Disclosure of Misattributed Paternity - American Academy of ...
←
→
Page content transcription
If your browser does not render page correctly, please read the page content below
Disclosure of Misattributed Paternity
Moshe Y. Prero, MD,a Meghan Strenk, MS, CGC,b Jeremy Garrett, PhD,b Ann Kessler, LISW-S,a Jonathan M. Fanaroff, MD, JD,a
John D. Lantos, MDb
One of the most common dilemmas faced by physicians and genetic abstract
counselors is the discovery of misattributed paternity. In this article, we
present a case in which misattributed paternity was discovered as an
incidental finding. Experts analyze the competing moral obligations that
might dictate disclosure or nondisclosure.
In the age of genomics, there will be chromosome. Additionally,
more and more secondary (or chromosomal microarray testing will
“incidental”) findings. Doctors need to detect a familial relationship between
think about how they will deal with the parents. This was carefully
these findings and need to inform explained to the mother. She consented
patients (and parents) about the to the test.
possibilities, the choices, and their
implications. In this Ethics Rounds, we Rebecca’s mother and father are both
present a case to illustrate some of the 22 years old, are not married, and have
dilemmas that might arise as a result of no other children. Rebecca’s mother
test results that suggest both cares for her full-time while her father
a
works at a local manufacturing Department of Pediatrics, Case Western Reserve University,
misattributed paternity and Cleveland, Ohio; and bDepartment of Pediatrics, Children’s
consanguinity. We then present company. Rebecca is under his Mercy Hospital, Kansas City, Missouri
comments by a physician lawyer, insurance.
Drs Prero, Fanaroff, Garrett, and Lantos, Ms Strenk,
a genetic counselor, and a philosopher. and Ms Kessler contributed to the design of this
Microarray testing confirmed that the
article and the drafting and review of the
mother was heterozygous for the manuscript; and all authors approved the final
THE CASE CLDN1 variant, whereas the father had version of the manuscript as submitted.
no CLDN1 mutations, making it likely DOI: https://doi.org/10.1542/peds.2018-3899
Rebecca was born at term and was
that he is not the biologic father (the
noted to have “thickened skin.” Accepted for publication Dec 13, 2018
only other possibility is that it could be
Additionally, she had poor weight gain Address correspondence to John D. Lantos, MD,
a new mutation). The testing indicated
and dehydration with hypernatremia. Children’s Mercy Hospital, 2401 Gillham Rd, Kansas
a likely third-degree to fourth-degree City, MO 64108. E-mail: jlantos@cmh.edu
Geneticists suspected neonatal
relatedness between the parents.
ichthyosis-sclerosing cholangitis PEDIATRICS (ISSN Numbers: Print, 0031-4005; Online,
syndrome (NISCH), an autosomal 1098-4275).
When genetics first discussed the
recessive condition. Clinical features of implications of the results with the Copyright © 2019 by the American Academy of
NISCH syndrome include ichthyosis, Pediatrics
mother, she stated, “I don’t want to talk
leukocyte vacuolization, hypertrichosis about it right now.” The team counseled FINANCIAL DISCLOSURE: The authors have indicated
and/or alopecia, and sclerosing they have no financial relationships relevant to this
the mother about disclosing the testing
article to disclose.
cholangitis. Exome sequencing revealed results, but to date, she has not done so.
a variant associated with NISCH. FUNDING: No external funding.
Both parents have the right and ability
Subsequent exon array testing to access the medical record. POTENTIAL CONFLICT OF INTEREST: The authors have
confirmed that Rebecca has 2 copies of indicated they have no potential conflicts of interest
to disclose.
the CLDN1 gene. The next step in The medical team requested an ethics
genetic testing is chromosomal consult. They asked if they have an
microarray, which can further detect if ethical obligation to tell the putative To cite: Prero MY, Strenk M, Garrett J, et al.
Disclosure of Misattributed Paternity. Pediatrics.
there are any large regions of deletion father that he is not the biologic father
2019;143(6):e20183899
or duplication within the patient’s or to find and notify the biologic father.
Downloaded from www.aappublications.org/news by guest on February 8, 2021
PEDIATRICS Volume 143, number 6, June 2019:e20183899 ETHICS ROUNDSAdditionally, they wondered what to The American Academy of Pediatrics determine parentage, and it should be
do about the evidence of Policy Statement on ethical and policy explained to the parties that “possible
consanguinity. issues in genetic testing and misattributed parentage will not be
screening of children recommends disclosed unless the participants
Dr Fanaroff Comments that misattributed paternity may be themselves request explanations of
Misattributed paternity occurs when uncovered incidentally and that “this genetic variation.”
it is determined incidentally from risk should be discussed, and a plan
about disclosure or nondisclosure In this case, we would recommend
a genetic test that the assumed father
should be in place before testing.”5 In that the genetic counselor fully
is likely not the biological father.1
2013, the American College of disclose the genetic results, including
There is controversy within the
Genetics and Genomics technical carrier status and noncarrier status,
bioethical, medical, and research
report on the ethical and policy issues but not address parentage. This
communities on how to handle this
in genetic testing and screening of provides the couple with the accurate
difficult situation, and it is largely
children recommended that when information needed to make
considered an unsettled debate. Some
findings are suggestive of autonomous reproductive choices.
even argue that the power and great
The genetic counselors will have
burden in such a decision “exceeds misattributed paternity, it must be
verified by formal testing.6 upheld their duty to tell the truth, as
the moral authority vested in the
the couple is fully informed of the
profession.”2
Palmor and Fiester7 have advocated genetic risks carried by any future
The purpose of the genetic testing in a method to avoid this conflict progeny the couple may have
this case is twofold. First, it serves to altogether by adopting a policy of together. This would also inform the
elucidate the genetic mutations in the universal nondisclosure. They argue man and the woman of their personal
pediatric patient. Secondly, it that if parents want further genetic risk should they have children
determines the reproductive and information of parentage, then they with other partners.
genetic risk of the couple’s future should be counseled to obtain it via
offspring. The informed consent elective parentage testing.7 Consanguinity raises concern for
process should include informing the potential sexual abuse or incest.3 The
parents that the test may reveal Lucast8 maintains that respect for mother should be directly informed
misattributed paternity.3,4 The finding equality can be honored and that, of the misattributed paternity given
of misattributed paternity is when possible, a counselor should that there exists concern for her well-
incidental in the sense that it is not provide the man with his own genetic being and safety as well as the safety
information sought by the parties information without mentioning the of the child. The concern for
ordering the test or by the parties issue of misattributed paternity. If consanguinity, and potential abuse or
being tested. It represents an that is impossible, the counselor will incest, should be discussed with the
asymmetric risk to the mother in that have to weigh the risks of disclosure mother alone in a sensitive and
only she is at risk for being exposed to the mother and to the child.8 supportive manner with the focus on
of having an extramarital the well-being and safety of the
There are no empirical data to mother and child and their
relationship.3,4
support the likely outcome of environment. Proper social and
Opinions about the duty to disclose disclosure.9 Therefore, it is emotional supports should be offered
vary among professional societies and impossible to predict whether and made available to the mother to
bioethicists. In 1994, the Committee disclosure will have a positive or help her exercise her autonomy in
on Assessing Genetic Risks of the negative effect on the relationship of determining how to best address this
Institute of Medicine (now the the partners to each other or the information in the context of her
National Academy of Medicine) relationship of the partners to the relationship.
recommended that genetic testing child.5 Ross10 maintains that both
should not be disruptive to the family, partners should be informed of the Is there a duty to find and inform the
and therefore, in most cases, only the results of misattributed parentage biologic father, who is a carrier, and
mother should be informed of this because genetic counselors have how do we balance the need to
result.4 In 2015, the American Society a moral duty of truth telling and preserve patient confidentiality of the
of Human Genetics position statement avoiding active deception of their mother? There have been a number of
recommended that health care clients. In regard to tests used by court cases in which health care
providers avoid disclosure of transplant surgeons to determine providers have been found
misattributed parentage unless there living donor and recipient responsible for failure to warn third
is a clear medical benefit that compatibility, Ross11 argues that parties of harm.12,13 The Standards
outweighs the potential harms.3 those tests are not adequate to for Privacy of Individually Identifiable
Downloaded from www.aappublications.org/news by guest on February 8, 2021
2 PRERO et alHealth Information regulate health health professionals must consider him includes accurate knowledge of
care information disclosures. There whether child abuse has occurred. his carrier status of a CLDN1 variant,
are public interest exceptions, They also must know whether such and therefore his risk of having future
specifically when there is a serious pairings are legal in the family’s affected children, which could have
and imminent threat to the health or jurisdiction. An American Society of long-term consequences on his
safety of a third party.14 In this case, Human Genetics position statement reproductive decisions. Potential
the seriousness and immanency of from 2015 differentiates legal harms of disclosure include the
the threat of the biologic father’s obligations, “Practitioners have a duty fracturing of this family.
genetic makeup is unclear. Moreover, to report suspected child abuse.
because the identity of the biologic Health care providers do not have It could be argued that truthful and
father is unknown to the health care a responsibility to report incest appropriate counseling could be
team, it can be argued that they are involving consenting adults, even provided to Rebecca’s putative father
under no obligation to seek him out. though this might be illegal in their without disclosing the presumed
However, depending on the jurisdiction.” Given that Rebecca’s misattributed paternity. This could be
circumstances, the mother could be mother was not a minor at the time accomplished by simply informing
encouraged to tell the biologic father Rebecca was conceived, the medical him that he did not have the same
the truth and encourage him to obtain team is not obligated to report the CLDN1 variant that was identified in
genetic counseling. consanguinity to any regulatory the child. Although rare in autosomal
bodies. If no abuse is reported by the recessive disease, genetic mutations
Ms Strenk Comments mother, they are not obligated to can arise spontaneously. Additionally,
report consanguinity even if such gonadal mosaicism, in which a person
There are 2 primary issues here. First
a relationship is illegal in their has a gene mutation only in the egg or
is the issue of consanguinity; second
jurisdiction. sperm cell, is a well-described
is the issue of misattributed paternity.
phenomenon. Thus, from a genetic
Both of these issues raise questions
Regarding the issue of nonpaternity, and/or biologic perspective, it would
about disclosure. What should the
the American Society of Human not be inaccurate to tell the putative
health professionals do with this
Genetics position article from 2017 father that although he did not have
information? Finally, a secondary
estimates misattributed paternity to the same genetic variant identified in
issue is related to what, if anything, to
occur at a rate between 1% and 10%. the child, there are reasonable, if rare,
tell Rebecca’s biological father.
They recommend pretest counseling explanations for how and why this
Ideally, whenever a patient is about this possibility but discourage could occur, including but not limited
considering genetic testing, pretest providers from disclosing such to misattributed paternity.
counseling is done to provide the results in the absence of clear medial
patient and/or family with an Were I the genetic counselor working
benefit that outweighs the potential with this family, I would hesitate to
opportunity to be fully informed of harms. Thus, in our case, we must
the possible results of the testing. discuss the consanguinity with the
measure the medical benefit of putative father, as this could be
This discussion would include
disclosure against the potential harm. a breach of Rebecca’s mother’s
information about the possibility of
There is no medical benefit to confidentiality (in revealing
incidental findings. These include
Rebecca of informing the putative information of her previous sexual
consanguinity and misattributed
father of the misattributed paternity. history). Rebecca’s mother has been
paternity. From the information
Her diagnosis will not change informed of the microarray result,
provided, it is not clear whether this
whether her putative father has this including its suggestion of
discussion occurred with Rebecca’s
information. Potential harms to consanguinity, and she has declined
family.
Rebecca of disclosing include losing opportunities to talk more about it. I
Regarding the issue of consanguinity, her home, her medical insurance, her would inform her that the test results
Rebecca’s microarray indicated father, and a stable caregiver (should are accessible in the medical record
a third- or fourth-degree relationship her mother have to return to work). to health care providers and the
between her biological parents. This Harms of nondisclosure include loss putative father (who is still
suggests that, at most, Rebecca was of trust in the medical team should presumably the legal father) and that
the product of a pregnancy between her father discover this information should he have questions about this
first cousins. Although unusual in in the future. However, although result, the health care team would
most settings in the United States, Rebecca is technically our patient, in answer them truthfully.
first-cousin marriages are not genetics, we “treat” families, and her
unusual worldwide. Before putative father is also a stakeholder Additionally, I would inform both
considering the genetic implications, in this decision. Medical benefit to parents that Rebecca’s diagnosis of
Downloaded from www.aappublications.org/news by guest on February 8, 2021
PEDIATRICS Volume 143, number 6, June 2019 3CLDN1-related NISCH has been The key here is recognizing the need Instead, the medical team would do
confirmed by the identification of for a procedural approach to far better to work with and through
homozygous CLDN1 variants. I would decision-making. Such an approach the person who is clearly at the
counsel that Rebecca’s mother is would not seek to determine the best center of these disclosure decisions:
a carrier for 1 of these variants and solution. Instead, it would focus on Rebecca’s mother. She is the person
that her putative father is not. Thus, identifying and empowering the agent most likely to remain central to
should they have future pregnancies, who has moral authority to make Rebecca’s caregiving throughout her
their risk of recurrence would be low. decisions. The rationale for childhood. She is the person through
If the family had additional questions a procedural approach stems from whom any effective disclosure
about where the second CLDN1 the nature of the ethical questions process is going to need to work
variant came from, I would discuss being asked here. These questions do (particularly in identifying the likely
the various reasons a mutation may not directly pertain to or impact the biological father). And she has the
be present in a child but not the clinical management of the patient. most at stake in terms of the
father, including spontaneous Instead, they concern interests of the consequences of the decision.
mutation, gonadal mosaicism, and patient’s parents. Thus, this is not the
misattributed paternity. Additional kind of case in which the traditional There may well be a moral obligation
resources for official paternity testing principle of pediatric ethics (the best to disclose nonpaternity to the
could be offered if the family desired interest standard) provides guidance. putative father and (consanguine)
such testing. That is not what this is about. paternity to the biological father. But
Rebecca’s clinical care will be that does not mean that the moral
Finally, Rebecca’s biological father is obligation falls on the health
managed the same regardless of
assumed to be a carrier for the CLDN1 professionals. The moral obligation,
whether either the putative or
variant and may be unaware of this. I or the choice whether to perceive
biological father is informed by her
would encourage the mother to disclosure as a moral obligation, falls
mother or the clinicians about the
inform Rebecca’s biological father to Rebecca’s mother. Clinicians cannot
circumstances of her conception.
about his presumed carrier status insist that parents always fulfill their
and the option for carrier testing. I There are, of course, legitimate moral obligations even when these
would not take further action to track concerns about the wider pertain to their children who are
down or inform the absent parent. psychosocial and relational impacts of patients (ie, donate organs to save the
disclosure decisions in the case. child’s life). Clinicians certainly
Dr Garrett Comments However, these concerns are mostly cannot insist that parents fulfill their
An old proverb cautions that hard hypothetical, and they extend in every perceived moral obligations to adult
cases make for bad ethics. This is conceivable direction. If the clinicians nonpatients.
wise counsel. When encountering unilaterally disclose the truth to the
a highly idiosyncratic or genuinely putative father, will he abandon Understanding and appreciating the
dilemmatic case, we do well to resist Rebecca and her mother, leaving them right procedure here (that Rebecca’s
the temptation to abandon or without an income and without mother is the moral agent and any
fundamentally revise what generally health insurance? Potentially. But is obligations to disclose paternity are
works well. The world is a strange not this also a possibility if the hers) provides a basis for answering
place. No conceptual toolbox is uninformed putative father later the medical team’s questions. The
equipped to fully resolve all potential decides to consult Dr Google, examine medical team should not unilaterally
problems that might arise. It is better the medical record, or overhear disclose nonpaternity or
to accept the limitations of a fairly indiscrete clinicians discussing the consanguinity to either the putative
simple but generally useful toolbox case? These possibilities seem just as or biologic father.
than to outfit oneself with an likely (indeed, even the most cursory
Instead, clinicians should continue to
unwieldy set of ad hoc instruments. Internet search for Rebecca’s
counsel Rebecca’s mother to
extremely rare syndrome reveals the
At first glance, the case under recognize and appreciate the
high likelihood of consanguinity and,
consideration here looks like considerable case for disclosure and
hence, nonpaternity).
a paradigm example of just this kind risks of nondisclosure to the putative
of hard case: nonpaternity, Trying to make an unambiguous father. They should first seek to
consanguinity, rare disease, young determination of whether Rebecca’s determine how well she understands
unmarried couple, and so on. wider psychosocial and relational the decision and what worries she
However, despite all the moving parts, interests are better served by has about the consequences. Then,
the case may be more straightforward disclosure to 1 or both of the adult they should make sure that any
than it first appears. male stakeholders is a fool’s errand. misunderstandings are addressed
Downloaded from www.aappublications.org/news by guest on February 8, 2021
4 PRERO et aland all relevant scenarios considered educate patients and parents about of children. Pediatrics. 2013;131(3):
in advance. They also must make sure the possibility of incidental findings, 620–622
the mother knows that they will not including misattributed paternity. 6. Ross LF, Saal HM, David KL, Anderson
lie if asked about the nature and Then, patients and parents can make RR; American Academy of Pediatrics;
implications of Rebecca’s condition, their own informed choices about American College of Medical Genetics
so she needs to weigh this possibility, whether to seek such information and and Genomics. Technical report: ethical
and many others, in her decision- how to interpret test results. and policy issues in genetic testing and
making. The medical team should screening of children [published
All of the cases in Ethics Rounds are correction appears in Genet Med. 2013;
discuss concerns about consanguinity based on real events. Some incorporate
and nonconsensual intercourse with 15(4):321]. Genet Med. 2013;15(3):
elements of a number of different cases 234–245
Rebecca’s mother, especially because in order to better highlight a specific
Rebecca herself may be at risk for 7. Palmor M, Fiester A. Incidental findings
ethical dilemma.
sexual assault in the future. This of nonparentage: a case for universal
should, of course, be done with nondisclosure. Pediatrics. 2014;134(1):
considerable care and skill, but there 163–168
ABBREVIATION
is no overriding reason not to have 8. Lucast EK. Informed consent and the
these discussions, and there are many NISCH: neonatal ichthyosis- misattributed paternity problem in
good reasons to do so. sclerosing cholangitis genetic counseling. Bioethics. 2007;
syndrome 21(1):41–50
At the end of the day, however,
disclosure is Rebecca’s mother’s 9. Lucassen A, Parker M. Revealing false
paternity: some ethical considerations.
decision to make. Clinicians do not
Lancet. 2001;357(9261):1033–1035
have the moral authority to REFERENCES
unilaterally guarantee that the “right” 10. Ross LF. Disclosing misattributed
decision gets made. They do have the 1. Lowe G, Pugh J, Kahane G, et al. How paternity. Bioethics. 1996;10(2):114–130
should we deal with misattributed
moral obligation to maintain an 11. Ross LF. Good ethics requires good
paternity? A survey of lay public
ongoing supportive relationship with attitudes. AJOB Empir Bioeth. 2017;8(4): science: why transplant programs
the primary decision-maker. In cases 234–242 should not disclose misattributed
like this, that is the best we can do. parentage. Am J Transplant. 2010;10(4):
2. Chandler J. Incidental findings of 742–746
Dr Lantos Comments nonparentage should be disclosed.
Pediatrics. 2015;135(1). Available at: 12. Weaver M. The double helix: applying an
Most of the ethical dilemmas www.pediatrics.org/cgi/content/full/ ethic of care to the duty to warn
associated with genetic testing 135/1/e284.1 genetic relatives of genetic information.
existed long before we could Bioethics. 2016;30(3):181–187
3. Botkin JR, Belmont JW, Berg JS, et al.
sequence genomes.15 Misattributed 13. McAbee GN, Sherman J, Davidoff-
Points to consider: ethical, legal, and
paternity has been around forever. psychosocial implications of genetic Feldman B. Physician’s duty to warn
What has changed today is that testing in children and adolescents. Am third parties about the risk of genetic
patients have greater access to J Hum Genet. 2015;97(1):6–21 diseases. Pediatrics. 1998;102(1, pt 1):
genetic information. They can look at 140–142
4. Andrews LB, Fullarton JE, Holtzman NA,
their own health records. They can Motulsky AG, eds. Assessing Genetic 14. Offit K, Groeger E, Turner S, Wadsworth
seek information about their own Risks: Implications for Health and EA, Weiser MA. The “duty to warn”
genomes without the need for Social Policy. Washington, DC: Institute a patient’s family members about
a physician intermediary. This lessens of Medicine; 1994 hereditary disease risks. JAMA. 2004;
the obligation of doctors to disclose 292(12):1469–1473
5. Committee on Bioethics; Committee on
information today compared with the Genetics; American College of Medical 15. Hercher L, Jamal L. An old problem in
days when only doctors had access to Genetics; Genomics Social, Ethical, Legal a new age: revisiting the clinical
that information. It also creates a new Issues Committee. Ethical and policy dilemma of misattributed paternity.
and important role for doctors: to issues in genetic testing and screening Appl Transl Genomics. 2016;8:36–39
Downloaded from www.aappublications.org/news by guest on February 8, 2021
PEDIATRICS Volume 143, number 6, June 2019 5Disclosure of Misattributed Paternity
Moshe Y. Prero, Meghan Strenk, Jeremy Garrett, Ann Kessler, Jonathan M. Fanaroff
and John D. Lantos
Pediatrics 2019;143;
DOI: 10.1542/peds.2018-3899 originally published online May 16, 2019;
Updated Information & including high resolution figures, can be found at:
Services http://pediatrics.aappublications.org/content/143/6/e20183899
References This article cites 14 articles, 4 of which you can access for free at:
http://pediatrics.aappublications.org/content/143/6/e20183899#BIBL
Subspecialty Collections This article, along with others on similar topics, appears in the
following collection(s):
Ethics/Bioethics
http://www.aappublications.org/cgi/collection/ethics:bioethics_sub
Genetics
http://www.aappublications.org/cgi/collection/genetics_sub
Permissions & Licensing Information about reproducing this article in parts (figures, tables) or
in its entirety can be found online at:
http://www.aappublications.org/site/misc/Permissions.xhtml
Reprints Information about ordering reprints can be found online:
http://www.aappublications.org/site/misc/reprints.xhtml
Downloaded from www.aappublications.org/news by guest on February 8, 2021Disclosure of Misattributed Paternity
Moshe Y. Prero, Meghan Strenk, Jeremy Garrett, Ann Kessler, Jonathan M. Fanaroff
and John D. Lantos
Pediatrics 2019;143;
DOI: 10.1542/peds.2018-3899 originally published online May 16, 2019;
The online version of this article, along with updated information and services, is
located on the World Wide Web at:
http://pediatrics.aappublications.org/content/143/6/e20183899
Pediatrics is the official journal of the American Academy of Pediatrics. A monthly publication, it
has been published continuously since 1948. Pediatrics is owned, published, and trademarked by
the American Academy of Pediatrics, 345 Park Avenue, Itasca, Illinois, 60143. Copyright © 2019
by the American Academy of Pediatrics. All rights reserved. Print ISSN: 1073-0397.
Downloaded from www.aappublications.org/news by guest on February 8, 2021You can also read
