International Journal of Pediatric Otorhinolaryngology 75 (2011) 425–429
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Outcomes of surgery for laryngotracheal stenosis—The parents perspective S. Polubothu, K.J. Blackmore *, H. Kubba Department of ENT, Royal Hospital for Sick Children, Dalnair Street, Glasgow G3 8SJ, United Kingdom
A R T I C L E I N F O
A B S T R A C T
Article history: Received 23 November 2010 Accepted 22 December 2010 Available online 26 January 2011
Reconstructive laryngeal surgery has been very successful in the treatment of laryngotracheal stenosis. Traditionally authors have used survival and decannulation rates as markers of success. Objective: To evaluate the outcome of paediatric laryngotracheal surgery from the parents perspective. Method: Retrospective audit of all children undergoing laryngotracheal reconstruction or cricotracheal resection for laryngotracheal stenosis in a three year period at the Royal Hospital for Sick Children, Glasgow. An open-ended questionnaire was used to assess parental concerns regarding their child’s airway, post-operative results and the current status of their voice, breathing and swallowing. Results: 30 patients were eligible for the inclusion of which there were 17 responses (56%). The most common concerns pre-operatively were difficulty breathing, tracheostomy and survival. Postoperatively parents felt that breathing, voice and a reduction in the number of lower respiratory tract infections were the main improvements. We also identified the high level of importance that the parents assigned to their child’s independence and ability to lead a normal life. Conclusion: This study has given us a unique insight into the concerns of parents who have children with laryngotracheal stenosis. It has also allowed us to evaluate our post-operative results from a qualitative perspective through the eyes of the parents. ß 2010 Elsevier Ireland Ltd. All rights reserved.
Keywords: Laryngotracheal stenosis Reconstructive laryngotracheal surgery Quality of life Voice Swallowing Respiration
1. Introduction The management of laryngotracheal stenosis is one of the most challenging problems for the paediatric otolaryngologist. Surgery in the form of laryngotracheal reconstruction (LTR) or cricotracheal resection (CTR) has been very successful in relieving the stenosis and surgeons often quote their success rates in their published case series. Traditionally ‘‘success’’ has been measured by endpoints such as survival and decannulation [1,2] but with increasing surgeon experience and raised expectations of the patients/parents it seems appropriate that these end points are revised. Quality of life is becoming increasingly recognised as an important outcome tool in clinical medicine and it reflects the World Health Organisation’s definition of health as ‘‘the state of complete mental and social wellbeing not merely the absence of disease or infirmity’’ [3]. To our knowledge there have not been any previous reports that have evaluated the parents perspective of their child’s airway surgery. The aim of this study was to gain an understanding of the parents view of their child’s airway pre and post operatively and gain an insight into the areas of most importance to the parent. We also wished to evaluate the current status of the child’s voice, breathing and swallowing from the parents viewpoints—3 areas
* Corresponding author. Tel.: +44 7590312257. E-mail address:
[email protected] (K.J. Blackmore). 0165-5876/$ – see front matter ß 2010 Elsevier Ireland Ltd. All rights reserved. doi:10.1016/j.ijporl.2010.12.021
which we feel are fundamental to a child’s development and quality of life. 2. Methods A retrospective audit was undertaken. Children that had undergone an LTR or CTR for laryngotracheal stenosis between January 2003 and December 2007 at the Royal Hospital for Sick Children, Glasgow, UK were included. Children who had since deceased were excluded. The patients were identified using the departmental airway database. An 11 point semi-structured questionnaire was sent to the parents with a repost to non-responders. The questionnaire consisted of open questions with respect to the parental pre-operative concerns and views of the postoperative results (both positive and negative). We also asked the parents to give us a subjective assessment of the current status of their child’s voice, breathing and swallowing. 3. Results 32 children underwent a LTR or CTR in the time period evaluated of which 30 children were suitable for inclusion. There were 17 respondents (56%) of which there were 9 female and 8 male patients. The cause of the stenosis is shown in Fig. 1.
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expected the open ended approach also revealed some concerns that we had not anticipated: ‘‘What did I do wrong during pregnancy?’’ ‘‘How is this going to affect my relationship with my son?’’
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Fig. 1. Aetiology of subglottic stenosis.
The second part of the questionnaire explored the parents views of their child’s health since the surgery. We asked them what aspects of their child’s health had improved and worsened since their surgery and encouraged them to list as many aspects as they felt appropriate. An improvement in their child’s breathing and speech were the two most frequently quoted benefits from the parents perspective. Also noted were a decrease in the number of infections, decannulation, improved general health and development, increased confidence and independence and the ability to lead a normal life. ‘‘He no longer needs constant supervision’’ ‘‘The end result gave James some freedom and a taste of life with his friends’’.
Fig. 2. Parents most worrying concern pre-operatively.
10 patients had a tracheostomy pre-operatively of which 3 remained tracheostomised post-operatively. The age at surgery ranged from 4.7 to 223.6 months (median 34.7 months) and the time from surgery to the questionnaire being received was 7.1–53.7 months (median 27.9 months). The first question focussed on the parents main concerns about their child’s airway before the surgery. This was structured as an open-ended question and asked the parents to list their concerns in ranking order. Ranked as the main number one concerns pre-operatively were difficulty breathing, survival, the need for a tracheostomy/long term tracheostomy (see Fig. 2). Concerns that also ranked highly were fears for the future, recurrent infections, poor voice and blockage of the tracheostomy. Whilst these concerns were all to be
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Areas of their child’s health that parent’s felt had worsened since surgery were few and most parents felt that there were only improvements. Two parents felt their child’s voice had declined-in one case it had become more hoarse and in the second case the child was unable to project as well post-operatively. Following surgery and decannulation one child had become more breathless on exertion but the parents felt one of the main benefits of the surgery had been the removal of the tracheostomy as this had been their greatest pre-operative concern. One child’s parents were disappointed the tracheostomy had to be reinserted and another about their prominent scar although they quoted: ‘‘Although Sarah’s scar is quite thick but she is not ashamed or tries to hide it in any way. We are proud of what’s been done and that scar is just a reminder’’. We asked the parents to list any aspects of the end result that they were disappointed with or they felt could have turned out better. 12 of the 17 respondents said there were no disappointments or areas that could have been better. Other parents quoted disappointment in the post-operative voice, need for a tracheostomy, shortness of breath, scar and an administrative error (see Fig. 3). We followed with a question asking the parents to list any aspect of the end result that they were particularly pleased with. Most frequently quoted were speech, lack of shortness of breath
Fig. 3. Aspects of surgery that parents were disappointed with or felt could have turned out better.
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Fig. 4. Aspects of surgery that parents were particularly pleased with.
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Fig. 5. Post-operative voice outcome.
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Fig. 6. Parents views of their child’s breathing post-operatively.
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and a good cosmetic result. Also frequently quoted were lack of tracheostomy, increased freedom and independence and the communication between parents and medical staff. See Fig. 4. The questionnaire then focussed on the child’s current status with respect to their voice, breathing and swallowing. We asked the parents to describe their child’s voice giving as much detail as they could. In order to help them we listed examples, e.g. quality of their cry, easily understood by strangers, use of sign/gesturing to be understood. 10 parents felt their child’s voice was normal. 3 parents reported their child’s voice was weak and had difficulty shouting. One child had a hoarse voice but had no problems at school and was able to make themselves understood. 2 children also had hearing loss and the parents felt it difficult to ascertain whether their speech delay was secondary to their hearing or due to their airway surgery. 2 children were under the care of the speech therapists. See Fig. 5. With regard to the current status of the child’s breathing the parents reported their child’s breathing was normal in 8 cases. 4 children had noisy breathing in certain situations, i.e. sleeping, eating, upper respiratory tract infections and 2 children were short of breath on exertion. 3 children had a tracheostomy and one child required CPAP at night. See Fig. 6. We asked the parents to describe the current status of their child’s swallowing. We listed several questions with regard to swallowing to help guide their answers, e.g. Do they eat and drink normally? Do they have trouble swallowing solid food? The parents reported that their child ate and drank without difficulty in 11 cases. 2 children were tube fed and in 3 cases the child would occasionally cough or splutter on eating or drinking. One child was slow at feeding when compared with her twin. See Fig. 7. As a general guide to the child’s airway and respiratory system we asked the parents to document the number of significant respiratory tract infections that their child had suffered from in the previous year. 14 children had no infections and 3 had developed between 1 and several infections. Finally we asked the parents if they had any additional comments they wished to make. Whilst many expressed gratitude some interesting comments were:
‘‘Johnathan states he felt worried and panicky. He was scared he wouldn’t cope without a trachy. . .it took him several months to become confident with no trachy and he has just blossomed’’.
‘‘after surgery at 7 months I became aware that I had been holding David at arm’s length due to fears of him not surviving the anaesthetic’’ ‘‘She is able to breathe significantly better enabling her to be like any active ‘‘normal’’ child of six years of age. She loves swimming which obviously a tracheostomy restricts’’.
Whilst this study has given us an insight into the parents perspective of their child’s airway surgery it does have several limitations. Firstly the small study population means it is difficult to draw conclusions from the results which could be generalised to a wider population. The low response rate to the questionnaire is disappointing and will affect the results as generally those most
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4. Discussion The aim of this study was to evaluate the parents views of their child’s compromised airway and subsequent surgery to gain their perspective of the condition and surgical intervention undertaken. Published case series typically quote ‘‘success rates’’ ranging from decannulation to objective assessments of voice quality in their reports but we wanted to get a better understanding of what the parents felt was important in order to improve the care we gave them. Our study revealed that the parents pre-operative concerns were largely as we had expected with fears of breathing difficulty, survival and tracheostomy being the commonest. We had not anticipated parental feelings of guilt and concerns over the parent– child bond to be raised. Being aware of these concerns will aid us in improving our pre-operative counselling and help alleviate parental anxiety. Following surgery the main areas of improvement documented were related to breathing, speech and decannulation but it was interesting to recognise the level of importance that the parents assigned to the improvement in their child independence and ability to lead a normal life-areas which again should be discussed in the surgical work-up. There are several published studies which have evaluated the post-operative outcomes of laryngotracheal surgery with respect to the child’s voice [4,5] and swallowing [6] but these have used objective quantitative measures. Our aim, by using open questions, was to gain insight into what the parents viewed as important within each of the domains of breathing, swallowing and voice rather than drawing conclusions from quantitative data. Naturally this led to comparisons with siblings, being able to keep up with peers and difficulty within the school environment. For example: ‘‘She attends main stream primary school and has not problems with communicating her teacher says’’.
Fig. 7. Parental views of post-operative feeding.
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likely to respond are in cases with an extreme view, i.e. very happy or unhappy with the outcome thus resulting in bias. Additionally, it is possible that the parents felt unable to speak freely about their views of their child’s surgery for fear of causing offense and affecting future consultations with their ENT surgeon. Whilst the covering letter clearly stated that their responses would not influence future care of their child this may not have alleviated their concerns enough to be honest in their responses. 5. Conclusion This study has given us a unique insight into the concerns of parents who have children with laryngotracheal stenosis. It has also allowed us to evaluate our post-operative results from a qualitative perspective through the eyes of the parents. We plan to use the results as a basis for constructing a validated structured quality of life assessment tool to be used after airway surgery.
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6. Confidentiality All patient names have been changed to protect their identity. References [1] S.S. Choi, G.H. Zalzal, Pitfalls in laryngotracheal reconstruction, Arch. Otolaryngol. Head Neck Surg. 125 (1999) 650–653. [2] H. Alvarez-Neri, J. Penchyna-Grub, J.D. Porras-Hernandez, R. Blanco-Rodriguez, M. Rutter, Primary cricotracheal resection with thyrotracheal anastomosis for the treatment of severe subglottic stenosis in children and adolescents, Arch. OtolRhinolLaryngol. 114 (2005) 2–6. [3] World Health Organisation, Constitution of the World Health Organisation, WHO, Geneva, 1947. [4] C.J. MacArthur, G.H. Kearns, G.B. Healy, Voice quality after laryngotracheal reconstruction, Arch. Otolaryngol. Head Neck Surg. 120 (6) (1994) 617–641. [5] C.M. Bailey, R.A. Clary, A. Pengilly, D.M. Albert, Voice quality following laryngotracheal reconstruction, Int. J. Pediatr. Otorhinolaryngol. 32 (Suppl) (1995) S93– S95. [6] S.M. Andreoli, S.A. Nguyen, D. White, Feeding status after pediatric laryngotracheal reconstruction, Otolayngol. Head Neck Surg. 143 (2) (2010) 210–213.