Esmée Tensen

Digital dermatology in general practice Past, present and future Esmée Tensen

Digital dermatology in general practice Past, present and future Esmée Tensen

COLOFON Digital dermatology in general practice: Past, present and future PhD thesis, University of Amsterdam, Amsterdam, the Netherlands ISBN 978-90-74013-16-1 Een uitgave van Tensen Scientific Provided by thesis specialist Ridderprint, Printing: Ridderprint Layout and design: Wiebke Keck, Copyright © 2024 Esmée Tensen, Amsterdam, the Netherlands All rights reserved. No part of this thesis may be reproduced, stored or transmitted in any form or by any means without prior permission of the author or copyright-owning journals for published chapters. Financial support by Ksyos for the publication of this thesis is gratefully acknowledged.

Digital dermatology in general practice Past, present and future 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 Aula der Universiteit op vrijdag 9 februari 2024, te 11.00 uur door Esmée Tensen geboren te Amsterdam

Promotiecommissie Promotor: prof. dr. M.W.M. Jaspers AMC-UvA Copromotor: dr. L.W.P. Dusseljee-Peute AMC-UvA Overige leden: prof. dr. M.W. Bekkenk Vrije Universiteit Amsterdam dr. ir. R. Cornet AMC-UvA dr. R. Marcilly Lille University prof. dr. J. Bont AMC-UvA prof. dr. R.B. Kool Radboud Universiteit prof. mr. dr. M.C. Ploem AMC-UvA Faculteit der Geneeskunde

CONTENTS Chapter 1 General introduction 7 Part I: Status of digital dermatology worldwide Chapter 2 Two decades of teledermatology: current status and integration in national healthcare systems 29 Part II: Value of store-and-forward teledermoscopy in Dutch GP practice Chapter 3 Eleven years of teledermoscopy in the Netherlands: a retrospective quality and performance analysis of 18,738 consultations 49 Chapter 4 The value of teledermoscopy to the expertise of general practitioners diagnosing skin disorders based on ICD-10 coding 69 Chapter 5 To refer or not to refer in teledermoscopy: retrospective study 83 Part III: GPs’ perspectives about store-and-forward digital dermatology care Chapter 6 The Store-and-Forward Telemedicine Service User-satisfaction Questionnaire: development and validation of a questionnaire to monitor and assess health care providers’ experiences 101 Chapter 7 General practitioners’ perspectives about remote dermatology care during the COVID-19 pandemic in the Netherlands: questionnairebased study 125 Chapter 8 General discussion 179 Appendices 203 Summary Nederlandse samenvatting Curriculum vitae Portfolio List of publications Authors’ contributions Dankwoord (Acknowledgments)

CHAPTER 1 General introduction

8 CHAPTER 1 TELEMEDICINE IN DERMATOLOGY The worldwide aging population and the rise in patients with chronic diseases have increased the healthcare demands and corresponding healthcare costs [1-3]. In the Netherlands, expenditure on healthcare and welfare as a percentage of the gross domestic product increased from 10% in 1998 to 13.2% in 2022 [4]. These expenditures on healthcare will triple between now and 2060 if healthcare is not reformed [5]. Furthermore, healthcare personnel shortages exists and currently one out of seven Dutch employees are already working in the healthcare sector [6]. In addition, there is an increasing shortage and outflow of general practitioners (GPs) [7] and an increasing number of tasks have been shifted to primary care. If nothing changes, it is estimated that one out of three employees will be working in healthcare in 2040 [6] resulting in a shortage of staff in other social sectors such as education, construction, and energy [8]. Therefore, healthcare organizations, such as GP practices, need to reorganize and transform their existing care flow and innovate by use of digital health solutions to avoid a standstill in healthcare [1,9]. Digital transformations of sectors and businesses are a part of everyday life at the time of this writing and already widely applied in banking, shopping, traveling, consuming media, communication etcetera but are lagging behind in healthcare. Reasons for this delay are interwoven and complex, such as lack of interoperability standards and synergy between countries and healthcare organizations. This leads to a very difficult process to upscale innovations in digital healthcare transformations [10]. Telemedicine – the delivery of health care services where patients and care providers or two or more care providers are digitally brought together – has been one of the first internet-based solutions in the digital transformation of healthcare and is one of the more successfully adopted transformations in healthcare worldwide. Telemedicine, which can be applied in various care settings, provides opportunities to keep healthcare accessible and affordable and of high quality [11]. The visual features of dermatology (skin lesions have specific colors or shapes) make it well-suited for the application of telemedicine [12-14]. Telemedicine in dermatology, also called teledermatology, digital dermatology, or remote dermatology care, was one of the first telemedicine services that became available worldwide. Teledermatology aims “to provide the highest quality of dermatologic care more efficiently by moving patient information rather than patients” [15]. In other words, teledermatology includes medical specialist (diagnostic) care close to the patient (for example, at the patient’s home or in a GP practice) under remote supervision by a (tele) dermatologist (TD) [16]. The first publications in PubMed related to teledermatology date back to 1995 [17-20]. In the meantime, articles about teledermatology are being published worldwide [14]. Research on teledermatology solutions have proven that these services are effective, save the wait and travel time of the patient, cut healthcare and travel costs and reduce the number of unnecessary physical referrals of patients [12,14,21-25]. Furthermore,

9 GENERAL INTRODUCTION systematic reviews show that patients and care providers are satisfied with teledermatology services [26,27]. However, due to the heterogeneity between studies in the literature, different statements are made regarding the diagnostic reliability and referral management accuracy with these services compared to traditional in-person dermatology consultation [28,29]. Teledermatology has been aided by the evolution of consumer digital cameras and mobile phones as photo-capturing devices for GPs and by the emergence of the Internet. The expectations were that the use of smartphones in teledermatology services for patients and GPs would become increasingly integrated into healthcare like smartphones became indispensable in everyday life [30]. Literature nevertheless shows that several teledermatology implementation barriers (for example reimbursement issues, communication challenges, the lack of clinical information such as missing patient history or images in the consultation, and technological impediments) hampered the frequent use of teledermatology in daily practice worldwide before the COVID-19 pandemic [22,24,31]. The pandemic, on the other hand, stimulated the relaxations around laws and regulations and triggered the extension of reimbursement for teledermatology consultations [24,31]. As a result, the sudden need for digital care during the pandemic accelerated the implementation and adoption of several teledermatology services as healthcare solutions around the world [24,31,32]. In contrast to other countries, the Netherlands was the first country where teledermatology services had already been integrated into Dutch GP practices nationally and reimbursed by health insurance companies before the pandemic (since 2006) [33]. Results of a largescale implementation of teledermatology in Dutch daily GP practices in 2011 already showed that teledermatology reduces the number of physical referrals and delivers efficient and affordable care [33]. Despite these proven successes and expectations regarding the potential of teledermatology to address current healthcare challenges, the number of deployed digital dermatology consultations stabilized before the pandemic and the growth of this promising service stopped in the Netherlands [34]. Figure 1.1 shows the number of digital dermatology consultations registered by a telemedicine organization in the Netherlands delivering teledermatology services. One of the causes of this stagnation in usage may be an improvement in dermatological knowledge GPs acquire and their learning curve of diagnosing skin lesions after frequent use of these services [33,35]. This learning curve and gained expertise in dermatology might have reduced the need for GPs to utilize teledermatology for subsequent patients with similar lesions. Moreover, certain obstacles could hamper the upward trend in the use of digital dermatology consultations. It is unknown how GPs experienced the existing digital dermatology services during the COVID-19 pandemic and which facilitators and barriers GPs encountered in the use of these services. Therefore, this thesis investigates the status of digital dermatology in Dutch GP practices and the use of the service during the COVID-19 pandemic. More specifically, the added value of digital dermatology consultation for GPs, the challenges 1

10 CHAPTER 1 GPs face in the use of the service, and the reasons for the stagnation in the use of digital dermatology on a national level are studied. Figure 1.1: Number of digital dermatology consultations registered by a telemedicine organization in the Netherlands delivering teledermatology services. The first section of this general introduction discusses the setup of standard dermatology care in Dutch GP practices. The second section provides an overview of the involved actors and delivery modalities of teledermatology. The third section zooms in on the teledermatology flow of a telemedicine organization in the Netherlands delivering teledermatology services. The fourth section addresses the use of the service during the COVID-19 pandemic and the implementation complexity of teledermatology. The last section of this chapter specifies the aims of the studies and the outline of this thesis. STANDARD DERMATOLOGY CARE IN DUTCH GP PRACTICES In the Netherlands, GPs act as gatekeepers for secondary care, which includes specialist dermatology care. GPs are the first-line contact for patients seeking specialist dermatology care and in this way play an essential role in triaging patients with skin lesions. Patients thus only have access to a dermatologist through a referral by a GP. GPs assess patients’ skin lesions and decide whether further diagnostics (such as a digital dermatology consultation, skin culture or biopsy) are needed. If further diagnostics are indicated, they might directly refer the patient to a dermatologist or start a digital dermatology consultation. If no further diagnostics are needed, they manage the skin

11 GENERAL INTRODUCTION conditions themselves in their practice with a wait-and-see policy or medical treatment. Fourteen percent of patient complaints in GP practice are related to the skin [36]. It is estimated that GPs manage or treat 86% of these skin conditions themselves and that they refer 14% of the patients to a dermatologist [37]. With more than 34 referrals per 1000 enrolled patients per year (in 2022), dermatology is the largest referral specialism of GPs [38]. The top-3 International Classification of Primary Care (ICPC) codes for dermatology referrals from 2022 showed that most of these GP referrals were related to “Skin disease, other” (S99), “Nevus/mole” (S82), or “Malignant neoplasm of skin” (S77) [38]. Patients wait on average 35 days for a scheduled consultation appointment in the outpatient dermatology department and 18 days for a consultation in case of a suspicious lesion (September 2023) [39], while GPs receive a diagnosis and advice by a TD about a patient’s skin lesion within 2 working days. Furthermore, the incidence of skin cancer is increasing worldwide due to excessive sun exposure (Ultraviolet (UV) light) on tropical holidays and leisure activities, the growing and aging population, and patients’ awareness of skin cancer [40]. GPs experience an increase in patients who request assessments of nevi and other skin lesions [40]. This growing need for dermatology care and the rise in suspicious lesions combined with the aging population further enlarges the pressure on Dutch healthcare [41,42]. As the pivot of primary care, GPs experience high work pressure and have little consultation time for each patient [1]. In the fixed consultation time of 10-15 minutes, the GP has to identify the patient’s purpose of consultation and original request for help, take an anamnesis, perform a physical examination, discuss diagnosis and treatment management policy with the patient, start a referral or prescribe medication, and write a report in the GP Information System (Dutch: Huisarts Informatie Systeem (HIS)) [40]. In addition, for skin neoplasms, the GP needs to decide whether the skin condition is benign or malignant. However, GPs experience challenges in diagnosing skin disorders, especially in discriminating (pre) malignant and benign skin lesions, and diagnosing skin cancer [40,43-45]. This results not only in referrals of patients with malignant skin lesions to a dermatologist, but also of patients with mild benign skin lesions (for example, seborrheic keratosis, vascular lesions, and benign nevus) [42-44]. Teledermatology could thus support GPs in this patient referral process for low-complex dermatology questions so that mainly patients with complex or severe skin lesions or patients that require specialized care outside the scope of the GP are referred to the dermatologist. Teledermatology is therefore an example of a digital dermatology intervention that can be used in addition to standard care to support GPs in diagnosing skin lesions in primary practice and improve GP’s diagnostic accuracy by TD advice. 1

12 CHAPTER 1 TELEDERMATOLOGY: INVOLVED ACTORS AND DELIVERY MODALITIES Teledermatology can be availed by various actors: patients, GPs, and dermatologists (Figure 1.2). In primary teledermatology, the patient directly exchanges photographs taken by himself and information of the skin disorder via a digital consultation request with the healthcare provider (in the Netherlands mostly the GP) [12,46]. In secondary teledermatology, currently the most prevalent used method, the GP seeks TD triage advice or a diagnosis by sending information of the skin disorder and photographs taken by the GP to a TD [12,46]. The aim of this secondary teledermatology is to avoid unnecessary physical patient referrals to the dermatologist or to accelerate patient referrals to the dermatologist for urgent skin lesions that would otherwise not have been directly referred. Tertiary teledermatology involves direct collaboration between dermatologists, for example, a second opinion of a skin lesion or to seek advice from a specialized (academic) dermatologist [12,46,47]. Finally, patientassisted teledermatology concerns direct contact between the patient and the TD such as a follow-up or monitoring [12,46]. Figure 1.2: Involved actors teledermatology [49]. Furthermore, teledermatology can be delivered store-and-forward (asynchronous), real-time (synchronous or live interactive), or hybrid (combination of store-and-forward and real-time) [12,46,48]. Store-and-forward is the commonly used delivery modality of teledermatology in which (high-resolution) digital clinical images are obtained by a GP and exchanged asynchronously between the patient and the GP, between the GP and the TD, or between two dermatologists. Store-and-forward teledermatology consultation is time and space independent. The patient, GP and TD can start or assess the consultation at any time. In contrast, the real-time teledermatology service uses (lowresolution) videoconferencing and facilitates direct interaction between the patient and the GP or between the GP and TD from various physical locations. This latter real-time synchronous teledermatology format is time-dependent and labor-intensive. Finally, hybrid teledermatology is a combination of store-and-forward and real-time consultation. High-resolution digital images are assessed and at the same time, direct video interaction takes place between the patient and the GP or between the GP and TD.

13 GENERAL INTRODUCTION This thesis focuses on store-and-forward primary and secondary teledermatology in GP practices (in this thesis also referred to and explained further below as dermatology home consultation and teledermatology or teledermoscopy, respectively). This means that patient information and digital images of a skin lesion are asynchronously exchanged between a patient and his/her GP or between a GP and a TD. TELEDERMATOLOGY FLOW OF A DUTCH TELEMEDICINE ORGANIZATION Healthcare organizations that facilitate digital services such as teledermatology are either mostly traditional healthcare institutions or innovative ICT providers. In contrast, Ksyos (formerly also known as Ksyos Telemedical Center), is a digital hospital that provides lowcomplex medical specialist care using innovative ICT techniques [50]. Ksyos operates on a national scale and is the largest telemedicine organization providing store-and-forward teleconsultation in the Netherlands. “[The telemedicine organization] is an innovative colleague who helps to improve the digital means of communication between primary and secondary care” (Respondent questionnaire, 2019) Ksyos contracts medical specialists, paramedics, and GPs to perform digital health services through a platform in fields such as dermatology, cardiology, ophthalmology, mental health, and somnology. Research in this thesis was conducted in collaboration with Ksyos within the path of dermatology care and from the perspective of the GPs. Approximately half of all Dutch GPs are affiliated with Ksyos and have been registered for the digital dermatology service. In this thesis Ksyos will be referred to as the telemedicine organization. This Dutch telemedicine organization facilitates three types of store-andforward digital dermatology consultation: (1) teledermatology, (2) teledermoscopy, and (3) dermatology home consultation (Figure 1.3). In the Netherlands, with nearly 18 million inhabitants, there were in 2022 approximately 4900 GP practices and 11,754 operating GPs (i.e., self-employed GP practice owners, GPs employed at a GP practice (Dutch: Huisarts in dienst van een huisarts), and freelance GPs (Dutch: waarnemers)) available [52,53]. In 2022, in about one out of five GP practices, GPs had performed a teledermatology, teledermoscopy, or digital dermatology home consultation in the Dutch telemedicine organization’s platform. Between 2005 and 2022, a total of 203,862 teledermatology consultations, 25,891 teledermoscopy, and 17,142 digital dermatology home consultations had been performed via the Dutch digital dermatology platform (Figure 1.4). The total number of performed digital dermatology consultations in the Netherlands might be higher because there are more digital dermatology providers on the market. 1

14 CHAPTER 1 Figure 1.3: Teledermatology, teledermoscopy, and digital dermatology home consultation process [51]. GP = General practitioner; TD = Teledermatologist. Figure 1.4: Number of teledermatology, teledermoscopy, and dermatology home consultations in the telemedicine organization’s digital dermatology platform per year.

15 GENERAL INTRODUCTION Teledermatology The first type of store-and-forward digital dermatology consultation that GPs can perform through the telemedicine organization is regular teledermatology (Figure 1.3). To start a teledermatology consultation, GPs directly log in through a single sign-on from their GP Information System (Dutch: Huisarts Informatiesysteem (HIS)) in the telemedicine platform or they sign in on the telemedicine organization’s website. The GP uses a digital camera or ubiquitous smartphone or tablet to take detailed and overview photographs of the patient’s skin lesion. A maximum of four obtained images can be uploaded to a standardized consultation request after oral consent from the patient. The GP completes the consultation request with an anamnesis (for example, additional patient information, patient medication, prehistory of skin cancer, etc.), optionally enters a primary (differential) diagnosis and selects a TD for assessment. After sending the consultation to a TD for assessment, for each consultation the GP is asked through an embedded evaluation question on the platform, whether the GP would have referred the patient without the availability of teledermatology (Box 1.1). Within two working days, the TD assesses the images of the skin lesion in the consultation request, provides a mandatory primary diagnosis and advice to a GP. If the TD cannot assess the skin lesion based on the pictures added, there is an option for the TD to choose “no diagnosis,” “non-assessable,” or “no abnormalities.” Both GP and TD diagnoses in the teledermatology platform are automatically coded according to the International Classification of Diseases, 10th revision (ICD-10) [54]. This worldwide used ICD-10 classification contributes to a consistent way of reporting and monitoring diseases and health-related conditions. Since July 2015, Dutch specialized care organizations are obliged by the Dutch Healthcare Authority (Dutch: Nederlandse Zorgautoriteit, NZa) to register diagnoses in compliance with such an ICD-10 code. This classification standard is controlled by the World Health Organization and facilitates, for example, data analysis on provided diagnoses over time. In this thesis, we compared the GPs and TDs diagnoses with each other and analyzed the TD diagnoses based on the derived ICD-10 codes. Furthermore, we used the ICD-10 codes to map the TD diagnoses into three diagnosis groups (benign, premalignant, and malignant). The TD diagnoses were used as a diagnosis measure in our studies as not all TD diagnoses were face-to-face or histopathologically confirmed. In addition, face-to-face or histopathological diagnoses determined after teledermatology were out of our study scope as these were not registered into the digital dermatology platform and interoperability and privacy restrictions of data limited the exchange of this data between the electronic patient records of the dermatologists and the teledermatology platform. In addition to providing a diagnosis via the digital dermatology platform, the TD responds through an evaluation question whether a physical referral to a dermatologist is necessary (Box 1.1). The GP receives the TD response in the teledermatology platform and (if preferred by the GP, automatically) in the GP’s HIS. Afterwards, the GP completes three evaluation questions in the teledermatology platform (Box 1.1). 1

16 CHAPTER 1 The mandatory basic insurance for all Dutch inhabitants usually fully covers all patient visits in GP practices and applies deductible excess for visits in secondary care. Nowadays, most Dutch health insurance companies reimburse digital dermatology consultations without deductible excess. This means that as long as the patient is not physically referred to the dermatologist and remains treated in GP care, the patient does not have to pay excess costs. GPs can declare these consultations as a Modernization and Innovation (M&I) service (code 13009). This M&I financing form has been introduced for special procedures in general practice, among others, to promote the quality of GP care and to substitute care from secondary to primary care [55]. TDs receive financial compensation by the telemedicine organization for each assessed consultation. Box 1.1: Evaluation questions asked to general practitioners (GPs) and teledermatologists (TDs) before, during, and after teledermatology and teledermoscopy assessment. Before sending consultation: Q1 (GP): Would you have referred this patient if teledermatology/teledermoscopy was not available? During consultation: Q2 (TD): Is a visit to the specialist necessary? After consultation: Q3 (GP): Do you refer this patient to a specialist? Q4 (GP): Are you and your patient helped with this teledermatology/teledermoscopy consultation? Q5 (GP): Have you learned from this teledermatology/teledermoscopy consultation? Teledermoscopy The second type of store-and-forward digital dermatology consultation is teledermoscopy (Figure 1.3). Teledermoscopy (or teledermatoscopy) is an extension of teledermatology and has been facilitated by the telemedicine organization since 2009. The teledermoscopy flow is similar to the regular teledermatology flow: the GP submits skin photographs to a TD for diagnosis or advice. However, in addition to detailed and overview photographs, the GP takes one or more digitalized dermoscopic images of the patient’s skin lesion with a dermoscope plus digital camera or a smartphone plus dermoscopy attachment. Dermoscopy is a noninvasive diagnostic tool for evaluating skin lesions. It allows GPs and dermatologists to closely visualize the skin and to gain a magnified view of details beneath the skin surface that is not visible to the naked eye. This technique is often used for the diagnosis of suspicious skin lesions, improves diagnostic efficacy for the screening of skin cancers, and can help GPs to discriminate between benign skin lesions or potentially life-threatening skin malignancies (melanoma) [56-58]. Early detection of melanoma is critical because the stadium of detection and treatment is one of the determinants for the 5-year survival of the patient [59].

17 GENERAL INTRODUCTION Dermatology home consultation The third type of store-and-forward digital dermatology consultation provided by the telemedicine organization is dermatology home consultation (Figure 1.3). Digital dermatology home consultation is a free-of-charge primary teledermatology service whereby patients with skin complaints take photographs with their own smartphone and directly exchange information with their GP. In this way, the patient stays at home and does not have to physically visit a GP practice. During the pandemic this also meant no risk of exposure to the coronavirus (SARS-CoV-2). In this modality, the patient receives an email from a GP(assistant) with a link to a structured and secured digital request. The patient opens the digital request, preferably on his/her smartphone or tablet, takes and uploads two to four images of the skin lesion, and completes a closed-ended questionnaire. In the digital request, the patient receives basic instructions on how to take high-quality images (take images which are sharp and have a clear contrast, use good lighting, use no flash, and do not zoom in). Thereafter, the patient sends the consultation request back to the GP. The GP evaluates the skin condition himself/herself, provides feedback to the patient, and saves the information recorded in the dermatology home consultation request (automatically) into his/her HIS. If necessary, the GP can convert the home consultation request into an additional teledermatology consultation for advice and diagnosis by a TD. The GP can choose to add only images taken by the patient, or to take additional photographs in the GP practice in presence of the patient and add these photographs just like the regular teledermatology consultation flow. COVID-19 PANDEMIC AND TELEDERMATOLOGY IMPLEMENTATION COMPLEXITY The COVID-19 pandemic has been a worldwide driver to use digital dermatology as a solution to unburden standard face-to-face dermatology care [24,31,32]. Simultaneously, there was an exponential growth in “teledermatology” publications in the first (2020) and second (2021) COVID-19 pandemic year (Figure 1.5). The number of “teledermatology” “teledermoscopy” and “teledermatoscopy” publications grew steadily before the pandemic (864 publications between 1995-2019) and almost doubled to 1540 publications by the end of 2022. 1

18 CHAPTER 1 Figure 1.5: The number of publications in PubMed with search query “teledermatology or teledermoscopy or teledermatoscopy” (September 2023). This large worldwide uptake of digital dermatology services during the COVID-19 pandemic contrasted sharply with the long history of slow adoption before. The limited adoption of digital dermatology services before the pandemic was probably caused by the fact that telemedicine services are complex and versatile health systems and challenges remained in the use of these services. Implementation of these services is challenging because it transcends the process of merely installing a technology tool or platform and involves interactions between various technological and social human system aspects and workflow changes [60-62]. These aspects include for example, the perceived ease of use or usefulness of the digital dermatology platform technology, serviceable equipment adapted to technology needs, adequate and up-to-date training, technological assistance and support of patients, GPs and dermatologists, integration of the service within the current workflow, interaction and communication between digital dermatology provider and GPs or dermatologists and among GPs and dermatologists, insurance eligibility, reimbursement of care, equipment costs, etc. [22,62-67]. Furthermore, all of these aspects are closely interrelated with each other, which means that modifications to one aspect will affect changes elsewhere. The digital dermatology services function most effectively when these social and technical components work together and are “jointly optimized” [68,69]. Understanding how the different interrelated components in the digital dermatology services are experienced by the GPs in their day-to-day work processes is essential to further improve the services to the needs of the GPs and for the sustainability of the service [70]. Therefore, to continuously apply digital dermatology services and to support the major challenges of healthcare delivery by these services, it is important to regularly evaluate the status of these services, how these services are used and experienced by stakeholders, and to examine their possible added value. More specifically, given the impact of sociotechnical factors on implementation success and use of these services, research on the facilitators and barriers GPs examine will give insight into which improvements are required

19 GENERAL INTRODUCTION to guarantee the quality, continuity and higher uptake of the digital dermatology services in the Netherlands. AIMS OF THIS THESIS The general objective of this thesis is to evaluate the value of store-and-forward teledermatology, teledermoscopy, and dermatology home consultation for Dutch GP practice. Furthermore, this thesis aims to explore the facilitators and barriers in the uptake of these digital dermatology services by GPs and to provide recommendations for future practice and research. First, we aimed to gain insight into the worldwide status of two decades (1995 – 2015) of teledermatology by performing a literature review (Part I). Second, we aimed to understand the impact and added value of performing store-and-forward teledermoscopy for GPs in Dutch GP practice (Part II). Lastly, our objective was to determine GPs’ perspectives about store-and-forward teledermatology, teledermoscopy, and dermatology home consultation in Dutch GP practice and to examine those factors that facilitate or impede the successful implementation and use of the services. To realize this aim, we developed, validated, and applied a quality feedback tool for store-and-forward telemedicine services (Part III). OUTLINE OF THIS THESIS Part I: Status of digital dermatology worldwide Chapter 2 reviews two decades of worldwide teledermatology research in the literature concerning: the actors involved in the teledermatology process, purposes and subspecialties of teledermatology services, delivery modalities and technologies used, business models used, the integration of teledermatology into national health infrastructures, preconditions and requirements for implementation, and the surplus merits. Part I will give an overview of the status worldwide before we zoom in on teledermoscopy use in Dutch GP practice in part II. Part II: Value of store-and-forward teledermoscopy in Dutch GP practice Part II includes three retrospective studies and focuses on the added value of the storeand-forward teledermoscopy consultations for GPs in Dutch primary care. For these studies, we use for example data on the GP and TD self-reported evaluation questions and TD diagnoses embedded in the digital dermatology platform. In Chapter 3 we evaluate teledermoscopy quality and performance outcomes in Dutch primary care based on 11 years of teledermoscopy data (February 2009 – February 2020). Quality outcomes are operationalized as the percentage of GPs’ second opinion requests to TDs, percentage of extra teledermoscopy referrals, percentage of GPs following up on TDs’ referral advice, and percentages of GPs who valued the teledermoscopy consultation as helpful and instructive. Performance outcomes are defined as the percentage of patients that would 1

20 CHAPTER 1 have been referred to the dermatologist if teledermoscopy would not have been available, the overall referral percentage in teledermoscopy, and the time GPs and TDs need to send in and respond to a teledermoscopy consultation. In Chapter 4 and Chapter 5, we further examine the merits of teledermoscopy for GPs. Chapter 4 assesses the added value as the GPs’ gained expertise in diagnosing skin disorders after continued use of teledermoscopy. In this chapter, we specifically focus on experienced GPs and compare the GP pre-diagnosed skin disorders to the final provided TD diagnoses. Chapter 5 measures the added value as to whether the availability of teledermoscopy changes GPs’ (non)referral decisions before and after teledermoscopy for skin lesions diagnosed by the TD as benign, premalignant and malignant. Part III: GPs’ perspectives about store-and-forward digital dermatology care Part III elaborates on GPs’ perspectives about store-and-forward digital dermatology care and examines how teledermatology and teledermoscopy are incorporated into GPs’ work practice. Chapter 6 focuses on the development and validation of the Store-and-Forward Telemedicine Service User-satisfaction Questionnaire (SAF-TSUQ). This quality feedback tool monitors health care providers’ experiences with store-and-forward contracted telemedicine services. This means that SAF-TSUQ assesses experiences of both the referring health care providers, such as GPs, and the performing health care providers, such as dermatologists. This quality feedback tool aims to provide insight into why and what motivates health care providers to embrace or reject these innovative telemedicine services and how store-and-forward telemedicine organizations can enhance their services. Chapter 7 uses the quality feedback tool from Chapter 6 and extends the questionnaire with open and closed-ended questions on the use of digital dermatology care in general practice and questions related to the COVID-19 pandemic. This combined questionnaire was sent to GPs to investigate how satisfied GPs were with the three types of digital dermatology consultation in the Netherlands during the COVID-19 pandemic. In addition, we used a socio-technical model to understand the interrelations between the factors that facilitate and hamper the successful implementation and use of the services. Finally, Chapter 8 provides a summary of the main findings, a general discussion, and a conclusion on how teledermatology, teledermoscopy, and dermatology home consultation are integrated and received or rejected in Dutch GP practice. Furthermore, we will discuss the added value and challenges contributing to the successful implementation and use of these services in the Netherlands and provide suggestions for future research.

21 GENERAL INTRODUCTION REFERENCES 1. Ministerie van Volksgezondheid Welzijn en Sport. Integraal Zorgakkoord: Samen werken aan gezonde zorg, 2022. URL: rapporten/2022/09/16/integraal-zorgakkoord-samen-werken-aan-gezonde-zorg/integraalzorg-akkoord.pdf [accessed 2023-09-18]. 2. Organisation for Economic Co-operation and Development (OECD). URL: https://data.oecd. org/ [accessed 2023-09-18]. 3. Hilderink H, Verschuuren M. Rijksinstituut voor Volksgezondheid en Milieu. Volksgezondheid Toekomst Verkenning 2018: een gezond vooruitzicht. Synthese. URL: synthese-vtv-2018-een-gezond-vooruitzicht [accessed 2023-09-18]. 4. Zorguitgaven; kerncijfers. Statistics Netherlands (CBS). URL: nl/dataset/84047NED/table [accessed 2023-09-18]. 5. Vonk R, Hilderink H, Plasmans M, Kommer G, Polder J. Toekomstverkenning zorguitgaven 2015-2060: Kwantitatief vooronderzoek in opdracht van de Wetenschappelijke Raad voor het Regeringsbeleid (WRR). Deel 1: toekomstprojecties, RIVM-Rapport 2020-0059, Bilthoven: Rijksinstituut voor Volksgezondheid en Milieu. Doi:10.21945/RIVM-2020-0059. 6. Wetenschappelijke Raad voor het Regeringsbeleid. Kiezen voor houdbare zorg. Mensen, middelen en maatschappelijk draagvlak, WRR-Rapport 104, Den Haag: WRR, 2021. 7. Batenburg R, Bosmans M, Versteeg S, Vis E, van Asten B, Vandermeulen L, et al. Balans in vraag en aanbod huisartsenzorg. Utrecht: Nivel, Prismant, 2018. ISBN 978-94-6122-531-3. URL: [accessed 2023-09-18]. 8. Letter to Parliament about new labor market forecast for healthcare and welfare, 2023. URL: [accessed 2023-09-18]. 9. Social and Economic Council of the Netherlands. Zorg voor de toekomst. Over de toekomstbestendigheid van de zorg, Den Haag: Sociaal-Economische Raad, 2020. URL: https://www. [accessed 2023-09-18]. 10. Greenhalgh T, Wherton J, Papoutsi C, Lynch J, Hughes G, A’Court C, et al. Beyond adoption: a new framework for theorizing and evaluating nonadoption, abandonment, and challenges to the scale-up, spread, and sustainability of health and care technologies. J Med Internet Res 2017;19(11):e367. 11. van der Vaart R, van Tuyl LHD, Versluis A, Wouters MJM, van Deursen L, Standaar L, et al. E-healthmonitor 2022: stand van zaken digitale zorg. Bilthoven: Rijksinstituut voor Volksgezondheid en Milieu, 2022. 12. Tognetti L, Fiorani D, Russo F, Lazzeri L, Trovato E, Flori ML, et al. Teledermatology in 2020: past, present and future perspectives. Ital J Dermatol Venerol 2021;156(2):198-212. 13. Coates SJ, Kvedar J, Granstein RD. Teledermatology: from historical perspective to emerging techniques of the modern era: part I: history, rationale, and current practice. J Am Acad Dermatol 2015;72(4):563-574. 14. Trettel A, Eissing L, Augustin M. Telemedicine in dermatology: findings and experiences worldwide - a systematic literature review. J Eur Acad Dermatol Venereol 2018;32(2):215-224. 15. Pak HS. Teledermatology and teledermatopathology. Semin Cutan Med Surg 2002;21(3):179-189. 1

22 CHAPTER 1 16. Noordzij M, Witkamp L. Digitale Transformatie: nog grote stappen te maken. Topic 2022;28. 17. Gonçalves L, Cunha C. Telemedicine project in the Azores Islands. Arch Anat Cytol Pathol 1995;43(4):285-287. 18. Crichton C, Macdonald S, Potts S, Syme A, Toms J, McKinlay J, et al. Teledermatology in Scotland. J Telemed Telecare 1995;1(3):185. 19. Perednia DA, Brown NA. Teledermatology: one application of telemedicine. Bull Med Libr Assoc 1995;83(1):42-47. 20. Menn ER, Kvedar JC. Teledermatology in a changing health care environment. Telemed J 1995;1(4):303-308. 21. Lee KJ, Finnane A, Soyer HP. Recent trends in teledermatology and teledermoscopy. Dermatol Pract Concept 2018;8(3):214-223. 22. Dovigi E, Kwok EYL and English JC. A framework-driven systematic review of the barriers and facilitators to teledermatology implementation. Curr Dermatol Rep 2020;9(4):353-361. 23. Landow SM, Mateus A, Korgavkar K, Nightingale D, Weinstock MA. Teledermatology: key factors associated with reducing face-to-face dermatology visits. J Am Acad Dermatol 2014;71(3):570-576. 24. Farr MA, Duvic M, Joshi TP. Teledermatology during COVID-19: an updated review. Am J Clin Dermatol 2021;22(4):467-475. 25. López-Liria R, Valverde-Martínez MÁ, López-Villegas A, Bautista-Mesa RJ, Vega-Ramírez FA, Peiró S, et al. Teledermatology versus face-to-face dermatology: an analysis of costeffectiveness from eight studies from Europe and the United States. Int J Environ Res Public Health 2022;19(5):2534. 26. Mounessa JS, Chapman S, Braunberger T, Qin R, Lipoff JB, Dellavalle RP, Dunnick CA. A systematic review of satisfaction with teledermatology. J Telemed Telecare 2018;24(4):263-270. 27. Hadeler E, Gitlow H, Nouri K. Definitions, survey methods, and findings of patient satisfaction studies in teledermatology: a systematic review. Arch Dermatol Res 2021;313(4):205-215. 28. Bourkas AN, Barone N, Bourkas MEC, Mannarino M, Fraser RDJ, Lorincz A, et al. Diagnostic reliability in teledermatology: a systematic review and a meta-analysis. BMJ Open 2023;13:e068207. 29. Chuchu N, Dinnes J, Takwoingi Y, Matin RN, Bayliss SE, Davenport C, et al. Teledermatology for diagnosing skin cancer in adults. Cochrane Database of Syst Rev 2018;12(12):CD013193. 30. Nami N, Massone C, Rubegni P, Cevenini G, Fimiani M, Hofmann-Wellenhof R. Concordance and time estimation of store-and-forward mobile teledermatology compared to classical faceto-face consultation. Acta Derm Venereol 2015;95(1):35-39. 31. Yeboah CB, Harvey N, Krishnan R, Lipoff JB. The impact of COVID-19 on teledermatology: a review. Dermatol Clin 2021;39(4):599-608. 32. Loh CH, Chong Tam SY, Oh CC. Teledermatology in the COVID-19 pandemic: a systematic review. JAAD Int 2021;5:54-64. 33. Van der Heijden JP, De Keizer NF, Bos JD, Spuls PI, Witkamp L. Teledermatology applied following patient selection by general practitioners in daily practice improves efficiency and quality of care at lower cost. Br J Dermatol 2011;165(5):1058-1065. 34. Tensen E, van der Heijden JP, Jaspers MWM, Witkamp L. Teledermatologie. Bijblijven 2017;33(2):101-113.

23 GENERAL INTRODUCTION 35. Mohan GC, Molina GE, Stavert R. Store and forward teledermatology improves dermatology knowledge among referring primary care providers: a survey-based cohort study. J Am Acad Dermatol 2018;79(5):960-961. 36. De Vries, E. Huidaandoeningen bij huisarts en dermatoloog. Ned Tijdschr Geneeskd 2013;157:B1930. 37. Hansen J, Schepman S. Op de huid: inventarisatie zorgvraag en marktgebied huidtherapie. Utrecht: Nivel, 2009. 38. Heins M, Bes J, Weesie Y, Davids R, Winckers M, Korteweg L, et al. Zorg door de huisarts. Nivel Zorgregistraties Eerste Lijn: jaarcijfers 2022 en trendcijfers 2018-2022. Utrecht: Nivel, 2023. 39. ZorgkaartNederland. URL: [accessed 202309-21]. 40. Wakkee M, Eekhof J. Huidkankerzorg door de huisarts. Ned Tijdschr Dermatol Venereol 2020;30(3):16-19. 41. Schreuder K, de Groot J, Hollestein L, Louwman M. Huidkanker in Nederland - Cijfers Uit 30 Jaar Kankerregistratie, 2019. URL: iknl/media/pdfs/kankersoorten/iknl_huidkanker-in-nl_rapport_nkr.pdf [accessed 2023-09-21]. 42. Koelink CJL, Kollen BJ, Groenhof F, van der Meer K, van der Heide WK. Skin lesions suspected of malignancy: an increasing burden on general practice. BMC Fam Pract 2014;15:29. 43. Ahmadi K, Prickaerts E, Smeets JGE, Joosten VHMJ, Kelleners-Smeets NWJ, Dinant GJ. Current approach of skin lesions suspected of malignancy in general practice in the Netherlands: a quantitative overview. J Eur Acad Dermatol Venereol 2018;32(2):236-241. 44. van Rijsingen MCJ, Hanssen SCA, Groenewoud JMM, van der Wilt GJ, Gerritsen MP. Referrals by general practitioners for suspicious skin lesions: the urgency of training. Acta Derm Venereol 2014;94(2):138-141. 45. Vestergaard T, Prasad SC, Schuster A, Laurinaviciene R, Bygum A, Munck A, et al. Introducing teledermoscopy of possible skin cancers in general practice in Southern Denmark. Fam Pract 2020;37(4):513-518. 46. Pasquali P, Sonthalia S, Moreno-Ramirez D, Sharma P, Agrawal M, Gupta S, et al. Teledermatology and its Current Perspective. Indian Dermatol Online J 2020;11(1):12-20. 47. van der Heijden JP, Spuls PI, Voorbraak FP, De Keizer NF, Witkamp L, Bos JD. Tertiary teledermatology: a systematic review. Telemed J E Health 2010;16(1):56-62. 48. Whited JD. Teledermatology. Med Clin North Am 2015;99(6):1365-1379. 49. Tensen E, van der Heijden JP, Jaspers MW, Witkamp L. Two decades of teledermatology: current status and integration in national healthcare systems. Curr Dermatol Rep 2016;5:96-104. 50. Ksyos homepage. Ksyos. URL: [accessed 2023-09-30]. 51. Tensen E, Kuziemsky C, Jaspers MW, Peute LW. General practitioners’ perspectives about remote dermatology care during the COVID-19 pandemic in the Netherlands: QuestionnaireBased Study. JMIR Dermatol 2023;6:e46682. 52. Batenburg R, Flinterman L, Vis E, van Schaaijk A, de Geit E, Kenens RJ, et al. Cijfers uit de Nivel-registratie van huisartsen en huisartsenpraktijken, Een actualisering voor de periode 2020-2022. Utrecht: Nivel, 2022. 53. Bevolkingsteller. Statistics Netherlands (CBS). URL: dashboard-bevolking/bevolkingsteller [accessed 2023-09-30]. 1

24 CHAPTER 1 54. World Health Organization ICD-10 Browser. URL: [accessed 2023-09-30]. 55. Nederlandse Zorgautoriteit. Prestatie- en tariefbeschikking huisartsenzorg en multidisciplinaire zorg 2023 - TB/REG-23617-03. URL: [accessed 2023-09-30]. 56. Chappuis P, Duru G, Marchal O, Girier P, Dalle S, Thomas L. Dermoscopy, a useful tool for general practitioners in melanoma screening: a nationwide survey. Br J Dermatol 2016;175(4):744-750. 57. Ferrándiz L, Ojeda-Vila T, Corrales A, Martín-Gutiérrez FJ, Ruíz-de-Casas A, Galdeano R, et al. Internet-based skin cancer screening using clinical images alone or in conjunction with dermoscopic images: a randomized teledermoscopy trial. J Am Acad Dermatol 2017;76(4):676682. 58. Jones OT, Jurascheck LC, van Melle MA, Hickman S, Burrows NP, Hall PN, et al. Dermoscopy for melanoma detection and triage in primary care: a systematic review. BMJ Open 2019;9(8):e027529. 59. Saginala K, Barsouk A, Aluru JS, Rawla P, Barsouk A. Epidemiology of Melanoma. Medical Sciences 2021; 9(4):63. 60. Basu A, Kuziemsky C, de Araújo Novaes M, Kleber A, Sales F, Al-Shorbaji N, et al. Telehealth and the COVID-19 pandemic: international perspectives and a health systems framework for telehealth implementation to support critical response. Yearb Med Inform 2021;30(1):126-133. 61. Champion C, Kuziemsky C, Affleck E, Alvarez GG. A systems approach for modeling health information complexity. Int J Inf Manag 2019;49:343-354. 62. Alami H, Gagnon MP and Fortin JP. Some multidimensional unintended consequences of telehealth utilization: a multi-project evaluation synthesis. Int J Health Policy Manag 2019;8(6):337-352. 63. Moore MA, Coffman M, Jetty A, Petterson S, Bazemore A. Only 15% of FPs report using telehealth; training and lack of reimbursement are top barriers. Am Fam Physician 2016;93(2):101. 64. Lin CC, Dievler A, Robbins C, Sripipatana A, Quinn M, Nair S. Telehealth in health centers: key adoption factors, barriers, and opportunities. Health Aff 2018;37(12):1967-1974. 65. Kruse CS, Karem P, Shifflett K, Vegi L, Ravi K, Brooks M. Evaluating barriers to adopting telemedicine worldwide: a systematic review. J Telemed Telecare 2018;24(1):4-12. 66. Segato F and Masella C. Telemedicine services: how to make them last over time. Health Policy Technol 2017;6(3):268-278. 67. Orruño E, Gagnon MP, Asua J, Ben AA. Evaluation of teledermatology adoption by healthcare professionals using a modified technology acceptance model. J Telemed Telecare 2011;17(6):303-307. 68. Di Maio, P. Towards a metamodel to support the joint optimization of socio technical systems. Systems 2014,2(3):273-296. 69. Olsen, G, Wolfe D, Hellewell J, Ize-Ludlow D, Bledsoe J, Srivastava R. Building technology solutions in context. Qual Manag Health Care 2022;31(1):46-48. 70. Meurs M, Keuper J, Sankatsing V, Batenburg R, van Tuyl L. “Get used to the fact that some of the care Is really going to take place in a different way”: general practitioners’ experiences with e-health during the COVID-19 pandemic. Int J Environ Res Public Health 2022;19(9):5120.


PART I Status of digital dermatology worldwide

CHAPTER 2 Two decades of teledermatology: current status and integration in national healthcare systems Authors: Esmée Tensen Job P. van der Heijden Monique W.M. Jaspers Leonard Witkamp Current Dermatology Reports. 2016;5:96-104. DOI: 10.1007/s13671-016-0136-7.

30 CHAPTER 2 ABSTRACT Teledermatology, originating in 1995, has been one of the first telemedicine services to see the light of day. Two decades of teledermatology research is summarized in this review. A literature search was conducted in PubMed. Search terms included “teledermatology,” “teledermoscopy,” “tele wound care,” “telederm*,” “(dermatology OR dermoscopy OR wound care OR skin) AND (telemedicine OR ehealth or mhealth OR telecare OR teledermatology OR teledermoscopy).” Inclusion criteria were (i) Dutch or English written papers and (ii) publication year from 2011 to present or (iii) (systematic) reviews with publication year before 2011. One hundred fourteen publications and 14 (systematic) reviews were included for full text reading. Focus of this review is on the following outcomes: (i) actors (primary, secondary, tertiary), (ii) purposes (consultation, triage, follow-up, education) and subspecialties (tele-wound care, burn care, teledermoscopy (teledermatoscopy), teledermatopathology, and mobile teledermatology), (iii) delivery modalities and technologies (store-and-forward, real-time interactive, and hybrid modalities using web-based systems, email, mobile phones, tablets, or videoconferencing equipment), (iv) business models, (v) integration of teledermatology into national healthcare systems, (vi) preconditions and requirements for implementation (security, ethical issues, responsibility, reimbursement, user satisfaction, technique, and technology standards), and (vii) added value. To conclude, teledermatology is an efficient and effective healthcare service compared to in-person care. Teledermatology reduces patients’ travel time and waiting time, avoids (unnecessary) dermatologic visits, and improves access of care to underserved patients.