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Genetic risk assessment and haemoglobinopathy counselling: two case studies

02 December 2019
Volume 27 · Issue 12

Abstract

Background

The author works as an antenatal and newborn (ANNB) screening coordinator in a hospital designated as an area of low prevalence for sickle cell and thalassaemia (SCT). As such, the opportunity for an ANNB coordinator to gain competency in prenatal counselling of individuals with SCT is limited.

Method

Two case studies are used to demonstrate social, cultural and ethical issues arising from counselling individuals with SCT.

Findings

Information regarding genetic risk and haemoglobinopathy status can be threatening for individuals and families, as well as transforming, depending on the context in which information is provided.

The role of the antenatal and newborn (ANNB) coordinator is to work as part of a team to deliver the six ANNB programmes commissioned by the NHS, with oversight and quality assurance provided by Public Health England (PHE). One of these programmes is the Sickle Cell and Thalassaemia (SCT) screening programme (PHE, 2018). In order to have assurance of safety and quality, PHE have a mandatory requirement for all ANNB coordinators to undertake genetic risk assessment and counselling training. While educational and sociological debates exist regarding the pedagogic discourse of knowledge transmission (Berstein, 1973) and how competency and performance in midwifery practice can be assessed (Chenery-Morris, 2017), two reflective counselling episodes are used to demonstrate how ANNB coordinators can meet the requirements of the Royal College of Nursing ([RCN], 2011) SCT competency framework. This framework ensures high quality counselling and engagement in shared decision making with individuals and families. This is especially important when the opportunity for ANNB coordinators to engage and gain confidence in genetic and haemoglobinopathy counselling on a regular basis is limited.

Working within a predominantly rural geographical area with a population of around 280 000 people, the local trust has been identified as an area of low prevalence for SC—determined by less than 1% of the booking bloods resulting in a screen positive result for SCT (PHE, 2018). Risk for SCT is assessed through the use of a screening form; a family origin questionnaire (FOQ), which results in only those individuals identified as originating from an area at risk for SCT, having their blood tested for SCT.

Globally, inherited haemoglobin disorders, including thalassemia and sickle cell disease, are common and life-threatening disorders with an estimated 1–5% of the population identified as carriers for a genetic thalassemia mutation worldwide (Taher et al, 2018). Whilst the epidemiology of the various clinical forms is not fully understood (Galanello et al, 2013), prevalence is strongly linked to ethnicity, with sickle cell disease predominantly affecting individuals from African and Caribbean origin (Ware et al, 2017) and thalassaemia affecting predominantly individuals from sub-Saharan Africa, the Mediterranean region, the Middle East, India and East to South East Asia (Taher et al, 2018). Screening and prevention programmes internationally are dependent on regional prevalence as well as cultural and economic factors (Barrett et al, 2017).

Case study 1

The first episode of care relates to a woman and her partner who were both carriers for sickle cell disease who were counselled and subsequently declined the offer of prenatal diagnosis (PND) for their pregnancy and had a baby born with sickle cell anaemia. This episode was chosen as I was new to the role of ANNB screening coordinator when I first met the couple; it was during my induction period, while I was gaining insight into the ANNB role within local trusts. Reflections are made regarding personal growth and local development for future practice. Pseudonyms are used to maintain confidentiality.

Sickle cell disease is a term that relates to a collection of inherited blood disorders which result from a sickle mutation on the sixth codon of the beta-globin gene and is a disease of haemoglobin synthesis (PHE, 2017). Haemoglobin (Hb) is the substance within erythrocytes (red blood cells) and has three main functions: carrying and releasing oxygen throughout the body, transporting carbon dioxide from tissues to lungs, and the buffering of hydrogen ions to ensure maintenance of blood pH. In an individual with sickle cell disease, the erythrocytes have a tendency to change into sickle, or crescent shapes, resulting in the Hb being de-oxygenated. These misshapen and rigid erythrocytes can then result in vaso-occlusion, a disruption of the blood flow in small vessels with ischemia and inflammation manifesting clinically as acute pain. Over time, repeated sickling of the erythrocytes and associated haemolytic anaemia can lead to injury and organ damage resulting in morbidity and early mortality (Ware et al, 2017).

Overall survival and health of individuals born with sickle cell disease has improved since the introduction of newborn screening programmes (introduced in the UK in 2006) and prophylactic use of penicillin, alongside wider use of hydroxycarbamide, that acts to pharmacologically induce foetal haemoglobin (HbF), known for its high affinity for oxygen transportation and inhibition of intracellular HbS polymerisation (Ware et al, 2017). Additionally, patient and professional educational programmes about disease complications have enhanced care pathways for affected individuals. However, morbidity from the disease itself or treatment side effects can be severe for affected individuals and families (Mayo-Gamble et al, 2017) with the World Health Organization ([WHO], 2010) declaring sickle cell disease as a public health priority.

The consultation

Ten weeks into her first pregnancy, Mia booked with a community midwife who utilised the FOQ to ascertain Mia and her partner, Eze, were of Nigerian origin, where sickle cell carrier prevalence is estimated to be 20–30% (Adewoyin, 2015). As screening coordinator, I was contacted by the laboratory to advise Mia was a carrier for HbS and a request was made for urgent partner testing. This is required as sickle cell disease is an autosomal recessive disorder whereby for a child to inherit the condition, both parents need to be a carrier of the gene (NHS National Genetics and Genomics Education Centre, 2019).

Following partner testing, Eze was also identified as a carrier for HbS and confirmatory testing was conducted on both maternal and paternal samples. This finding identified there was a 25% chance that the fetus would be affected with the most common and severe form of homozygous HbSS, sickle cell anaemia, resulting in a 25% chance of having a baby not affected, and a 50% chance of having a baby who is a sickle cell carrier (NHS National Genetics and Genomics Education Centre, 2019).

‘As screening coordinator, I was contacted by the laboratory to advise Mia was a carrier for HbS and a request was made for urgent partner testing’

Mia and Eze were invited into the hospital to discuss the implications for the pregnancy by the consultant haematologist and ANNB screening coordinator. This appointment was situated in a haematology clinic situated within an oncology unit, and, in hindsight, was very poorly suited to facilitate counselling. For counselling to be effective, it should be in an unhurried environment, calming in atmosphere and where individuals can provide empathy and warmth, and concentrated attention (Harper, 2010). Additionally, Cappellini et al (2008) identifies that the environment should be in an atmosphere that will sustain hope and optimism, and reduce feelings of depression. As such, the environment of the oncology unit, with the consultant situated behind a desk and appointment scheduled amongst a routine clinic resulted in the time pressure of an overrunning clinic and the feeling of a closed negative conversation.

Informed decision-making

Related specifically to counselling for antenatal screening, the functions of both health education information and decision-making support are required to enable informed decision-making (Martin et al, 2015). Crucially, active listening techniques and a reciprocal trusting therapeutic relationship should be formed where the counsellor is able to step back and listen to how both parents make sense of the information, separately and together (Martin et al, 2015). Additionally, antenatal counselling is embodied in the need for preference-sensitive decisions to be made regarding psychosocial issues relating to an individual's views on parenthood, disabled life, and social, cultural and religious influences to opt or decline screening (Khuse and Singer, 1985). Ethical issues related to genetic counselling fundamentally require counsellors to be unbiased and non-judgemental, with the need for considered verbal and non-verbal communication, and affective behaviour of the counsellor and counselling duration known to be important for facilitating parental decision-making where both parents have low decisional regret and long-term confidence in (Nuffield Council on Bioethics, 2017).

Reflection on this counselling episode identifies that traditional medical hierarchies were present (Kirkham, 2006). The consultant haematologist was very much directing the conversation with information predominantly limited to the genetic and scientific basis of sickle cell disease and a disease prevention model discussing the pathological nature of the disease relating to physical morbidity and mortality. While I was present in the counselling room, due to my limited knowledge and experience, I offered little in the way to facilitate discussion about the ontological and epistemological changes in diseases, that is what we understand specifically about a certain disease, and how we know about a disease (Timmermans and Buchbinder, 2012). It includes how the same disease can shift and change in populations, with the nature of the disease clinically manifesting differently in individuals with the same disease ranging from mild to severe (Traeger-Synodinos, 2017).

I was also unable to offer any information about broader psychosocial implications or possible advancements in treatment or implementation of molecular medicine (Ribeil et al, 2017). This information, if meaningfully conveyed in a manner understood by the parents (Martin et al, 2016), may have provided a broader, more informed basis for the parents to consider their care options, namely of opting for or declining invasive PND for sickle cell through amniocentesis, which carries an associated risk of miscarriage of 1% (Royal College of Obstetricians and Gynaecologists, 2010).

Understanding the decision

Mia and Eze were unaware of their sickle cell carrier status and Eze had three children from another partner before moving to the UK, with no history of sickle cell anaemia. Mia had two female adult cousins with sickle cell anaemia who both had experience of episodes of severe pain and treatment through blood transfusions.

Mia and Eze informed us during this appointment that they would decline the option for PND, identifying they did not wish to risk miscarriage. Assumptions were possibly made by myself and the haematologist that an informed decision had been made due to family history of sickle cell anaemia and their decision was not explored further. However, Oni (2007) identifies many factors influence decision-making in at risk pregnancies. While concern regarding miscarriage often prevails, religion and enculturation are also highly influential, with cognitively constructed cultural attitudes varying significantly between cultures which may lead to significant cognitive dissonance for individuals (Oni, 2007).

Reducing concerns

There is potential for a screening programme expansion in the future to offer non-invasive prenatal diagnosis (NIPD) for single gene disorders and diseases such as sickle cell (Barrett et al, 2012). Such options may be empowering in reducing concerns regarding miscarriage and for situations when partner testing is not an option, although ethical questions relating to revealing carrier status of partners exist (Deans et al, 2013).

Indeed, Hill et al (2014), through an empirical qualitative focus group and one-to-one interviews with a sample of 28 individuals from the UK, identified that NIPD may result in the increased uptake of prenatal testing for sickle cell disease. Whilst common for qualitative studies to use small numbers of participants, these findings should be viewed with caution.

It is fundamental for health professionals involved in genetic counselling to acknowledge full bioethical outcomes of the possible introduction of such tests, as well as future potential for genome editing to make changes to the DNA of embryos to avoid genetic diseases such as SCT (Timmermans and Buchbinder, 2012; Nuffield Council of Bioethics, 2016; Cappellini et al, 2018). The benefits of reducing the impact of sickle cell disease worldwide, including long-term financial treatment costs, are well recognised alongside the reduction of individual physical morbidity and multiple psychosocial effects, such as isolation and associated stigma of having a disease (Blake et al, 2018).

‘It is fundamental for health professionals involved in genetic counselling to acknowledge full bioethical outcomes of the possible introduction of such tests’

However, it is necessary to consider the judgements on which the ‘benefits’ or ‘public health success’ of any future SCT screening programme, testing and genomic editing are based on. It can be argued in line with the UK National Screening Committee ([UKNSC], 2018) principles for all screening programmes (UKNSC 2018), any public health intervention success should be defined not at the individual level, but rather population level, and have transparency of moral perspectives and subsequent ethical, financial, psychological and cultural implications of future genetic tests (Stillwell, 2016; Nuffield Council on Bioethics, 2016).

The problem with stigmatisation

Individuals affected with sickle cell disease often already experience stigmatisation (Jegede, 1998), defined by Goffman's (1963) social theory of stigma as a phenomenon where rejection of an individual is due to an attribute or behaviour that is deeply discredited by society. However, it is arguable that introduction of NIPD or genetic editing for sickle cell disease, with the aim of reducing incidence in a targeted population with high prevalence in a specific ethnic community, may inadvertently result in an even deeper and more profound impact on individuals with sickle cell anaemia currently within society for having a socially discredited medical condition (Blake et al, 2018). This is compounded by the fact that discrimination based on ethnicity alone within the UK is a well-known phenomenon (Haywood et al, 2013).

It is fundamental to acknowledge future decisions which promote reproductive choice and autonomy need to minimise any unintentional psychosocial and broader ethical societal harms and ultimately support an equal society. Similarities can be drawn between NIPD for sickle cell with the proposed non-invasive prenatal test (NIPT) for congenital trisomies, which is now due to be rolled out in the NHS in the UK in 2020. The legal battle raised by individuals with Down syndrome (Nuffield Council on Bioethics, 2017) has been finalised and the plan is for NIPT to be rolled out once a new procurement by the NHS is completed. This identifies that differing values, priorities and experiences can strongly influence moral perspectives.

Case study 2

The second counselling episode relates to a woman named Lana and her partner Lao, both of Cyprian origin. Initial SCT screening at nine-weeks' gestation identified Lana as a beta-thalassaemia carrier.

Beta-thalassaemia is an autosomal recessive blood condition, resulting in mutations in the beta-globin gene characterised by either deficient or absent synthesis of the beta-globin subunit of adult Hb (Cappellini et al, 2018). In excess of 300 beta-thalassaemia alleles exist, and it is the co-inheritance of a beta-thalassaemia allele alongside a structural Hb variant, that results in diverse clinical phenotypes with clinical manifestations ranging from asymptomatic through to severe, chronic life-limiting disease. Conservative estimates worldwide indicate 40 000 babies are born each year with a clinically significant beta-thalassaemia with the financial cost in the UK per patient around £483 454 over 50 years (Cappellini et al, 2018).

Concerned partner

This counselling episode was specially chosen as Lao, during the first meeting at 10 weeks into the pregnancy to discuss Lana's beta-thalassaemia carrier status, became significantly agitated and angry during the appointment. As an experienced midwife, I was familiar with partners becoming upset or angry during care episodes when care did not fulfil patient expectations and where partners especially can feel a sense of loss of control or anxiety for their loved one that subsequently manifests as anger (Kirkham, 2006).

However, on this episode I was taken aback by the extent of anger displayed by Lao which continued to develop and exacerbate throughout the appointment. Lana's initial blood result identified hypochromic, microcytic anaemia. Laboratory diagnostic algorithms using red cell indices, reticulocyte counts and Hb levels had been used to differentiate thalassaemia trait from iron deficiency anaemia. As such, I began our conversation by describing how thalassaemia carrier status could possibly translate into a significant thalassaemia condition for the unborn fetus.

I felt I was clear in describing that for the significance of the carrier status to be determined, further testing was recommended, of partner blood testing for any presence of thalassaemia trait, and potentially for DNA genotyping to obtain a definitive diagnosis. Initially, I perceived Lao's anger to be directed to concern that molecular analysis results, if required, would take 2–3 weeks. I empathetically identified that, during notification of a presumptive serious health condition, it was recognised that frustration or anger is a common occurrence, hoping this would offer Lao an opportunity to rationalise and stablise his emotions (Harper, 2010).

Harper (2010) identifies that displays of anger are more likely when perceived helplessness or a lack of understanding of the information provided exists. As Lao's anger did not diminish, I offered multiple opportunities for the couple to discuss concerns and asked them to confirm their understanding of the information already provided, aware that after receiving negative or uncertain clinical information, individuals can appear confused even after receiving complete information. Alongside using nationally recommended literature (PHE, 2018a), I further described treatment options for individuals based on the severity of the clinical phenotype, for both transfusion-dependent or non-transfusion-dependent thalassaemia. I also made reference to how the demands of treatment may impact on quality of life emotionally, socially and physically (Cappellini et al, 2008), and provided brief information about novel strategies for the treatment of beta-thalassaemia (Taher et al, 2018). Aware that individuals may perceive having a carrier status as a crisis of identity, or even consider it as being ‘tainted’ with a sense of self or others as transferring from having insider status to outsider status in society (Kirkham, 2006), I attempted to use neutral language to discuss gene inheritance. However, crucially the more information I provided, the further Lao became agitated.

Looking back

Reflecting on this experience, using new knowledge gained from SCT literature and undertaking the counselling course provided me with a valuable opportunity to identify much resonance exists between the blood conditions of SCT and a personal experience I have with my five-year-old son, who was diagnosed with an acute blood condition – acute lymphoblastic leukaemia (ALL) – at age two years.

During the last three years, I have been living with the demands of him having multiple and significant episodes of clinical treatment, including regular invasive procedures and blood transfusions, which has had important psychological implications for our whole family, and significant physical implications for him living with the disease over the course of his life.

As a health professional with experience in academia and undertaking empirical research, my own personal coping mechanisms in acceptance of the disease and its implications have been an overwhelming desire to learn as much scientific and evidenced-based information as possible, to develop myself as an expert patient in order to advocate for him on each care episode.

However, reflection on this SCT counselling episode has strongly prompted me to consider how my own values, beliefs and personal experiences may impact on the level and type of information I provide during SCT counselling sessions. Significantly, I have identified that particularly at the first counselling session to communicate carrier status, a full and detailed explanation of a disease or condition before its overall significance (or absence of clinical significance) has been fully determined, may in fact cause unintentional harm to an individual.

Information overload (Barr and Skirton, 2013) and the phenomenon of over-diagnosis in health screening is now widely accepted (Gilbert Welch et al, 2011; McCartney, 2012; Raffle et al, 2019), with an awareness that screening itself has the potential for psychological distress and burden to patients if a condition is potentially identified. Yet, clinical significance or positive benefit from knowing about a screening status is unclear or later identified as of minimal significance. I have recognised whilst I personally desire a significant depth of health information to make decisions about my own son, it is fundamental I am fully self-aware and able to self-reflect to remain emotionally detached from individuals I am counselling.

Deep awareness of any personal ‘blind spots' through regular supervision may provide opportunity to assess personal strengths and limitations, and how they impact on my ability to effectively counsel individuals who are carriers of SCT (Raffle et al, 2019). Indeed, this self-awareness may enable counsellors to use their own life experiences to provide enhanced levels of empathy, compassion and understanding, and provides an ideal setting to communicate in a supportive way (Barr et al, 2018).

Self-reflection has identified that alongside a risk assessment and situational awareness of the context of each counselling episode, it is fundamental to provide a unique dialogue suited to individual needs, values, knowledge and beliefs, experiences, and cultural preferences. Further, Karetti et al (2004) promotes psychological harm of informing individuals about a thalassaemia carrier status may be minimised by informing individuals of possible advantages, as well as the risks associated with carrier status. These include the protective effect of beta-thalassaemia carrier status against both malaria and coronary heart disease.

Conclusion

Nuffield Council of Bioethics (2017) identifies that information regarding genetic risk can be threatening as well as transforming, depending on the context in which information is provided. Two episodes of counselling have been used to demonstrate the vital role that counsellors have in establishing communication that has awareness of situational and environmental context, is sensitive to the timing and depth of information required and respectful of the patient's educational, social, and cultural values and beliefs.

The first episode identified an example where information was potentially lacking to provide unbiased and meaningful understanding of a condition which provided realistic hopes (Cappellini et al, 2008) at an emotional as well as pathological level. Based on this reflection, a change in local practice has now occurred and all women identified as a carrier status for SCT are invited for a face-to-face discussion with the ANNB coordinator in an environment more suited to counselling. The second example demonstrated that genetic counsellors as gatekeepers of information must be self-aware of their own frame of reference, background and experiences, and how these may impact on the counselling episode influencing long-term psychosocial, cultural, ethical and moral burden.

Reflection has identified the responsibility involved in genetic counselling and demonstrated the importance of establishing a therapeutic relationship between health professionals and patients. Such relationships are known to be extremely beneficial and may permanently impact negatively or positively, any subsequent therapeutic relationships and a sense of balance in future conversations and decision-making regarding SCT (Cappellini et al, 2008). Reflecting against the eight standards of the genetics education framework for nurses (Kirk, 2013; Kirk et al, 2013) RCN (2011) framework and the genomics education programme (NHS England, 2019a) has led to greater proficiency and confidence when involved in counselling parents myself or alongside a haematology consultant.

Key points

  • Antenatal and Newborn coordinators require specialised training to ensure competency is gained in genetic risk assessment and haemoglobinopathy counselling
  • Individuals identified as a carrier of a haemoglobinopathy may face significant social, cultural and ethical dilemmas about both themselves and the wellbeing of their unborn fetus
  • Antenatal genetic counselling is embodied in the need for preference-sensitive decisions relating to an individual's views on parenthood, disabled life and social, cultural and religious influences to opt or decline screening
  • Sickle cell and thalassaemia screening has the potential for psychological distress to an individual if a condition is potentially identified, yet clinical significance or positive benefit from knowing about a screening status is unclear or later identified as of minimal significance
  • Genetic testing and future screening options which promote reproductive choice and autonomy need to minimise any unintentional psychosocial and broader ethical societal harms
  • CPD reflective questions

  • Do you feel equipped to offer haemoglobinopathy screening to newly pregnant parents and discuss the potential impact that a positive screening result may have?
  • Do you feel confident that you are self-aware of your own frames of reference, background and experiences when talking to pregnant parents, and how these may impact on conversations regarding haemoglobinopathy screening?
  • Reflect on how you would communicate a potential haemoglobinopathy carrier status to a pregnant individual and the recommendation for partner testing. How will you ensure you provide appropriate information to parents without adding to their anxiety?
  • Which aspects of genetic testing, that promote reproductive choice and autonomy, should be addressed by future research and screening options?
  • How have these case studies influenced you in your practice going forward?