 2003;50(2):363–80. [7] Niedzielska G, Glijer E, Niedzielski A. Acoustic analysis of voice in children with noduli vocales. Int J Pediatr Otorhinolaryngol 2001;60(2):119–22. [8] Rahbar R, Healy GB. Voice disorders in the pediatric population. In: Rubin JS, Sataloff RT, Korovin GS, editors. Diagnosis and treatment of voice disorders. 3rd ed. San Diego, CA: Plural Publishing, Inc; 2006. p. 371–80. [9] Vaghela HM, Fergie N, Slade S, et al. Speech therapist led voice clinic: which patients may be suitable? Logoped Phoniatr Vocol 2005;30(2):85–90.
[10] Nicollas R, Giovanni A, Triglia JM. Dysphonia in children. Arch Pediatr 2008;15(6):1133–8. [11] Lecoq M, Drape F. Epidemiological survey of dysphonia in children at primary school entry. Rev Laryngol Otol Rhinol (Bord) 1996;117(4):323–5. [12] Arias Marsal C, Estapé i Vilà M. Disfonía infantil: diagnóstico y tratamiento. Barcelona: Ars Médica; 2005 [230 p.]. [13] Cornut G, Trolliet-Cornut A. Les dysphonies de l’enfant : aspects cliniques et thérapeutiques. Rééducation orthophonique : les pathologies vocales chez l’enfant, 194. Paris: Fédération Nationale des Orthophonistes (FNO); 1998. p. 9–17. [14] Kilic¸ MA, O˘güt F, Dursun G, et al. The effects of vowels on voice perturbation measures. J Voice 2004;18(3):318–24. [15] Merati AL, Keppel K, Braun NM, et al. Pediatric voice-related quality of life: findings in healthy children and in common laryngeal disorders. Ann Otol Rhinol Laryngol 2008;117(4):259–62. [16] Bonet M, Casan P. Evaluation of dysphonia in a children’s choir. Folia Phoniatr Logop 1994;46(1):27–34. [17] Roland NJ, Bhalla RK, Earis J. The local side effects of inhaled corticosteroids: current understanding and review of the literature. Chest 2004;126(1): 213–9. [18] Cornut G. Genèse de la voix de l’enfant. JFORL J Fr Otorhinolaryngol Audiophonol Chir Maxillofac 1971;2:411–6. [19] Giovanni A. Les dysphonies de l’enfant. In: Garabedian EN, Bobin S, Monteil JP, et al., editors. ORL de l’enfant. 2e ed. Paris: Éditions Flammarion MédecineSciences; 2006. p. 266–77. [20] Dejonckere PH, Bradley P, Clemente P, et al. A basic protocol for functional assessment of voice pathology, especially for investigating the efficacy of (phonosurgical) treatments and evaluating new assessment techniques. Guideline elaborated by the Committee on Phoniatrics of the European Laryngological Society (ELS). Eur Arch Otorhinolaryngol 2001;258(2):77–82. [21] Heuillet-Martin G, Garson-Bavard H, Legre A. Les enfants dysphoniques. Une voix pour tous, Tome 2, 2e ed. Paris: Éditions Solal; 1997. p. 54–76. [22] Coulombeau B. Étude acoustique de la voix. In: Cornut G, et al., editors. Moyens d’investigation et pédagogie de la voix chantée Lyon: Éditions Symétrie; 2002. p. 57–66. [23] Brenot C, Kowalik-Cucciniello L. Étude du fondamental laryngé dans la voix parlée chez l’enfant de sept à dix ans. Lyon: Mémoire d’orthophonie; 1989. [24] Magne S, Oudot S, Voisin C. Étude du fondamental laryngé dans la voix parlée entre 10 et 20 ans. Lyon: Mémoire d’orthophonie; 1991. [25] Woisard V, Percodani J, Serrano E, et al. La voix de l’enfant : évolution morphologique du larynx et ses conséquences acoustiques. Rev Laryngol Otol Rhinol (Bord) 1996;117(4):313–7. [26] Boersma P, Weenink D. Praat: doing phonetics by computer. Praat – http://www.fon.hum.uva.nl/praat/. [27] Campisi P, Tewfik TL, Manoukian JJ, et al. Computer-assisted voice analysis: establishing a pediatric database. Arch Otolaryngol Head Neck Surg 2002;128(2):156–60. [28] Van Lieshout P. Praat short tutorial – http://oraldynamicslab.ca/. [29] Wuyts FL, De Bodt MS, Molenberghs G, et al. The dysphonia severity index: an objective measure of vocal quality based on a multiparameter approach. J Speech Lang Hear Res 2000;43(3):796–809. [30] Dejonckere PH. Techniques de base d’évaluation de la voix. Cabay: Louvain-laNeuve; 1985. p. 1–296. [31] Wuyts FL, Heylen L, Mertens F, et al. Effects of age, sex, and disorder on voice range profile characteristics of 230 children. Ann Otol Rhinol Laryngol 2003;112(6):540–8. [32] Jacobson BH, Johnson A, Grywalski C, et al. The voice handicap index (VHI): development and validation. Am J Speech Lang Pathol 1997;6(3):66–70. [33] Verdonck-de Leeuw IM, Kuik DJ, De Bodt M, et al. Validation of the voice handicap index by assessing equivalence of European translations. Folia Phoniatr Logop 2008;60(4):173–8. [34] Schindler A, Capaccio P, Maruzzi P, et al. Preliminary considerations on the application of the voice handicap index to paediatric dysphonia. Acta Otorhinolaryngol Ital 2007;27(1):22–6. [35] Schindler A, Tiddia C, Ghidelli C, et al. Adaptation and validation of the Italian pediatric voice handicap index. Folia Phoniatr Logop 2011;63(1):9–14. [36] Wolf M, Primov-Fever A, Amir O, et al. The feasibility of rigid stroboscopy in children. Int J Pediatr Otorhinolaryngol 2005;69(8):1077–9. [37] Cornut G, Bouchayer M. Assessing dysphonia: the role of videostroboscopy. An interactive videotextbook. Gibraltar: The 3 Ears Company Ltd; 1998. [38] Hartnick CJ, Zeitels SM. Pediatric video laryngostroboscopy. Int J Pediatr Otorhinolaryngol 2005;69(2):215–9. [39] Hernández Sandemetrio R, Nieto Curiel P, Dalmau Galofre J, et al. What is the contribution of stroboscopy in the diagnosis of voice disorders? Acta Otorrinolaringol Esp 2010;61(2):145–8. [40] Mortensen M, Schaberg M, Woo P. Diagnostic contributions of videolaryngostroboscopy in the pediatric population. Arch Otolaryngol Head Neck Surg 2010;136(1):75–9.
Available online at
ScienceDirect www.sciencedirect.com
Review
Assessment of chronic childhood dysphonia E. Mornet a , B. Coulombeau b , P. Fayoux c , J.-P. Marie d , R. Nicollas e , D. Robert-Rochet f , R. Marianowski a,∗ a
Service ORL et chirurgie cervico-faciale, hôpital Morvan, 2, avenue Foch, 29609 Brest cedex, France Service ORL et chirurgie cervico-faciale, hôpital Femme-Mère-Enfant, 59, boulevard Pinel, 69677 Bron cedex, France Service ORL et chirurgie cervico-faciale, hôpital Jeanne-de-Flandre, avenue Eugène-Avinée, 59037 Lille, France d Service ORL et chirurgie cervico-faciale, hôpital Charles-Nicolle, 1, rue de Germont, 76031 Rouen cedex, France e Service ORL et chirurgie cervico-faciale, hôpital d’enfant de la Timone, 264, rue Saint-Pierre, 13385 Marseille cedex 05, France f Service ORL et chirurgie cervico-faciale, hôpital de la Timone Adulte, 264, rue Saint-Pierre, 13385 Marseille cedex 5, France b c
a r t i c l e
i n f o
Keywords: Voice Dysphonia Children Endoscopic examination
a b s t r a c t Chronic childhood dysphonia is a common condition in the school-age period. Perceived functional disorder is subjective and the alert is usually given by a person not belonging to the child’s immediate environment. History-taking often suggests a malformation or acquired lesion. Functional assessment helps measure and diagnose the vocal impairment. Physical and endoscopic assessment in consultation is the key examination: it is only rarely impossible in children and can often found diagnosis. Additional examinations are sometimes necessary. © 2014 Elsevier Masson SAS. All rights reserved.
1. Introduction Chronic childhood voice disorder is a frequent condition, especially in the school-age period [1–4]. When there are no associated symptoms, consultation is usually triggered by the child’s school or music teachers rather than by family members. It impacts on school, family, and social life [4,5]. Impaired communication may impair social integration (difficulty in making oneself heard, remarks about vocal timbre or intensity, etc.) [6]; it may be a source of anxiety for parents and/or cause physical discomfort that may in some cases be painful. The present article describes the epidemiology of chronic childhood voice disorder and the diagnostic approach, which must be multidisciplinary [6–9]. We shall deal only with laryngeal conditions liable to induce isolated chronic childhood voice disorder, excluding acute dysphonia of infectious onset and vocal disorder induced by endocrine pathology. 2. Epidemiology The few studies of the frequency of childhood dysphonia report prevalence of 6% to 38% in school-age children [1,10,11].
∗ Corresponding author. E-mail addresses: [email protected], [email protected] (R. Marianowski). http://dx.doi.org/10.1016/j.anorl.2013.02.001 1879-7296/© 2014 Elsevier Masson SAS. All rights reserved.
Two large-scale systematic studies found a peak incidence between 8 and 10 years (43–44%), although about 30% of patients were aged 7 or under [12,13]. Age at onset is often difficult to determine precisely. In the absence of associated symptoms, the interval between onset and consultation tends to be more than 1 year. There is a strong male predominance of about 60% in dysphonia [1,2,11–13]; this is less marked before 7 years of age, followed by a male predominance which equalizes by 11 years and then turns into a female predominance as of 13 years [5,12,13]. Nodular pathology is the most frequent (55–68%), followed by congenital lesions (27–41%).
3. Clinical presentation 3.1. Functional impairment Assessment criteria for dysphonia vary greatly [14,15]. A recent transverse study of a large cohort of 8 years old from the UK [1] reported a prevalence of 6% when assessment was made by a clinician and of almost 12% when made by the child’s parents. Almost 5% of children were assessed by clinicians as being dysphonic despite their parents having noticed no signs of dysphonia. It is unusual (1%) for consultation to be the child’s own request; it is more often (about 60%) that of an outside party (family physician, speech therapist, music teacher, etc.) and in about 40% of cases that of a close friend or family member [4,16].
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3.2. Anamnesis and history 3.2.1. Anamnesis The first consultation includes history-taking with the child and parents [6,12]. A fundamental point, which might at first sight seem unimportant, concerns the person who reported or noticed the dysphonia and triggered consultation. This is an important indication when the time comes to suggest the optimal form of treatment. The interview determines the history of the dysphonia, and notably the age at onset of vocal disturbance. Dysphonia may be reported by friends or relatives as early as the first years or even months of life, without the parents themselves being at all worried by what they take to be simply the child’s own particular voice. Vocal disorder may be noticed when the child begins school, at around 3 or 4 years of age, after a more or less clear period in which the child’s voice had been considered normal. Schematically, longstanding dysphonia going back to the child’s first vocalizations, although not pathognomic, is suggestive of congenital pathology. Onset later than the beginning of schooling is suggestive of acquired pathology. A correlation with onset of ENT inflammation may also be a trigger. Evolution, and especially variation in dysphonia according to varying vocal demand, provides useful information for both diagnosis and treatment. Improvement at weekends or school holidays suggest that the child’s vocal behaviour is an important factor. Dysphonia, however severe, that is stable over the year or presenting variations unrelated to any vocal effort is suggestive of congenital malformation of vocal fold structure. 3.2.2. History Prematurity, neonatal intensive care, a particularity about the first cry or familial history of dysphonia are suggestive of a congenital lesion, especially in case of associated respiratory symptoms. Any psychomotor developmental abnormalities and the chronology of language acquisition are to be noted. Previous or current swallowing impairment and associated ENT (infection, auditory disorder, allergy), pneumologic (asthma, allergy) or digestive pathology (gastro-oesophageal reflux) should be screened for [1,8]. Finally, the interview should cover corticosteroid inhalation therapy [17] and history of tracheal intubation or ENT surgery. 3.2.3. General presentation It is essential to obtain details of the child’s personality and environment. It needs to be established whether the child is introverted or extraverted, carefree or anxious, sociable or solitary and how easily he or she communicates with adults and other children in the family and school environment. The existence of siblings and the patient’s position among them, a noisy home environment and a family habit of talking loudly are factors for functional dysphonia. School level, school-related and sports activities (individual or team sports) and musical or arts activities shed light on the child’s personality and help assess the degree of personal trouble. Finally, very young children may be mimicking a dysphonic parent [18]. 4. Voice function assessment Laryngeal examination, although central, is not the only clinical examination to be performed in consultation for childhood
dysphonia. Vocal characteristics should be noted, observing and assessing the attitudes the child adopts when using the voice [4]. Such assessment may be made, in whole or in part, during each of the examinations performed and is especially important at the first examination, setting an initial benchmark from which to assess the impact of the vocal disorder and the potential investment and motivation of child and family in any intended therapy; later, it will serve to assess treatment benefit and guide subsequent indications. Direct listening to the voice, by ear [19], is fundamental and is one of the most precise and reliable means of perceptual assessment when performed by an experienced examiner. It begins during the interview with the child and the parents, with a general assessment of the impact of the dysphonia on the child’s communication. Subsequently, more systematic assessment is required [20], including if at all possible recordings that can be kept for reference; these may be digital (computerized or not) or analogic. They should allow easy comparison during successive consultations. Various samples of voice use should be recorded [21]: reading a simple, fairly short text, if the child can read; singing voice (with a well-known children’s song); increased-volume speaking voice (e.g., counting as loudly as possible) and possibly prolonged vowelsounds (/a/ /e/ /u/). Ideally, collecting and recording samples should be associated to measuring the mean intensity of each on a sonometer. These recordings allow the main acoustic features of the voice to be assessed: tone, volume and pitch. Pitch is assessed as the mean fundamental frequency for the sample [22]. It will vary greatly according to age and type of expression [8,12,21,23–25] but, for the conversational voice or the reading voice after 6 years of age, averages 320 Hz around 3 years, 280–300 Hz around 4–5 years and 250–270 Hz around 6–7 years; it also depends on gender, averaging 240–260 Hz in boys and 250–270 Hz in girls around 11 years. With puberty-induced voice changes, tone varies radically in boys (120–140 Hz) but little in girls (220–250 Hz). It can be assessed by ear, with the help of a keyboard, with a little practice, or by asking the child to prolong the final syllable for a count of three [12]. It can also by measured on a frequency analysis system [26–28]. Volume is measured in dBSPL , using a sonometer. It generally approximates 65–70 dB for the speaking voice and 90 dB for the increased-volume speaking voice [12]. It can be useful to note the minimum sustainable intensity for a continuous vowel-sound; this is one of the parameters used in calculating the DSI (dysphonia severity index) [29]. Tone is “characteristic” of each individual, and largely results from transformation of the sound emitted from the laryngeal vestibule by crossing resonance cavities. It is the hardest aspect of voice quality to assess. The human ear is able to discern voice pitch and its variations with great finesse, but the difficulty lies in describing these observations. The most widely used scale in phoniatrics is Hirano’s [4,20], rating 5 voice pathology parameters on 4 levels (0 = normal to 3 = severe): G (grade), R (roughness), B (breathiness), A (asthenicity), S (strain). It was usefully supplemented by a 6th parameter (I: instability) by Dejonckere et al. [30]. Finally, although it is not always feasible to obtain a voice range profile [31] in due form in consultation, a rough assessment of vocal ambit (range between the lowest and highest sound the child can emit) can easily be made. The highest producible frequency is also a parameter in the DSI [29]. Objective instrumental voice assessment has developed spectacularly over the last two decades and numerous parameters have been reported. Their interest for diagnosis and follow-up in everyday clinical practice is debatable, but three are worth assessing and are relatively accessible.
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Maximum phonation time [20] for a given vowel-sound, usually /a/ or /e/, may if possible be associated to phonation flow rate, which approximates 60–100 mL/s for a comfortable intensity [19]. Jitter is the mean frequency variation on a given periodic sound, measured cycle to cycle; it is easy to determine for a vowel-sound on a frequency analysis system [19,26–28]. Both maximum phonation time and jitter are used in the DSI [29]. The s/z ratio between the longest /s/ and /z/ sounds the child can sustain is easy to determine: it usually approximates 1 and increases in case of poor laryngeal vibratory function. During the interview and the above examinations, the child’s vocal behaviour should also be assessed. Posture, verticality, seated and standing balance, face and neck muscle attitudes, respiratory gestures and the child’s commitment help complete the general assessment of vocal dysfunction, adapt treatment and subsequently assess benefit. The patient’s own subjective assessment [20] is becoming a reference in clinical practice, quantifying the disability in everyday, social, school and family life induced by the dysphonia. The Voice Handicap Index [32] has now been validated in several European languages [33] and is widely used in clinical practice. It has been suggested that it should be adapted for use with children [34,35].
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breathing) and getting used to the contact of the endoscope on the tongue. Local anesthesia will then be superfluous [37] (although this has been disputed [38]). After these preliminaries, the examination itself is quite quick. Unlike the recommended procedure in adults [37], the examiner holds the child’s tongue without pulling. The choice between a 70◦ and a 90◦ endoscope basically depends on the examiner’s habits: a 70◦ lens provides better visualization of the anterior commissure and avoids the uvula, whereas a 90◦ model enables better assessment of the laryngopharynx as a whole and leaves the head in a more “physiological” position. As soon as possible, stroboscopic examination of laryngeal vibration should be associated, providing precious diagnostic information [20,39,40]. In young children, the first examination may often provide images that are too brief for stroboscopy to be associated [13,37]. In older children, over successive examinations, proper and interpretable stroboscopy can be performed. Exceptionally, laryngeal examination proves impossible [13]; the child may then be examined often much more easily after a fairly short delay (of a few weeks, if possible) [21]. Systematic video-tape or computerized recording is especially interesting when images are short, allowing finer analysis of the sequences obtained and helping in explaining matters to the child and family. As with voice recording, comparison with subsequent examinations is often very useful.
5. Physical examination Physical examination is often quite easy to perform during consultation if one takes time to explain things to the child, especially when he or she is young. In some 20% of cases, there can be real difficulties, although only in 2% of cases does it prove impossible to obtain sufficient imaging for precise diagnosis. Examination is often informative, as it is very unusual (0.7–1.5% [12,13]) to find normal vocal folds in a dysphonic child, unlike in adults. 5.1. Rigid endoscopy Although often intuitively thought to be difficult [36] or impossible in children [2], rigid endoscopy is in fact usually feasible as of 4½ to 5 years of age, or even younger [4,37]. It is essential for the child to be prepared so as to ensure cooperation, briefly trying out each step of the examination (sonorization, tonality, mouth
5.2. Flexible endoscopy Flexible endoscopy can use adult caliber fibers if the child is big enough, but pediatric fibers are often preferable [4], with a diameter of about 2 mm, being better tolerated, although the reduced image definition and the impossibility of associating stroboscopy may impair the precision of diagnosis. The development and marketing of digital 3 mm flexible endoscopes are making it possible to get round these limitations and obtain better quality images and analyzable stroboscopic examinations [38]. It is not rare for flexible endoscopy to be poorly tolerated, especially by younger children, and it should be then replaced by rigid endoscopy whenever possible. Flexible endoscopy, however, is of unquestioned interest for assessing pharyngolaryngeal behaviour, especially in child singer [12], or when blowing in a musical instrument.
Chronic childhood dysphonia
syndromic
functional
Isolated lesion congenital
-Anterior commissure synechia / glottic stenosis -Epidermoid cyst – sulcus / open cyst – mucosal bridge
-Impaired laryngeal mobility
acquired -Papillomatosis -Nodular lesion
-Psychogenic or psychological dysphonia (and dysphonia)
- Adolescent voice disorder
-Polyp -Mucus retention cyst or glandular cyst
-Sulcus / vergeture
-Glottic stenosis
- Diastema
- Tumor
Fig. 1. Etiologies of chronic childhood dysphonia.
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5.3. “Classic” ENT examination Systematic oro-pharyngoscopy, otoscopy and anterior rhinoscopy complete assessment, seeking risk factors and associated factors. 6. Endoscopy under general anesthesia Endoscopy under general anesthesia is seldom required for diagnosis. It can, however, determine the topography of stenoses and tumours. Diagnostic biopsy of papillomatous and tumoral lesions can be performed during endoscopy. 7. Imaging Etiologic diagnosis is mainly clinical and endoscopic. Ultrasound, CT and MRI are indicated for tumoral syndrome assessment. Stenosis assessment requires CT. Virtual endoscopy has been suggested and should be assessed as a replacement for classical endoscopy during follow-up. 8. Etiologies In isolated dysphonia, at the end of the clinical examination, a laryngeal malformation or acquired lesion is usually diagnosed [12,13]. Functional disorder is less often suggested (Fig. 1). 9. Conclusion Assessment of childhood dysphonia should determine the cause and also the severity and the impact on the child’s life. The interview and clinical examination are usually enough to allow diagnosis. Sometimes complementary examinations and endoscopy under general anesthesia allow more precise diagnosis, enabling the dysphonia to be treated. Disclosure of interest The authors declare that they have no conflicts of interest concerning this article. References [1] Carding PN, Roulstone S, Northstone K, et al. The prevalence of childhood dysphonia: a cross-sectional study. J Voice 2006;20(4):623–30. [2] Connelly A, Clement WA, Kubba H. Management of dysphonia in children. J Laryngol Otol 2009;123(6):642–7. [3] Dejonckere PH. Voice problems in children: pathogenesis and diagnosis. Int J Pediatr Otorhinolaryngol 1999;49(Suppl. 1):S311–4. [4] Kaufmann I, Lina-Granade G, Truy E, et al. La dysphonie chronique de l’enfant. Mise au point à la lumière d’une série personnelle de 54 cas. Pediatrie 1992;47:313–9. [5] Connor NP, Cohen SB, Theis SM, et al. Attitudes of children with dysphonia. J Voice 2008;22(2):197–209. [6] McMurray JS. Disorders of phonation in children. Pediatr Clin North Am 2003;50(2):363–80. [7] Niedzielska G, Glijer E, Niedzielski A. Acoustic analysis of voice in children with noduli vocales. Int J Pediatr Otorhinolaryngol 2001;60(2):119–22. [8] Rahbar R, Healy GB. Voice disorders in the pediatric population. In: Rubin JS, Sataloff RT, Korovin GS, editors. Diagnosis and treatment of voice disorders. 3rd ed. San Diego, CA: Plural Publishing, Inc; 2006. p. 371–80. [9] Vaghela HM, Fergie N, Slade S, et al. Speech therapist led voice clinic: which patients may be suitable? Logoped Phoniatr Vocol 2005;30(2):85–90.
[10] Nicollas R, Giovanni A, Triglia JM. Dysphonia in children. Arch Pediatr 2008;15(6):1133–8. [11] Lecoq M, Drape F. Epidemiological survey of dysphonia in children at primary school entry. Rev Laryngol Otol Rhinol (Bord) 1996;117(4):323–5. [12] Arias Marsal C, Estapé i Vilà M. Disfonía infantil: diagnóstico y tratamiento. Barcelona: Ars Médica; 2005 [230 p.]. [13] Cornut G, Trolliet-Cornut A. Les dysphonies de l’enfant : aspects cliniques et thérapeutiques. Rééducation orthophonique : les pathologies vocales chez l’enfant, 194. Paris: Fédération Nationale des Orthophonistes (FNO); 1998. p. 9–17. [14] Kilic¸ MA, O˘güt F, Dursun G, et al. The effects of vowels on voice perturbation measures. J Voice 2004;18(3):318–24. [15] Merati AL, Keppel K, Braun NM, et al. Pediatric voice-related quality of life: findings in healthy children and in common laryngeal disorders. Ann Otol Rhinol Laryngol 2008;117(4):259–62. [16] Bonet M, Casan P. Evaluation of dysphonia in a children’s choir. Folia Phoniatr Logop 1994;46(1):27–34. [17] Roland NJ, Bhalla RK, Earis J. The local side effects of inhaled corticosteroids: current understanding and review of the literature. Chest 2004;126(1): 213–9. [18] Cornut G. Genèse de la voix de l’enfant. JFORL J Fr Otorhinolaryngol Audiophonol Chir Maxillofac 1971;2:411–6. [19] Giovanni A. Les dysphonies de l’enfant. In: Garabedian EN, Bobin S, Monteil JP, et al., editors. ORL de l’enfant. 2e ed. Paris: Éditions Flammarion MédecineSciences; 2006. p. 266–77. [20] Dejonckere PH, Bradley P, Clemente P, et al. A basic protocol for functional assessment of voice pathology, especially for investigating the efficacy of (phonosurgical) treatments and evaluating new assessment techniques. Guideline elaborated by the Committee on Phoniatrics of the European Laryngological Society (ELS). Eur Arch Otorhinolaryngol 2001;258(2):77–82. [21] Heuillet-Martin G, Garson-Bavard H, Legre A. Les enfants dysphoniques. Une voix pour tous, Tome 2, 2e ed. Paris: Éditions Solal; 1997. p. 54–76. [22] Coulombeau B. Étude acoustique de la voix. In: Cornut G, et al., editors. Moyens d’investigation et pédagogie de la voix chantée Lyon: Éditions Symétrie; 2002. p. 57–66. [23] Brenot C, Kowalik-Cucciniello L. Étude du fondamental laryngé dans la voix parlée chez l’enfant de sept à dix ans. Lyon: Mémoire d’orthophonie; 1989. [24] Magne S, Oudot S, Voisin C. Étude du fondamental laryngé dans la voix parlée entre 10 et 20 ans. Lyon: Mémoire d’orthophonie; 1991. [25] Woisard V, Percodani J, Serrano E, et al. La voix de l’enfant : évolution morphologique du larynx et ses conséquences acoustiques. Rev Laryngol Otol Rhinol (Bord) 1996;117(4):313–7. [26] Boersma P, Weenink D. Praat: doing phonetics by computer. Praat – http://www.fon.hum.uva.nl/praat/. [27] Campisi P, Tewfik TL, Manoukian JJ, et al. Computer-assisted voice analysis: establishing a pediatric database. Arch Otolaryngol Head Neck Surg 2002;128(2):156–60. [28] Van Lieshout P. Praat short tutorial – http://oraldynamicslab.ca/. [29] Wuyts FL, De Bodt MS, Molenberghs G, et al. The dysphonia severity index: an objective measure of vocal quality based on a multiparameter approach. J Speech Lang Hear Res 2000;43(3):796–809. [30] Dejonckere PH. Techniques de base d’évaluation de la voix. Cabay: Louvain-laNeuve; 1985. p. 1–296. [31] Wuyts FL, Heylen L, Mertens F, et al. Effects of age, sex, and disorder on voice range profile characteristics of 230 children. Ann Otol Rhinol Laryngol 2003;112(6):540–8. [32] Jacobson BH, Johnson A, Grywalski C, et al. The voice handicap index (VHI): development and validation. Am J Speech Lang Pathol 1997;6(3):66–70. [33] Verdonck-de Leeuw IM, Kuik DJ, De Bodt M, et al. Validation of the voice handicap index by assessing equivalence of European translations. Folia Phoniatr Logop 2008;60(4):173–8. [34] Schindler A, Capaccio P, Maruzzi P, et al. Preliminary considerations on the application of the voice handicap index to paediatric dysphonia. Acta Otorhinolaryngol Ital 2007;27(1):22–6. [35] Schindler A, Tiddia C, Ghidelli C, et al. Adaptation and validation of the Italian pediatric voice handicap index. Folia Phoniatr Logop 2011;63(1):9–14. [36] Wolf M, Primov-Fever A, Amir O, et al. The feasibility of rigid stroboscopy in children. Int J Pediatr Otorhinolaryngol 2005;69(8):1077–9. [37] Cornut G, Bouchayer M. Assessing dysphonia: the role of videostroboscopy. An interactive videotextbook. Gibraltar: The 3 Ears Company Ltd; 1998. [38] Hartnick CJ, Zeitels SM. Pediatric video laryngostroboscopy. Int J Pediatr Otorhinolaryngol 2005;69(2):215–9. [39] Hernández Sandemetrio R, Nieto Curiel P, Dalmau Galofre J, et al. What is the contribution of stroboscopy in the diagnosis of voice disorders? Acta Otorrinolaringol Esp 2010;61(2):145–8. [40] Mortensen M, Schaberg M, Woo P. Diagnostic contributions of videolaryngostroboscopy in the pediatric population. Arch Otolaryngol Head Neck Surg 2010;136(1):75–9.