Abstract
Introduction: Despite the manifold symptoms women with inherited bleeding disorders experience, whether they receive timely screening and specialist referral remains unclear. While literature on this topic is sparse, studies have found that, irrespective of the hemostatic challenges women face, they are often unlikely to be referred to hematologists, subsequently remaining undiagnosed and under-treated. Recognition and early diagnosis remain paramount for management and avoidance of complications, particularly as women with bleeding disorders are at increased risk for heavy menstrual bleeding, anemia, post-partum hemorrhage, and decreased quality of life. However, to our knowledge, no studies have systematically assessed whether women face unique barriers to receiving specialist care and timely treatment. This systematic review sought to better characterize literature on access to care for women with inherited bleeding disorders.
Methods: A systematic review was conducted to assess the literature on access to care for women with inherited bleeding disorders. This was conducted following the Cochrane Handbook, and reported as per Preferred Reporting for Systematic Reviews and Meta-Analyses (PRISMA) guidelines. In consultation with a health services librarian, we designed a search strategy in order to identify published papers reporting on all forms of bleeding diatheses. Inclusion criteria included all studies examining bleeding disorders and equity of care in women after the year 2000; exclusion criteria included studies written in a non-English language or only enrolling men. Two review authors independently extracted data and assessed included studies for risk of bias. Risk of bias was assessed using the Newcastle-Ottawa guidelines. The GRADE approach was used to assess quality of evidence. If required, additional data was sought from study authors.
Results: Of 526 screened abstracts, only a small subset (N=12) explicitly focused on women's health, with the majority focusing on gynecologic and obstetric complications associated with von Willebrand Disease (VWD). There was significant heterogeneity in study methodology and populations enrolled. Only two studies explicitly studied self-reported barriers to hemophilia care, with one study largely enrolling men with hemophilia A of moderate-severe severity. Within the literature investigating barriers to care, themes that emerged included greater difficulties surrounding access to treatment for women, a tendency for hemophilia carriers to encounter dismissive healthcare provider attitudes, and ignorance around bleeding disorders in women. These studies proposed that unmet needs, particularly for women with inherited bleeding disorders, require further evaluation. However, risk of bias was found to be high, and grade of evidence low.
Discussion: We sought to better characterize the literature on access to care for women with inherited bleeding disorders, and found that literature on this topic is particularly sparse. Given our lack of understanding of women's experiences, and particularly the experiences of hemophilia carriers, ongoing efforts are needed to appreciate where inequities in care may exist. Using qualitative research methodology, we will explore the experiences of women, as well as other understudied populations, who live with bleeding disorders. By identifying possible demographic, geographic, and systems level factors hindering patients' access to care, we hope to increase healthcare provider awareness around existing barriers and, through targeted interventions, promote patient self-advocacy.
No relevant conflicts of interest to declare.
