Shared Decision Making and Rehabilitation IMPROVING PERSONALIZED MEDICINE IN HEAD AND NECK ONCOLOGY A.N. Heirman
IMPROVING PERSONALIZED MEDICINE IN HEAD AND NECK ONCOLOGY: ‘Shared Decision Making and Rehabilitation’ Anne Nadine Heirman
Author: Anne Nadine Heirman Cover design and layout: © evelienjagtman.com Printing: Ridderprint Funding for this research The research described in this thesis was performed at the Netherlands Cancer Institute -Antoni van Leeuwenhoek, Amsterdam, The Netherlands. The Netherlands Cancer Institute receives a research grant from ATOS Medical Sweden which contributes to the existing intrastructure for health-related quality of life research of the department of Head and Neck Oncology and Surgery. De uitgave van dit proefschrift werd mede mogelijk gemaakt door financiële steun van: Atos Medical B.V. I ChipSoft I ANW Nederland I CityClinics I prof. dr. Eelco Huizinga Stichting I Dos Medical I ACTA I Onderzoeksschool Oncologie Amsterdam I Michel Keyzerfonds I Aegis Advocaten I ALK I Straumann I Reconcept I MDS ISBN: 978-94-6506-439-0 Copyright 2024 Anne Heirman. All rights reserved
IMPROVING PERSONALIZED MEDICINE IN HEAD AND NECK ONCOLOGY: ‘Shared decision making and rehabilitation’ ACADEMISCH PROEFSCHRIFT ter verkrijging van de graad van doctor aan de Universiteit van Amsterdam op gezag van de Rector Magnificus prof. dr. ir. P.P.C.C. Verbeek ten overstaan van een door het College voor Promoties ingestelde commissie, in het openbaar te verdedigen in de Agnietenkapel op dinsdag 10 december 2024, te 13.00 uur door Anne Nadine Heirman geboren te GRONINGEN
Promotiecommissie Promotores: prof. dr. M.W.M. van den Brekel Universiteit van Amsterdam dr. G.B. Halmos Rijksuniversiteit Groningen Copromotores: dr. R. Dirven NKI-AvL dr. L. van der Molen NKI-AvL Overige leden: prof. dr. D.T. Ubbink Universiteit van Amsterdam prof. dr. B.L. van Leeuwen Rijksuniversiteit Groningen prof. dr. A.J.M. Balm Universiteit van Amsterdam prof. dr. B.F.A.M. van der Laan Rijksuniversiteit Groningen prof. dr. L.E. Smeele Universiteit van Amsterdam prof. dr. G. Elwyn Dartmouth College Faculteit der Tandheelkunde
Table of Contents Prologue 7 Chapter 1. General introduction 13 Part 1. Shared Decision Making in Head and Neck Oncology Chapter 2. Decisional Conflict and Decision Regret in Head and Neck Oncology – A Systematic Review and Meta-Analysis 39 Chapter 3. Exploring the State of Shared Decision-Making in Head and Neck Oncology: Assessing Treatment Communication 73 Chapter 4. Decisional Conflict in Patients with Advanced Laryngeal Carcinoma – A Multicenter Study 103 Chapter 5. The Impact of a Patient Decision Aid for Patients with Advanced Laryngeal Carcinoma – A Multicenter Study 123 Chapter 6. The Development of a Decision Aid for Patients with Operable Oropharyngeal Carcinoma in the Netherlands – A Mixed Methods Study 147 Part 2. Rehabilitation after Total Laryngectomy Chapter 7. Does Prophylactic Replacement of Voice Prosthesis Make Sense? – A Study to Predict Prosthesis Lifetime 173 Chapter 8. The Acceptance and Voice Quality of a New Voice Prosthesis “Vega High Performance” – A Feasibility Study 191 Chapter 9. Maximal Cardiopulmonary Exercise Testing in Laryngectomized Patients using Different HMEs – Feasibility and Exercise Responses 213 Chapter 10. Singing after Total Laryngectomy 235 Chapter 11. General Discussion and Future Perspectives 241 Chapter 12 12.1 English summary 261 12.2 Nederlandse samenvatting 265 Appendices Authors and affiliations 273 Author contributions 277 PhD portfolio 280 Dankwoord 282 About the author 290
Prologue
Prologue | 9 P Prologue This is the story of mr. T., a 66-year-old man. He is healthy, although he has a known high blood pressure for which he takes medication. He works as a janitor at a high school, but he is nearing retirement. In his free time, he enjoys being at home with his wife and dog, and playing cards at the community center. They have two daughters, and one grandson. However, recent months have brought unwelcome health concerns. Persistent hoarseness and worsening coughing have disrupted his routine, making communication increasingly difficult as his voice weakens, impacting both his personal and professional life. Concurrently, he experiences discomfort in his throat, occasionally extending to his left ear. Initially, his general practitioner prescribed antibiotics, suspecting a throat or airway infection, but unfortunately, these provided no relief. Consequently, Mr. T. was referred to the ENT department, where a thorough examination uncovered concerning abnormalities in his vocal cords during an endoscopy procedure. Recognizing the urgency of the situation, the ENT specialist promptly referred him to the Head and Neck Oncology department, where he was scheduled for evaluation the following day. During this evaluation, it was revealed that Mr. T. had been a heavy smoker since the age of 16, with unsuccessful attempts at quitting. Additionally, he regularly indulged in alcohol, consuming five cans of beer post-work and whiskey during Friday card games. Following extensive evaluation, Mr. T. and his wife received devastating news from the doctor: laryngeal carcinoma, which had metastasized to his lymph nodes. After consultations with medical experts, it was determined that a total laryngectomy offered the best chance of survival. Radiation treatment was considered as an alternative, but with a significant risk of rendering his larynx nonfunctional, ultimately leading to the need for a total laryngectomy. Back home, they feel overwhelmed and full of questions, after consultations with the head and neck surgeon, radiation oncologist, and oncologist. “What is metastasis anyway?” asks his wife. “And will you never be able to talk again?” “I think I can talk again, the doctor mentioned something about a voice prosthesis and a humidifier or something,” says Mr. T. “But what should I do? Everything sounds terrible. Am I going to die?”
10 | Prologue Mr. T. and his wife are lost. Ultimately, they agree with the surgery since the doctor said it offers the best chance of survival for him. The surgery is performed, and Mr. T. is hospitalized for 19 days. He hadn’t expected it to be so hard. Pain, coupled with difficulty communicating through the voice prosthesis, fuels anxiety. Upon returning home, familiar surroundings offer little comfort as he navigates a changed existence. Months later, tentative adjustment begins. Managing his stoma and communicating with the voice prosthesis gradually become routine. He has opted for early retirement. Initially, he mentioned dropping by occasionally, yet he does not dare to. Card games, once a source of enjoyment, are now abandoned. Participating in group conversations is hard, and how is he supposed to speak with a voice prosthesis while holding all his cards? His life is far from what it once was. Every day, he doubts his decision. What if he had chosen radiation? Perhaps his larynx would have remained functional, sparing him the life-changing side effects of a laryngectomy. But then again, could life have taken a turn for the worse? Or would it have been better to have done nothing at all and faced the inevitable outcome of cancer? After all, he had enjoyed a great life. Well, either way.. He needs to go to the hospital; his voice prosthesis is leaking. Again..
Chapter 1 General introduction
Introduction | 15 1 General introduction The experience of patients with head and neck cancer (HNC), like Mr. T.’s story, begins a challenging journey. Initial encounters often involve consultations with a head-and-neck surgeon, a radiation oncologist, and occasionally, a medical oncologist and dentist. Additionally, patients will interface with various allied health professionals, including speech-language therapists (SLPs), dieticians, and head-and-neck oncology nurses. Given the aggressive nature of HNC, there exists a pressing sense of urgency. In the midst of numerous consultations with various specialists, patients face the heavy burden of their diagnosis and its emotional impact, while considering their treatment options, which could even include choosing not to undergo treatment at all. In this thesis, we delve into the multifaceted landscape of HNC care, exploring the challenges faced by patients and healthcare professionals alike. Through an exploration of shared decision-making dynamics and rehabilitation strategies, we aim to contribute to a deeper understanding of HNC management. With this introduction, I will guide you through some key aspects crucial for comprehending the contents of this thesis. Epidemiology of head and neck cancer HNC is a rare type of cancer with around 3000 diagnoses per year in the Netherlands1. It constitutes a diverse group of cancers affecting the oral cavity, pharynx, larynx, paranasal sinuses, nasal cavity, salivary glands and the unknown primary tumor. The epidemiology of head and neck tumors is intricate and influenced by various factors, including geographical location, lifestyle choices, and exposure to risk factors. The most common risk factors associated with HNC include tobacco use, alcohol consumption, human papillomavirus (HPV) infection, and Epstein-Barr virus (EBV) infection. Men are diagnosed at a significantly higher rate than women, with ratios typically falling between 2:1 and 4:12. HNC is more common in older individuals, with a mean age of 62 years. However, due the rising incidence of HPV-associated oropharyngeal tumors, the number of younger individuals is also rising3. In the Netherlands, more than 1000 patients decease each year due to HNC4. Survival is related to tumor stage, where the lower the stage at diagnosis, the higher the survival rates are. The TNM staging system, ranging from I to IV, plays a pivotal role in assessing the extent of disease and guiding treatment decisions in HNC5. For most
16 | Chapter 1 head and neck tumors, the 5-year relative survival rate has been increasing over the years, although survival remains low for hypopharyngeal cancer. The survival rate for laryngeal cancer remains approximately constant over time6. We will delve into two specific HNC types that we studied in this thesis. Oropharyngeal carcinoma The oropharyngeal carcinoma is a relatively rare form of cancer that originates in the mucous membrane of the middle throat cavity. The oropharynx is a critical anatomical area within the throat, encompassing structures such as the base of the tongue, the soft palate, the tonsillar regions, and the pharyngeal walls. Given the complexity of this anatomical area, treatment approaches must be precise and conservative to preserve critical functions such as speech and swallowing while minimizing morbidity. Maintaining the patient’s quality of life is paramount, underscoring the importance of tailored treatment strategies that address both the cancer and its potential impact on essential physiological processes7–9. Symptoms of oropharyngeal carcinoma can vary but often include persistent sore throat, difficulty swallowing (dysphagia), ear pain, a lump in the neck (due to enlarged lymph nodes), and changes in voice or hoarseness. Diagnosis typically involves a comprehensive assessment, including physical examination, imaging studies such as CT scans or MRIs, and biopsy for pathological analysis. Risk factors for oropharyngeal cancer include smoking, alcohol consumption, and HPV infection. There are notable differences between HPV-positive and HPV-negative tumors. HPV-positive tumors, often found in younger individuals, has a relatively favorable prognosis compared to HPV-negative tumors, which are more commonly associated with traditional risk factors such as tobacco and alcohol use10. In the Netherlands, as elsewhere, the epidemiology oropharyngeal cancer has evolved, notably influenced by human papillomavirus (HPV) prevalence11, making HPV+ tumors more common. This shift has implications for treatment decisions and outcomes. Treatment for oropharyngeal tumors varies according to the stage of the cancer. For advanced stages (Stage III and IV), where the tumor has spread to nearby lymph nodes or tissues, treatment typically involves a combination of surgery, radiation, and chemotherapy. Chemoradiation is often the primary approach for locally advanced tumors to shrink the tumor and target any spread cancer cells. Surgery may still be considered in certain cases, either before or after chemoradiation, to address remaining tumor or affected lymph nodes12. For recurrent or metastatic disease, treatment options may include surgery, radiation therapy, chemotherapy, targeted therapy, or immunotherapy, with a focus on palliative care to alleviate symptoms and improve quality of life13.
Introduction | 17 1 In early-stage tumors (Stage I and II), localized and small tumors may be treated with either surgery or radiation therapy. Surgical options like transoral robotic surgery (TORS) or transoral laser microsurgery (TLM) aim to remove the tumor while preserving healthy surrounding tissue14. Alternatively, radiation therapy alone or combined with chemotherapy (chemoradiation) may be used, especially if surgery is not feasible or preferred by the patient. Surgery and radiation have shown to give similar outcomes, but different side-effects15. Early stage oropharyngeal carcinoma Early-stage oropharyngeal cancer, identified at stages I or II, signifies that the cancer is localized within the oropharynx without extensive spread to surrounding tissues or distant sites. Transoral minimally invasive surgery, may include different surgical techniques, most commonly transoral robotic surgery (TORS) or transoral laser surgery (TOLS). It presents a treatment option for early-stage oropharyngeal cancer that can precisely target and remove the tumor while minimizing damage to surrounding healthy tissues. This approach is particularly suited for patients whose tumors are located in areas accessible through the mouth, allowing for direct visualization and resection of the cancer16. Transoral surgery’s benefits include a potential reduction in the need for more invasive procedures, a lower risk of disfigurement, and, importantly, the preservation of organ function17,18. In the Netherlands it is most often used to avoid radiation if possible. However, when surgery results in too close or positive margins, or when more than very limited neck disease is present, adjuvant radiation therapy or even chemoradiation is necessary to eliminate any residual cancer cells and reduce recurrence risk. Radiation therapy remains a cornerstone in the management of early-stage oropharyngeal cancer, either as a primary treatment modality or adjuvant following surgery to address microscopic disease. Radiation therapy aims to eradicate cancer cells while preserving as much normal tissue as possible. However, it’s associated with toxicities such as dry mouth, dysphagia, and potential long-term changes in salivary function, underscoring the importance of careful treatment planning avoiding large fields and sparing critical structures19,20. Because of the side effects of radiotherapy and chemoradiation, de-intensification strategies for eligible patients are being studied widely21,22. Controversies persist in determining the optimal treatment approach, yet most researchers concur on one point: if a single modality proves effective, it should be favored over multimodality
18 | Chapter 1 treatment. In the case of early stage oropharyngeal tumors, this means that in case of a small, well accessible primary without or with minimal neck disease, surgery is a very good alternative for radiotherapy without risking the long term toxicity of radiotherapy23. The choice between radiation and surgery—or the combination thereof—is influenced by several factors, including tumor size, location, neck disease, HPV status, and patient preference. The impact of these treatments on patients varies. TORS, while less invasive than traditional surgery, may still lead to post-operative pain, temporary changes in swallowing, or speech difficulties, though these are generally less severe and resolve more quickly than with open surgical approaches15. Radiation therapy, while non-invasive, can cause acute side effects like mucositis, which typically resolve after treatment completion, but it may also lead to long-term changes in saliva production and taste sensation, swallowing problems and long tern fibrosis and atherosclerosis15. Choosing the appropriate treatment for early-stage oropharyngeal cancer involves careful consideration of the tumor’s size, location, N stage and the HPV status, as well as the patient’s overall health and personal preferences. Both TORS and radiation therapy offer the potential for cure with a focus on organ preservation and functional outcomes. Decisions regarding the optimal approach should be made in collaboration with a multidisciplinary team of specialists, ensuring that treatment plans are tailored to achieve the best possible balance between effective cancer control and preservation of quality of life. Laryngeal cancer Laryngeal cancer develops in the tissues of the larynx, which is commonly referred to as the voice box. The larynx plays a crucial role in breathing, swallowing, and speaking. Laryngeal cancer can manifest in various parts of the organ, including the vocal cords, supraglottis, glottis and subglottis. Risk factors for laryngeal cancer include tobacco use, particularly smoking, and excessive alcohol consumption. Chronic exposure to irritants such as industrial chemicals and asbestos, as well as gastroesophageal reflux disease (GERD), may also increase the risk of developing laryngeal cancer. Symptoms of laryngeal cancer can vary depending on the location and extent of the tumor but often include persistent hoarseness or changes in voice quality, difficulty swallowing (dysphagia), throat pain, ear pain, a lump or mass in the neck, and persistent coughing or coughing up blood24.
Introduction | 19 1 Figure 1. Overview of anatomy of the Head and Neck Region. This image has been used with permission from Atos Medical AB Treatment options for laryngeal cancer depend on factors such as the stage and location of the tumor, as well as the overall health and preferences of the patient. Treatment for early-stage laryngeal cancer typically involves a combination of surgical and nonsurgical interventions. Surgical options like transoral laser microsurgery (TLM) or partial laryngectomy are commonly employed to remove localized tumors while preserving laryngeal function25. Additionally, radiation therapy may be utilized either as a primary treatment modality or in combination with surgery to effectively target cancer cells.
20 | Chapter 1 However, in cases of advanced-stage laryngeal cancer, more aggressive approaches such as total laryngectomy or chemoradiation therapy may be necessary to achieve optimal outcomes26,27. Treatment decisions should be tailored to each patient’s specific circumstances, and require collaboration among a multidisciplinary team of specialists. Figure 2. Sagittal overview of the pharynx with a laryngeal tumor 1. tongue, 2. hyoid bone, 3. lingual tonsil, 4. epiglottis, 5. thyroid cartilage, 6. false vocal cord, 7. tumor, 8. true vocal cord, 9. cricoid cartilage, 10. trachea, 11. Esophagus Used with the permission of the author; Maartje Kunen, Medical Visuals, 2018 Advanced laryngeal cancer Advanced laryngeal carcinoma represents a severe stage of cancer affecting the larynx, categorized as stage III or IV28. Symptoms vary depending on the tumor’s location but can include persistent hoarseness, difficulty swallowing, unexplained weight loss, and
Introduction | 21 1 in more severe cases, breathing difficulties. The supraglottic area tend to show fewer early symptoms, leading to a higher likelihood of diagnosis at an advanced stage and is particularly prone to lymphatic spread due to the rich lymphatic network, contrasting with glottic cancers, which are often discovered earlier due to voice changes. Subglottic tumor are quite seldom and often precipitates symptoms of dyspnea and coughing, particularly in cases where the tumor attains considerable size, thereby impeding airflow within the respiratory tract. Treatment options are diverse, with larynx preserving strategies on the one hand, and total laryngectomy (TL) on the other hand. TL, the surgical removal of the entire larynx, is often recommended for extensive tumors, especially T4 or with impaired function at diagnosis or as salvage when other treatments have failed29,30. This procedure can effectively control the cancer but results in the loss of natural voice and necessitates a permanent stoma for breathing31–33. Respiration is then carried out through a tracheal stoma, created by bringing the trachea to the skin in the lower, front part of the neck. This procedure effectively separates the upper portion of the airway from the lower, leading to permanent loss of voice and smell. Organ-preserving strategies like chemoradiation (CRT, a combination of cisplatin based chemotherapy and radiation therapy) and radiation therapy alone are preferred when feasible, as they aim to maintain laryngeal function34. CRT is particularly used for tumors that are locally or regionally advanced, offering a potential for cure while preserving the larynx35. The impact of treatments on patients with advanced laryngeal carcinoma varies significantly. TL, while potentially curative, profoundly affects speech, swallowing, and breathing, necessitating significant lifestyle adjustments and rehabilitation31. In most cases, postoperative radiotherapy is still needed. In the Netherlands almost all patient receive a voice prosthesis (VP) during surgery, also called a primary puncture36. After rehabilitation most patients are able to use the VP and speak. Conversely, organ-preserving approaches may lessen these functional impacts but come with their own set of challenges, including acute side effects like mucositis, dysphagia, and altered taste, as well as long-term risks such as radiation-induced fibrosis, swallowing issues and poor laryngeal functioning as well as potentially decreased efficacy in controlling the cancer compared to surgical options26,37,38. So, how do you make the right treatment decision?
22 | Chapter 1 Figure 3. Overview of anatomy after a total laryngectomy. This image has been used with permission from Atos Medical AB Shared Decision Making Shared Decision Making (SDM) has evolved from a novel concept to a core component of patient-centered care, emphasizing the partnership between patients and healthcare providers in making health decisions. Initially emerging in the late 20th century in response to a growing recognition of patient autonomy and the ethical imperative for informed consent, SDM has grown in importance as healthcare has become more complex, with an increased focus on patient rights and the recognition that patients’
Introduction | 23 1 values and preferences should play a central role in healthcare decisions39–41. The benefits of SDM include enhanced patient satisfaction, improved treatment adherence, and a more personalized healthcare experience, particularly in conditions where multiple treatment options exist. However, challenges include time constraints in busy clinical settings, the need for healthcare providers to be skilled in communication and negotiation, and potential increased anxiety in some patients42,43. SDM in the medical field is most appropriately applied when patients are faced with decisions involving complex treatment options, multiple possible outcomes, and uncertainty regarding the best course of action. This approach empowers patients to actively participate in their healthcare by collaborating with healthcare professionals to weigh the risks and benefits of different options, leading to decisions that align with their individual circumstances and preferences. It is especially valuable in chronic conditions, end-of-life care, and situations where there are trade-offs between treatment benefits and potential harms. The success of SDM in improving patient outcomes underscores the importance of further research and implementation support41,44–47. An established model of SDM consists of three essential components: “team talk,” emphasizing support for patients when introducing options and eliciting their goals; “option talk,” involving the comparison of alternatives using risk communication principles; and “decision talk,” focusing on reaching decisions that reflect informed patient preferences, guided by healthcare professionals’ experience and expertise. This model provides a structured framework for collaboration and deliberation in the decision-making process48. Decisional conflict arises when individuals face uncertainty and struggle to make choices among various options, particularly when they feel uninformed or uncertain about which option aligns best with their values and preferences. This internal struggle can lead to stress, anxiety, and indecision, hindering the decision-making process and potentially resulting in suboptimal choices49,50. Decision regret, on the other hand, occurs when individuals experience negative emotions or dissatisfaction with a decision they have made, often in hindsight. This regret may stem from a perception that an alternative option would have yielded a better outcome or that the chosen course of action did not align with their expectations or values51,52. Both decisional conflict and decision regret underscore the importance of effective decision-making processes that prioritize informed choice, active participation, and consideration of individual values and preferences. Shared Decision Making (SDM) aims to address these challenges by fostering collaboration between patients and healthcare providers, providing patients with the information and support they need to make decisions that are aligned with their goals and values, thereby reducing decisional conflict and minimizing the likelihood of decision regret53.
24 | Chapter 1 Figure 4. Shared decision making model as developed by Elwyn et al. (2017)48 Patients facing HNC treatments should therefore make preference-sensitive decisions, where the trade-offs between treatment efficacy, side effects, and impacts on quality of life are complex and deeply personal. Despite the recognized value of SDM in these cases, research on its application in head and neck oncology is scarce, highlighting a gap in understanding how best to support these patients in their treatment decisions54. As mentioned earlier, early-stage oropharyngeal cancer poses preference-sensitive choices between radiation and transoral surgery, while advanced laryngeal cancer presents treatment options with notable side effects and survival trade-offs. We therefore have chosen to investigate these conditions in the context of Patient Decision Aids (PDAs). While our research has primarily focused on the development of a PDA for early-stage oropharyngeal cancer, we have also tested the impact of an earlier developed PDA for advanced laryngeal cancer55.
Introduction | 25 1 Patient decision aids Patient Decision Aids (PDAs) are instrumental in facilitating Shared Decision Making (SDM), offering a structured approach for patients and healthcare providers to engage in informed discussions about treatment options. PDAs are designed to present evidencebased information about the risks, benefits, and alternatives of treatments, helping patients to make decisions that align with their personal values and preferences56. The use of PDAs has been associated with improved knowledge, reduced decisional conflict, and greater satisfaction with the decision-making process. Moreover, PDAs support the implementation of clinical practice guidelines by ensuring that patient choices are informed by the best available evidence57,58. Despite benefits, challenges like time constraints and health literacy hinder widespread adoption. Discrepancies exist between positive outcomes and real-world implementation, highlighting a gap between evidence and practice59. Additionally, PDAs’ availability and efficacy vary, necessitating adherence to International Patient Decision Aid Standards (IPDAS) for evidence-based, patient-centered development60. Challenges include managing costs, achieving stakeholder consensus, involving a multidisciplinary team, clinical implementation, and keeping PDAs updated58,61. Addressing these demands careful planning, collaboration, and continuous evaluation. There’s a noted discrepancy between the positive outcomes associated with PDA use and the actual implementation of these tools in everyday clinical settings, underscoring a gap between evidence and practice58,61. The development of Patient Decision Aids (PDAs) adheres to the International Patient Decision Aid Standards (IPDAS) criteria60, ensuring evidence-based content, clarity, and patient-centeredness. However, challenges arise, including managing costs effectively, achieving consensus among stakeholders, involving a multidisciplinary team, implementing the PDA into clinical practice seamlessly, and keeping it up to date with evolving evidence and patient preferences. Overcoming these hurdles requires careful planning, collaboration, and continuous evaluation processes. In head and neck oncology, the lack of PDAs underscores a pressing necessity for tailored tools that address the distinctive decision-making challenges faced by patients. Bridging this gap is imperative for propelling patient-centered care forward in oncology, enabling patients to be fully informed and empowered in their treatment decisions.
26 | Chapter 1 Once a treatment plan, such as a total laryngectomy for advanced laryngeal cancer, has been chosen, patients will enter a rehabilitation phase. The following section of this introduction will explore particular aspects of rehabilitation tailored to laryngectomized patients. Rehabilitation after Total Laryngectomy Rehabilitation after TL presents a profound challenge for patients, affecting their ability to speak, breathe, and swallow. This multifaceted rehabilitation process aims to address these challenges, focusing on restoring respiratory function, facilitating communication, and ensuring nutritional support. In addition, physical rehabilitation is also an important aspect to take into account. Essential components of the first challenges involves the use of voice prostheses (VP) and Heat and Moisture Exchangers (HMEs)62 Voice prosthesis Vocal rehabilitation after TL is a crucial component of post-surgical care, aimed at restoring the patient’s ability to communicate verbally. In the Western world, tracheoesophageal puncture (TEP) with voice prosthesis (VP) insertion has emerged as the gold standard for voice restoration following TL. This method significantly surpasses other techniques in providing patients with more intelligible speech and better voice quality63. Indwelling voice prostheses, including the Provox, Fahl and Blom-Singer models, are the most common types used, categorized into regular VPs for standard cases and problem-solving VPs for those with specific issues such as short device lifetimes or periprosthetic leakage36,64. The device lifetime of VPs varies, typically ranging from 2 to 6 months, depending on several factors such as the type of VP, patient’s ability to maintain the device, biological factors like biofilm formation on the prosthesis as well as reimbursement and other barriers for the patient. The primary reason for VP replacement is transprosthetic leakage, occurring in 55% to 80% of cases, followed by periprosthetic leakage affecting 5% to 30% of patients64. These frequent replacements can significantly impact a patient’s quality of life, leading to feelings of insecurity and unplanned hospital visits65,66. Despite the challenges associated with VPs, the use of tracheoesophageal voice prosthesis remains a highly effective method for vocal rehabilitation post-TL. Research and development continue to aim for VPs with longer lifetimes and fewer complications, improving patient satisfaction and quality of life. The ongoing innovation in this field underscores the importance of tailored approaches to vocal rehabilitation, ensuring that each patient receives the most suitable device based on their individual needs and circumstances67.
Introduction | 27 1 Heat and Moisture Exchangers Respiratory rehabilitation after TL is essential due to the separation of the upper and lower respiratory tracts, fundamentally altering the airway dynamics. After the surgery, the natural functions of the upper airway—warming, humidifying, and filtering the inhaled air—are bypassed as breathing is redirected through a stoma directly into the lower airways. This loss leads to increased pulmonary symptoms such as involuntary coughing, mucus retention, and forced expectoration. Furthermore, patients are at a higher risk of airway infections and inflammations, affecting not only their physical health but also their sleep, social interactions, and overall quality of life68–72. To mitigate these challenges, two primary devices have been developed: stoma cloth covers (bib) and Heat and Moisture Exchangers (HMEs). While bibs are a cost-effective solution offering potentially good air conditioning, they pose challenges in stoma occlusion for speech and are generally less favored by patients73. Consequently, HMEs have become the preferred choice in many developed countries due to their efficiency in retaining heat and moisture from exhaled air and transferring it to the inhaled air, significantly improving the tracheal climate, reducing coughing and sputum production, and enhancing patients’ quality of life68,74–76.These devices, attached via an adhesive baseplate or cannula over the stoma, have shown efficacy in long-term use by preventing the deterioration of tracheal cells and even restoring them. Despite the benefits, the performance of HMEs is balanced against their resistance, which can vary based on the device’s size and internal specifications. Higher resistance HMEs, while more effective in air conditioning, may cause discomfort during physical activities, potentially discouraging physical exercise and impacting overall health and quality of life negatively77. The choice of HME, therefore, should consider individual patient needs, lifestyle, and the specific level of activity to ensure optimal compliance and enhance the quality of life. With a range of HMEs available, tailored to offer varying levels of resistance and humidification, it’s crucial for patients to find a balance that supports their respiratory health without compromising their activity levels and overall well-being78. Cardiopulmonary exercise testing Maximal Cardiopulmonary Exercise Testing (CPET) is a valuable tool in rehabilitation, offering insights into patients’ cardiovascular and pulmonary health, as well as overall physical fitness. It is typically conducted in a controlled clinical setting under the supervision of healthcare professionals. During the test, patients are instructed to perform incremental exercise on a stationary cycle or treadmill. The intensity of exercise gradually increases in predetermined stages or through continuous ramp protocols79.
28 | Chapter 1 Figure 5. Overview of a laryngectomized patient with a voiceprosthesis and HME. This image has been used with permission from Atos Medical AB. Throughout the test, various physiological parameters are continuously monitored. These include heart rate, oxygen consumption (VO2), carbon dioxide production (VCO2), minute ventilation (VE), respiratory exchange ratio (RER) and electrocardiography (ECG). A key aspect of maximal CPET is the utilization of a face mask or mouthpiece connected to a metabolic cart, which allows for the collection of expired gases. This equipment enables the accurate measurement of respiratory parameters, providing insights into the efficiency of gas exchange during exercise80.
Introduction | 29 1 Despite its proven benefits, maximal CPET has not found widespread use in laryngectomy rehabilitation. This may be attributed to the intensive focus on other aspects of rehabilitation, such as vocal rehabilitation, which often take precedence due to their immediate impact on quality of life. However, with advancements in medical care, laryngectomy patients are now surviving longer, leading to an increased need for comprehensive rehabilitation strategies. Incorporating maximal CPET into laryngectomy rehabilitation could provide crucial information about patients’ aerobic capacity and exercise tolerance, aiding in the optimization of their overall functional capacity and quality of life. Nevertheless, integrating this presents unique challenges. One significant obstacle is the use of a face mask during the test, which is essential for accurate measurement of respiratory parameters but is not useful for laryngectomy patients since their airway ends in their neck after surgery. This difficulty in using traditional CPET equipment highlights the need for innovative approaches and adaptations to make maximal CPET accessible and beneficial for laryngectomy patients. Overcoming these challenges could unlock the potential of maximal CPET as a valuable tool in optimizing the rehabilitation outcomes and long-term well-being of laryngectomy patients. Overview and aims of this thesis This thesis is divided into two parts, each dedicated to distinct yet interconnected aspects of head-and-neck cancer (HNC) care. The first part focuses on shared decisionmaking dynamics, specifically examining advanced laryngeal cancer and operable oropharyngeal carcinoma. It aims to provide insights into the prevalence of decisional conflict and decision regret among head-and-neck oncology patients, exploring whether enhanced counseling can reduce these issues while also augmenting knowledge. Additionally, this part investigates the development and impact of patient decision aids (PDAs) in facilitating informed decision-making processes. The second part centers on rehabilitation strategies tailored to patients undergoing Total Laryngectomy (TL). The aim of this part is to contribute to the advancement of knowledge regarding rehabilitation techniques and assistive devices, with the ultimate goal of improving outcomes for TL patients. Part 1: Shared Decision Making in Head and Neck Oncology Chapter 2 presents a systematic review and meta-analysis, delving into the current literature on the prevalence of decisional conflict and decision regret among individuals diagnosed with head-and-neck cancer. Through an exhaustive examination of existing research, we aimed to provide a comprehensive understanding of the challenges faced by patients in making treatment decisions. In Chapter 3, we shifted our focus inward
30 | Chapter 1 to explore the level of observed and perceived shared decision-making among head-andneck surgeons, patients, and their relatives within our own institute. By assessing the dynamics of decision-making processes in a localized setting, we aimed to gain insights into potential areas for improvement and optimization of patient care. Moving forward to Chapter 4, our attention turned specifically to patients with advanced laryngeal carcinoma who are confronted with curative treatment options. This investigation is a component of a larger multicenter study aimed at evaluating the impact of a patient decision aid tailored to this particular patient cohort. By examining the level of decisional conflict experienced by these individuals, we sought to ascertain the efficacy of interventions designed to support informed decision-making. Chapter 5 of this thesis investigated the impact of a Patient Decision Aid (PDA) for individuals facing advanced laryngeal cancer. We assessed how this tool influences treatment knowledge, decisional conflict and perceived shared decision-making. Finally, Chapter 6 details our efforts in the development of a PDA tailored specifically for individuals diagnosed with operable oropharyngeal carcinoma. By customizing decision support tools to address the unique needs and preferences of this patient population, we aimed to empower patients to actively participate in the decisionmaking process regarding their treatment options. Part 2: Rehabilitation after Total Laryngectomy Voice prostheses are crucial for laryngectomized patients, but they come with a limited device lifetime. In Chapter 7, we thoroughly explored the feasibility of prophylactic replacement strategies to address this challenge. Chapter 8 examined a newly developed voice prosthesis, meant to overcome biofilm destruction of the valve, focusing on its acceptance and voice quality among laryngectomized patients, providing valuable insights into its efficacy and usability. Moving forward to Chapter 9, we delved into the feasibility of Maximal Cardiopulmonary Exercise testing in laryngectomized patients, aiming to understand the physiological responses to exercise within this population. Additionally, we investigated the influence of Heat and Moisture Exchangers (HMEs) on exercise responses, shedding light on potential optimization strategies for respiratory health postlaryngectomy. And even after total laryngectomy, patients can still possess remarkable abilities. In Chapter 10, we shared an uplifting anecdote through a letter to the editor, highlighting the inspiring story of a laryngectomized patient who defies expectations by demonstrating an extraordinary ability to sing and play guitar.
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