A Systematic Review of the Cost-Effectiveness of Cleft Care in Low- and Middle-Income Countries: What is Needed?

OBJECTIVE: The objective of this paper is to conduct a systematic review that summarizes the cost-effectiveness of cleft lip and/or palate (CL/P) care in low- and middle-income countries (LMICs) based on existing literature. DESIGN: We searched eleven electronic databases for articles from January 1, 2000 to December 29, 2020. This study is registered in PROSPERO (CRD42020148402). Two reviewers independently conducted primary and secondary screening, and data extraction. SETTING: All CL/P cost-effectiveness analyses in LMIC settings. PATIENTS, PARTICIPANTS: In total, 2883 citations were screened. Eleven articles encompassing 1,001,675 patients from 86 LMICs were included. MAIN OUTCOME MEASURES: We used cost-effectiveness thresholds of 1% to 51% of a country's gross domestic product per capita (GDP/capita), a conservative threshold recommended for LMICs. Quality appraisal was conducted using the Joanna Briggs Institute (JBI) checklist. RESULTS: Primary CL/P repair was cost-effective at the threshold of 51% of a country's GDP/capita across all studies. However, only 1 study met at least 70% of the JBI criteria. There is a need for context-specific cost and health outcome data for primary CL/P repair, complications, and existing multidisciplinary management in LMICs. CONCLUSIONS: Existing economic evaluations suggest primary CL/P repair is cost-effective, however context-specific local data will make future cost-effectiveness analyses more relevant to local decision-makers and lead to better-informed resource allocation decisions in LMICs.

Patient-Proxy and Societal Perspectives of Quality-of-Life Utilities in Children With Cleft Lip and Palate Managed With Surgical Repair vs No Repair in Ethiopia.

Importance: In Ethiopia, more than 70% of infants with cleft lip and/or palate (CL/P) lack access to surgery. Infants who are untreated can experience severe malnutrition and extreme social stigma resulting in abandonment. Utilities are standardized measures of health-related quality of life (HRQOL) that inform health care resource allocation. However, CL/P utilities are missing from low- and middle-income countries (LMICs). Objective: To elicit utilities for untreated and surgically treated children with CL/P with consideration for social determinants of health from patient-proxy and societal participants. Design, Setting, and Participants: This cross-sectional study used patient proxies and societal participants in Addis Ababa, Ethiopia, from July 1, 2019, to January 30, 2020. Eligible patient proxies were caregivers of children younger than 18 years with nonsyndromic CL/P who were untreated or received surgery. Proxies were necessary as most patients were 0 to 4 years old and cannot reliably self-report. Eligible societal participants were 18 years and older with no family history of CL/P. Exposures: Surgical treatment and social determinants of health. Main Outcomes and Measures: Participants measured utilities using the visual analog scale (VAS), time trade-off (TTO), and standard gamble (SG). Results: In this study, 312 patient proxies and 135 societal participants were recruited. Mean (SD) utilities for untreated CL/P ranged from 0.57 (0.23) to 0.70 (0.22) from patient proxies and from 0.35 (0.21) to 0.8 (0.23) from societal participants, depending on utility instrument and cleft type. Surgical treatment was associated with a better HRQOL from the patient-proxy perspective (VAS, 0.17; 95% CI, 0.09 to 0.26; TTO, 0.15; 95% CI, 0.05 to 0.25) from the societal perspective (VAS, 0.21; 95% CI, 0.16 to 0.26; TTO, 0.17; 95% CI, 0.13 to 0.22; SG, 0.11; 95% CI, 0.06 to 0.15). Social determinants of health that were associated with patient-proxy utilities were income above the national mean (VAS, 0.10; 95% CI, 0.02 to 0.17; TTO, 0.11; 95% CI, 0.02 to 0.20), and religion (Christian vs other: TTO, -0.10; 95% CI, -0.17 to -0.03). From the societal perspective, the association between treatment and utilities was smaller in females compared with males (TTO, -0.05; 95% CI, -0.10 to -0.01). Conclusions and Relevance: The findings of this study suggest that CL/P disease severity and surgical impact in Ethiopia were undervalued by previous estimates from high-income countries and were associated with social determinants of health. Utility studies from participants from LMICs are feasible and necessary for representing HRQOL in LMICs and addressing health inequalities.

Aromatase Inhibitor-Induced Carpal Tunnel Syndrome and Stenosing Tenosynovitis: A Systematic Review.

BACKGROUND: Although aromatase inhibitors are the first-line treatment in postmenopausal women with hormone receptor-positive breast cancer, there is increasing evidence that they can induce carpal tunnel syndrome and stenosing tenosynovitis. This systematic review summarizes the risk factors, incidence, and management for patients with aromatase inhibitor-induced carpal tunnel syndrome and stenosing tenosynovitis compared to tamoxifen or placebo. METHODS: A Preferred Reporting Items for Systematic Reviews and Meta-Analyses-guided systematic review of PubMed/MEDLINE, Ovid Embase, and the Cochrane Central Register of Controlled Trials was conducted (to March 19, 2020), supplemented with Google Scholar, Plastic and Reconstructive Surgery, and The Journal of Hand Surgery. Two reviewers independently completed the primary and secondary screens and the quality appraisal. RESULTS: This study reviewed 577 abstracts and included 19 studies. Risk factors for aromatase inhibitor-induced carpal tunnel syndrome or stenosing tenosynovitis included hormone replacement therapy before trial entry, history of musculoskeletal symptoms, age younger than 60 years, prior chemotherapy, and body mass index greater than 25 kg/m2. The incidence can be increased up to 10 times compared to tamoxifen. Patient discontinuation of aromatase inhibitor treatment because of carpal tunnel syndrome and stenosing tenosynovitis was reported. Nonsurgical management led to complete resolution of carpal tunnel syndrome symptoms in up to 67 percent of cases. Although most aromatase inhibitor-induced stenosing tenosynovitis original studies were low quality, all recommended surgical release for symptom resolution. CONCLUSIONS: This study provides current knowledge of the associated risk factors, management options, and quality of literature for aromatase inhibitor-induced carpal tunnel syndrome and stenosing tenosynovitis. Early recognition can prevent self-discontinuation of an aromatase inhibitor and long-term sequelae of poorly treated carpal tunnel syndrome and stenosing tenosynovitis.

Cost-Utility Analysis of Dolutegravir- Versus Efavirenz-Based Regimens as a First-Line Treatment in Adult HIV/AIDS Patients in Ethiopia.

BACKGROUND: In several countries, the dolutegravir (DTG)-based regimen is generally preferred as first-line antiretroviral therapy (ART) over the efavirenz (EFV)-based regimen, but the evidence in low-income countries is limited. OBJECTIVE: Our study aimed to evaluate the cost effectiveness of DTG- versus EFV-based first-line human immunodeficiency virus (HIV) treatment in Ethiopia. METHODS: We developed a microsimulation model for the progression of HIV/acquired immune deficiency syndrome (AIDS) to examine the cost effectiveness of DTG-based first-line ART compared with an EFV-based regimen from a healthcare payer perspective. We used a lifetime horizon with a 1-month cycle length and a 3% annual discount rate. The primary outcomes were a lifetime cost in US dollars ($), quality-adjusted life-months (QALMs) that converted to QALYs using the formula QALY = QALM/12, and incremental cost-effectiveness ratio (ICER). Deterministic sensitivity analysis was conducted to account for parameter uncertainty. RESULTS: Compared with the EFV-based regimen, the DTG-based regimen was associated with an expected lifetime cost of $12,709 (vs. $12,701) and expected QALYs of 15.3 (vs. 14.7 QALYs) per patient, resulting in an ICER value of $13.33 per QALY. From an alternative analysis with a 5-year time horizon, DTG-based ART was found to be dominant, with expected gains of 0.17 QALYs at a lower cost of $1 per patient. The deterministic sensitivity analysis depicted that the maximum increase in ICER value was $72 per QALY, and all ICER values were below the estimated threshold value. CONCLUSIONS: The DTG-based first-line regimen appears to be cost effective compared with the EFV-based regimen for the treatment of HIV/AIDS patients in an Ethiopian setting.

The Impact of Social Stigma for Children with Cleft Lip and/or Palate in Low-resource Areas: A Systematic Review.

There are still children with cleft lip and/or palate (CLP) in low-resource areas who face social rejection. This stigma disadvantages children in education, employment, marriage, and community, and is exacerbated by barriers to care. Our study objective was to conduct a systematic review of the impact of social stigma of CLP for children in low-resource areas. We followed the Preferred Reporting Items for Systematic Reviews and Meta-analyses guidelines. A systematic search was conducted of 3 databases: Ovid Embase, Ovid Medline, and the African Journal Online from 2000 to October 5 2018. Common themes were identified using a grounded theory approach and quantitatively summarized. The Joanna Briggs Institute criteria were used to evaluate the risk-of-bias assessments. Four hundred seventy-seven articles were screened; 15 articles were included that focused on the impact of social stigma on CLP in low-resource areas. This was limited to English articles. The majority of studies originated in Nigeria or India. Themes were reported as follows: societal beliefs (n = 9; 60%), social impact (n = 7; 46%), marriage (n = 7; 46%), education (n = 6; 40%), employment (n = 5; 33%), and psychological distress (n = 3; 20%). Causes include the effect of "God's will," supernatural forces, evil spirits or ancestral spirits, exposure to an eclipse, black magic, or a contagion. Further, children with CLP may not be worth a full name or considered human and killed. Awareness of the impact of social stigma for children with CLP in low-resource areas generates support toward national education and awareness in low-resource areas.

Innovate Global Plastic and Reconstructive Surgery: Cleft Lip and Palate Charity Database.

BACKGROUND: There is an emerging interest in global surgery. The Lancet Commission on Global Surgery recognizes the important role that nongovernmental organizations (NGOs) play in the delivery of cleft lip and/or palate (CLP) surgical care. To better address the unmet burden of surgical disease, the commissioners propose the use of a centralized registry to maximize coordination of global surgical volunteerism efforts. This study aims to create a comprehensive database of CLP organizations. METHODS: A systematic search of the following resources was conducted: The Plastic Surgery Foundation, Smile Train, Wikipedia, Google, and lists of surgical NGOs. A secondary review of each organization's website was performed to verify inclusion criteria and to extract data. Organizations were classified as providing surgical or nonsurgical care. RESULTS: Thirty-one organizations providing CLP care were reviewed, with 30 that met inclusion criteria. Of the 20 surgical NGOs, 50% use a diagonal approach of international outreach, 40% a vertical one-way approach, and 10% a horizontal approach. All 10 of the nonsurgical NGOs provide care through a horizontal approach. Their offices are distributed across North America (43%), Asia (27%), Europe (23%), and Australia (7%). Forty-three percent of the organizations provide CLP surgeries or services in more than 1 country; 93% do so with a multidisciplinary team. A majority of the organizations established collaborations with host institutions (80%). CONCLUSION: To the authors' best knowledge, this database includes the largest collection of CLP organizations. This list will be made publicly available to inform surgical care planning, facilitate collaboration, and promote further research.

The Role for International Outreach in Hand Surgery.

Internationally, surgery is making its mark as a necessary priority in Low and Middle Income Countries (LMICs). In 2013, Paul Farmer, MD, and international leaders from 14 countries founded the Lancet Commission of Global Surgery to develop and assemble the best evidence on the state of surgery worldwide, to study the economics of surgical and anesthesia care delivery, and to develop strategies for improving access. Concurrently, there is growing interest in hand surgery. The Journal of Hand Surgery (European Volume) recently called for LMIC research with the goal of using their resources to improve the end product. International outreach was further prioritized at the American Society for Surgery of the Hand, with Scott Kozin, MD, and colleagues subsequently launching the Touching Hands Program (THP). Insight into current models, proven benefits, emphasis on quality, and research in international hand surgery will help invested volunteers provide high-quality, safe, and innovative solutions in the global hand surgery landscape.

Plastic and Reconstructive Surgery in Global Health: Let's Reconstruct Global Surgery.

Since the inception of the Lancet Commission in 2013 and consequent prioritization of Global Surgery at the World Health Assembly, international surgical outreach efforts have increased and become more synergistic. Plastic surgeons have been involved in international outreach for decades, and there is now a demand to collaborate and address local need in an innovative way. The aim of this article was to summarize new developments in plastic and reconstructive surgery in global health, to unify our approach to international outreach. Specifically, 5 topics are explored: current models in international outreach, benefits and concerns, the value of research, the value of international surgical outreach education, and the value of technology. A "Let's Reconstruct Global Surgery" network has been formed using Facebook as a platform to unite plastic and reconstructive surgeons worldwide who are interested in international outreach. The article concludes with actionable recommendations from each topic.

Pediatric Hand Surgery in Global Health: The Role for International Outreach.

PURPOSE: There is emerging interest in hand surgery and global health. This was emphasized at the 2015 presidential address at the American Society for Surgery of Hand. Children are prioritized because of their increased risk for trauma and higher potential for better outcomes. This study aims to identify how hand surgical volunteer programs can benefit the pediatric hand surgical landscape in global health. There has been no literature review to date. METHODS: This institutional review board-approved review systematically searched PubMed, Embase, Medline, African Journal Online, and the Journal of Hand Surgery. A scoping review methodology was selected to allow mapping of a body of literature by topic, include a greater range of study designs, and provide a descriptive overview of the reviewed material. All studies published between 2000 to March 2016 relevant to pediatric hand surgery in global health were included. Preferred Reporting Items for Systematic Reviews and Meta-Analyses was used to record the search results. RESULTS: Six hundred sixty-eight citations were reviewed, with 10 studies that satisfied the inclusion criteria. Hand trauma (70%), congenital anomalies (30%), tumors (20%), surgical technique (50%), and international outreach recommendation (30%) were common themes. Targeting prevention (50%), international outreach education (30%), and building on previous studies to validate findings study (10%) were identified as gaps.