Practice points for lymphoedema care in low- and middle- income countries developed by nominal group technique

Abstract

Background

Lymphoedema is a common, distressing, and debilitating condition affecting more than 200 million people globally. There is a small body of evidence to guide lymphoedema care which underpins several lymphoedema clinical practice guidelines developed for high-income countries (HIC). Some of these recommendations are unlikely to be feasible in low-resource settings.

Aim

To develop practice points for healthcare workers that optimise lymphoedema care in low- and middle-income countries (LMIC).

Methods

A nominal group technique (NGT) was undertaken to gain consensus on which content from HIC guidelines was important and feasible to include in practice points for LMIC, and other important advice or recommendations. Participants included experts, clinicians, and volunteers involved in lymphoedema care in LMIC. The NGT followed five key stages: silent ‘ideas’ generation, round-robin rationale, clarification, refinement and verification. The first, fourth and fifth stages were completed via email, and the second and third during a video meeting in order to generate a series of consensus based prevention, assessment, diagnosis, and management of lymphoedema in LMIC practice points.

Results

Of sixteen participants invited, ten members completed stage 1 of the NGT (ideas generation), of whom six contributed to stages 2 (round-robin) and 3 (clarification). All those who completed stage 1 also completed stages 4 (refinement) and 5 (verification). Practice points unanimously agreed on included Complex Decongestive Therapy (CDT) and good skin care, with management to be determined by lymphoedema stage. For podoconiosis-endemic areas, the use of socks and shoes was identified as very important in the prevention of non-filarial lymphoedema and other lymphoedema-causing conditions. Participants indicated that diagnosing lymphoedema using the lymphoscintigraphy and Indocyanine green (ICG) fluorescent lymphography was not possible due to unavailability and cost in LMIC. Surgical procedures for lymphoedema management were unanimously eliminated due to the unavailability of technology, limited workforce, and expensive cost in LMIC.

Conclusion

The consensus-based practice points generated by this project provide healthcare workers with guidance on caring for people with lymphoedema in LMIC. Further development of workforce capacity is needed.

Background

Lymphoedema is a chronic and debilitating condition, often referred to as the “forgotten complication” because it can be mistaken for oedema, venous insufficiency or heart disease [1,2,3]. Globally, more than 200 million people are affected by lymphoedema [4], the majority of whom live in low- and middle-income countries (LMIC). The condition impacts negatively on quality of life, interferes with activities of daily living and ability to work, and is traumatic for the individuals affected and their families, lymphoedema alters their “sense of identity” and reduces their ability to participate in social events [5, 6].

Compared to people in high-income countries (HIC), those living in LMIC are often diagnosed at a later stage and with more advanced forms of lymphoedema [7]. They also have fewer treatment options, which increases their risk of developing complications and poorer health [8]. Unlike in HIC where cancer treatment is the major cause of lymphoedema, the most common cause of lymphoedema in LMIC is infectious lymphatic filariasis, while others suffer from the “swollen limb syndrome” [9, 10]. The World Health Organisation (WHO) reported that there has been a rapid reduction (74%) in lymphatic filariasis since the beginning of the ‘Global Programme to Eliminate Lymphatic Filariasis’ in 2000, limiting the impact of the disease in most areas [9, 11]. However, lymphoedema from other causes such as cancer, podoconiosis, idiopathy and trauma remains a major problem.

A recent systematic review [12] identified ten lymphoedema guidelines for HIC but only one for LMIC: the WHO Wound and Lymphoedema Management [13], which focuses exclusively on lymphoedema caused by infectious filariasis and wounds, and has very limited content on diagnosis and assessment. Unfortunately, recommendations in HIC guidelines are not always transferable to LMIC, especially when access to specialist services is limited and healthcare workers lack high levels of expertise to provide optimal care [14]. In many LMIC, awareness of lymphoedema by the general public is limited and those affected are often stigmatised due to cultural beliefs [15]. Finally, financial constraints for many people in LMIC negatively affect their capacity to pay for medical supplies and professional support.

In the absence of robust evidence to guide the prevention, identification and management of lymphoedema in LMIC, ‘practice points’ can fill this gap and are often highly valued by health workers. Practice points are sets of recommendations that are developed through expert opinion when evidence is insufficient or outside the scope of systematic reviews [16].

The current research aimed to develop practice points for healthcare workers that optimise lymphoedema care in LMIC.

Methods

Design

A nominal group technique (NGT) using a formal consensus process, was adopted as an interpretative method for generating a series of lymphoedema care practice points [17, 18].

A NGT is a technique of interviewing where group members independently provide ideas without interrupting each other [19]. The technique emphasizes considering all participants' views equally and enhancing group dynamics [18]. The NGT has been used in the past to evaluate priorities for health services, and formulate policies both in treatment of health conditions including cancer and in other health settings [20, 21]. The group deliberation and discussion of specific topics used in NGT help to minimise misunderstanding on a topic and provide highly suitable responses [20]. Where evidence is limited like in the area of lymphoedema care in LMIC, NGT consensus provides a useful method in developing clinical practice points.

Participants and setting

To be eligible to contribute, participant needed to be: a) expert on the diagnosis, assessment and/or management of lymphoedema from any cause, as denoted by authorship on a relevant journal article and/or b) health professional or caregiver in a World Bank Group [22] LMIC in the ‘paid’ or voluntary capacity.

Recruitment strategy

Participants were sampled using two non-probabilistic methods: purposive and snowballing methods [23, 24]. Initial approach was via email invitation to authors of relevant journal articles identified by a systematic review of lymphoedema studies in LMIC [25], personal and professional networks, and through organizations with an interest in lymphoedema care in LMIC, as well as to participants of previous research on lymphoedema care in LMIC [26].

Email invitations to individuals were followed up with a reminder after a week, to ensure that people had ample opportunity to respond without unduly ‘pestering’ them. A non-response to the follow up email was considered a decline to participate [27].

Participants who completed the first stage on the idea generation survey via email were invited to participate in stages 2 to 5.

Data collection

The NGT consensus was used to identify which key topic areas should be prioritised for simple and practical guidance on lymphoedema care for clinicians in LMIC. This work followed an adapted version of McMillan et al. [28] five NGT stages, as summarised below:

Stage 1

Stage 1 (silent generation) is an idea generation stage. Participants were emailed a list of topic areas from the HIC guidelines asking them to individually reflect on which ones they think are most important and feasible for LMIC as well as to add any further ideas they have for such guidance. Each participant returned their ideas to the first investigator (ET) for him to identify areas of agreement and disagreement.

Stage 2

In a ‘round robin’ stage, participants attending a meeting were asked to explain the rationale for their ‘top choice’ for inclusion in the clinician guidance. If a participant’s top choice had already been put forward by someone else, (s)he was invited to choose another. The round robin continued until everyone’s choices were exhausted. No group discussion or commentary by the facilitator occurred at this stage. This stage and stage 3 were completed at a video meeting facilitated by the second author (TL), with notes taken by ET.

Stage 3

The next stage focused on clarification of ideas via group discussion, in which participants were asked to identify areas of overlap or synergy between the selected topics to create themes, ensuring that the list was coherent and not repetitive. At this stage, participants could also agree to exclude or alter ideas, as well as generate new ideas. Feasibility in terms of cost for each recommendation was appraised subjectively by NGT participants based on their own experience rather than using a pre-specified monetary threshold.

Stage 4

In the fourth stage (refinement), the group choices from the list created at Stage 3 for each domain of the guidance document were sent via email to participants for verification. Participants were allowed to comment on the guidance document individually rather than as a group to ensure all participants’ opinions were captured. Where a practice point was suggested by a participant that was additional to those in the HIC guidelines, a Medline database search was conducted to check the evidence available either in support or against the suggestion. Where evidence was considered favourable or absent, the suggestion was put forward for discussion and consensus. Consensus was defined as a 100% agreement from all participants.

Recommendations were graded based on the systems as reported by the International Lymphoedema Framework [29]. Levels of evidence for each recommendation were defined by the relevant guideline(s) from which it was sourced. All of the guidelines except two [30, 31] used the grading system described by the International Lymphoedema Framework [27]:

• Grade A—clear research evidence obtained from meta-analysis or systematic review of randomized controlled trials;

• Grade B—limited supporting research evidence; and

• Grade C—experienced common sense judgement (consensus) [29].

Stage 5

In the final stage (verification), results from the fourth Stage were presented back to the group for discussion and consensus, again via email. Where a participant suggested a review, the new suggestion was made and returned via email to all participants for another round of verification until there were no more additional information. ET synthesised all the feedbacks and identified areas of agreement and consensus and presented the practice points for lymphoedema prevention, assessment, diagnosis and management.

Analysis

Analysis of Stage 1 data involved simple counts of the number of participants identifying each topic as important and/or feasible. Analysis for Stages 3 and 5 used a simple qualitative approach, with themes inductively derived from participant discussion and verified with participants then modified according to feedback [32]. At the end of Stage 4, descriptive statistics of median and range were used to indicate consensus among participants for each topic based on survey results [33, 34].

Ethical considerations

Projects building consensus among experts are not required to obtain formal ethical approval according to policy at the University of Technology Sydney provided participants agree to be identified on reports. It was made clear during recruitment that participation was voluntary for ethical consideration, and that completion of Stage 1 and/or attendance at the meeting for Stages 2 to 5 would be taken as evidence for consent. Participation included an understanding that names would be published, in accordance with common practice for consensus studies [35]. However, each participant’s responses to the survey remained anonymous. They also reserved the right to discontinue participation at any time. These terms were repeated at commencement of the meeting.

The NGT consensus was conducted between December 2021 and May 2022.

Out of the sixteen participants invited, ten completed Stage 1 (idea generation), and six attended the meeting to complete Stages 2 (round-robin) and 3 (clarification) and reach a consensus on contents for inclusion in the practice points. Participants were representatives from the disciplines of physiotherapy (n = 4), nursing (n = 1), and medicine (n = 1). All participants had more than ten years of experience of providing lymphoedema care in LMIC. All experts also had advanced education (masters or doctorate), and were affiliated to either a higher academic, research and/or health institution. The experts had expertise in providing care in the following LMIC; Bangladesh, Brazil, Cooks Islands, Egypt, Ethiopia, Ghana, Haiti, India, Ivory Coast, Papua New Guinea, and the Solomon Islands.

Consensus

Consensus was reached on twenty-five of the thirty-three items included in stages 2–5 of the NGT. The highest level of consensus was reached for lymphoedema prevention, diagnosis and non-surgical management (Refer to Tables 1 and 2 for outcome of Stage 1 of the NGT consensus), while no consensus was achieved for the surgical management of lymphoedema in LMIC. There were very low levels of support for promoting costly preventative, diagnostic or surgical interventions that required specialist equipment or capabilities. Diagnostic techniques such as indocyanine green, bioimpedance spectroscopy and lymphoscintigraphy along with the surgical procedures recommended in HIC, such as liposuction, lymphovenous anastomosis and vascularised lymph node transfer were all excluded from the lymphoedema practice points for LMIC.

Table 1 Outcome of Stage 1 of the NGT consensus on lymphoedema prevention and diagnosis

Table 2 Outcome of Stage 1 of the NGT consensus on surgical and non-surgical management

Results

Across prevention, diagnosis, assessment and management, eighteen practice points were recommended, each with accompanying key areas for the simple, practical guidance, as summarised below and detailed in Table 3. A list of societies and organisations guidelines used in the development of the practice points have been provided in Supplementary Table 1 with their web pages.

Table 3 Practice points: lymphoedema prevention, diagnosis, non-surgical and surgical management in LMIC

Of the eighteen practice points developed, five were generated under lymphoedema prevention, six under assessment, two under lymphoedema diagnosis and four under management.

Discussion

This consensus process has generated eighteen affordable and feasible practice points to support better lymphoedema care in LMIC. These practice points detail the actions LMIC health care professionals can readily implement to prevent lymphoedema, ensure it is diagnosed early, and manage the condition to prevent or reduce disability and unnecessary suffering and optimise the person’s quality of life. The NGT provided a quick and efficient method of group decision-making and ensured consensus was reached on important topics for practice point generation.

Practice points for prevention require a whole of sector approach to increase community awareness and education regarding lymphoedema, and the importance of meticulous skin care. Drawing on the Ottawa Charter, the three main health promotion strategies required to prevent lymphoedema in LMIC are: 1) advocacy to create the essential conditions to prevent lymphoedema; 2) enabling all people to achieve their full health potential; and 3) mediating between the different interests in society in the pursuit of health [45, 46]. Positive public health policies are needed to increase community awareness of lymphoedema including strengthening community action against lymphoedema [46]. Combatting lymphoedema related stigma requires the creation of an environment free from all forms of prejudice [46].

People at risk of lymphoedema, need to be provided with the skills and resources required to implement simple prevention interventions like meticulous skin care, including hand-hygiene and wearing shoes. A recent meta-analysis of lymphoedema interventional studies found that education focused on hygiene-based techniques in lymphatic filariasis-endemic areas prevented the transmission of lymphatic filariasis through proper hand washing [47], which is equally relevant to people with cancer related and other forms of lymphoedema. For the above actions lymphoedema prevention strategies to be effective, the health systems and services of LMIC require a re-orientation – an effort which should be welcomed by all levels of care (individual, community and health service). Similar to the findings of a previous research on lymphoedema care in LMIC, this NGT consensus identified that increasing community and healthcare workers awareness on lymphoedema was important first step to improving lymphoedema care outcomes in LMIC [26].

Complex decongestive therapy (CDT) is the mainstay of lymphoedema management and is critical to improving function for people living with lymphoedema, regardless of the setting. This involves manual mobilising and drainage of fluid, with meticulous skin care, which ensures a well-moisturised, healthy, clean and intact skin [48].

Practice points for lymphoedema diagnostic techniques generated through our NGT involve comparing circumferential or limb volume measurement of affected and unaffected limbs in unilateral limb lymphoedema or undertaking a baseline measurement of the affected body part. While other diagnostic techniques such as lymphoscintigraphy and Indocyanine green (ICG) florescent lymphography are effective, their prohibitive costs and limited availability makes them unsuitable for use in most LMIC [49]. It is important to promote the less expensive and more straightforward lymphoedema diagnostic methods, such as the use of the volumetric measurement, circumferential measurement as well as palpation in identifying lymphoedema suitable for use in LMIC.

Accurate diagnosis of lymphoedema was one of the major findings of our NGT consensus. Similar to the recommendation of the WHO Wound and Lymphoedema Management [13], accurate diagnosis of lymphoedema requires a detailed history, physical examination and correct measurement of limb volumes. Healthcare workers in LMIC should be taught appropriate ways of confirming if 10% of the affected limb is affected by lymphoedema (positive diagnosis) [50] so that they are able to accurately measure and interprete lymphoedema volume measurements.

Practice points generated for lymphoedema assessment include detailed subjective (history taking) and objective assessments. According to Svensson et al. [51], lymphoedema assessment should involve performing a detailed report of presenting signs and symptoms, which were consistent with our study findings that the assessment should include performing Stemmer’s test and physical examination. Despite the bioimpedance spectroscopy is widely known for measuring total fluid volume, our findings indicate that it is expensive, and not readily available for lymphoedema assessment in LMIC.

Another important finding of this study is staging of the lymphoedema. Participants indicated that for proper treatment outcomes, lymphoedema should be classified according to the stages as presented by the International Society of Lymphology [8]. Lymphoedema staging is vital for diagnosing and useful in implementing treatment interventions. A comparative study of the similarities and differences of lymphoedema between HIC and LMIC observed that healthcare workers in both countries are unaware of the disease classification system [14]. It is recommended that healthcare workers will put in efforts to understand the staging system and to implement it in the assessment and diagnosing of lymphoedema in LMIC to improve care for people living with lymphoedema.

In endemic areas of lymphatic filariasis, participants recommended that an ICT be performed. The ICT is a simple and fast method used in detecting Wuchereria bancrofti infections responsible for lymphoedema caused by lymphatic filariasis [52]. It is advised that, healthcare workers should request for ICT in cases of podoconiosis to rule out any infection of lymphatic filariasis and to aid early identification and treatment. A review of studies on non-filarial lymphoedema or podoconiosis in Africa indicated that a combination of both laboratory test and geochemical investigations are important in understanding the pathogenesis of the condition, and subsequently developing mechanisms to eradicate lymphoedema [53].

Our NGT confirmed that lymphoedema management in LMIC requires a set of coordinated effort from the multidisciplinary team of healthcare workers. Main management techniques identified in this NGT include meticulous skin care, exercise, modified lymphatic massage, and compression therapy (wraps) based on the stage of the condition. These were not different from CDT, the key management strategy for lymphoedema [31]. The main difference in LMIC is that specialist skills may be limited, and some resources may not be available for its implementation. Based on this factor, experts of our NGT suggest the use of improvised materials such as the wraps and simple treatment modalities. Additionally, this NGT consensus indicated that the management of lymphoedema should involve proper foot and skin hygiene, and wearing of appropriate foot wears in podoconiosis endemic areas. Where appropriate, lymphoedema treatment should include pelvic floor muscle exercises for people living with genital lymphoedema.

Despite the importance of surgical procedures such as lymphaticovenular anastomosis or vascularised lymph node transfer which increase limb volumes [54], these procedures require well-trained surgeons and experienced healthcare workers which are inaccessible in LMIC due to a lack of suitable facilities or cost. It is hoped that, with the advance of technology and health systems globally, LMIC will be able to secure appropriate facilities and skills to help optimise conditions for surgery. A considerable strength of these lymphoedema care practice points are the use of an international multi-disciplinary consensus process and the development of guidance that is cost-effective and feasible for all health professionals in LMIC to apply.

Limitations

Our inability to identify and include a larger number of healthcare workers providing lymphoedema care in LMIC is a limitation. However, the participants who contributed were lymphoedema experts with diverse experience across most LMIC regions.

Another limitation was not specifying specific management for various etiologies including primary, genetic or secondary lymphoedema. Instead, we provided practice points suitable for treating all kinds of lymphoedema no matter the stage of the condition. Further research should consider providing targeted treatments for genetic and non-genetic causes of lymphoedema. Finally, there is likely to have been a sampling bias and we were not able to ascertain reasons for non-response to invitations to take part in the NGT.

Implications for future research

Ideally, the practice points developed through the consensus process reported in this paper should be evaluated in research, with the gold standard being randomised controlled trials.

Implementation of these practice points will also need a concerted effort that should be underpinned by research aimed at evaluating their impacts and informing similar efforts for other guidance in the future. According to the Guidelines International Network, new guidelines should be accompanied by campaigns aimed at raising awareness, taking into account cultural and social contexts of the LMIC in which they will be implemented. Practice points should also be made readily accessible to all healthcare workers through open access journals and websites of non-governmental agencies that focus on LMIC healthcare delivery.

Lymphoedema education for healthcare workers could not be underestimated in the prevention of lymphoedema in LMIC. However, most healthcare workers lack appropriate knowledge and professional information required for lymphoedema management of people living with lymphoedema. It is appropriate that, in-service training and structured workshops with focus on increasing healthcare workers knowledge on lymphoedema be instituted to support care in LMIC.

Conclusion

Healthcare workers require increased knowledge and awareness to improve lymphoedema care in LMIC. Eighteen practice points have been developed to optimise lymphoedema care in LMIC. Key lymphoedema care strategies identified in this NGT consensus include meticulous skin care, exercise, modified lymphatic massage, and use of compression bandages or wraps. These practice points require dissemination to enhance lymphoedema care and improve clinical practice on management in LMIC.