An alternative strategy for perinatal verbal autopsy coding: single versus multiple coders

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An alternative strategy for perinatal verbal autopsy coding: single versus multiple coders C. Engmann 1 , I. Jehan 2 , J. Ditekemena 3 , A. Garces 4 , M. Phiri 5 , M. Mazariegos 6 , E. Chomba 5 , O. Pasha 2 , A. Tshefu 3 , E. M. McClure 7 , V. Thorsten 7 , H. Chakraborty 7 , R. L. Goldenberg 8 , C. Bose 1 , W. A. Carlo 9 and L. L. Wright 10 1 University of North Carolina, Chapel Hill, NC, USA 2 The Aga Khan University, Karachi, Pakistan 3 Kinshasa School of Public Health, Kinshasa, Democratic Republic of Congo 4 IMSALUD San Carlos University, Guatemala City, Guatemala 5 University Teaching Hospital, Lusaka, Zambia 6 Institute of Nutrition for Central America and Panama, Guatemala City, Guatemala 7 Research Triangle Institute, Durham, NC, USA 8 Drexel University, Philadelphia, PA, USA 9 University of Alabama, Birmingham AL, USA 10 Eunice Kennedy Shriver National Institute of Child Health and Human Development, Bethesda, MD, USA Summary objective To determine the comparability between cause of death (COD) by a single physician coder and a two-physician panel, using verbal autopsy. methods The study was conducted between May 2007 and June 2008. Within a week of a perinatal death in 38 rural remote communities in Guatemala, the Democratic Republic of Congo, Zambia and Pakistan, VA questionnaires were completed. Two independent physicians, unaware of the others decisions, assigned an underlying COD, in accordance with the causes listed in the chapter headings of the International classification diseases and related health problems, 10th revision (ICD-10). Cohen’s kappa statistic was used to assess level of agreement between physician coders. results There were 9461 births during the study period; 252 deaths met study enrolment criteria and underwent verbal autopsy. Physicians assigned the same COD for 75% of stillbirths (SB) (K = 0.69; 95% confidence interval: 0.61–0.78) and 82% early neonatal deaths (END) (K = 0.75; 95% confidence interval: 0.65–0.84). The patterns and proportion of SBs and ENDs determined by the physician coders were very similar compared to causes individually assigned by each physician. Similarly, rank order of the top five causes of SB and END was identical for each physician. conclusion This study raises important questions about the utility of a system of multiple coders that is currently widely accepted and speculates that a single physician coder may be an effective and economical alternative to VA programmes that use traditional two-physician panels to assign COD. keywords verbal autopsy, perinatal death, comparing coders Introduction Understanding population-based causes of perinatal death (stillbirths and newborn deaths in the first 7 days of life) is critical to the development of an effective perinatal health policy (Lopez & Mathers 2006). Because there will always be competing demands for healthcare resources, a well- established system for identifying all perinatal deaths and assigning a medically determined cause of death (COD) for each death is highly desirable (Engmann et al. 2009b). In many high-income countries, there is complete recording of deaths and for over 90% of these, medical certification is provided (Mathers et al. 2005). By contrast, fewer than 3% of all perinatal deaths in low- and middle-income countries (LMIC) have medical certification of COD (Lawn et al. 2005). Many of these countries have the highest burden of poverty and disease and continue to lack routine, representative and high-quality information on the levels and causes of death (Setel et al. 2007). Part of the explanation for this may be that over half of all births and perinatal deaths occur in the home and are frequently unrecorded in vital registration systems (Lawn et al. 2008). Increasing numbers of LMIC are using verbal autopsy (VA) as a cost-effective and sustainable alternative to a Tropical Medicine and International Health doi:10.1111/j.1365-3156.2010.02679.x volume 16 no 1 pp 18–29 january 2011 18 ª 2010 Blackwell Publishing Ltd

Transcript of An alternative strategy for perinatal verbal autopsy coding: single versus multiple coders

An alternative strategy for perinatal verbal autopsy coding:

single versus multiple coders

C. Engmann1, I. Jehan2, J. Ditekemena3, A. Garces4, M. Phiri5, M. Mazariegos6, E. Chomba5, O. Pasha2, A. Tshefu3,

E. M. McClure7, V. Thorsten7, H. Chakraborty7, R. L. Goldenberg8, C. Bose1, W. A. Carlo9 and L. L. Wright10

1 University of North Carolina, Chapel Hill, NC, USA2 The Aga Khan University, Karachi, Pakistan3 Kinshasa School of Public Health, Kinshasa, Democratic Republic of Congo4 IMSALUD ⁄ San Carlos University, Guatemala City, Guatemala5 University Teaching Hospital, Lusaka, Zambia6 Institute of Nutrition for Central America and Panama, Guatemala City, Guatemala7 Research Triangle Institute, Durham, NC, USA8 Drexel University, Philadelphia, PA, USA9 University of Alabama, Birmingham AL, USA10 Eunice Kennedy Shriver National Institute of Child Health and Human Development, Bethesda, MD, USA

Summary objective To determine the comparability between cause of death (COD) by a single physician coder

and a two-physician panel, using verbal autopsy.

methods The study was conducted between May 2007 and June 2008. Within a week of a perinatal

death in 38 rural remote communities in Guatemala, the Democratic Republic of Congo, Zambia and

Pakistan, VA questionnaires were completed. Two independent physicians, unaware of the others

decisions, assigned an underlying COD, in accordance with the causes listed in the chapter headings of

the International classification diseases and related health problems, 10th revision (ICD-10). Cohen’s

kappa statistic was used to assess level of agreement between physician coders.

results There were 9461 births during the study period; 252 deaths met study enrolment criteria and

underwent verbal autopsy. Physicians assigned the same COD for 75% of stillbirths (SB) (K = 0.69;

95% confidence interval: 0.61–0.78) and 82% early neonatal deaths (END) (K = 0.75; 95% confidence

interval: 0.65–0.84). The patterns and proportion of SBs and ENDs determined by the physician coders

were very similar compared to causes individually assigned by each physician. Similarly, rank order

of the top five causes of SB and END was identical for each physician.

conclusion This study raises important questions about the utility of a system of multiple coders that

is currently widely accepted and speculates that a single physician coder may be an effective and

economical alternative to VA programmes that use traditional two-physician panels to assign COD.

keywords verbal autopsy, perinatal death, comparing coders

Introduction

Understanding population-based causes of perinatal death

(stillbirths and newborn deaths in the first 7 days of life) is

critical to the development of an effective perinatal health

policy (Lopez & Mathers 2006). Because there will always

be competing demands for healthcare resources, a well-

established system for identifying all perinatal deaths and

assigning a medically determined cause of death (COD) for

each death is highly desirable (Engmann et al. 2009b). In

many high-income countries, there is complete recording of

deaths and for over 90% of these, medical certification is

provided (Mathers et al. 2005). By contrast, fewer than

3% of all perinatal deaths in low- and middle-income

countries (LMIC) have medical certification of COD

(Lawn et al. 2005). Many of these countries have the

highest burden of poverty and disease and continue to lack

routine, representative and high-quality information on the

levels and causes of death (Setel et al. 2007). Part of the

explanation for this may be that over half of all births and

perinatal deaths occur in the home and are frequently

unrecorded in vital registration systems (Lawn et al. 2008).

Increasing numbers of LMIC are using verbal autopsy

(VA) as a cost-effective and sustainable alternative to a

Tropical Medicine and International Health doi:10.1111/j.1365-3156.2010.02679.x

volume 16 no 1 pp 18–29 january 2011

18 ª 2010 Blackwell Publishing Ltd

thorough medical diagnostic evaluation as a source of data

to inform mortality surveillance systems (Hill et al.

2007).To determine the cause of foetal or infant mortality,

the VA method relies on information obtained from a

standardized interview with the primary caregiver (usually

the mother) of the deceased. During this process, the

symptoms, signs and behaviours during the illness of the

deceased, or of the mother in the case of foetal death, are

recorded. Trained coders review these data and apply

diagnostic algorithms to determine COD. Typically, two or

three trained physician coders review the data and inde-

pendently assign a COD (Soleman et al. 2006). Any

discrepancies between the COD assigned by each physician

member of the panel are resolved by discussion and review

of the VA data, and a final consensus COD is agreed upon

by the physician panel (Setel et al. 2005). The use of

multiple physician coders in VA has been used to prevent

random and systematic errors. Some researchers have urged

that physicians should be encouraged to assign more than

just a single COD, and that discussion of discrepant cases

among a panel of physicians to reach consensus be

considered less appropriate than allowing all physician

diagnoses to contribute to the COD profile, whether

individual physician diagnoses agree (Joshi et al. 2009b).

Other authors have suggested methods for simultaneous

analysis of COD (King & Lu 2008). Alternatively, COD can

be assigned by the use of predetermined criteria ⁄ algorithms

or computer simulations, a method that does not require the

presence of a physician (Soleman et al. 2006).

A recent report from a general population in India

suggests that one trained physician determining COD

facilitated by a series of algorithms developed for the

Sample Registration System may be as effective as a

physician panel in coding COD (Joshi et al. 2009a,b). We

sought to determine the potential effectiveness of using a

single physician coder to assign the cause of perinatal

deaths by comparing COD assigned by two members of a

physician panel. Each panel was based in rural districts in

one of four low-income countries.

Methods

Setting, subjects and study design

This prospective observational study was nested within an

ongoing, cluster randomized, controlled trial, the FIRST

BREATH Trial, conducted by the Global Network

McClure et al. 2007). The FIRST BREATH trial investi-

gated the effects of implementing a package of newborn

care practices, using the WHO Essential Newborn Care

(ENC) programme, and a neonatal resuscitation training

programme, a simplified version of the American Academy

of Pediatrics Neonatal Resuscitation Program, in commu-

nity settings. As part of this study, birth attendants were

trained to collect basic maternal, foetal and neonatal

outcomes data, which included demographics, mode of

delivery, birthweight, gestational age, receipt of resuscita-

tion and adverse events. All birth attendants were trained

to check for foetal and neonatal vital signs on every baby

by auscultating the abdomen of every pregnant woman

before delivery, and after delivery by feeling the umbilical

cord of the neonate for a pulse, auscultating lungs for

breath sounds and assessing for any movement (Engmann

et al. 2009a,b). Birth weights were measured within 48 h

of delivery using UNICEF spring Salter Scales (UNICEF

model 145555) provided for the study.

This study included sites in Guatemala (Chimaltenango

province), the Democratic Republic of Congo (Equateur

province), Zambia (Kafue district) and Pakistan (Thatta

district). Within these sites, 38 communities participated in

this study. Each community comprised a cluster of villages

with approximately 300 deliveries per year. Data describ-

ing births were collected by birth attendants and reviewed

by trained nurses or health workers assigned to each

community and designated as Community Coordinators.

Within 1 week of an early neonatal death (END) or

stillbirth (SB), birth attendants notified Community Coor-

dinators who then visited the family, determined eligibility

for the study and requested consent from eligible mothers.

Perinatal deaths were excluded if they occurred in a

hospital, if a birth attendant was absent at delivery, if the

mother was unavailable for any reason (including peri-

partum death) or attempts to enrol the mother did not

occur within 7 days of death. A 7-day window within a

perinatal death was chosen to reduce the variability in the

quality of reporting introduced by recall bias (Soleman

et al. 2006; Lee et al. 2008; Fottrell & Byass 2010).

Because the conventional perinatal verbal autopsy respon-

dents are mothers, we elected to enrol only those subjects

whose mothers were available for interview. Informed

consent was obtained from mothers in a private and

confidential setting. The consent form was read to all

mothers who then provided their signatures or, if they were

illiterate, thumbprints.

Training and VA methodology

All Community Coordinators and physicians participat-

ing in this study received standardized training in VA

methodology (Engmann et al. 2009a). Community

Coordinators were trained to interview mothers using the

VA questionnaire. To assign COD, physicians were

trained in ICD-10 classification, rules and guidelines

(WHO 2005).

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C. Engmann et al. Perinatal verbal autopsy

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Uniform data describing the circumstances surrounding

a perinatal death were collected from each mother using a

standardized VA questionnaire developed specifically for

this study from a validated VA tool (Engmann 2009a;

Mswia et al. 2006. See Appendices 1 & 2). The question-

naire was administered by the Community Coordinators

who then sent these data separately to two local physicians

who independently assigned a COD. All physicians were

provided with demographic and other descriptive data

collected as part of the FIRST BREATH Trial. Each

physician assigned one underlying COD, a final COD and

contributing causes of death. Underlying COD was defined

as the single most important disease or condition that

initiated the train of morbid events leading directly to

foetal or neonatal death. Underlying COD was assigned in

a non-hierarchical manner by physicians familiar with

prevailing local diseases and health conditions ⁄ patterns

(Thatte et al. 2009). After the COD was assigned and

entered independently, any discrepancy in assignment of

COD between physicians was discussed and a consensus

underlying COD assigned. In all cases, the two physicians

were able to reach consensus after discussion.

Data collection and analysis

Data were collected between May 2007 and June 2008.

Data were entered and transmitted electronically to the

data coordinating centre (RTI: Research Triangle Institute

International, Research Triangle Park, NC, USA) where

data edits, including inter- and intra form consistency

checks, were performed. The study was reviewed and

approved by the institutional ethics review committees of

the Research Triangle International, the University of

North Carolina at Chapel Hill and local institutional

review boards.

The level of agreement between physician coders for

underlying COD was calculated using Cohen’s kappa

statistic (K). Levels of agreement based on ranges of kappa

values were defined as follows: 0.81–0.99 almost perfect

agreement, 0.61–0.80, substantial agreement, 0.41–0.6

moderate agreement and <0.4 slight to fair agreement.

(Viera & Garrett 2005). Data were analysed using sas

(SAS ⁄ STAT� Software version 9.0). Descriptive statistics

were generated for participant demographics and circum-

stances surrounding the deaths. Relationships between

categorical variables were evaluated by examining cross-

tabulations. Relationships between continuous variables

were evaluated by examining means, standard deviations,

medians and ranges.

Results

There were 9461 infants born in the designated commu-

nities during the study period (Figure 1). Among these,

there were 518 SB and END. The SB, END and perinatal

mortality rates were 30 ⁄ 1000 births, 25 ⁄ 1000 live births

and 55 ⁄ 1000 births, respectively. Of the 518 deaths, 81

were ineligible for the study because the delivery occurred

in a hospital (79) or the birth attendant was absent at the

time of delivery (2). Among eligible deaths, 185 were not

enrolled because the mother was not available for inter-

view within 7 days after the death (145) or did not provide

consent (40). This study includes data describing deliveries

of 241 women which resulted in 252 perinatal deaths (134

SBs and 118 ENDs).

The five major causes of END were attributable to

infections (45%), birth asphyxia (26%), prematurity

(17%), tetanus (4%) congenital malformations (3%) and

other ⁄ unknown causes (5%). Major causes of SB were

attributable to infections (37%), obstructed ⁄ prolonged

labour (11%), antepartum haemorrhage (10%), prematu-

rity (7%) and cord complications such as prolapse (6%).

For 12% of SBs, a COD could not be determined.

Agreement among coders

Physician coders assigned the same COD for 82% of END

and 75% of SB. The kappa statistic for overall inter-coder

05

1015202530354045

Antepartumhemorrhage

Maternalinfection/sepsis

Pre-term Maternalaccident

Prolongedlabor

Cord prolapse/complication

Pro

port

ion

of d

eath

s (%

)

Physician 1

Physician 2

Physician consensus

Figure 1 Proportion of stillbirths assigned an underlying cause of death by each of two physician coders and by physician consensus.

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C. Engmann et al. Perinatal verbal autopsy

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agreement was 0.75 (0.65, 0.84) for END and 0.69 (0.61,

0.78) for SB.

Early neonatal death

Table 1 compares physician coder 1 and physician coder 2

responses for END. Overall, physicians agreed on the same

COD for 97 of 118 (82%) END. Table 2 is a comparison

of physician coder 1 vs. physician coder 2 responses for

specific causes of END. As an example, physicians agreed

109 times of 118 (92%) on prematurity as a COD. They

agreed that prematurity was a COD 13 times, and that

prematurity was not the COD 96 times. The kappa value

(level of agreement) between physicians was 0.7 (95% CI

0.51–0.88).

Stillbirth

Table 3 compares physician 1 and physician coder 2

responses for SB. Overall, physicians agreed on the same

Table 1 Comparison of physician coder 1 and physician coder 2 assigned cause of early neonatal death (n = 118)

Physician 1 responses

Physician 2 responses

Physician 1

Total n (%)

Physician

consensus

n (%)Preterm Infection

Birth

asphyxia

Congenital

malform Tetanus

Unknown ⁄ no

cause Other

Preterm 13 1 1 0 0 0 1 16 (14) 20 (17)

Neonatal infection 2 44 4 0 1 1 0 52 (44) 52 (44)

Birth asphyxia 3 3 28 0 0 1 0 35 (30) 31 (26)Congenital malformation 0 0 0 4 0 0 0 4 (3) 4 (3)

Tetanus 0 0 0 0 5 0 0 5 (4) 5 (4)

Unknown ⁄ no cause 0 0 0 0 0 2 0 2 (2) 4 (3)Other 1 1 1 0 0 0 1 4 (3) 2 (2)

Physician 2

Total n (%)

19 (16) 49 (42) 34 (28) 4 (3) 6 (5) 4 (3) 2 (2) 118

The bold numbers along the diagonal indicate agreement reached independently by the two physicians. Per cent agree-

ment = 97 ⁄ 118 = 82.2%. The percentages in parenthesis, provided along the Physician 1 total column, indicate how often Physician 1reported the cause of death (COD) out of the total. Similarly, the percentages in parenthesis, provided along the bottom Physician 2 total

row, indicate how often Physician 2 reported the COD out of the total.

The percentages provided in parenthesis in the extreme right Physician Consensus column refer to how often the Physician Consensus

reported the COD out of the total. Thus, taking infection as an example, Physician 1 and Physician 2 concluded neonatal infection was theCOD 52 times. Physician 1 and Physician 2 initially agreed that infection was the underlying COD for 44 of the 52 cases. After discussing

the 13 discrepant cases where one but not both attributed the underlying COD to infection, the physicians came to final consensus that 8

of the 13 cases had an underlying COD of neonatal infection. Other causes of early neonatal death were hypothermia, low birth weight

and birth trauma.

Table 2 Comparison of physician coder 1 and physician coder 2 responses for specific causes of early neonatal deaths (n = 118)

Underlying cause

of death

Physician response – n (%)

Kappa with

corresponding

95% CI

Physicians agreedPhysicians disagreed

(one physician notedthe condition positive

and other noted the

condition negative)

Condition noted

as underlying cause

Condition not noted as

underlying cause Total

Preterm 13 96 109 (92%) 9 (8%) 0.70 (0.51, 0.88)

Neonatal Infection 44 61 105 (89%) 13 (11%) 0.78 (0.66, 0.89)

Birth asphyxia 28 77 105 (89%) 13 (11%) 0.73 (0.60, 0.87)Congenital malformation 4 114 118 (100%) 0 1.00 (1.00, 1.00)

Tetanus 5 112 117 (99%) 1 (1%) 0.90 (0.72, 1.00)

Unknown ⁄ no cause 2 114 116 (98%) 2 (2%) 0.66 (0.22, 1.00)

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C. Engmann et al. Perinatal verbal autopsy

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underlying COD at the chapter-heading level of ICD-10 for

101 of 134 (75%) of SBs.

Table 4 compares physician coder 1 vs. physician coder

2 responses for specific causes of SB. Using maternal

infection as an example, physicians agreed 120 times of

134 (90%). They agreed that maternal infection was the

cause of SB 42 times and that maternal infection was not

the cause of SB 78 times. Physicians disagreed 14 times on

the designation of infection as a cause of SB. The kappa

level of agreement was 0.78 (0.67, 0.89).

Table 3 Comparison of physician coder 1 and physician coder 2 responses for cause of stillbirth (n = 134)

Physician 1responses

Physician 2 responses

Physician 1

Totaln (%)

Physician

consensusn (%)

Antepartumhaemorrhage

Maternalinfection Preterm

Maternalaccident

Prolongedlabour

Cord

prolapse ⁄complication

Unknown ⁄no cause Other

Antepartumhaemorrhage

8 3 0 0 0 0 0 1 12 (8) 13 (10)

Maternal

infection

0 42 0 0 0 0 0 1 43 (32) 50 (37)

Preterm 1 3 5 0 0 0 4 0 13 (10) 9 (7)

Maternal

accident

0 0 0 6 1 0 0 0 7 (5) 7 (5)

Prolonged labour 0 0 0 0 11 1 0 0 12 (9) 15 (11)Cord prolapse ⁄complication

0 0 0 0 0 5 3 0 8 (6) 8 (6)

Unknown ⁄ no

cause

1 2 1 0 0 0 11 0 15 (11) 16 (12)

Other 0 5 1 0 3 2 0 13 24 (18) 16 (12)

Physician 2

Total n (%)

10 (7) 55 (41) 7 (5) 6 (4) 15 (11) 8 (6) 18 (13) 15 (11) 134

The bold numbers along the diagonal indicate agreement reached independently by the two physicians. Per cent agree-

ment = 101 ⁄ 134 = 75.4%. The percentages provided in parenthesis along the Physician 1 total column indicate how often Physician 1reported the cause of death (COD) out of the total. Similarly, the percentages provided in parenthesis along the total row indicate how

often Physician 2 reported the COD out of the total.

The percentages provided in parenthesis in the extreme right Physician Consensus column refer to how often the Physician Consensusreported the COD out of the total. Other causes of stillbirth were identified as malpresentation, folic acid deficiency, hypertension, post-

term delivery, multiple birth, polyhydramnios and multipara.

Table 4 Comparison of physician coder 1 and physician coder 2 responses for specific causes of stillbirth (n = 134)

Underlying cause of SB

Physician response – n (%)

Kappa with

corresponding

95% CI

Physicians agreedPhysicians disagreed

(one physician notedcondition positive and

other noted condition

negative)

Conditionnoted as

underlying

cause

Condition notnoted as

underlying

cause Total

Antepartum haemorrhage 8 120 128 (96%) 6 (4%) 0.70 (0.48, 0.93)

Maternal infection 42 78 120 (90%) 14 (10%) 0.78 (0.67, 0.89)

Preterm 5 119 124 (93%) 10 (7%) 0.46 (0.19, 0.74)Maternal accident 6 127 133 (99%) 1 (1%) 0.92 (0.76. 1.00)

Prolonged labour 11 118 129 (96%) 5 (4%) 0.79 (0.62, 0.97)

Cord prolapse ⁄ complication 5 123 128 (96%) 6 (4%) 0.60 (0.31, 0.89)Unknown ⁄ no cause 11 112 123 (92%) 11 (8%) 0.62 (0.42, 0.83)

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Individual assignment of COD and consensus COD

The proportion of SBs and ENDs determined by the two

physicians were very similar, as were the patterns derived

from the consensus process, compared to causes individ-

ually assigned by each physician. Similarly, the rank order

of the top 5 leading causes of SB and END was identical for

each physician (Figures 1 and 2).

Discussion

After preparation using a standardized VA training

programme, two physicians were in substantial agreement

when assigning the major causes of END. There was

almost perfect agreement when tetanus and congenital

malformations were the causes of END. Any discrepan-

cies noted in the COD assigned to individual cases had

little impact on their rank order or the overall pattern of

reported mortality. Substantial agreement between phy-

sicians was observed in the assignment of the major

causes of SB (antepartum haemorrhage, maternal infec-

tion and prolonged labour). There was only moderate

agreement on the assignment of cord prolapse and

prematurity as a cause of SB, while there was almost

perfect agreement when maternal accident was assigned

as the COD.

Few studies have evaluated the impact of different

methods for assigning cause of neonatal death using VA. In

a recent paper, Joshi et al. (2009a) compared the assign-

ment of the COD in 45 villages in Southern India by single

vs. multiple coders. This was a study of mortality in a

general population of all ages, and fewer than 1% of the

deaths occurred in children 0–28 days of age. They

reported that physician coders agreed on the same diag-

nosis 94% of the time, with overall kappa values of 0.93

suggesting almost perfect agreement among physician

coders. Among deaths in children aged 0–28 days, they

reported kappa values of 1.0, although there were only 11

cases. Our study examined the comparability of the

assignment of causes by two physicians for perinatal deaths

only. For the three most important causes of END

(infections, birth asphyxia and prematurity), physicians

agreed on the same COD approximately 90% of the time,

suggesting substantial agreement. For two other causes of

END, congenital malformations and tetanus, physician

agreement was nearly 100%. Similar results were reported

by Edmond et al. (2008) on levels of agreement among

three physicians determining cause of 590 neonatal deaths

from verbal autopsies in rural Ghana. There was sub-

stantial agreement among three physicians for prematurity,

birth asphyxia and infections (kappa values 0.8, 0.77 and

0.72). In contrast to our study, they reported a kappa value

of 0.63 for congenital abnormalities as a COD.

In our study, physicians showed substantial agreement

for certain causes of SBs (antepartum haemorrhage,

maternal infection and prolonged labour), and almost

perfect agreement for maternal accidents. There was only

moderate agreement for prematurity and cord prolapse.

When the diagnostic accuracy of VA as determined by

three experienced community paediatricians to determine

cause of SBs from rural Ghana was compared to a hospital

reference standard, VA performed poorly for causes of SB

diagnosis such as congenital abnormalities and maternal

haemorrhage, while accuracy was higher for intrapartum

obstetric complications and antepartum maternal disease

(Edmond et al. 2008).

Even in settings where placental examinations, autop-

sies, cultures, karyotypes, x-rays, MRIs and other imaging

are available, up to 60% of SBs are unexplained, high-

lighting the inherent difficulties that understanding and

obtaining agreement over cause of SB can pose (McClure

et al. 2006; Silver et al. 2007). In our study, the low rate of

‘unknown’ COD may be an artefact of the study during

which coders may have perceived some pressure to assign a

05

101520253035404550

Pre-term Birth asphyxia Infection/tetanus Malformation

Pro

port

ion

of d

eath

s (%

)

Physician 1

Physician 2

Physician consensus

Figure 2 Proportion of early neonatal deaths assigned an underlying cause of death by each of two physician coders and by physician

consensus.

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C. Engmann et al. Perinatal verbal autopsy

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cause of SB. Nonetheless, the rate of concurrence between

coders also suggests that VA may be a useful tool in

determining population-based causes of SB.

There are economic and resource implications of the

results of this study. The cost of programmes using VA to

assign COD could be substantially reduced by switching to

a system of single coding. Joshi et al. estimate that with

deaths coded only once, the cause-of-death assignment

costs can be halved and project management costs reduced

by one-third. They also suggest that funds currently used

for duplicate coding could be reassigned to conduct

validation studies that compare COD assignments from

single coders against COD derived from reliable medical

records, diagnosis by autopsy or physician-diagnosed

deaths in the community. Because VA is most typically

used within weak health systems that suffer a shortage of

physicians, utilizing fewer physicians and provide stan-

dardized training to them to code VA and redeploying

them to other clinical tasks could be a more appropriate

use of scarce human resources.

A major strength of this study is the standardized VA

training and tools programme which we have reported on

previously. After initial training, a train-the-trainer model

was used to spread it in the different countries within the

GN. This strategy increases knowledge, promotes owner-

ship, builds capacity and enables sustainability of pro-

grammes (Enweronu-Laryea et al. 2009). In contrast to

other studies which delay interview, we performed them

within 1 week and found mothers eager to discuss their

baby’s death. Early interviews may also yield more

accurate diagnoses. There are also limitations to our study.

It is possible that the duplicate coding process may be a

poor method for detecting systematic errors in the assign-

ment of causes of death. Also, poor training of coders

could also result in a bias towards a particular COD

assignment, which could be repeated by subsequent coders.

Each coder was tested after training in the VA programme,

making these potential biases less likely. Another potential

limitation may be bias towards certain diagnoses resulting

from prior knowledge of the coders of disease patterns in

their community. Therefore, use of coders from the

community in which the deaths occur would be expected to

result in a high level of agreement, but with less certainty of

the correct assignment of COD. Although the VA tool has

been validated previously in hospital settings, its applica-

tion in a community setting where deaths occur outside of

hospitals and the formal health care system has not been

validated. Therefore, we cannot be certain of the accurate

assignment of COD. However, even if tests of validity

discovered incorrect assignment of COD for particular

causes, it is unlikely that such a problem would affect

agreement between coders.

Although inter-observer and intra-observer variations

have been recognized over the years, the impact of these

variations has not been studied in detail (Fauveau 2006;

Garenne & Fauveau 2006). The findings from our study

suggest that a single physician coder may be as effective as

two coders in determining cause of SB and END when

trained in a standardized VA programme. This study also

raises important questions about the utility of a system of

multiple coders that is currently widely accepted and

speculates that a single physician coder may be an effective

and economical alternative to VA programmes that use

traditional two-physician panels to assign COD.

Acknowledgements

Funding was provided by grants from the National

Institutes of Child Health and Human Development and

the Bill and Melinda Gates Foundation.

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Corresponding Author Cyril Engmann, Division of Neonatal-Perinatal Medicine, Department of Pediatrics, University of North

Carolina, School of Medicine, CB # 7596, 4th Floor, UNC Hospitals, UNC-Chapel Hill, Chapel Hill, NC 27599-7596, USA. E-mail:

[email protected]

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C. Engmann et al. Perinatal verbal autopsy

ª 2010 Blackwell Publishing Ltd 25

Appendix1: Verbal autopsy questionnaire

Verbal Autopsy Study

OVERVIEW. I am froma study to find out why newborns die. I am very sorrythat the baby you delivered died and would like to talkto you about the death. If you do not want to talkabout it now, I can come back on another day.

9. How is the child’s mother now? (If mother, ask ‘how are you now’)

10. Was the birth more difficult than usual?

11. Did the mother have fits before giving birth? (If mother, ask ‘did you have fits before giving birth’)

12. Did/does the mother have high blood pressure? (If mother, ask ‘did/do you have high blood pressure’)

13. Did the mother have a fever at time of labor or delivery? (If mother, ask ‘did you have a fever at the time of labor or delivery’)

14. Did the mother suffer from any of the following conditions? (If mother, ask ‘did you suffer from any of the following conditions’). Circle all that apply.

15. Did the mother recieve any antenatal care during her pregnancy? (If mother, ask ‘did you recieve any antenatal care during your pregnancy’) (this excludes care by a TBA)

16. Where did the mother give birth? (If mother, ask ‘Where did you give birth’)

17. Who assisted the birth?

18. Had the mother recieved Tetanus toxoid vaccination (TT) since she

was first pregnant? (If mother, ask‘have you recived Tetanus Toxoidvaccination (TT) during or since yourfirst pregnancy’)

1- Fine2- Sick3- Don’t know

1- Yes2- No3- Don’t know

1- Yes2- No3- Don’t know

1- Yes2- No3- Don’t know

1- Yes2- No3- Don’t know

1- Yes2- No3- Don’t know

1- Yes2- No3- Don’t know

1- Home2- Health facility3- Intrained4- Other place5- Don’t know

1- None2- Unrained TBA3- Trained TBA4- Nurse, midwife5- Physician6- Don’t know

1- None2- Diabites3- Heart disease4- TB5- Epllepsy6- Abdominal trauma or accidnet7- syphills8- Malaria9- Other10- Don’t know

1. Date of Interview

dd mm yyyy

1. Date of death?

8. History of events leading to death(Write summary of what the respondent tells you)

2. Interviewer name:

3. Respondent (Circle one):

4. Who responded first? (Circle one):

5. Was a birth attendant present at time of death or stillbirth?

6. Place of death/stillbirth? (for stillbirth, place of delivery) (Circle one)

1- Birth attendant2- Mother

1- Birth attendant2- Mother

1- Yes2- No

1- Home2- Health facility

Specify:3- Other

Specify:

We are conducting.

STUDY ID Version: 1.0Version Date: 16-APR-07

dd mm yyyy

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Verbal Autopsy Study

EVENTS DURING THE BIRTH OF THE CHILD

19. Did any of the following occur during labor or delivery? Circle all that apply.

1 - None

1 - Singieton2 - Twin9 - Don’t know

1 - Yes2 - No9 - Don’t know

1 - Yes2 - No9 - Don’t know

1 - Yes2 - No9 - Don’t know

1 - Yes2 - No9 - Don’t know

1 - Yes2 - No9 - Don’t know

1 - Yes

1 - Yes

2 - No

2 - No (Skip to Q 29)

9 - Don’t know

1 - Yes2 - No9 - Don’t know

months

hours ORDays

a.b.

weeks

1 - Yes2 - No9 - Don’t know

1 - Yes2 - No9 - Don’t know

1 - Yes2 - No9 - Don’t know

1 - Yes2 - No9 - Don’t know

9 - Don’t know

1 - Yes2 - No (Skip to Q.38)9 - Don’t know

1 - Yes2 - No (Skip to Q.40)9 - Don’t know

Kgs.

1 - Yes2 - No (Skip to Q.45)9 - Don’t know

1 - Yes2 - No (Skip to Q.47)9 - Don’t know

1 - anencephaly2 - myelomeningocoele3 - cleft Ilp/palate4 - limb defects5 - Other9 - Don’t know

Ack the following questions if the child was born alive.Skip to Q78 if the child was born dead.

1 - Yes2 - No (Skip to Q.49)9 - Don’t know

1 - Normal2 - Purple3 - Pale

1 - Child2 - Mother

1 - Yes2 - No9 - Don’t know

9 - Don’t know

1 - Yes2 - No9 - Don’t know

1 - Yes2 - No9 - Don’t know

9 - Don’t know

1 - Tiny/very small2 - Smaller than usual3 - About average4 - Larger than usual

4 - Breech presentation’child delivered feet first5 - Retained placenta8 - Other, specify

20. Was the child a

33. Did the baby cry or breathe at all after delivery?

34. Was anything done to try to help the baby breathe?

37. If Q36 -yes, was the problem with the child or the mother?

39. If Q38 -yes, how much did the child weigh?

40. Were there any bruises or signs of injury on the child’s body after delivery?

41. What was the color of the child’s skin after being born?

42. Did the child’s arms/legs have stength?

43. Was the baby dead at birth?

44. If stilbrith, was the baby’s body mecerated (skin and tissue was pulpy and peeling)? Show picture

45. Did the child have any malformation at birth

46. If Q45 - Yes, Show pictures Circle if there were any similarities

48. If Q47 - Yes, how many days after being Days born?

47. Did the eye color change to yellow (Jaundice)?

38. Was the child weighed after being born?

35. Did the umblical cord come out before the baby was born?

36. If born alive, did the child have difficulty breastfeeding soon after birth?

21. Was it a forceps or vacuum/delivery?

22. Was it a caesarian delivery?

23. Was it a prolonged/obstructed labor? (Prolonged labor is more than 1 day)

24. Did waters break 1 day or more before delivery of the baby?

25. Was there a vaginal odor?

26. Was there an odor to the baby at birth?

27. Was the child premature?

29. Was the child post-term?

31. Did the baby play or move in the womb before labor?

31. When did mother last feel the baby move/ (if mother ask, ‘When did you last feel the baby move?’)

30. How many months/weeks was the baby at delivery?

28. If premature, show pichure (Circle one)

9 - Don’t know

2 - Severe abdominal pain 3 - Heavt vaginal bleeding in the week before or during labor

STUDY ID Version: 1.0Version Date: 16-APR-07

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50. Did the child have a fever?

70. Did the child have an injury or accident?

72. During the illness that led to death, did the child become unconscious?

71. If Q.70 is yes, what kind of injury of accident?

73. Was baby cold to the touch?

76. Was the informant reliable?1 - Yes2 - No9 - Don’t know

1 - Yes

Verbal Autopsy Study STUDY ID Version: 1.0Version Date: 16-APR-07

2 - No9 - Don’t know

1 - Yes2 - No9 - Don’t know

1 - Yes2 - No9 - Don’t know

1 - Yes2 - No (Skip to Q.56)9 - Don’t know

1 - Yes2 - No (Skip to Q.58)9 - Don’t know

1 - Yes2 - No (Skip to Q.60)9 - Don’t know

1 - Yes2 - No (Skip to Q.62)9 - Don’t know

1 - Yes2 - No (Skip to Q.64)9 - Don’t know

Days

Days

Days

Days

Days

Days

Days

Days

Days

1 - Yes2 - No (Skip to Q.66)9 - Don’t know

1 - Yes2 - No (Skip to Q.68)9 - Don’t know

1 - Yes2 - No (Skip to Q.70)9 - Don’t know

1 - Yes2 - No (Skip to Q.52)9 - Don’t know

1 - Yes2 - No (Skip to Q.72)9 - Don’t know

1 - Yes2 - No9 - Don’t know

1 - Yes2 - No9 - Don’t know

1 - Yes2 - No9 - Don’t know

74. For family only, did health care worker tel you the cause of death?

51. If Q50 -yes, for how many days?

52. Did the child have fits?

54. Was the child coughing?

If Q74 -yes, what did s/he say?

The folowing is not asked duringInterview -respond re: youropinion of the informant:

75. What do you think was the cause of the death or stillbirth?55. If Q.54 -yes, how many days?

57. If Q.56 -yes, how many days?

58. Did the child have fast breathing?

59. If Q.58 -yes, how many days?

61. If Q.60 -yes, how many days?

62. Was the child vomiting?

63. If Q.62 -yes, how many days?

65. If Q.54 -yes, how many days?

67. If Q.66 -yes, how many days?

69. If Q.68 -yes, how many days?

68. Was there a bulge in the child’sfontanel?

66. Was the child unable to breastfeed when s/he was ill?

64. Did s/he have diarhea?

60. Did s/he have indrawing of the chest while breathing?

56. Did the child have difficulty breathing?

53. During the period of illness did s/he have areas of skin that were red or peeling, or a skin rash with bilsters contalning pus?

49. Did the child have an umblical stump that was red or draining pus?

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Appendix 2: Case definitions for maternal and

neonatal ⁄ foetal underlying, final, and contributing

causes of death

Fetal ⁄ neonatal causes

Preterm ⁄ complications of prematurity:

Infant born before 37 completed weeks of pregnancy

(includes maternal subjective assessment of born early).

Fetal ⁄ neonatal infection ⁄ sepsis ⁄ pneumonia:

Congenital or acquired invasion and multiplication of

germs (bacteria, fungi or viruses).

Birth asphyxia ⁄ intrapartum asphyxia:

Failure to initiate and sustain breathing at birth.

Congenital malformation:

A major structural defect which causes the baby to die.

Birth trauma (during labor):

Injuries to the infant during the process of birth.

Fetal trauma:

Injuries affecting the fetus before it is born.

Neonatal accident: Any injury or trauma affecting the

infant after it is born.

Tetanus:

Neonates with a normal ability to suck and cry during

the first 2 days of life, and who, between day 3 and 28

cannot suck normally, and become stiff or have fits ⁄ con-

vulsions (i.e. jerking of muscles) or both.

Diarrhea:

The passage of loose or liquid stools more frequently

than is normal for babies.

Hypothermia:

A neonate who feels cold to touch, or who has a

temperature below 36.5 �C.

Low birth weight:

Birthweight <2500 g, or smaller in size than expected for

the baby’s gender, genetic heritage and gestational age

(includes mother’s opinion of small).

Jaundice:

Yellow coloration of the skin or whites of the eyes.

Maternal causes

Antepartum haemorrhage:

Heavy bleeding in the last week of pregnancy (excludes

‘spotting’)

Maternal infection ⁄ sepsis:

Invasion and multiplication of germs (bacteria, fungi and

viruses). This definition includes malaria, and excludes

minor infections such as colds.

Preterm delivery:

Labor and delivery before 37 completed weeks of

pregnancy

Maternal accident:

Any injury occurring to the mother after 20 weeks of

pregnancy that might affect the fetus.

Obstructed ⁄ prolonged labor:

Any condition that results in the labor process being

obstructed e.g. because the fetus is too large for the birth

canal or the presentation of the fetus prevents the mother

from pushing the baby out of the birth canal.

Multiple delivery:

Birth of two or more babies (even if one of the babies is

dead).

Hypertensive disorder ⁄ eclampsia: High blood pressure

that occurs before or during pregnancy. This may be

associated with generalized swelling, headaches or seizures.

Cord prolapse ⁄ complications: When the baby’s umbili-

cal cord falls into the birth canal ahead of the baby’s head

or other parts.

Malpresentation: When the baby is in a difficult position

for the birth process.

Post-term: any baby born after 42 completed weeks of

pregnancy.

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