Neglected tropical diseases morbidity and disability (NTD) toolkit validation: A cross-sectional validation study of the EMIC stigma scale – affected persons in Papua, Indonesia.

Background

There is a lack of information about the burden of neglected tropical diseases (NTDs) in terms of morbidity and disability. In order to overcome this gap and create a set of agreed instruments to assess the morbidity and disability aspects of NTDs, a cross-NTD Morbidity and Disability (NMD) toolkit was developed. This toolkit is composed of existing questionnaires, and needs to be validated across different cultural settings, languages and NTDs. This study is a first phase validation study of one of the questionnaires of the NMD toolkit, the EMIC stigma scale for affected persons (EMIC-AP). This study was done in Papua, Indonesia, which is one of the hotspots for NTDs. Well-known examples of NTDs that occur in Papua are leprosy and lymphatic filariasis. Therefore, the research question is: "How valid is the EMIC-AP of the NMD toolkit among persons affected by the NTDs leprosy or lymphatic filariasis living in Papua, Indonesia?".

Methodology

A cross-sectional design was used for the instrument validation of this study. The study population consists of people affected by the NTDs leprosy or lymphatic filariasis living in the northern part of Papua. The EMIC-AP was translated into Bahasa as part of this study and then back translated to check the first translation. After this, a pilot of seven persons was conducted with an interview-administrated version of the EMIC-AP. Changes were made to the questionnaire where needed. After this pilot study, the data gathering started with the finalized version of the EMIC-AP. The total sample included 15 participants with leprosy and 15 participants with lymphatic filariasis. The data collection tools and analyses were based on a revised version of the Herdman framework of cultural equivalence testing. This framework defines five categories of equivalence that can be used for the validation of culturally adapted instruments: conceptual-, item-, semantic-, operational- and measurement equivalence. This first phase validation study focussed on item-, semantic- and operational equivalence. In this study the item equivalence was based on the acceptability of the participants with the topics and the relevance of the questions to their experience with their NTD. The semantic equivalence was based on the understanding of the questions. The operational 4

equivalence was based on the opinion of the participants regarding the answer options and on the input of participants on the improvement of the questionnaire

Results

In the category of item equivalence, 90% of the participants answered that they were comfortable with the topics, and 93% of the participants said that the questions where suitable given their experience with their NTD. In the category of the semantic equivalence, 57% answered to have no troubles in understanding the questionnaire. The understanding of the questionnaire was related to education level and place of residence. A higher educational level was related to better understanding of the questionnaire. The questionnaire was also better understood if the place of residence was in an urban area compared to a rural area. In the category of the operational equivalence, 73% of the participants were positive about the answer options, and 87% said to have nothing to add or change in the questionnaire.

Discussion and conclusion

The results of the item equivalence were positive, as all items seemed acceptable and relevant. The results of the operational equivalence suggest that the format of the EMIC-AP worked well with the addition of an answer option flash card. The semantic equivalence showed possible room for improvement. One of the questions regarding self-respect lost its meaning, and therefore was not validated during this study. The question regarding the NTD causing problems in marriage seemed to cause some confusion with the participants, and needs revision for future research. There were participants that indicated that they had difficulties understanding the questionnaire. The main reason that was found for this was the language usage. In the province of Papua there are over a 250 different languages, which makes validation in the area of semantic equivalence of any questionnaire for the population of Papua difficult. For future research a second phase validation study is recommended, with some qualitative concerns that also need to be addressed.