Resource overutilization in the diagnosis of lymphedema praecox.

PURPOSE: Primary lymphedema presenting in adolescence is known as lymphedema praecox. Older children presenting with leg swelling are often subjected to a myriad of diagnostic tests. The purpose of this study is to review a large-cohort of patients with lymphedema praecox to determine the fiscal impact of diagnostic testing on these patients. METHODS: A 13-year review was performed of patients with lymphedema praecox. Information was obtained on demographic parameters, diagnostic studies performed, and clinical outcomes. RESULTS: Forty-nine patients were identified. The median age was 14 (range: 7-21) years. Participants were predominantly female (n = 40, 81.6%). 19 patients had bilateral disease and 30 had unilateral disease. The diagnosis was made on clinical exam only in 14 patients. 35 patients had imaging which consisted of plain X rays, Doppler ultrasound (DUS), lymphoscintigraphy (LSG) or MRI as the sole imaging study (n = 28) or in combination with others (n = 7). The charges for plain X-rays, DUS, LSG, and MRI with contrast were $335, $1715, $1269, and $6006 respectively. CONCLUSION: We believe that in the adolescent female with physical findings consistent with lymphedema praecox, diagnostic imaging should be limited to a Doppler ultrasound to rule out a secondary cause of the swelling. LEVEL OF EVIDENCE: IV TYPE OF EVIDENCE: Case series with no comparison group.

Superior Clinical, Quality of Life, Functional, and Health Economic Outcomes with Pneumatic Compression Therapy for Lymphedema.

BACKGROUND: Pneumatic compression therapy is one of several options for the management of lymphedema. The lack of clarity around clinical outcomes, quality of life, cost of care, and its proper application, as a function of lymphedema complexity, limit its use in clinical practice. This is compounded by difficulties associated with insurance approval and uncertainty about the role of this modality in the treatment algorithm. The purpose of this study is to elucidate the healthcare economics and value of pneumatic compression therapy for lymphedema. METHODS: All patients who underwent treatment for lymphedema at a single institution were followed prospectively over a 2-year period. Patient demographics, comorbidities, treatment modality, and treatment efficacy were determined. Direct costs over the 2-year period, inclusive of hospitalization and device costs, SF-36 quality of life, and leg lymphedema complexity score (LLCS), were measured. RESULTS: A total of 128 patients were enrolled over a period of 3 years for a total of 232 extremities treated for secondary lymphedema. Pneumatic compression therapy was utilized for all patients and led to a 28% decrease in absolute limb volume (P < 0.001), decrease in body mass index (BMI) (P < 0.001), significant improvement in SF-36 quality of life in 7 out of 8 domains (P < 0.001), and a significant improvement in LLCS (P < 0.001) at 1 year. A subsequent decrease in hospitalization for lymphedema-associated complications saved over $3,200 per patient per year. CONCLUSIONS: Pneumatic compression therapy leads to improved clinical outcomes, quality of life, and functional status for clinically significant lymphedema. Significant per capita direct cost savings, a beneficial impact on pay for performance measures, and a reduction in lymphedema-related complications suggest that earlier adoption of this treatment modality may offer a superior value proposition to patients, physicians, hospitals, and the healthcare system.

Lymphoedema management by independent hospices: a cohort study.

OBJECTIVES: To consider the type and cost of clinical services delivered for patients with lymphoedema. DESIGN: Clinical cohort. SETTING: Independent hospices in the North East of England. PARTICIPANTS: All those attending lymphoedema services delivered by the independent hospice sector 2017/2018. RESULTS: 13 914 lymphoedema appointments were recorded across four independent hospices. Twelve thousand nine hundred and sixty-five were attended, which equates to an approximate cost of £1.56 million. Those with lymphoedema were predominately aged over 65 (54.5%) years with females across all age groups being more predominant (3.3:1). Where the cause was recorded, 66% of activity related to lymphoedema was not secondary to cancer. CONCLUSION: Independent hospices are providing a specialist lymphoedema service, which is high in volume and largely invisible. This service is delivered at not insignificant cost. In contrast to previous work, in the North East of England, lymphoedema sufferers are more likely to be female and not have the condition in association with cancer. The availability of rigorous data collection will allow the independent hospices to understand better the delivery and associated costs of lymphoedema services.

Critical examination of skin care self-management in lymphoedema.

Lymphoedema is a long-term chronic condition that results from lymphatic insufficiency and may cause skin changes, which can cause discomfort and impact patient' quality of life. The four cornerstones of lymphoedema management are exercise, lymphatic massage, compression and skin care. This article critically examines self-management in lymphoedema focusing on skin care. Patients may initially demonstrate enthusiasm to undertake the required skin care independently. However, psychosocial factors, such as financial burden and physical restraints, can affect motivation and behaviours over time. A patient's knowledge of the importance of skin care self-management should not be limited to the initial assessment, but should be continually assessed throughout their lymphoedema journey. The ongoing provision of patient-centred care may have psychological and behavioural benefits and help optimise skin care self-management.

Cost-effectiveness and social outcomes of a community-based treatment for podoconiosis lymphoedema in the East Gojjam zone, Ethiopia.

BACKGROUND: Podoconiosis is a disease of the lymphatic vessels of the lower extremities that is caused by chronic exposure to irritant soils. It results in leg swelling, commonly complicated by acute dermatolymphangioadenitis (ADLA), characterised by severe pain, fever and disability. METHODS: We conducted cost-effectiveness and social outcome analyses of a pragmatic, randomised controlled trial of a hygiene and foot-care intervention for people with podoconiosis in the East Gojjam zone of northern Ethiopia. Participants were allocated to the immediate intervention group or the delayed intervention group (control). The 12-month intervention included training in foot hygiene, skin care, bandaging, exercises, and use of socks and shoes, and was supported by lay community assistants. The cost-effectiveness analysis was conducted using the cost of productivity loss due to acute dermatolymphangioadenitis. Household costs were not included. Health outcomes in the cost-effectiveness analysis were: the incidence of ADLA episodes, health-related quality of life captured using the Dermatology Life Quality Index (DLQI), and disability scores measured using the WHO Disability Assessment Schedule 2.0 (WHODAS 2.0). RESULTS: The cost of the foot hygiene and lymphoedema management supplies was 529 ETB (69 I$, international dollars) per person per year. The cost of delivery of the intervention as part of the trial, including transportation, storage, training of lay community assistants and administering the intervention was 1,890 ETB (246 I$) per person. The intervention was effective in reducing the incidence of acute dermatolymphangioadenitis episodes and improving DLQI scores, while there were no significant improvements in the disability scores measured using WHODAS 2.0. In 75% of estimations, the intervention was less costly than the control. This was due to improved work productivity. Subgroup analyses based on income group showed that the intervention was cost-effective (both less costly and more effective) in reducing the number of acute dermatolymphangioadenitis episodes and improving health-related quality of life in families with monthly income <1,000 ETB (130 I$). For the subgroup with family income ≥1,000 ETB, the intervention was more effective but more costly than the control. CONCLUSIONS: Whilst there is evident benefit of the intervention for all, the economic impact would be greatest for the poorest.

Drivers of cost differences between US breast cancer survivors with or without lymphedema.

PURPOSE: Breast cancer-related lymphedema is an adverse effect of breast cancer surgery affecting nearly 30% of US breast cancer survivors (BCS). Our previous analysis showed that, even 12 years after cancer treatment, out-of-pocket healthcare costs for BCS with lymphedema remained higher than for BCS without lymphedema; however, only half of the cost difference was lymphedema-related. This follow-up analysis examines what, above and beyond lymphedema, contributes to cost differences. METHODS: This mixed methods study included 129 BCS who completed 12 monthly cost diaries in 2015. Using Cohen's d and multivariable analysis, we compared self-reported costs across 13 cost categories by lymphedema status. We elicited quotes about specific cost categories from in-person interviews with 40 survey participants. RESULTS: Compared with BCS without lymphedema, BCS with lymphedema faced 122% higher mean overall monthly direct costs ($355 vs $160); had significantly higher co-pay, medication, and other out-of-pocket costs, lower lotion costs; and reported inadequate insurance coverage and higher costs that persisted over time. Lotion and medication expenditure differences were driven by BCS' socioeconomic differences in ability to pay. CONCLUSIONS: Elevated patient costs for BCS with lymphedema are for more than lymphedema itself, suggesting that financial coverage for lymphedema treatment alone may not eliminate cost disparities. IMPLICATIONS FOR CANCER SURVIVORS: The economic challenges examined in this paper have long been a concern of BCS and advocates, with only recent attention by policy makers, researchers, and providers. BCS identified potential policy and programmatic solutions, including expanding insurance coverage and financial assistance for BCS across socioeconomic levels.

Out-of-pocket payments, vertical equity and unmet medical needs in France: A national multicenter prospective study on lymphedema.

Out-of-pocket payments might threaten the vertical equity of financing and generate unmet medical needs. The main objective was to assess the vertical equity of outpatient out-of-pocket payments for lymphedema patients in France. Twenty-seven centres, among which 11 secondary care hospitals and 16 primary care practices participated in this prospective national multicenter study. We measured the lymphedema-specific outpatient out-of-pocket payments over 6 months. The vertical equity of out-of-pocket payments was examined using concentration curves, the Gini coefficient for income, the Kakwani index, and the Reynolds-Smolensky index. We included 231 lymphedema patients aged 7 years or more, living in metropolitan France, and being able to use Internet and email. After voluntary health insurance reimbursement, the mean out-of-pocket payment was equal to 101.4 Euros per month, mainly due to transport (32%) and medical devices (26%). Concentration curves indicated regressivity of out-of-pocket payments. Total out-of-pocket payments represented 10.1% of the income by consumption unit for the poorest quintile and 3.5% for the wealthiest (p<0.05). The Kakwani index for out-of-pocket payments was equal to -0.18. Regarding outpatient health care, French lymphedema patients face significant and regressive out-of-pocket payments, associated with an increased risk of unmet medical needs. Such results shed light on significant socioeconomic inequalities and bring into question the current financing arrangements of outpatient health care in France. Trial registration: ClinicalTrials.gov ID: NCT02988479.

LIMPRINT in Australia.

Background and Study Objective: Australia was one of nine participating countries in the epidemiology Phase II Lymphoedema Impact and Prevalence - International (LIMPRINT) project to determine the number of people with chronic edema (CO) in local health services. Methods and Results: Data collection occurred through questionnaire-based interviews and clinical assessment with provided LIMPRINT tools. Four different types of services across three states in Australia participated. A total of 222 adults participated with an age range from 22 to 102 years, and 60% were female. Site 1 included three residential care facilities (54% of participants had swelling), site 2 was community-delivered aged care services (24% of participants had swelling), site 3 was a hospital setting (facility-based prevalence study; 28% of participants had swelling), and site 4 was a wound treatment center (specific patient population; 100% of participants had swelling). Of those with CO or secondary lymphedema, 93% were not related to cancer, the lower limbs were affected in 51% of cases, and 18% of participants with swelling reported one or more episodes of cellulitis in the previous year. Wounds were identified in 47% (n = 105) of all participants with more than half of those with wounds coming from the dedicated wound clinic. Leg/foot ulcer was the most common type of wound (65%, n = 68). Conclusions: Distances between services, lack of specialized services, and various state funding models contribute to inequities in CO treatment. Understanding the high number of noncancer-related CO presentations will assist health services to provide timely effective care and improve referral pathways.

LIMPRINT: A Sociological Perspective on "Chronic Edema".

Background: Chronic edema is a condition that is biologically complex, distressing for patients and sociopolitically weak. Like many other complex and chronic conditions, it has a low status within health care. The result is that it has a low priority in health policy and consequently is undervalued and undertreated. While evidence-based practice promotes a hierarchy of evidence, it is also the case that clinical practice is influenced by a hierarchy of social status. These are as much political as they are scientific. Methods and Results: This article will provide an explanation for why chronic edema is a low priority. It will do this through a critical review of the literature. We examine this through the theoretical lens of Pierre Bourdieu. The sociology of Bourdieu frames an understanding of power relations through habitus, field, and capital. We will employ these theoretical tools to understand the way that chronic edema is situated within the policy arena. We identify a number of social mechanisms that affect the status of chronic edema, including diagnostic uncertainty, social capital, scientific capital, cultural capital and economic capital. Conclusion: We argue that a whole system approach to care, based on human need rather than unequal power relations, is a prerequisite for the delivery of good health care. The specialty of chronic edema is not a powerless group and we identify some of the ways that the social mechanism that acts as barriers to change, can also be employed to challenge them.

Lymphedema Impact and Prevalence International Study: The Canadian Data.

Background: Chronic edema/lymphedema is defined as edema present for more than 3 months. It is underrecognized and undertreated. The International Lymphedema Framework developed an international study, Lymphedema Impact and Prevalence International (LIMPRINT), to estimate the prevalence and impact of chronic edema in heterogeneous populations. Canada participated in this study. Methods and Results: Participants were recruited from an outpatient chronic wound management clinic. At a study visit, the following tools were administered: The Core Tool, Demographics and Disability assessment (WHODAS 2.0), Quality-of-life assessment (LYMQOL + EQ-5D), Details of swelling, Wound assessment, and Cancer. Data were entered into an international database (Clindex), and country-specific data were analyzed. Sixty-eight subjects were enrolled. Fifty-seven percent were males and 43% females. More than 90% were older than 45 years. Only 7.35% had primary lymphedema. Most had lower extremity edema (65 of 68). Over half (47.06%) were morbidly obese with body mass index of >40. The most common underlying condition was venous disease. Only 8 of 68 had a history of cancer. While 72.06% had a history of cellulitis, only 10.2% had been hospitalized in the past year. 39.71% had an open wound. More than 75% had received multilayer bandaging, compression garments, wound dressings, and extensive counseling. Few had received manual lymphatic drainage, which is not funded. Disability was less than expected. Conclusion: Chronic edema/lymphedema is an underrecognized condition. These data and the wider LIMPRINT study are important tools to advocate for wider recognition and funding of treatment by health care systems.

The social, physical and economic impact of lymphedema and hydrocele: a matched cross-sectional study in rural Nigeria.

BACKGROUND: Lymphatic filariasis (LF) is a mosquito-borne parasitic disease and a major cause of disability worldwide. To effectively plan morbidity management programmes, it is important to estimate disease burden and evaluate the needs of patients. This study aimed to estimate patient numbers and characterise the physical, social and economic impact of LF in in rural Nigeria. METHODS: This is a matched cross-sectional study which identified lymphedema and hydrocele patients with the help of district health officers and community-directed distributors of mass drug administration programmes. A total of 52 cases were identified and matched to 52 apparently disease-free controls, selected from the same communities and matched by age and sex. Questionnaires and narrative interviews were used to characterise the physical, social and economic impact of lymphedema and hydrocele. RESULTS: Forty-eight cases with various stages of lower limb lymphedema, and 4 with hydrocele were identified. 40% of all cases reported feeling stigma and were 36 times (95% CI: 5.18-1564.69) more likely to avoid forms of social participation. Although most cases engaged in some form of income-generating activity, these were low paid employment, and on average cases spent significantly less time than controls working. The economic effects of lower income were exacerbated by increased healthcare spending, as cases were 86 times (95% CI: 17.48-874.90) more likely to spend over US $125 on their last healthcare payment. CONCLUSION: This study highlights the importance of patient-search as a means of estimating the burden of LF morbidity in rural settings. Findings from this work also confirm that LF causes considerable psychosocial and economic suffering, all of which adversely affect the mental health of patients. It is therefore important to incorporate mental health care as a major component of morbidity management programmes.

"It still affects our economic situation": long-term economic burden of breast cancer and lymphedema.

PURPOSE: Financial toxicity after breast cancer may be exacerbated by adverse treatment effects, like breast cancer-related lymphedema. As the first study of long-term out-of-pocket costs for breast cancer survivors in the USA with lymphedema, this mixed methods study compares out-of-pocket costs for breast cancer survivors with and without lymphedema. METHODS: In 2015, 129 breast cancer survivors from Pennsylvania and New Jersey completed surveys on demographics, economically burdensome events since cancer diagnosis, cancer treatment factors, insurance, and comorbidities; and prospective monthly out-of-pocket cost diaries over 12 months. Forty participants completed in-person semi-structured interviews. GLM regression predicted annual dollar amount estimates. RESULTS: 46.5% of participants had lymphedema. Mean age was 63 years (SD = 8). Average time since cancer diagnosis was 12 years (SD = 5). Over 98% had insurance. Annual adjusted health-related out-of-pocket costs excluding productivity losses totaled $2306 compared to $1090 (p = 0.006) for those without lymphedema, or including productivity losses, $3325 compared to $2792 (p = 0.55). Interviews suggested that the cascading nature of economic burden on long-term savings and work opportunities, and insufficiency of insurance to cover lymphedema-related needs drove cost differences. Higher costs delayed retirement, reduced employment, and increased inability to access lymphedema care. CONCLUSIONS: Long-term cancer survivors with lymphedema may face up to 112% higher out-of-pocket costs than those without lymphedema, which influences lymphedema management, and has lasting impact on savings and productivity. Findings reinforce the need for actions at policy, provider, and individual patient levels, to reduce lymphedema costs. Future work should explore patient-driven recommendations to reduce economic burden after cancer.

Health and economic benefits of advanced pneumatic compression devices in patients with phlebolymphedema.

OBJECTIVE: Phlebolymphedema (chronic venous insufficiency-related lymphedema) is a common and costly condition. Nevertheless, there is a dearth of evidence comparing phlebolymphedema therapeutic interventions. This study sought to examine the medical resource utilization and phlebolymphedema-related cost associated with Flexitouch (FLX; Tactile Medical, Minneapolis, Minn) advanced pneumatic compression devices (APCDs) relative to conservative therapy (CONS) alone, simple pneumatic compression devices (SPCDs), and other APCDs in a representative U.S. population of phlebolymphedema patients. METHODS: This was a longitudinal matched case-control analysis of deidentified private insurance claims. The study used administrative claims data from Blue Health Intelligence for the complete years 2012 through 2016. Patients were continuously enrolled for at least 18 months, diagnosed with phlebolymphedema, and received at least one claim for CONS either alone or in addition to pneumatic compression (SPCDs or APCDs). The main outcomes included direct phlebolymphedema- and sequelae-related medical resource utilization and costs. RESULTS: After case matching, the study included 86 patients on CONS (87 on FLX), 34 on SPCDs (23 on FLX), and 69 on other APCDs (67 on FLX). Compared with CONS, FLX was associated with 69% lower per patient per year total phlebolymphedema- and sequelae-related costs net of any pneumatic compression device-related costs ($3839 vs $12,253; P = .001). This was driven by 59% fewer mean annual hospitalizations (0.13 vs 0.32; P < .001) corresponding to 82% lower inpatient costs and 55% lower outpatient hospital costs. FLX was also associated with 52% lower outpatient physical therapy and occupational therapy costs and 56% lower other outpatient-related costs. Compared with SPCDs, FLX was associated with 85% lower total costs ($1153 vs $7449; P = .008) driven by 93% lower inpatient costs ($297 vs $4215; P = .002), 84% lower outpatient hospital costs ($368 vs $2347; P = .020), and 85% lower other outpatient-related costs ($353 vs $2313; P = .023). Compared with APCDs, FLX was associated with 53% lower total costs ($3973 vs $8436; P = .032) because of lower outpatient costs and lower rates of cellulitis (22.4% vs 44.9% of patients; P = .02). CONCLUSIONS: This analysis indicates significant benefits attributable to FLX compared with alternative compression therapies that can help reduce the notable economic burden of phlebolymphedema.

Community-Based Treatment for Chronic Edema: An Effective Service Model.

BACKGROUND: Chronic edema care is patchy and of variable quality internationally. This study was undertaken to develop and evaluate a system of care that would provide for patients within a geographical area of London (Wandsworth), United Kingdom. METHODS AND RESULTS: A prospective cohort design with intervention of a new service design following a 6-month baseline period. Patients were identified through health professionals. A stratified random sample was drawn from all patients and an implementation strategy developed. Clinical assessment combined with questionnaires evaluated clinical, patient, and health service outcomes at 6-month periods. In all, 312 patients were identified in community and acute services giving a crude ascertainment rate of 1.16 per 1000 population. The random sample of 107 was mostly female (82%) with mean (standard deviation) age of 72.9 (12.4) in men and 68.6 (15.0) years in women. Mean reductions in limb volume achieved statistical differences at 6-12 months after implementation (difference [d] = 115 mL, p = 0.0001). Incidence of cellulitis dropped from 41.5/100 patient years at baseline to zero at 6-12 months. Quality of life showed greatest improvements between baseline and 6 months postimplementation, the largest differences being in role physical (d = 32.7, p = 0.0001) and role emotion (d = 24.0, p < 0.0001). EuroQol increased following implementation by a mean score of 0.05 (p = 0.007). There was a reduction in 6 monthly healthcare costs from £50171 per 100 patients at baseline to £17618 between 6 and 12 months. CONCLUSIONS: This process of implementation improves health outcomes while reducing healthcare costs in patients with lymphedema.

Evaluation of the economic impact of a national lymphoedema service in Wales.

Lymphoedema Network Wales has focused on maximising the impact of its service through the effective use of available resources to ensure high-quality and consistent care for people with lymphoedema across Wales. The aim of this evaluation was to estimate the economic impact of a national lymphoedema service on the NHS Wales budget. Work was undertaken to determine the care pathway within Lymphoedema Network Wales and develop a hypothetical 'world without' the service as a comparator. The four groups of patients that made up the pathways were group 0: 'at risk', group 1-2: 'uncomplicated lymphoedema', group 3: 'complicated/complex' and group 4: 'palliative care'. Overall resource utilisation between 6 months pre- and 6 months post-entry indicated that there were significant cost reductions to be seen after lymphoedema service entry for all patients in each group. This evaluation provides estimates that suggest that the service is likely to be cost saving when people with lymphoedema are managed within Lymphoedema Network Wales rather than in a 'world without' the service.

Cost-of-illness of patients with lymphoedema.

BACKGROUND: Chronic lymphoedema is characterized by a continuous need for medical treatment, many comorbidities and impaired quality of life. In Germany, about 4.5 million patients are affected by lymphoedema. Thus, lymphoedema causes high direct and indirect costs, even more in case of complications such as erysipelas and ulcers. OBJECTIVE: The aim of this study was to determine the costs of illness of community lymphoedema patients living in the metropolitan area of Hamburg, Germany. METHODS: An observational cross-sectional study in patients with lymphoedema and combined lipolymphoedema of any origin was performed analysing direct and indirect costs for the patients, the statutory health insurance and society. RESULTS: In total, 348 patients (90.8% female) were examined and interviewed. The mean age of the patients was 57.3 ± 14.5 years. On average, the total costs per patient and year were € 5784, of which € 4445 (76.9%) were direct costs and € 1338 indirect costs. Within the direct medical costs, € 3796 were accounted for the statutory health insurances and € 649 for the patient. The main cost drivers were costs for manual decongestive therapy and disability costs. CONCLUSION: Chronic lymphoedema is associated with high direct and indirect costs. This community-based study is the first cost analysis of chronic lymphoedema and combined lipolymphoedema giving insights to economic impact of lymphoedema treatment. There is a high need for structured disease management programs in order to diagnose and treat lymphoedema early and to avoid complications, thus limiting socio-economic burden.

Financial cost of lymphedema borne by women with breast cancer.

OBJECTIVE: Our study examines the financial cost of lymphedema following a diagnosis of breast cancer and addresses a significant knowledge gap regarding the additional impact of lymphedema on breast cancer survivors. METHODS: An online national survey was conducted with 361 women who had either breast cancer without lymphedema (BC) (group 1, n = 209) or breast cancer with lymphedema (BC+LE) (group 2, n = 152). Participant recruitment was supported by the Breast Cancer Network Australia and the Australasian Lymphology Association. RESULTS: Both breast cancer and lymphedema result in significant out-of-pocket financial costs borne by women. Of patients with BC+LE, 80% indicated that their breast cancer diagnosis had affected them financially compared with 67% in the BC group (P < .020). For patients with lymphedema, over half (56%) indicated that this specific additional diagnosis to their breast cancer affected them financially and that costs increased with lymphedema severity. The cost of compression garments formed a large proportion of these costs (40.1%). The average number of attendances to a therapist each year was 5.8 (range, 0-45). Twenty-five patients (16.4%) had an episode of cellulitis in the past year. The incidence of cellulitis was 7.7% in 91 patients with subclinical or mild lymphedema compared with 29.5% of 61 patients with more extensive lymphedema (P < .001). The average out-of-pocket financial cost of lymphedema care borne by women was A$977 per annum, ranging from A$207 for subclinical lymphedema to over A$1400 for moderate or severe lymphedema. CONCLUSIONS: This study identifies an additional detrimental effect of lymphedema on women in terms of financial costs.