Time tracking and comparison of genetic counseling tasks in inpatient and outpatient settings.

Genetic counselors (GCs) practice in critical care settings. Some GCs have full-time inpatient roles, while most GCs who see inpatients do so as needed or on a rotating schedule in addition to seeing patients in an outpatient setting. Few studies have tracked and compared the amount of time it takes GCs to perform tasks in the inpatient and outpatient settings. Genetic counselors were invited to participate in this study via the National Society of Genetic Counselors research listserv. Participants completed an online survey asking how their role is structured and what types of support are available to them while seeing inpatients. They also performed time tracking for 16 tasks known to be associated with inpatient and/or outpatient care via RedCap. These tasks include direct patient care, care coordination, and other tasks which encapsulate a new patient encounter from beginning to end. Forty-two inpatient encounters and 26 outpatient encounters were analyzed. The total average time spent on an inpatient consult (3 h and 38.5 min) was significantly higher than the time spent on an outpatient consult (2 h and 24.7 min; p < 0.05). Individually, genetic counselors spent significantly more time on the following tasks in an inpatient setting: direct follow-up encounters, multidisciplinary team communication, results disclosure encounters, results disclosure documentation, traveling, and waiting. Follow-up encounters, traveling, and waiting happen almost exclusively in inpatient settings. Short answer prompts regarding structure of GC role and available support revealed themes including lack of inpatient role structure, challenges with balancing between inpatient and outpatient tasks, and varied institutional support. These results promote further discussion about how to support GCs who see inpatients as these roles expand. Some suggestions include increased FTE/protected time and/or GCA support specific to the inpatient role.

Characterizing the research mentorship experience of genetic counseling students.

The Accreditation Council for Genetic Counseling's Practice-Based Competencies include research-related skills, which are taught in master's level genetic counseling programs through didactic coursework and completion of mentored research experiences. It is known that research mentors can impact student work environments, create positive perceptions of the research process, and increase students' likelihood of future involvement in research. However, few studies have characterized the experiences of GC students in receiving research mentorship. It is crucial to understand these experiences from student perspectives to better inform stakeholders about factors that impact mentorship. Using a mixed-methods approach, this study explored GC students' experiences receiving research mentorship and their thoughts regarding the successful qualities of research mentors. GC students (N = 165) who graduated between 2019 and 2022 responded to an online survey measuring the mentorship relationship, defined by the Advisory Working Alliance Inventory (AWAI). On average, participants scored 3.96/5 on the AWAI, where higher scores indicate stronger working alliances. When asked to describe their overall research experience in three words, 75.7% of participants used at least one negatively connotated descriptor. Thematic analysis of semi-structured interviews obtained via purposive sampling of highest and lowest scoring participants on the AWAI (N = 14) revealed the following five themes related to successful qualities of a research mentor: (1) communication; (2) rapport building and relationship; (3) engagement and guidance; (4) expertise and connections; and (5) mentors with different roles. Of note, many of these qualities are foundational skills in genetic counseling. Thus, genetic counselors who may be strong in these areas who do not identify as "researchers" ought to consider becoming a research committee member. Additionally, education programs could consider implementing research committee member evaluations and/or student research self-efficacy surveys to evaluate how these relationships may be shaping research experiences for students within their program.

Provision and availability of genomic medicine services in Level IV neonatal intensive care units.

PURPOSE: To describe variation in genomic medicine services across level IV neonatal intensive care units (NICUs) in the United States and Canada. METHODS: We developed and distributed a novel survey to the 43 level IV NICUs belonging to the Children's Hospitals Neonatal Consortium, requesting a single response per site from a clinician with knowledge of the provision of genomic medicine services. RESULTS: Overall response rate was 74% (32/43). Although chromosomal microarray and exome or genome sequencing (ES or GS) were universally available, access was restricted for 22% (7/32) and 81% (26/32) of centers, respectively. The most common restriction on ES or GS was requiring approval by a specialist (41%, 13/32). Rapid ES/GS was available in 69% of NICUs (22/32). Availability of same-day genetics consultative services was limited (41%, 13/32 sites), and pre- and post-test counseling practices varied widely. CONCLUSION: We observed large inter-center variation in genomic medicine services across level IV NICUs: most notably, access to rapid, comprehensive genetic testing in time frames relevant to critical care decision making was limited at many level IV Children's Hospitals Neonatal Consortium NICUs despite a significant burden of genetic disease. Further efforts are needed to improve access to neonatal genomic medicine services.

Exploring collaboration models between geneticists and intensivists for implementing rapid genome sequencing in critical care settings.

The availability of rapid genome sequencing (rGS) for children in a critical-care setting is increasing. This study explored the perspectives of geneticists and intensivists on optimal collaboration and division of roles when implementing rGS in neonatal and pediatric intensive care units (ICUs). We conducted an explanatory mixed methods study involving a survey embedded within an interview with 13 genetics and intensive care providers. Interviews were recorded, transcribed, and coded. Geneticists endorsed higher confidence in performing a physical exam and interpreting/communicating positive results. Intensivists endorsed highest confidence in determining whether genetic testing was appropriate, communicating negative results, and consenting. Major qualitative themes that emerged were: (1) concerns with both "genetics-led" and "intensivist-led" models with workflows and sustainability (2) shift the role of determining rGS eligibility to ICU medical professionals, (3) continued role of geneticists to assess phenotype, and (4) include genetic counselors (GCs) and neonatal nurse practitioners to enhance workflow and care. All geneticists supported shifting decisions regarding eligibility for rGS to the ICU team to minimize time cost for the genetics workforce. Exploring models of geneticist-led phenotyping, intensivist-led phenotyping for some indications, and/or inclusion of a dedicated inpatient GC may help offset the time burden of consenting and other tasks associated with rGS.

Seizures in trisomy 18: Prevalence, description, and treatment.

Changes in medical intervention over the last decade have improved outcomes for individuals with trisomy 18, the second most common human aneuploidy syndrome at birth. As children with trisomy 18 live longer, a shared concern of medical experts and parents is the occurrence and treatment of seizures. Previously published surveillance guidelines for this condition have not addressed seizure management. Using parent-reported data collected as part of the Tracking Rare Incidence Syndromes project, we report on the prevalence, course, and management of seizures in individuals with trisomy 18. Twenty-eight percent (52/186) of individuals diagnosed with trisomy 18 in our retrospective cohort experienced generalized, focal, or mixed seizures at some point in their lifetime. For many individuals, seizures were effectively managed by broad-spectrum anti-seizure medications. Correlation analysis showed that focal and generalized seizures were more likely to occur in individuals who had previously experienced infantile spasms or central apnea. Electroencephalogram testing should be considered as part of a standard screening approach in individuals with trisomy 18 to enable early diagnosis and treatment of seizures. An international registry that incorporates parent-reported and clinical data for patients with trisomy 18 may facilitate ongoing research and recruitment into clinical trials for seizure management.

Variants in AQP11 may result in autosomal recessive bilateral cystic renal dysgenesis.

Congenital renal cystic dysplasia is a rare disease that occurs in approximately 1 in 4000 children and is often discovered in the antenatal period by ultrasound. It is commonly associated with oligohydramnios in utero and/or renal insufficiency or failure in the postnatal period. Aquaporins are membrane proteins that serve as transport channels in the transfer of water or small solutes across cell membranes. They play a role in the development of renal cysts. Aquaporin 11 (AQP11) deficient mice develop polycystic kidney disease in utero due to disruption of polycystin-1. Here we describe a case of bilateral cystic kidney disease in a patient with novel compound heterozygous variants in AQP11: c.780G>T (p. Trp260Cys) and c.472C>T (p.Pro158Ser) (NM_173039.2) identified by whole genome sequencing. These findings suggest, for the first time, the potential role of AQP11 in congenital renal cystic dysplasia.

Re: "Next generation sequencing in neonatology: what does it mean for the next generation?"

Available evidence does not support limiting the use of rapid or ultra-rapid exome or genome sequencing in critically ill neonates to cases of predicted high diagnostic yield. Such testing is best positioned to improve neonatal care when test utilization is conceptualized within the total care of the family with a goal of rapid resolution of the diagnostic odyssey.

Evaluating a general pediatric/adult genetic counseling clinic in a Midwest medical center.

In 2018, the Munroe-Meyer Institute for Genetics & Rehabilitation (MMI) at the University of Nebraska Medical Center (UNMC) in Omaha, NE created a genetic counseling clinic (GCC) to increase access to genetics services and decrease the time spent between a referral and being seen in a general genetics outpatient clinic. In the GCC, genetic counselors led patient encounters and geneticists served as advisors, rather than primary providers. We conducted a chart review of 109 patients seen in the GCC from November 1, 2018, to March 16, 2020, and obtained information regarding patient demographics, indications, and clinical recommendations as a result of the visit. Most patients seen in this clinic were female (65.1%) and aged 19 years of age or older (54.1%). The primary indications for patients in this clinic included review genetic test results (42.2%), coordination of genetic testing for a known familial variant (30.2%), and concerns for personal or family history suspicious of a genetic condition without dysmorphic features (24.8%). The average patient wait time between referral date and appointment date in the GCC was 49.8 days. The two most common clinical recommendations made by genetic counselors in the GCC were genetic testing (56.1%) and/or follow-up with specialist (26.5%). These specialists primarily included endocrinology (n = 5), neurology (n = 4), cardiology (n = 4), ophthalmology (n = 3), and audiology (n = 3). We found that the GCC model may be appropriate for patients with (1) genetic test results requiring interpretation, (2) a known familial variant or (3) genetic testing recommended by a specialist physician. Descriptions of the indications and recommendations for patients seen in this GCC provide a framework for potential implementation of a GCC in other regions across the nation.

Ethical Considerations for Equitable Access to Genomic Sequencing for Critically Ill Neonates in the United States.

Rare diseases impact all socio-economic, geographic, and racial groups indiscriminately. Newborn screening (NBS) is an exemplary international public health initiative that identifies infants with rare conditions early in life to reduce morbidity and mortality. NBS theoretically promotes equity through universal access, regardless of financial ability. There is however heterogeneity in access to newborn screening and conditions that are screened throughout the world. In the United States and some other developed countries, NBS is provided to all babies, subsidized by the local or federal government. Although NBS is an equitable test, infants admitted to neonatal intensive care units (NICUs) may not receive similar benefits to healthier infants. Newborns in the NICU may receive delayed and/or multiple newborn screens due to known limitations in interpreting the results with prematurity, total parenteral nutrition, blood transfusions, infection, and life support. Thus, genomic technologies might be needed in addition to NBS for equitable care of this vulnerable population. Whole exome (WES) and genome sequencing (WGS) have been recently studied in critically ill newborns across the world and have shown promising results in shortening diagnostic odysseys and providing clinical utility. However, in certain circumstances several barriers might limit access to these tests. Here, we discuss some of the existing barriers to genomic sequencing in NICUs in the United States, explore the ethical implications related to low access, consider ways to increase access to genomic testing, and offer some suggestions for future research in these areas.

Telehealth genetic services during the COVID-19 Pandemic: Implementation and patient experiences across multiple specialties in Nebraska.

The COVID-19 pandemic has altered the delivery of genetics services. In response to the pandemic, our genetics department offered telehealth visits to all outpatients, regardless of their physical proximity to Omaha, Nebraska. Traditionally, our department did not offer telehealth visits to patient's homes or to patients who lived in close proximity to a genetics clinic. Therefore, we designed a survey to gain insight into the patient experience with remote genetic counseling appointments during the pandemic. Any patient referred to see a genetics provider in pediatrics, prenatal, adult, or cancer between March 16, 2020 and October 28, 2020 was eligible for the study. The survey included both quantitative and qualitative measures to assess patient demographics, patient experience, stressors during the COVID-19 pandemic, and anxiety and depression. We hypothesized that patients would report they received quality care by telehealth despite the presence of COVID-19-related stressors or anxiety/depression. From the 143 survey participants, 80% had their first telehealth appointment during the pandemic. The vast majority (96%) reported that they felt like they received quality care by telehealth. Additionally, more than 93% of participants strongly or somewhat agreed that their genetic providers were attentive to their emotional needs, medical needs, and privacy. Since March 2020, participants reported experiencing several COVID-19-related stressors including fear of illness (86%), feelings of isolation (45%), and safety concerns (33%). Relatively low levels of depressive and anxiety symptoms were recorded using the HADS questionnaire. Despite the prevalence of COVID-19 stressors, depression, and/or anxiety, our participants felt they received quality care via telehealth. In fact, 51% agree that they prefer to receive future genetics services virtually. These results suggest the value of telehealth as an alternative service delivery model, even for local patients, and should be offered for future appointments, beyond the COVID-19 pandemic.

Effect of Whole-Genome Sequencing on the Clinical Management of Acutely Ill Infants With Suspected Genetic Disease: A Randomized Clinical Trial.

Importance: Whole-genome sequencing (WGS) shows promise as a first-line genetic test for acutely ill infants, but widespread adoption and implementation requires evidence of an effect on clinical management. Objective: To determine the effect of WGS on clinical management in a racially and ethnically diverse and geographically distributed population of acutely ill infants in the US. Design, Setting, and Participants: This randomized, time-delayed clinical trial enrolled participants from September 11, 2017, to April 30, 2019, with an observation period extending to July 2, 2019. The study was conducted at 5 US academic medical centers and affiliated children's hospitals. Participants included infants aged between 0 and 120 days who were admitted to an intensive care unit with a suspected genetic disease. Data were analyzed from January 14 to August 20, 2020. Interventions: Patients were randomized to receive clinical WGS results 15 days (early) or 60 days (delayed) after enrollment, with the observation period extending to 90 days. Usual care was continued throughout the study. Main Outcomes and Measures: The main outcome was the difference in the proportion of infants in the early and delayed groups who received a change of management (COM) 60 days after enrollment. Additional outcome measures included WGS diagnostic efficacy, within-group COM at 90 days, length of hospital stay, and mortality. Results: A total of 354 infants were randomized to the early (n = 176) or delayed (n = 178) arms. The mean participant age was 15 days (IQR, 7-32 days); 201 participants (56.8%) were boys; 19 (5.4%) were Asian; 47 (13.3%) were Black; 250 (70.6%) were White; and 38 (10.7%) were of other race. At 60 days, twice as many infants in the early group vs the delayed group received a COM (34 of 161 [21.1%; 95% CI, 15.1%-28.2%] vs 17 of 165 [10.3%; 95% CI, 6.1%-16.0%]; P = .009; odds ratio, 2.3; 95% CI, 1.22-4.32) and a molecular diagnosis (55 of 176 [31.0%; 95% CI, 24.5%-38.7%] vs 27 of 178 [15.0%; 95% CI, 10.2%-21.3%]; P < .001). At 90 days, the delayed group showed a doubling of COM (to 45 of 161 [28.0%; 95% CI, 21.2%-35.6%]) and diagnostic efficacy (to 56 of 178 [31.0%; 95% CI, 24.7%-38.8%]). The most frequent COMs across the observation window were subspecialty referrals (39 of 354; 11%), surgery or other invasive procedures (17 of 354; 4%), condition-specific medications (9 of 354; 2%), or other supportive alterations in medication (12 of 354; 3%). No differences in length of stay or survival were observed. Conclusions and Relevance: In this randomized clinical trial, for acutely ill infants in an intensive care unit, introduction of WGS was associated with a significant increase in focused clinical management compared with usual care. Access to first-line WGS may reduce health care disparities by enabling diagnostic equity. These data support WGS adoption and implementation in this population. Trail Registration: ClinicalTrials.gov Identifier: NCT03290469.

Conscience clauses in genetic counseling: Awareness and attitudes.

Conscience clauses are laws that allow healthcare providers to refuse to participate in legal medical services based on moral or ethical objections. Genetic counselors encounter a variety of ethical and moral issues, including counseling about abortions. Currently, three states (Oklahoma, Nebraska, and Virginia) have genetic counseling conscience clause laws that allow genetic counselors to refuse to counsel about abortions. Conscience clause laws applying to physicians and pharmacists have been studied; however, they have not been studied in genetic counseling to date. We conducted an exploratory study assessing conscience clause awareness, attitudes, perceived obligations if utilizing a conscience clause, and alignment with the National Society of Genetic Counseling (NSGC) Code of Ethics. Genetic counselors (n = 274) currently practicing in the United States completed an online survey recruited through the NSGC listserv. The majority of participants were not aware that conscience clauses exist for genetic counseling (90%). There was uncertainty about whether genetic counselors had the right to utilize a conscience clause in practice (24% said yes, 31% said no, and 45% were unsure/needed more information). The majority reported an obligation to refer a patient if implementing a conscience clause (90%), although there were discrepancies among what constitutes an appropriate referral. When asked about the interaction between conscience clauses and the NSGC Code of Ethics, 45% believe they are separate and one does not supersede the other, 31% felt the Code of Ethics supersedes, 8% felt conscience clauses supersede, and 16% were unsure. Our study shows overall uncertainty with how conscience clause laws may be applied in clinical practice. Further clarity and education, especially in states where these laws exist, is critical to navigate the interaction between conscience clause laws and genetic counseling practice.

Surveillance guidelines for children with trisomy 13.

Trisomy 13 is one of the three most common aneuploidy syndromes in live-born infants. It is associated with mortality rates as high as 90% within the first year of life, in large part, due to the high prevalence of severe congenital abnormalities that increase mortality and morbidity. However, life-saving and life-prolonging medical interventions are being performed at a higher rate for these infants, resulting in increased rates of survival. Although cardiac complications have been well described in infants with trisomy 13, these patients also experience other complications such as respiratory, neurological, genitourinary, abdominal, otolaryngologic, and orthopedic complications that can impact their quality of life. The goal of this review is to present a comprehensive description of complications in children with trisomy 13 to aid in the development of monitoring and treatment guidelines for the increasing number of providers who will be caring for these patients throughout their lives. Where the evidence is available, this review presents screening recommendations to allow for more rapid detection and documentation of these complications.

Surveillance guidelines for children with trisomy 18.

Trisomy 18 is the second most common aneuploidy syndromes in live born infants. It is associated with high mortality rates, estimated to be 75%-95% in the first year of life, as well as significant morbidity in survivors. The low survival is largely due to the high prevalence of severe congenital anomalies in infants with this diagnosis. However, interventions to repair or palliate those life-threatening anomalies are being performed at a higher rate for these infants, resulting in increased rates of survival beyond the first year of life. While it is well documented that trisomy 18 is associated with several cardiac malformations, these patients also have respiratory, neurological, neoplastic, genitourinary, abdominal, otolaryngologic, and orthopedic complications that can impact their quality of life. The goal of this review is to present a comprehensive description of complications in children with trisomy 18 to aid in the development of monitoring and treatment guidelines for the increasing number of providers who will be caring for these patients throughout their lives. Where the evidence is available, this review presents screening recommendations to allow for more rapid detection and documentation of these complications.

Experiences of a Multidisciplinary Genomic Tumor Board Interpreting Risk for Underlying Germline Variants in Tumor-Only Sequencing Results.

PURPOSE: Although analyzing germline and tumor samples concurrently provides the best opportunity for differentiating between germline and somatic mutations, tumor-only sequencing is becoming increasingly common in clinical care. The purpose of this study is to assess how a multidisciplinary genomic tumor board (MGTB) evaluated patients' tumor-only sequencing results and made genetics referrals. With limited professional society guidance on how to manage pathogenic mutations identified via tumor-only sequencing, this study contemplates the professional knowledge and skills necessary to have represented on an MGTB to interpret these results in context of potential germline findings. METHODS: Qualitative interviews with MGTB members and an ethnographic case study of a breast cancer MGTB at a National Cancer Institute cancer center were examined. RESULTS: This MGTB discussed 34 cases of women with advanced-stage breast cancer over 13 months. Interviews and observations of MGTB meetings indicated that members of the MGTB contemplated whether variants were germline or somatic and potential for identification of germline cancer predisposition. On the basis of existing professional society guidelines, 18 patients would be eligible for germline testing. However, the MGTB only referred 11 patients (61%) for additional germline testing, and the remaining seven patients (39%) were not referred, raising questions about the kind of genomic expertise needed on an MGTB to optimize results interpretation and referrals. CONCLUSION: To ensure adequate interpretation, recommendation, and communication of tumor sequencing results, an MGTB should include professionals with knowledge and experience in clinical translation of tumor sequencing, testing methodology, molecular pathology, cancer biology, genomic pathways, germline variant interpretation, evaluation of family history, and application of professional recommendations for germline testing after tumor-only sequencing. These skills may not be held by a single professional on an MGTB.

Knowledge, motivations, expectations, and traits of an African, African-American, and Afro-Caribbean sequencing cohort and comparisons to the original ClinSeq® cohort.

PURPOSE: Racial minority populations are underrepresented in genomics research. This study enrolled African-descended individuals in a sequencing study and reported their characteristics. METHODS: We purposively recruited 467 individuals self-identified as African, African American, or Afro-Caribbean to the ClinSeq® study and surveyed them about knowledge, motivations, expectations, and traits. Summary statistics were calculated and compared with data from the study's original cohort, which was primarily White and self-referred. RESULTS: Recruitment took five years and 83% of enrollees completed the survey. Participants had modest knowledge about benefits and limitations of sequencing (x̅s = 5.1, ranges: 0-10), and less than the original cohort (x̅ = 7.5 and 7.7, respectively). Common motivations to enroll were learning information relevant to personal health (49%) or family members' health (33%), and most had realistic expectations of sequencing. Like the original cohort, they had high levels of optimism, openness, and resilience. CONCLUSION: Early adopters may have relatively consistent personality traits irrespective of majority/minority status and recruitment methods, but high levels of genomics knowledge are not universal. Research should determine whether recruitment and consent procedures provide adequate education to promote informed choices and realistic expectations, which are vital to ethical research and increasing genomics research participation in underrepresented communities.

Outcomes of Counseling after Education about Carrier Results: A Randomized Controlled Trial.

In-person education and counseling for all people receiving genetic results is the predominant model of disclosure but is challenged by the growing volume of low-impact results generated by sequencing. Evidence suggests that web-based tools may be as effective as in-person counseling at educating individuals about their low-impact results. However, the effects of counseling have not been assessed. To evaluate its utility, carrier results were returned to 459 post-reproductive participants from the ClinSeq cohort within a randomized controlled trial. Participants received education and were randomized to receive counseling or not. Primary outcomes included risk worry, test-related positive experiences, attitudes, and decisional conflict. Secondary outcomes were satisfaction, preferences, and counseling value. There were no differences between participants who received counseling and those who did not in the primary outcomes. Participants who received counseling were more satisfied than those who did not (x¯ = 10.2 and 9.5, respectively, p < 0.002, range: 3-12), although overall satisfaction was high. Most participants (92%) randomized to counseling preferred it and valued it because it provided validation of their reactions and an opportunity for interpersonal interaction. Web-based tools address the challenge of returning low-impact results, and these data provide empiric evidence that counseling, although preferred and satisfying, is not critical to achieving desired outcomes.

Web Platform vs In-Person Genetic Counselor for Return of Carrier Results From Exome Sequencing: A Randomized Clinical Trial.

Importance: A critical bottleneck in clinical genomics is the mismatch between large volumes of results and the availability of knowledgeable professionals to return them. Objective: To test whether a web-based platform is noninferior to a genetic counselor for educating patients about their carrier results from exome sequencing. Design, Setting, and Participants: A randomized noninferiority trial conducted in a longitudinal sequencing cohort at the National Institutes of Health from February 5, 2014, to December 16, 2016, was used to compare the web-based platform with a genetic counselor. Among the 571 eligible participants, 1 to 7 heterozygous variants were identified in genes that cause a phenotype that is recessively inherited. Surveys were administered after cohort enrollment, immediately following trial education, and 1 month and 6 months later to primarily healthy postreproductive participants who expressed interest in learning their carrier results. Both intention-to-treat and per-protocol analyses were applied. Interventions: A web-based platform that integrated education on carrier results with personal test results was designed to directly parallel disclosure education by a genetic counselor. The sessions took a mean (SD) time of 21 (10.6), and 27 (9.3) minutes, respectively. Main Outcomes and Measures: The primary outcomes and noninferiority margins (δNI) were knowledge (0 to 8, δNI = -1), test-specific distress (0 to 30, δNI = +1), and decisional conflict (15 to 75, δNI = +6). Results: After 462 participants (80.9%) provided consent and were randomized, all but 3 participants (n = 459) completed surveys following education and counseling; 398 (86.1%) completed 1-month surveys and 392 (84.8%) completed 6-month surveys. Participants were predominantly well-educated, non-Hispanic white, married parents; mean (SD) age was 63 (63.1) years and 246 (53.6%) were men. The web platform was noninferior to the genetic counselor on outcomes assessed at 1 and 6 months: knowledge (mean group difference, -0.18; lower limit of 97.5% CI, -0.63; δNI = -1), test-specific distress (median group difference, 0; upper limit of 97.5% CI, 0; δNI = +1), and decisional conflict about choosing to learn results (mean group difference, 1.18; upper limit of 97.5% CI, 2.66; δNI = +6). There were no significant differences between the genetic counselors and web-based platform detected between modes of education delivery in disclosure rates to spouses (151 vs 159; relative risk [RR], 1.04; 95% CI, 0.64-1.69; P > .99), children (103 vs 117; RR, 1.07; 95% CI, 0.85-1.36; P = .59), or siblings (91 vs 78; RR, 1.17; 95% CI, 0.94-1.46; P = .18). Conclusions and Relevance: This trial demonstrates noninferiority of web-based return of carrier results among postreproductive, mostly healthy adults. Replication studies among younger and more diverse populations are needed to establish generalizability. Yet return of results via a web-based platform may be sufficient for subsets of test results, reserving genetic counselors for return of results with a greater health threat. Trial Registration: clinicaltrials.gov Identifier: NCT00410241.