The Principle Methods of Identifying Twins for Research

1
STARTING FROM SCRATCH IN SRI LANKA
Sisira Siribaddana Sri Lankan Twin Registry
2
Sri Lanka

• Has a literacy rate over 92.5 in males and 87.9
  in females.
• The life expectancy is around 74 years. Crude
  death rate is 5.9 per 1000 population and
  maternal mortality rate of 3.5 per 10,000 live
  births.
• Infant mortality is 15.4 per 1000 live births. In
  the world health report 2000,
• Sri Lanka is ranked at 76 in overall performance
  compared to India 112 and china 144 (WHO, 2000).

3
Background

• We had absolutely nothing other than interest
• determination
• courage
• experience left behind by the giants in
• the field
• Basic knowledge on the subject was vital, but
  relevant books / journals were scarce in Sri Lanka

4
The way forward - 1

• Networking with experienced twin
• researchers.
• Collaboration with the west
• A partnership model based on mutual
• respect, trust and scientific benefits
• not
• on economically cheaper and easier research
  in the developing world. (outsourcing)

5
The way forward - 2

• A multi-disciplinary team back home
• Awareness raising on the concept
• of twin research
• Twins beyond obstetricians and
• paediatricians.

6
Funding was the hardest 90/10 divide in
research funding International funding
without prior work in twin research and
credentials A serious set back for a major
initiative
Funds
7
No charitable local research funds
Compelled to seek help from commercial
establishments. Potential conflicts of
interests Registry - academic interest Vs
Business establishment commercial interest
A letter writing campaign seeking assistance
support from leading newspapers in
Sri Lanka
Local Funds
8
Debate on a donation - a goodwill gesture -
from abroad Potential dangers and abuses
Concerns exporting DNA ownership of
data, DNA and national heritage
ethical standards on the ownership However we
were clear on our mission.
Concerns and the purists
9
Proactive than reactive A priority No
existing ethical framework for genetic and
twin research to safeguard the Sri Lankan
interests Moral rights and wrongs are
not absolute but vary with the culture
Development of guidelines on bio-ethics
10
Policies guidelines

• Funding and accepting donations
• Collection and access to the twin database
• Ownership of human biological material
• Commercial exploitation and patenting,
• Consent, international collaboration and
  authorship

11
Vulnerability of participants in the
developing world Asymmetry in knowledge and
authority between researchers and
participants Authoritative position held by
the clinicians Registry to ensure freely
given informed consent and ombudsman to be
present More important with illiterate subjects
Informed Consent
12
What have we done ?

• TWIN STATSTICS
• Volunteer cohorts
• Birth record based regional cohorts
• Community based cohort
• Birth record based whole island cohort

13

National Volunteer Register
Quads



02

02



Twin Research 2000 3 202-202.
14
Maternity Hospitals cohorts (Based on Birth
Registers)

• Can trace young twins
• Twin Research 2001 4459-463
• Difficult to trace older twins
• Twin Research 2003 667-71

15
A cohort of twins traced from the Registrar
Generals Department of Births and Deaths
Registration - Based on birth records
2500-3500 twins per year.
Whole Island younger twin cohort (1992-1997)
16
Whole Island prospective infant cohort (2001- .)

• The Registrar General of Births and Deaths
  Registration officially agreed to provide the
  details of multiple births on a monthly return
  basis from 2001.
• Around 200-300 twins a month.

17

What else have we done ?

• Adaptation and preliminary validation of a
  zygosity questionnaire, used a novel method for
  adaptation
• (Twin Research 2000 3 205-212)

18
Adaptation and validation was carried out in 3
steps. Firstly, we used a nominal group to
translate the English version and to assess the
extent of agreement (consensus measurement) on
the appropriateness of the translation and
resolve disagreement (consensus development).
19
Secondly we used a qualitative interview with 25
mothers of twins. The 3 main stems of the
translated questionnaire were used as a
semi-structured interview. The responses were
noted verbatim. These were categorised and
analysed. Then the translated full
questionnaire, was presented as closed questions
with fixed choice responses. The categorised
responses generated during the qualitative
interview were compared with the responses to the
fixed choices in the full questionnaire.
20
Third step was the appraisal of the questionnaire
by seventeen bilingual parents of twins. The
source and translated version of the
questionnaire were given to them at least 2 days
apart. The responses were rated and the total
scores were computed to determine the zygosity.
This step was carried out to measure the
validity and reliability of the Sinhala version.
Zygosity by the source and adapted
questionnaires revealed a perfect correlation
between the original and adapted version with a
kappa of 1.
21
Question number one The stem of the original
questionnaire was, were your twins as like as
two peas in a pod. It needed a culturally
meaningful translation because it is an idiom.
One phrase proposed by a panellist ( EKA VALLE
POLL- in Sinhala) could be back translated as
like coconuts of the same bunch. Although it
conveyed a meaning closer to the original (have a
semantic validity), the group decided against it.
The reason was its idiomatic meaning did not fit
the original construct. This idiom in Sinhala is
used with somewhat negative connotation to
introduce people with similar attitudes and
character rather than the physical resemblance.
(People of the same boat)
22
The alternative suggested by another panellist
( KAPAPU PALUA WAGE- in Sinhala) could be back
translated as Like the split half of. The
significance of this idiomatic phrase is, its
usage by lay Sinhala indicate similarity between
two persons. Therefore the group, agreed it was
the best culturally meaningful, idiomatic phrase
that represent the semantic equivalent of the
peas of the pods.
23
Original question were your twins as like as
two peas in a pod. The Sinhala equivalent back
translated by the group is were your twins so
similar and difficult to differentiate one from
the other, as if one was the split half of the
other.
24
Multiple Birth Foundation Sri Lanka

• To raise awareness on their unique issues faced
  by the twins, the multiples and their families
• For service development based on research
  findings
• To work with professionals, statutory
  organisations to initiate service development
• To represent the interests of multiples

25
Our vision

• Is to establish a center of excellence for
  twin, sibling, family studies and genetic studies
  for the region, which brings together
  researchers from diverse disciples to carry out
  research.
• Twin Research 2000 3 202-202.

26
Sri Lankan Twin Registry
SLTR is an independent academic and research
institution founded with the aim of establishing
a register for twins in Sri Lanka, to facilitate
the study of twins. Twin Research 2002 5
424-426
27
Our mission

• Capacity building
• Establishing a genetic lab
• Carry out twin research
• Extend the collaborative links

28
CMD project

• Twin and singleton study of common mental
  disorders Suicidal Ideations Alcohol intake
  (18-65 years)
• 4000 twins 1000 singletons
• Unprecedented ethical approval WHO, IOP (UK),
  SJU (SL)
• 550 000 households surveyed

29
CMD project

• 11 000 twins ascertained
• WHO manuals and questionnaires translated
• Instruments developed by qualitative methods
• Molecular biology lab
• Funded by Wellcome trust

30
Administrative Structure of the Island
31
As Newton said, If we see further that is
because we stand on the shoulders of giants.
32
THANK YOU