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Outcomes of early infancy laryngeal reconstruction on health- and voice-related quality of life
International Journal of Pediatric Otorhinolaryngology 75 (2011) 351–355
Contents lists available at ScienceDirect
International Journal of Pediatric Otorhinolaryngology journal homepage: www.elsevier.com/locate/ijporl
Outcomes of early infancy laryngeal reconstruction on health- and voice-related quality of life Ahmed Geneid a,b,c,*, Niklas Pakkasja¨rvi d, Assi Aherto a, Risto Roine e, Harri Sintonen f, Harry Lindahl d, Anne Pitka¨ranta a,c a
Department of Otolaryngology and Phoniatrics, Helsinki University Central Hospital, Helsinki, Finland Department of Otolaryngology, Suez Canal University Hospital, Ismailia, Egypt Faculty of Medicine, University of Helsinki, Finland d Helsinki University Children’s Hospital, Helsinki, Finland e Helsinki and Uusimaa Hospital District, Research and Development, Helsinki, Finland f Department of Public Health, University of Helsinki, Helsinki, Finland b c
A R T I C L E I N F O
A B S T R A C T
Article history: Received 30 August 2010 Received in revised form 1 December 2010 Accepted 6 December 2010 Available online 7 January 2011
Objective: We aimed to study the voice, voice-related quality of life (VRQoL) and health-related quality of life (HRQoL) of children who in early infancy underwent a laryngeal split (cotton plasty operation) at the Helsinki University Central Hospital between January 1990 and December 2005. Methods: A retrospective review identified 17 children, of whom 10 fulfilled the inclusion criteria and participated in the study. Age- and gender-matched volunteers formed a control group. We used generic 16D and 17D questionnaires to assess HRQoL; the Pediatric Voice Outcomes Survey (PVOS) and the pediatric voice-related quality of life (PVRQoL) instrument served to assess VRQoL. The children underwent indirect laryngoscopy and perceptual voice assessment. Results: HRQoL and PVRQoL scores did not differ between subjects and controls. The subjects had lower PVOS scores than did the controls (P < 0.01). Perceptual assessment revealed that the subjects’ voices were worse off according to the following dimensions: voice is strained, voice is hoarse or husky, and voice is weak/does not resonate (P < 0.05). Conclusions: This study adds to the knowledge on the long-term effects of laryngeal split on children through assessment of their HRQoL and PVRQoL. The study subjects’ lower PVOS scores and findings of perceptual assessment point to the effect of cotton plasty on VRQoL and voice. ß 2010 Elsevier Ireland Ltd. All rights reserved.
Keywords: Cotton plasty Children Voice HRQoL VRQoL Perceptual assessment
1. Introduction Subglottic stenosis (SGS) is an important laryngeal abnormality that may be congenital or iatrogenic due to prolonged intubation. The incidence of SGS in infancy varies according to the definition of SGS and the criteria of the condition [1]. Because advances in neonatal intensive care have decreased mortality, the number of patients in need of treatment of the sequelae of prolonged intubation seems to be rising [2]. The Anterior Cricoid Split operation (ACS) was introduced in 1980 as an alternative to the tracheostomy to facilitate extubation in children with subglottic stenosis [3]. Layngotracheal reconstruction (LTR) in the form of ACS with or without grafts has revolutionized the management of
subglottic stenosis failing extubation. However, the long term effects of the ACS in relation to growth and function of the larynx remain poorly understood. A number of studies have already examined the outcome of LTR in terms of avoidance of tracheotomy or successful decanulation or both [4–7]. However, follow-up studies on the effect of early laryngeal surgery on the voice as well as on voice-related quality of life (VRQoL) and health-related quality of life (HRQoL) are scarce in the medical literature. To investigate the effects of laryngeal surgery on the function of the larynx, we analyzed the quality of life and voice quality in a group of children who underwent anterior cricoid split at an early postnatal age. 1.1. Aim of the study
* Corresponding author at: Phoniatrics Department, Ear, Nose and Throat Hospital, Helsinki University Central Hospital, P.O. Box 220, FI-00290 HUS, Helsinki, Finland. Tel.: +358 443304949; fax: +358 947175922. E-mail address: ahmed.geneid@hus.fi (A. Geneid). 0165-5876/$ – see front matter ß 2010 Elsevier Ireland Ltd. All rights reserved. doi:10.1016/j.ijporl.2010.12.001
To study the voice, HRQoL and VRQoL of children who underwent laryngeal reconstruction in early infancy at the Helsinki University Hospital between January 1990 and December 2005.
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A. Geneid et al. / International Journal of Pediatric Otorhinolaryngology 75 (2011) 351–355
2. Subjects and methods 2.1. Subjects We reviewed the children’s hospital records of the hospital district of Helsinki and Uusimaa retrospectively from 1 January 1990 through 31 December 2005 to verify the number of children who underwent LTR in addition to the indications and possible complications of their procedures. The review of the hospital charts revealed that 17 children underwent LTR during the abovementioned period. Children with permanent tracheostomies or acute upper respiratory tract infections at the time of the study were excluded from the study. Of the 17 children, 1 died and 3 children with a permanent tracheotomy were excluded. Consequently, the total number of study children was 13. The subjects participating in this research ranged in age from 2.7 to 15.9 years. They were divided into two age groups: one ranged from 2 to 11, and the other from 12 to 15, based on the agreement between the subjects’ age and the HRQoL questionnaires used in the study. In addition to the population controls, an age- and gender-matched control group was included in the study. The control group was selected from the children of the staff of the otorhinolaryngology hospital and other hospitals of Helsinki and Uusimaa hospital district. 2.2. Health-related quality of life The children’s HRQoL was measured with 16D [8] and 17D [9] questionnaires. These questionnaires were chosen because they are generic (i.e., not disease-specific); we aimed to assess how children with a history of LTR compare to the general population in addition to an age- and gender-matched control group in terms of QoL. Both questionnaires subscribe to the World Health Organisation’s definition of health as comprising physical, mental and social well-being. Both questionnaires are generic, multidimensional, standardized, sensitive, self-administered measures of HRQoL. The 16D has been developed for children aged 12–15 years and includes 16 dimensions (mobility, vision, hearing, breathing, sleeping, eating, speech, elimination, school and hobbies, mental function, discomfort and symptoms, depression, distress, vitality, appearance, friends). Each dimension is divided into five levels where the best level = 1 and the worst level = 5. The 16D is easy to use and can be completed within 5–10 min. Proxy administration is also possible. The 17D has essentially been developed for children aged 8–11 years, but with proxy administration, it can also be used for younger children. The questionnaire includes 17 dimensions (mobility, vision, hearing, breathing, sleeping, eating, speech, elimination, school and hobbies, learning and memory, discomfort and symptoms, depression, distress, vitality, appearance, friends, concentration). Each dimension has five ordinal levels. The questionnaire takes about 20–30 min to complete. Both instruments include a valuation algorithm by which the responses on the questionnaires are converted to an overall HRQoL score ranging from 1 (full health) to 0 (death).
(0.96; P < 0.001) and test–retest reliability (weighted K value, 0.8) [11]. Both the PVOS and PVRQoL were translated into Finnish by two accredited translators, and then back translated into English by two different accredited translators. Appropriate changes, mainly in the wording, were introduced to develop a questionnaire that is semantically and conceptually equivalent to the original English versions. 2.4. Voice samples Voice recordings were carried out for each child from both the control and study groups, with the exception of one child who had Down’s syndrome and had nearly no vocabulary at the time of data collection. The voice samples taken from each child consisted of either a reading sample (short children’s story) of 41 words or a spontaneous speech recorded while the child was playing with his mother. Spontaneous speech always contained at least 41 words. Recording was carried out using a portable hard-disk recorder (iRiver 140) with a sampling rate of 44.1 kHz. A head-mounted condenser microphone (AKG C444) was (per instructions) located on one side of the mouth at a distance of three centimeters from the lips. This distance was continuously monitored by one of the authors (A.A. or A.G.). The voice samples were later transferred to a personal computer where they were edited using an audio editor (Adobe Audition, Version 2.0 by Adobe Systems Incorporated, San Jose, CA, USA). 2.5. Perceptual assessment of voice Two voice clinicians assessed the recorded voice samples in a blinded manner. The voice clinicians used a visual analogue scale (VAS) that included the following six parameters: 1. Voice is overstrained, 2. Voice is hoarse or husky, 3. Voice is weak/does not resonate, 4. Voice is tense or feeling the need to make effort when speaking, 5. Voice is creaky, 6. Voice breaks during speaking. 2.6. Population data The 16D scores were compared to the scores of the population controls (n = 235) from a survey performed in different schools in the greater Helsinki area. 2.7. Statistical analysis The statistical analysis of the data was carried out using SPSS for Windows, Version 17.0.0 Statistical Software (SPSS inc., Chicago, IL, USA). Descriptive statistics served to describe the demographic characteristics of the study and control groups. Differences in HRQoL as well as in PVOS and PVRQoL results between the study and control groups were compared using the independent samples t-test. The perceptual assessment of voice samples was studied using the Wilcoxon signed-rank test. 2.8. Ethical considerations
2.3. Voice-related quality of life Voice related quality of life (VRQOL) was measured using the Pediatric Voice Outcomes Survey (PVOS) [10] and the pediatric voice-related quality of life (PVRQOL) instrument [11]. The PVOS is a four-item instrument designed to measure VRQOL. A previous study found this instrument to be valid (P = 0.004) and to have a Cronbach a value of 0.86 [10]. Low scores describe a relatively poor VRQOL, whereas high scores describe a better VRQOL. On the other hand, the PVRQOL is a ten-item instrument designed to measure VRQOL and was adapted from the adult VRQOL instrument. The reliability of the PVRQOL was established by its Cronbach a value
The study design was approved by the Ethics Committee of Opthalmology, Otorhinolaryngology, Neurology and Neurosurgery of the Helsinki and Uusimaa Hospital District (353/E9/07). The participants and their parents received information concerning the study, and the parents gave their written consent on participation of their children in the study. 3. Results Of the 13 children and adolescents eligible for the study, only ten returned the questionnaires, yielding a response rate of 77%. Those
A. Geneid et al. / International Journal of Pediatric Otorhinolaryngology 75 (2011) 351–355 Table 1 Descriptive characteristics of the study group. Characteristics Gender Male Female Age at surgerya Mean SD Range Weight at birthb Mean SD Range Weight at surgeryb Mean SD Range a b
Respondents (n = 10)
Non-respondents (n = 3)
10 (100%) 0 (0%)
1 (33.3%) 2 (66.6%)
51 38 8–148
150 225 9–409
1834 1313 630–3850
952 352 550–1205
2398 1816 825–6100
975 326 680–1325
Age in days. Weight in grams.
who failed to return the questionnaire were later reminded by telephone; they either explained their unwillingness to participate or simply chose not to return the questionnaires sent. Of the ten who participated in the study, five (who received the 17D questionnaires) ranged in age from 2.7 to 10.5 years. The other five (who received the 16D questionnaires) ranged in age from 12.3 to 15.9. The gender distribution, age at surgery, weight at birth, and laryngeal reconstruction of the respondents and non-respondents
[()TD$FIG]
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appear in Table 1. All of the children and adolescents who participated in the study were boys. Five of the respondents underwent laryngeal reconstruction in the form of a cricoid split with grafting, whereas all three of the nonrespondents underwent only a cricoid split. Three children suffered from asthma, one of whom was older than 11; the others were younger. Six children suffered language and phonological disorders, and one of them stuttered. Five children had concomitant congenital anomalies such as ventricular septal defect, undescended testicles, fallot tetralogy, and VATER association. In addition, ear, nose and throat surgeries were rather common. The difference in the total 16D score of the study group (0.936) and the control group (0.989) was 0.053, whereas the difference between the study group and the population controls (0.949) was 0.013. Two dimensions showed greater differences between the study group on the one hand, and the control group and population controls on the other. On both dimensions, the study group scored lower than the control group and the population controls. On the dimension of elimination, the study group scored lower than the population controls by 0.115, and the control group by 0.127. On the dimension of speech, the study group scored lower than the population controls by 0.092, and the control group by 0.136. The differences on both dimensions, however, were not statistically significant. One of the five children eligible for the 17D had Down’s syndrome and nearly no vocabulary at the time of data collection.
Fig. 1. The HRQoL of the study and control groups, in addition to the population controls, as measured with the 16D questionnaire. The independent samples t-test revealed no significant differences.
[()TD$FIG]
Fig. 2. The HRQoL of the study and control groups as measured with the 17D questionnaire. The independent samples t-test revealed no significant differences.
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The child’s scores were quite low and accordingly lowered the overall score to 0.869 in comparison to 0.972 for the control group. However, when the child’s scores were excluded along with the age-matched control, the scores of both groups changed. The total score of the study group (excluding the child previously referred to) became 0.902, and that of the control group became 0.969, thus reducing the difference to 0.067. Such a difference was not statistically significant (Figs. 1 and 2). 3.1. PVOS and PVRQOL Statistical analysis of the results of PVOS revealed a mean measurement and standard deviation of 96.9 3.3 for the control group, and 85.6 9.8 for the study group. The difference between both groups was statistically significant (P < 0.01). Statistical analysis of the PVRQOL results revealed no significant differences. 3.2. Perceptual voice assessment of voice samples Statistical analysis of the perceptual assessment of the voice samples showed a significant difference between the study and control groups (n = 18, P < 0.05). The study group have worse medians of scores on the following parameters: 1. Voice is overstrained is 2.6 for the study group in comparison to the control one that scored 1.35, 2. Voice is hoarse or husky is 3.7 in comparison to 1.6 for the control one, 3. Voice is weak/does not resonate is 2.7 in comparison to 1.05 for the control one. 4. Discussion In this study, we explored how laryngeal reconstruction performed in early infancy affects voice, HRQoL and VRQoL. To achieve this, we used generic questionnaires to assess HRQoL and VRQoL. We also carried out a perceptual assessment of voice recordings of the children who participated in the study. A genderand age-matched control group was included, and its scores and assessments were compared to those of both the study group and the population controls, when applicable. The results of this study are directed mainly towards the absence of previous studies on the long-term effects of laryngeal reconstruction on HRQoL and VRQoL. This study aims to provide insight into its long-terms effects. The response rate to both mailed questionnaires and the participation in the study in Helsinki was 77%. We consider this acceptable, taking into consideration possible constrains faced by the children’s parents, who wanted to participate in the study. This study offered neither financial benefit nor financial compensation to the parents of children participating in the study or who travelled from different parts of Finland to Helsinki, where the study took place. Overall, the male children comprised 85% of the non-respondents and respondents together and 100% of the latter group. The non-significant differences in the 17D scores may stem from the small number of participating children, especially after excluding the child noted previously (n = 4). However, such results may also indicate that the HRQoL of those children did not differ from that of their age- and gender-matched controls. Similar non-significant results for children under 16 and over 11 may also be due to the small number of children who underwent surgery or may refer to a normal HRQoL among them. Interestingly, the greater differences, although not significant, between the study group on the one hand and the control and population groups on the other in both dimensions of elimination and speech are noteworthy. This result draws attention to the rule of subglottal pressure in speech and elimination, with the latter being one of the primary functions of the larynx. The subglottal
pressure may be lower among the children who underwent surgery due to cricotracheal resection with or without grafting, resulting in increased subglottal volume. We did not measure the subglottal pressure in this study, but future studies can explore such hypotheses. Comparing our results with those of previous studies is almost impossible, especially since previous studies on the long-term effects of laryngotracheal construction focused mainly on its complications, failures and mortality [12]. When the authors assessed the VRQoL of the study group through the PVOS and the PVRQoL, only the former showed significant differences between the control and study groups. The PVOS generally correlates highly with the PVRQoL, especially in the physical-functional scores, and has a lower, but still significant, correlation with the social-emotional portion of it [11]. The fact that only the PVOS showed significant differences may be due to its robustness in comparison to the PVRQoL. The PVRQoL contains certain items that may be difficult or confusing to answer, especially for younger children. Items such as ‘‘My child has trouble doing his or her job or schoolwork (because of his or her voice)’’ or ‘‘My child has trouble using the telephone or speaking with friends in person’’ will confuse the parents of children under eight years of age. In addition, their answers will disrupt the results and may have rendered insignificant the differences between the study and control groups. On the other hand, the PVOS contains only four robust, easy-to-answer questions that are easily detectable parameters. The findings point to a lower VRQoL among the study children than in the controls. This result is in line with those of a previous study that validated and tested the Pediatric Voice Handicap Index (pVHI) [13], which is a modification of the adult VHI [14]. The study examined a group of 33 guardians of children undergoing a voice evaluation pre- or postlaryngotracheal reconstruction, in addition to a control group. The scores of the dysphonic group were higher (worse) than those of the control group. However, the dysphonic group included children both pre- and post-laryngeal reconstruction, and the main aim of the study was to adapt the VHI to the pediatric population and to validate it. That the voices of the study group scored lower perceptually than did those of the control group in three voice parameters draws attention to the long-term effects of laryngeal reconstruction on voice. Such long-term effects have seen little previous study. During the 1990s, when research interests began to shift from the effects of laryngeal reconstruction on respiratory functions to laryngeal functions, attention focused mainly on the short-term effects on vocal quality [15,16]. Other studies have found results similar to ours. Zalzal et al. [15] found a similar net result to what we found in this study and concluded that vocal quality is generally disturbed for those who have undergone laryngeal reconstruction. Their psychoacoustic evaluation revealed that five of the six children included in their study showed excessive pharyngeal muscular tension and aberrant respiratory patterns during phonation. The previously mentioned study was limited, however, by the absence of a control group and blinded psychoacoustic evaluation. Another study [16] of 16 children ranging in age from 2 to 12 years showed disturbed vocal quality post-operatively. Among the children studied, 2 had a breathy voice, 12 were hoarse, and 2 were mixed. In addition, the children in the study exhibited a lower than optimal pitch and a restricted pitch range was predominant. The results of both of the above-mentioned studies are in line with ours. The fact that our study included a control group and blinded perceptual assessment confirms and elaborates upon the results of previous studies. In general, the findings of our study suggest that laryngeal reconstruction affects vocal quality and VRQoL in the long term.
A. Geneid et al. / International Journal of Pediatric Otorhinolaryngology 75 (2011) 351–355
At the same time, the results suggest that these children have a normal overall HRQoL compared to that of their age- and gender-matched controls and population controls. The findings of greater, but not significant, differences on the dimensions of elimination and speech point to possibly low subglottal pressures, which lowered the scores for these two domains. 5. Conclusion This study provided a new dimension in studying the long-term effects of laryngeal reconstruction surgery on children through the assessment of their HRQoL and PVRQoL in addition to vocal quality through perceptual assessment. The limitations of the study are mainly due to the small number of study children. The same limitation faced previously published studies attempting to examine the short- and long-term effects of laryngeal reconstruction. Accordingly, the need for a multi-center study is evident and should be strived for. Declaration of interest The authors declare no conflicts of interest. The authors alone are responsible for the content and writing of the paper. Acknowledgements The authors would like to thank Dr. Pa¨ivi Salminen and Professor Erkki Vilkman for their helpful thoughts and input during the planning of this study. The authors are deeply grateful to Anne Rissanen, MNSc for her valuable work in the analysis of the results. Thanks also go to SLP Kaarina Ruusuvirta, and SLP Laura Lehto for their help in the perceptual assessment of the voice samples. This study was supported by a grant from the Ear, Nose and Throat Research Foundation in Finland, in addition to a special state subsidy (EVO) through the Helsinki and Uusimaa Hospital District.
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References [1] R. Leung, Incidence of severe acquired subglottic stenosis in newborns, Int. J. Pediatr. Otorhinolaryngol. 71 (5) (2007) 763. [2] J.A. Matute, M.A. Villafruela, M.D. Delgado, F.J. Berchi, J. Vazquez, Surgery of subglottic stenosis in neonates and children, Eur. J. Pediatr. Surg. 10 (5) (2000) 286–290 (accessed 08.03.10). [3] R.T. Cotton, A.B. Seid, Management of the extubation problem in the premature child. Anterior cricoid split as an alternative to tracheotomy, Ann. Otol. Rhinol. Laryngol. 89 (6 Pt 1) (1980) 508–511. [4] S. Baker, L. Kelchner, B. Weinrich, et al., Pediatric laryngotracheal stenosis and airway reconstruction: a review of voice outcomes, assessment, and treatment issues, J. Voice 20 (4) (2006) 631–641. [5] M.E. Wyatt, Laryngotracheal reconstruction in congenital laryngeal webs and atresias, Otolaryngol. Head Neck Surg. 132 (2) (2005) 232. [6] H. Alvarez-Neri, G. Blanco-Rodriguez, J. Penchyna-Grub, R. Gonzalez, J.D. PorrasHernandez, M.J. Rutter, Primary cricotracheal resection with thyrotracheal anastomosis for the treatment of severe subglottic stenosis in children and adolescents, Ann. Otol. Rhinol. Laryngol. 114 (1 I) (2005) 2–6 (accessed 12.03.10). [7] B.B. Cable, Pediatric airway reconstruction: principles, decision-making, and outcomes at the university of Iowa hospitals and clinics, Ann. Otol. Rhinol. Laryngol. 113 (4) (2004) 289. [8] M. Apajasalo, H. Sintonen, C. Holmberg, J. Sinkkonen, V. Aalberg, H. Pihko, et al., Quality of life in early adolescence: a sixteen-dimensional health-related measure (16D), Qual. Life Res. 5 (2) (1996) 205–211. [9] M. Apajasalo, J. Rautonen, C. Holmberg, J. Sinkkonen, V. Aalberg, H. Pihko, et al., Quality of life in pre-adolescence: a 17-dimensional health-related measure (17D), Qual. Life Res. 5 (6) (1996) 532–538. [10] C.J. Hartnick, Validation of a pediatric voice quality-of-life instrument: the pediatric voice outcome survey, Arch. Otolaryngol. Head Neck Surg. 128 (8) (2002) 919–922. [11] M.E. Boseley, M.J. Cunningham, M.S. Volk, C.J. Hartnick, Validation of the pediatric voice-related quality-of-life survey, Arch. Otolaryngol. Head Neck Surg. 132 (7) (2006) 717–720. [12] R.T. Cotton, J.N. Evans, Laryngotracheal reconstruction in children. Five-year follow-up, Ann. Otol. Rhinol. Laryngol. 90 (5 Pt 1) (1981) 516–520. [13] K.B. Zur, Pediatric voice handicap index (pVHI): a new tool for evaluating pediatric dysphonia, Int. J. Pediatr. Otorhinolaryngol. 71 (1) (2007) 77. [14] B.H. Jacobson, The voice handicap index (VHI): development and validation, Am. J. Speech Lang. Pathol. 6 (3) (1997) 66. [15] G.H. Zalzal, S.R. Loomis, M. Fischer, Laryngeal reconstruction in children. Assessment of vocal quality, Arch. Otolaryngol. Head Neck Surg. 119 (5) (1993) 504–507. [16] G.H. Zalzal, S.R. Loomis, C.S. Derkay, S.L. Murray, J. Thomsen, Vocal quality of decannulated children following laryngeal reconstruction, Laryngoscope 101 (4 Pt 1) (1991) 425–429.
Contents lists available at ScienceDirect
International Journal of Pediatric Otorhinolaryngology journal homepage: www.elsevier.com/locate/ijporl
Outcomes of early infancy laryngeal reconstruction on health- and voice-related quality of life Ahmed Geneid a,b,c,*, Niklas Pakkasja¨rvi d, Assi Aherto a, Risto Roine e, Harri Sintonen f, Harry Lindahl d, Anne Pitka¨ranta a,c a
Department of Otolaryngology and Phoniatrics, Helsinki University Central Hospital, Helsinki, Finland Department of Otolaryngology, Suez Canal University Hospital, Ismailia, Egypt Faculty of Medicine, University of Helsinki, Finland d Helsinki University Children’s Hospital, Helsinki, Finland e Helsinki and Uusimaa Hospital District, Research and Development, Helsinki, Finland f Department of Public Health, University of Helsinki, Helsinki, Finland b c
A R T I C L E I N F O
A B S T R A C T
Article history: Received 30 August 2010 Received in revised form 1 December 2010 Accepted 6 December 2010 Available online 7 January 2011
Objective: We aimed to study the voice, voice-related quality of life (VRQoL) and health-related quality of life (HRQoL) of children who in early infancy underwent a laryngeal split (cotton plasty operation) at the Helsinki University Central Hospital between January 1990 and December 2005. Methods: A retrospective review identified 17 children, of whom 10 fulfilled the inclusion criteria and participated in the study. Age- and gender-matched volunteers formed a control group. We used generic 16D and 17D questionnaires to assess HRQoL; the Pediatric Voice Outcomes Survey (PVOS) and the pediatric voice-related quality of life (PVRQoL) instrument served to assess VRQoL. The children underwent indirect laryngoscopy and perceptual voice assessment. Results: HRQoL and PVRQoL scores did not differ between subjects and controls. The subjects had lower PVOS scores than did the controls (P < 0.01). Perceptual assessment revealed that the subjects’ voices were worse off according to the following dimensions: voice is strained, voice is hoarse or husky, and voice is weak/does not resonate (P < 0.05). Conclusions: This study adds to the knowledge on the long-term effects of laryngeal split on children through assessment of their HRQoL and PVRQoL. The study subjects’ lower PVOS scores and findings of perceptual assessment point to the effect of cotton plasty on VRQoL and voice. ß 2010 Elsevier Ireland Ltd. All rights reserved.
Keywords: Cotton plasty Children Voice HRQoL VRQoL Perceptual assessment
1. Introduction Subglottic stenosis (SGS) is an important laryngeal abnormality that may be congenital or iatrogenic due to prolonged intubation. The incidence of SGS in infancy varies according to the definition of SGS and the criteria of the condition [1]. Because advances in neonatal intensive care have decreased mortality, the number of patients in need of treatment of the sequelae of prolonged intubation seems to be rising [2]. The Anterior Cricoid Split operation (ACS) was introduced in 1980 as an alternative to the tracheostomy to facilitate extubation in children with subglottic stenosis [3]. Layngotracheal reconstruction (LTR) in the form of ACS with or without grafts has revolutionized the management of
subglottic stenosis failing extubation. However, the long term effects of the ACS in relation to growth and function of the larynx remain poorly understood. A number of studies have already examined the outcome of LTR in terms of avoidance of tracheotomy or successful decanulation or both [4–7]. However, follow-up studies on the effect of early laryngeal surgery on the voice as well as on voice-related quality of life (VRQoL) and health-related quality of life (HRQoL) are scarce in the medical literature. To investigate the effects of laryngeal surgery on the function of the larynx, we analyzed the quality of life and voice quality in a group of children who underwent anterior cricoid split at an early postnatal age. 1.1. Aim of the study
* Corresponding author at: Phoniatrics Department, Ear, Nose and Throat Hospital, Helsinki University Central Hospital, P.O. Box 220, FI-00290 HUS, Helsinki, Finland. Tel.: +358 443304949; fax: +358 947175922. E-mail address: ahmed.geneid@hus.fi (A. Geneid). 0165-5876/$ – see front matter ß 2010 Elsevier Ireland Ltd. All rights reserved. doi:10.1016/j.ijporl.2010.12.001
To study the voice, HRQoL and VRQoL of children who underwent laryngeal reconstruction in early infancy at the Helsinki University Hospital between January 1990 and December 2005.
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2. Subjects and methods 2.1. Subjects We reviewed the children’s hospital records of the hospital district of Helsinki and Uusimaa retrospectively from 1 January 1990 through 31 December 2005 to verify the number of children who underwent LTR in addition to the indications and possible complications of their procedures. The review of the hospital charts revealed that 17 children underwent LTR during the abovementioned period. Children with permanent tracheostomies or acute upper respiratory tract infections at the time of the study were excluded from the study. Of the 17 children, 1 died and 3 children with a permanent tracheotomy were excluded. Consequently, the total number of study children was 13. The subjects participating in this research ranged in age from 2.7 to 15.9 years. They were divided into two age groups: one ranged from 2 to 11, and the other from 12 to 15, based on the agreement between the subjects’ age and the HRQoL questionnaires used in the study. In addition to the population controls, an age- and gender-matched control group was included in the study. The control group was selected from the children of the staff of the otorhinolaryngology hospital and other hospitals of Helsinki and Uusimaa hospital district. 2.2. Health-related quality of life The children’s HRQoL was measured with 16D [8] and 17D [9] questionnaires. These questionnaires were chosen because they are generic (i.e., not disease-specific); we aimed to assess how children with a history of LTR compare to the general population in addition to an age- and gender-matched control group in terms of QoL. Both questionnaires subscribe to the World Health Organisation’s definition of health as comprising physical, mental and social well-being. Both questionnaires are generic, multidimensional, standardized, sensitive, self-administered measures of HRQoL. The 16D has been developed for children aged 12–15 years and includes 16 dimensions (mobility, vision, hearing, breathing, sleeping, eating, speech, elimination, school and hobbies, mental function, discomfort and symptoms, depression, distress, vitality, appearance, friends). Each dimension is divided into five levels where the best level = 1 and the worst level = 5. The 16D is easy to use and can be completed within 5–10 min. Proxy administration is also possible. The 17D has essentially been developed for children aged 8–11 years, but with proxy administration, it can also be used for younger children. The questionnaire includes 17 dimensions (mobility, vision, hearing, breathing, sleeping, eating, speech, elimination, school and hobbies, learning and memory, discomfort and symptoms, depression, distress, vitality, appearance, friends, concentration). Each dimension has five ordinal levels. The questionnaire takes about 20–30 min to complete. Both instruments include a valuation algorithm by which the responses on the questionnaires are converted to an overall HRQoL score ranging from 1 (full health) to 0 (death).
(0.96; P < 0.001) and test–retest reliability (weighted K value, 0.8) [11]. Both the PVOS and PVRQoL were translated into Finnish by two accredited translators, and then back translated into English by two different accredited translators. Appropriate changes, mainly in the wording, were introduced to develop a questionnaire that is semantically and conceptually equivalent to the original English versions. 2.4. Voice samples Voice recordings were carried out for each child from both the control and study groups, with the exception of one child who had Down’s syndrome and had nearly no vocabulary at the time of data collection. The voice samples taken from each child consisted of either a reading sample (short children’s story) of 41 words or a spontaneous speech recorded while the child was playing with his mother. Spontaneous speech always contained at least 41 words. Recording was carried out using a portable hard-disk recorder (iRiver 140) with a sampling rate of 44.1 kHz. A head-mounted condenser microphone (AKG C444) was (per instructions) located on one side of the mouth at a distance of three centimeters from the lips. This distance was continuously monitored by one of the authors (A.A. or A.G.). The voice samples were later transferred to a personal computer where they were edited using an audio editor (Adobe Audition, Version 2.0 by Adobe Systems Incorporated, San Jose, CA, USA). 2.5. Perceptual assessment of voice Two voice clinicians assessed the recorded voice samples in a blinded manner. The voice clinicians used a visual analogue scale (VAS) that included the following six parameters: 1. Voice is overstrained, 2. Voice is hoarse or husky, 3. Voice is weak/does not resonate, 4. Voice is tense or feeling the need to make effort when speaking, 5. Voice is creaky, 6. Voice breaks during speaking. 2.6. Population data The 16D scores were compared to the scores of the population controls (n = 235) from a survey performed in different schools in the greater Helsinki area. 2.7. Statistical analysis The statistical analysis of the data was carried out using SPSS for Windows, Version 17.0.0 Statistical Software (SPSS inc., Chicago, IL, USA). Descriptive statistics served to describe the demographic characteristics of the study and control groups. Differences in HRQoL as well as in PVOS and PVRQoL results between the study and control groups were compared using the independent samples t-test. The perceptual assessment of voice samples was studied using the Wilcoxon signed-rank test. 2.8. Ethical considerations
2.3. Voice-related quality of life Voice related quality of life (VRQOL) was measured using the Pediatric Voice Outcomes Survey (PVOS) [10] and the pediatric voice-related quality of life (PVRQOL) instrument [11]. The PVOS is a four-item instrument designed to measure VRQOL. A previous study found this instrument to be valid (P = 0.004) and to have a Cronbach a value of 0.86 [10]. Low scores describe a relatively poor VRQOL, whereas high scores describe a better VRQOL. On the other hand, the PVRQOL is a ten-item instrument designed to measure VRQOL and was adapted from the adult VRQOL instrument. The reliability of the PVRQOL was established by its Cronbach a value
The study design was approved by the Ethics Committee of Opthalmology, Otorhinolaryngology, Neurology and Neurosurgery of the Helsinki and Uusimaa Hospital District (353/E9/07). The participants and their parents received information concerning the study, and the parents gave their written consent on participation of their children in the study. 3. Results Of the 13 children and adolescents eligible for the study, only ten returned the questionnaires, yielding a response rate of 77%. Those
A. Geneid et al. / International Journal of Pediatric Otorhinolaryngology 75 (2011) 351–355 Table 1 Descriptive characteristics of the study group. Characteristics Gender Male Female Age at surgerya Mean SD Range Weight at birthb Mean SD Range Weight at surgeryb Mean SD Range a b
Respondents (n = 10)
Non-respondents (n = 3)
10 (100%) 0 (0%)
1 (33.3%) 2 (66.6%)
51 38 8–148
150 225 9–409
1834 1313 630–3850
952 352 550–1205
2398 1816 825–6100
975 326 680–1325
Age in days. Weight in grams.
who failed to return the questionnaire were later reminded by telephone; they either explained their unwillingness to participate or simply chose not to return the questionnaires sent. Of the ten who participated in the study, five (who received the 17D questionnaires) ranged in age from 2.7 to 10.5 years. The other five (who received the 16D questionnaires) ranged in age from 12.3 to 15.9. The gender distribution, age at surgery, weight at birth, and laryngeal reconstruction of the respondents and non-respondents
[()TD$FIG]
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appear in Table 1. All of the children and adolescents who participated in the study were boys. Five of the respondents underwent laryngeal reconstruction in the form of a cricoid split with grafting, whereas all three of the nonrespondents underwent only a cricoid split. Three children suffered from asthma, one of whom was older than 11; the others were younger. Six children suffered language and phonological disorders, and one of them stuttered. Five children had concomitant congenital anomalies such as ventricular septal defect, undescended testicles, fallot tetralogy, and VATER association. In addition, ear, nose and throat surgeries were rather common. The difference in the total 16D score of the study group (0.936) and the control group (0.989) was 0.053, whereas the difference between the study group and the population controls (0.949) was 0.013. Two dimensions showed greater differences between the study group on the one hand, and the control group and population controls on the other. On both dimensions, the study group scored lower than the control group and the population controls. On the dimension of elimination, the study group scored lower than the population controls by 0.115, and the control group by 0.127. On the dimension of speech, the study group scored lower than the population controls by 0.092, and the control group by 0.136. The differences on both dimensions, however, were not statistically significant. One of the five children eligible for the 17D had Down’s syndrome and nearly no vocabulary at the time of data collection.
Fig. 1. The HRQoL of the study and control groups, in addition to the population controls, as measured with the 16D questionnaire. The independent samples t-test revealed no significant differences.
[()TD$FIG]
Fig. 2. The HRQoL of the study and control groups as measured with the 17D questionnaire. The independent samples t-test revealed no significant differences.
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The child’s scores were quite low and accordingly lowered the overall score to 0.869 in comparison to 0.972 for the control group. However, when the child’s scores were excluded along with the age-matched control, the scores of both groups changed. The total score of the study group (excluding the child previously referred to) became 0.902, and that of the control group became 0.969, thus reducing the difference to 0.067. Such a difference was not statistically significant (Figs. 1 and 2). 3.1. PVOS and PVRQOL Statistical analysis of the results of PVOS revealed a mean measurement and standard deviation of 96.9 3.3 for the control group, and 85.6 9.8 for the study group. The difference between both groups was statistically significant (P < 0.01). Statistical analysis of the PVRQOL results revealed no significant differences. 3.2. Perceptual voice assessment of voice samples Statistical analysis of the perceptual assessment of the voice samples showed a significant difference between the study and control groups (n = 18, P < 0.05). The study group have worse medians of scores on the following parameters: 1. Voice is overstrained is 2.6 for the study group in comparison to the control one that scored 1.35, 2. Voice is hoarse or husky is 3.7 in comparison to 1.6 for the control one, 3. Voice is weak/does not resonate is 2.7 in comparison to 1.05 for the control one. 4. Discussion In this study, we explored how laryngeal reconstruction performed in early infancy affects voice, HRQoL and VRQoL. To achieve this, we used generic questionnaires to assess HRQoL and VRQoL. We also carried out a perceptual assessment of voice recordings of the children who participated in the study. A genderand age-matched control group was included, and its scores and assessments were compared to those of both the study group and the population controls, when applicable. The results of this study are directed mainly towards the absence of previous studies on the long-term effects of laryngeal reconstruction on HRQoL and VRQoL. This study aims to provide insight into its long-terms effects. The response rate to both mailed questionnaires and the participation in the study in Helsinki was 77%. We consider this acceptable, taking into consideration possible constrains faced by the children’s parents, who wanted to participate in the study. This study offered neither financial benefit nor financial compensation to the parents of children participating in the study or who travelled from different parts of Finland to Helsinki, where the study took place. Overall, the male children comprised 85% of the non-respondents and respondents together and 100% of the latter group. The non-significant differences in the 17D scores may stem from the small number of participating children, especially after excluding the child noted previously (n = 4). However, such results may also indicate that the HRQoL of those children did not differ from that of their age- and gender-matched controls. Similar non-significant results for children under 16 and over 11 may also be due to the small number of children who underwent surgery or may refer to a normal HRQoL among them. Interestingly, the greater differences, although not significant, between the study group on the one hand and the control and population groups on the other in both dimensions of elimination and speech are noteworthy. This result draws attention to the rule of subglottal pressure in speech and elimination, with the latter being one of the primary functions of the larynx. The subglottal
pressure may be lower among the children who underwent surgery due to cricotracheal resection with or without grafting, resulting in increased subglottal volume. We did not measure the subglottal pressure in this study, but future studies can explore such hypotheses. Comparing our results with those of previous studies is almost impossible, especially since previous studies on the long-term effects of laryngotracheal construction focused mainly on its complications, failures and mortality [12]. When the authors assessed the VRQoL of the study group through the PVOS and the PVRQoL, only the former showed significant differences between the control and study groups. The PVOS generally correlates highly with the PVRQoL, especially in the physical-functional scores, and has a lower, but still significant, correlation with the social-emotional portion of it [11]. The fact that only the PVOS showed significant differences may be due to its robustness in comparison to the PVRQoL. The PVRQoL contains certain items that may be difficult or confusing to answer, especially for younger children. Items such as ‘‘My child has trouble doing his or her job or schoolwork (because of his or her voice)’’ or ‘‘My child has trouble using the telephone or speaking with friends in person’’ will confuse the parents of children under eight years of age. In addition, their answers will disrupt the results and may have rendered insignificant the differences between the study and control groups. On the other hand, the PVOS contains only four robust, easy-to-answer questions that are easily detectable parameters. The findings point to a lower VRQoL among the study children than in the controls. This result is in line with those of a previous study that validated and tested the Pediatric Voice Handicap Index (pVHI) [13], which is a modification of the adult VHI [14]. The study examined a group of 33 guardians of children undergoing a voice evaluation pre- or postlaryngotracheal reconstruction, in addition to a control group. The scores of the dysphonic group were higher (worse) than those of the control group. However, the dysphonic group included children both pre- and post-laryngeal reconstruction, and the main aim of the study was to adapt the VHI to the pediatric population and to validate it. That the voices of the study group scored lower perceptually than did those of the control group in three voice parameters draws attention to the long-term effects of laryngeal reconstruction on voice. Such long-term effects have seen little previous study. During the 1990s, when research interests began to shift from the effects of laryngeal reconstruction on respiratory functions to laryngeal functions, attention focused mainly on the short-term effects on vocal quality [15,16]. Other studies have found results similar to ours. Zalzal et al. [15] found a similar net result to what we found in this study and concluded that vocal quality is generally disturbed for those who have undergone laryngeal reconstruction. Their psychoacoustic evaluation revealed that five of the six children included in their study showed excessive pharyngeal muscular tension and aberrant respiratory patterns during phonation. The previously mentioned study was limited, however, by the absence of a control group and blinded psychoacoustic evaluation. Another study [16] of 16 children ranging in age from 2 to 12 years showed disturbed vocal quality post-operatively. Among the children studied, 2 had a breathy voice, 12 were hoarse, and 2 were mixed. In addition, the children in the study exhibited a lower than optimal pitch and a restricted pitch range was predominant. The results of both of the above-mentioned studies are in line with ours. The fact that our study included a control group and blinded perceptual assessment confirms and elaborates upon the results of previous studies. In general, the findings of our study suggest that laryngeal reconstruction affects vocal quality and VRQoL in the long term.
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At the same time, the results suggest that these children have a normal overall HRQoL compared to that of their age- and gender-matched controls and population controls. The findings of greater, but not significant, differences on the dimensions of elimination and speech point to possibly low subglottal pressures, which lowered the scores for these two domains. 5. Conclusion This study provided a new dimension in studying the long-term effects of laryngeal reconstruction surgery on children through the assessment of their HRQoL and PVRQoL in addition to vocal quality through perceptual assessment. The limitations of the study are mainly due to the small number of study children. The same limitation faced previously published studies attempting to examine the short- and long-term effects of laryngeal reconstruction. Accordingly, the need for a multi-center study is evident and should be strived for. Declaration of interest The authors declare no conflicts of interest. The authors alone are responsible for the content and writing of the paper. Acknowledgements The authors would like to thank Dr. Pa¨ivi Salminen and Professor Erkki Vilkman for their helpful thoughts and input during the planning of this study. The authors are deeply grateful to Anne Rissanen, MNSc for her valuable work in the analysis of the results. Thanks also go to SLP Kaarina Ruusuvirta, and SLP Laura Lehto for their help in the perceptual assessment of the voice samples. This study was supported by a grant from the Ear, Nose and Throat Research Foundation in Finland, in addition to a special state subsidy (EVO) through the Helsinki and Uusimaa Hospital District.
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