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Brandon A Kohrt, 1Duke Global Health Institute, Department of Psychiatry and Behavioral Sciences, Durham, NC, USA, 2Department of Psychology, Fordham University, New York, USA, 3Department of Anthropology, Department of Epidemiology, Emory University, Atlanta, GA, USA, 4Department of Mental Health, Bloomberg School of Public Health, Johns Hopkins University, Baltimore, MD, USA, 5Department of Psychology, Tribhuvan University, Kirtipur, Nepal, 6Butabika National Referral Mental and Teaching Hospital, Kampala, Uganda, 7AISSR, University of Amsterdam, The Netherlands and 8Massachusetts General Hospital and Harvard Medical School, Boston, MA, USA *Corresponding author. Duke Global Health Institute, Trent Hall #213, 310 Trent Drive, Duke University, Durham, NC 27710, USA. E-mail: Search for other works by this author on: Andrew Rasmussen,1Duke Global Health Institute, Department of Psychiatry and Behavioral Sciences, Durham, NC, USA, 2Department of Psychology, Fordham University, New York, USA, 3Department of Anthropology, Department of Epidemiology, Emory University, Atlanta, GA, USA, 4Department of Mental Health, Bloomberg School of Public Health, Johns Hopkins University, Baltimore, MD, USA, 5Department of Psychology, Tribhuvan University, Kirtipur, Nepal, 6Butabika National Referral Mental and Teaching Hospital, Kampala, Uganda, 7AISSR, University of Amsterdam, The Netherlands and 8Massachusetts General Hospital and Harvard Medical School, Boston, MA, USA Search for other works by this author on: Bonnie N Kaiser,1Duke Global Health Institute, Department of Psychiatry and Behavioral Sciences, Durham, NC, USA, 2Department of Psychology, Fordham University, New York, USA, 3Department of Anthropology, Department of Epidemiology, Emory University, Atlanta, GA, USA, 4Department of Mental Health, Bloomberg School of Public Health, Johns Hopkins University, Baltimore, MD, USA, 5Department of Psychology, Tribhuvan University, Kirtipur, Nepal, 6Butabika National Referral Mental and Teaching Hospital, Kampala, Uganda, 7AISSR, University of Amsterdam, The Netherlands and 8Massachusetts General Hospital and Harvard Medical School, Boston, MA, USA Search for other works by this author on: Emily E Haroz,1Duke Global Health Institute, Department of Psychiatry and Behavioral Sciences, Durham, NC, USA, 2Department of Psychology, Fordham University, New York, USA, 3Department of Anthropology, Department of Epidemiology, Emory University, Atlanta, GA, USA, 4Department of Mental Health, Bloomberg School of Public Health, Johns Hopkins University, Baltimore, MD, USA, 5Department of Psychology, Tribhuvan University, Kirtipur, Nepal, 6Butabika National Referral Mental and Teaching Hospital, Kampala, Uganda, 7AISSR, University of Amsterdam, The Netherlands and 8Massachusetts General Hospital and Harvard Medical School, Boston, MA, USA Search for other works by this author on: Sujen M Maharjan,1Duke Global Health Institute, Department of Psychiatry and Behavioral Sciences, Durham, NC, USA, 2Department of Psychology, Fordham University, New York, USA, 3Department of Anthropology, Department of Epidemiology, Emory University, Atlanta, GA, USA, 4Department of Mental Health, Bloomberg School of Public Health, Johns Hopkins University, Baltimore, MD, USA, 5Department of Psychology, Tribhuvan University, Kirtipur, Nepal, 6Butabika National Referral Mental and Teaching Hospital, Kampala, Uganda, 7AISSR, University of Amsterdam, The Netherlands and 8Massachusetts General Hospital and Harvard Medical School, Boston, MA, USA Search for other works by this author on: Byamah B Mutamba,1Duke Global Health Institute, Department of Psychiatry and Behavioral Sciences, Durham, NC, USA, 2Department of Psychology, Fordham University, New York, USA, 3Department of Anthropology, Department of Epidemiology, Emory University, Atlanta, GA, USA, 4Department of Mental Health, Bloomberg School of Public Health, Johns Hopkins University, Baltimore, MD, USA, 5Department of Psychology, Tribhuvan University, Kirtipur, Nepal, 6Butabika National Referral Mental and Teaching Hospital, Kampala, Uganda, 7AISSR, University of Amsterdam, The Netherlands and 8Massachusetts General Hospital and Harvard Medical School, Boston, MA, USA Search for other works by this author on: Joop TVM de Jong,1Duke Global Health Institute, Department of Psychiatry and Behavioral Sciences, Durham, NC, USA, 2Department of Psychology, Fordham University, New York, USA, 3Department of Anthropology, Department of Epidemiology, Emory University, Atlanta, GA, USA, 4Department of Mental Health, Bloomberg School of Public Health, Johns Hopkins University, Baltimore, MD, USA, 5Department of Psychology, Tribhuvan University, Kirtipur, Nepal, 6Butabika National Referral Mental and Teaching Hospital, Kampala, Uganda, 7AISSR, University of Amsterdam, The Netherlands and 8Massachusetts General Hospital and Harvard Medical School, Boston, MA, USA Search for other works by this author on: Devon E Hinton1Duke Global Health Institute, Department of Psychiatry and Behavioral Sciences, Durham, NC, USA, 2Department of Psychology, Fordham University, New York, USA, 3Department of Anthropology, Department of Epidemiology, Emory University, Atlanta, GA, USA, 4Department of Mental Health, Bloomberg School of Public Health, Johns Hopkins University, Baltimore, MD, USA, 5Department of Psychology, Tribhuvan University, Kirtipur, Nepal, 6Butabika National Referral Mental and Teaching Hospital, Kampala, Uganda, 7AISSR, University of Amsterdam, The Netherlands and 8Massachusetts General Hospital and Harvard Medical School, Boston, MA, USA Search for other works by this author on: Accepted: 04 October 2013 Published: 22 December 2013
Close Navbar Search Filter Microsite Search Term Search AbstractBackground Burgeoning global mental health endeavors have renewed debates about cultural applicability of psychiatric categories. This study’s goal is to review strengths and limitations of literature comparing psychiatric categories with cultural concepts of distress (CCD) such as cultural syndromes, culture-bound syndromes, and idioms of distress. Methods The Systematic Assessment of Quality in Observational Research (SAQOR) was adapted based on cultural psychiatry principles to develop a Cultural Psychiatry Epidemiology version (SAQOR-CPE), which was used to rate quality of quantitative studies comparing CCD and psychiatric categories. A meta-analysis was performed for each psychiatric category. Results Forty-five studies met inclusion criteria, with 18 782 unique participants. Primary objectives of the studies included comparing CCD and psychiatric disorders (51%), assessing risk factors for CCD (18%) and instrument validation (16%). Only 27% of studies met SAQOR-CPE criteria for medium quality, with the remainder low or very low quality. Only 29% of studies employed representative samples, 53% used validated outcome measures, 44% included function assessments and 44% controlled for confounding. Meta-analyses for anxiety, depression, PTSD and somatization revealed high heterogeneity (I2 > 75%). Only general psychological distress had low heterogeneity (I2 = 8%) with a summary effect odds ratio of 5.39 (95% CI 4.71-6.17). Associations between CCD and psychiatric disorders were influenced by methodological issues, such as validation designs (β = 16.27, 95%CI 12.75-19.79) and use of CCD multi-item checklists (β = 6.10, 95%CI 1.89-10.31). Higher quality studies demonstrated weaker associations of CCD and psychiatric disorders. Conclusions Cultural concepts of distress are not inherently unamenable to epidemiological study. However, poor study quality impedes conceptual advancement and service application. With improved study design and reporting using guidelines such as the SAQOR-CPE, CCD research can enhance detection of mental health problems, reduce cultural biases in diagnostic criteria and increase cultural salience of intervention trial outcomes. IntroductionIn 1904 Emile Kraepelin initiated the field of comparative psychiatry (vergleichende Psychiatrie) through investigation of dementia praecox in Java, and he later documented psychiatric presentations among Native Americans, African Americans and Latin Americans.1 A century later, active debate continues regarding the role of culture in mental disorders and the cross-cultural applicability of biomedical psychiatric diagnoses.2 Methodological limitations in cross-cultural psychiatric epidemiology have been cited as a primary reason why cultural differences have not translated into re-evaluating psychiatric concepts and treatment practices.3,4 For example, cultural differences in schizophrenia outcomes, which have been identified in three successive studies,5–10 have done little to alter conceptualizations or treatment of the disorder, and this is in part due to methodological problems in the cross-national studies.3,11–13 These studies, along with World Health Organization (WHO) World Mental Health Surveys,14 are typified by application of Western culturally developed biomedical psychiatric diagnoses that lack inclusion of cultural concepts of distress (CCD). To date there have not been large-scale cross-national global mental health epidemiology studies incorporating CCD. To address this gap in the research, a review of the literature on CCD was undertaken to examine the types of studies conducted, the methodological approaches and the association of CCD with psychiatric disorders. The goal is to identify best practices in cross-cultural psychiatric epidemiology to improve research on CCD and encourage application to mental health services. The term ‘cultural concept of distress’ is a new addition to the Diagnostic and Statistical Manual of Mental Disorders (DSM) series with the publication of DSM-5: ‘Cultural Concepts of Distress refers to ways that cultural groups experience, understand, and communicate suffering, behavioral problems, or troubling thoughts and emotions’.15 The term is a recent advance in the history of attempts to categorize psychological distress with demonstrable cultural influence that lacks one-to-one unity with biomedical psychiatric diagnoses (see Box 1 for exemplar CCD.) The attempt to label CCD dates back to Pow Meng Yap’s research in Hong Kong in the 1950–60s.16 Yap employed the term ‘culture-bound depersonalization syndrome’ to describe koro, a ‘state of acute anxiety with partial depersonalization’ associated with fear of the penis retracting into the body. The term ‘culture-bound syndrome’ has been used in cross-cultural psychiatry since and was included in the DSM-IV.17 Box 1. Examples of Cultural Concepts of Distress (CCD) Nervios-related conditions—In the Americas, nervios (nerves)-related conditions among Latino populations are the most commonly described CCD.126 Nervios starts with ‘a persistent idea that ‘is stuck to one's mind’ (‘idea pegada a la mente’), and these ‘particular idea[s] … invade the mind and accumulate … Affected individuals think so much about the ideas that the ideas ‘get stuck' to the brain’.94 Among Mexicans with nervios, 40% endorsed having an idea stuck to their mind. In nervios, feelings of humiliation lead to the slow deterioration of one’s mind, nerves and spirit and ‘may even cause death, if adequate help is not timely received’.127 The spectrum of nervios follows a gradient of behavioural control.80 One end of the spectrum begins with socially acceptable nervousness: ser una persona nerviosa (being a nervous person). Padecer de los nervios (suffering from nerves) is more serious. Ataques de nervios (attacks of nerves) have greater severity and are characterized by social stressors triggering loss of behavioural control, dissociation, violent acts toward oneself or others, anger and somatic distress.128 Severe nerve illness can lead to loco (madness). Nervios (nerves), padecer de nervios (suffering from nerves) and ataques de nervios (nerve attacks) have been studied in clinical samples in large-scale Latino representative community studies in Puerto Rico and the USA.70,71 Ataques de nervios overlap with some symptoms of panic attacks and panic disorder. However, they are distinct from panic attacks because of the centrality of interpersonal disputes in triggering episodes, dissociative features and an experience of relief among some individuals after an ataque.80,132 These nervios-related conditions are associated with unexplained neurological complaints, physical health problems and functional impairment independent of association with psychiatric disorders. Dhat—Dhat syndrome has been studied in South Asia and is rooted in Ayurvedic traditions about bodily production of semen as representing an end-product of energy demanding metabolism: 40 meals create 1 drop of blood, 40 drops of blood create 1 drop of semen.43 Dhat is recognized by a whitish discharge in urine assumed to be semen. Although sexually transmitted infections may be a source of such discharge, dhat sufferers do not appear to have greater frequency of STIs.69 Dhat sufferers do appear to have high rates of psychosexual dysfunction including premature ejaculation and erectile dysfunction: 42% of men with dhat had premature ejaculation in one study in India.64 Young males appear to be the most frequent demographic group presenting with dhat. Dhat has corollaries in Chinese medicine and European and American history with accounts of weakness, physical illness and mental illness related to the loss of semen.43,77 Koro—Koro was one of the first cultural concepts discussed in transcultural psychiatry literature.16 Koro epidemics have been reported in South Asia, and case reports have been reported throughout the world. Fear of the penis retracting into the body among men and retraction of breasts among women is a central feature. The majority of reported cases are among men. Brain fag—Brain fag has been studied for a half-century in Western Africa. The condition is characterized by distress from thinking too much, with students being a vulnerable population.86 The experience includes headaches and an experience of a worm crawling in the head. This is similar to the Nigerian cultural concept of distress, ode ori:84 the disorder ode ori (hunter in the head) affects the brain under the anterior fontanelle where the iye (senses) control mental functions through okun (strings) that project throughout the body and provide direct linkages among the brain, eyes, ears and heart. Khyal attacks and ‘wind’-related illnesses—The substance qi, (cf chi, chi’i, khí, khii, rlung, khyal) is associated with wind flow and wind balance. Wind-related illnesses are commonly described in East Asian populations including Tibetans, Cambodians, Vietnamese, Chinese and Mongolians.73,77,78,129,130 Shenjing shuairuo (neurological weakness, neurasthenia), studied by Kleinman in the 1970s and 80s, is associated with weakness, fatigue and social distress mediated by an alteration in qi.77 Yadargaa, a nervous fatigue described in Mongolia, is similarly viewed as an alteration in khii flow and balance.78 In the Vietnamese CCD ‘hit by wind’, shifts in ambient temperature, especially gusts of cold air, are associated with a range of physical complaints, traumatic memories, thinking too much, epilepsy and stroke.73 Similarly, in China, nerve weakness is associated with a fear of cold because it worsens nerve weakness.77 Among Cambodians, the wind-like substance khyal can be experienced as an attack associated with palpitations, asphyxia and dizziness.130 Khyal attacks can lead to rupture of blood vessels in the neck and spinning of the brain. Kufungisisa—The experience of thinking too much (Shona: kufungisisa) is associated with general psychological distress and common mental disorders in Zimbabwe. Thinking too much is considered both a symptom of distress and a cause of other physical and psychological health problems: thinking too much can cause pain and feelings of physical pressure on the heart.54 Hwa-Byung—Heat and fire are important elements in East Asian ethnopsychology. The condition hwa-byung (fire illness resulting from chronic accumulated anger) in Korea occurs when haan (a mixture of sorrow, regret, hatred, revenge and perseverance) builds up to create a pushing sensation in the chest, resulting in the inability to appropriately control one’s anger.85 Hwa-byung affects middle-aged women in Korea who have experienced years of interpersonal conflict, typically in the context of an abusive marital relationship. However, the term culture-bound syndrome has been associated with numerous limitations: findings of similar patterns of distress in disparate cultural settings, lack of cohesive symptom presentation characterizing a syndrome, and wide diversity in aetiological attributions, vulnerability groups and symptoms that influence cultural labels.18–22 Moreover, the combination of medical anthropology research, which documents the social construction of psychiatric disorders,23 with innovations in gene-by-environment and social neuroscience research, which illustrate that culture and biology are not neatly divisible categories,24–28 demonstrates that all psychological distress is culture bound. To acknowledge this, the DSM-5 includes text that ‘all forms of distress are locally shaped, including the DSM disorders’.15 Due to dissatisfaction with the term culture-bound syndrome, researchers have proposed other labels such as ‘idioms of distress’, ‘popular category of distress’, ‘cultural syndrome’ and ‘explanatory model’.29–33 The term ‘cultural concept of distress’ is an attempt to aggregate these different concepts without implying cultural exclusivity. There has been a tension in cultural psychiatry about comparing CCD with psychiatric disorders. Because CCD often incorporate culturally salient aetiological models, vulnerability expectations, wide-ranging associated symptoms and a mixture of lay and local professional attributions systems, comparison with psychiatric diagnoses has been criticized as forcing homogeneity onto CCD and losing key aspects of aetiology and vulnerability that are not incorporated in most psychiatric diagnoses.20,21,34 However, there is a growing body of epidemiology literature comparing CCD with psychiatric disorders for a variety of goals, such as validating psychiatric disorders against CCD, identifying vulnerable groups based on CCD status and identifying forms of distress and impairment not captured by psychiatric disorders. The goal of this review is to explore the methodological approaches of these epidemiological studies of CCD and psychiatric disorders, to identify limitations in the approaches and best practices for future work. We sought to develop specific criteria for evaluating epidemiological studies based on cultural psychiatry principles. With the expansion of global mental health research and scaling up of services,35–38 it is an ideal time to evaluate if and how CCD can be incorporated into community and clinical epidemiology to reduce suffering. Our review is divided into the following sections: identification of studies comparing CCD and psychiatric disorders; description of study objectives and methods including ranking epidemiological quality of these studies; examining summary effect sizes and sources of heterogeneity when comparing CCD and psychiatric disorders; and concluding with recommendations for incorporating CCD in global mental health research and services. MethodsInformational sourcesTo identify literature on CCD we searched MEDLINE/PubMed, applying the following keywords: ‘culture-bound’ or ‘culture bound’ or ‘idiom of distress’ or ‘idioms of distress’. To assure inclusion of popularly studied CCD, we combined the above search with a search of CCD listed in the DSM-5 glossary: ‘nervios’ or ‘dhat’ or ‘khyal’ or ‘kufungisisa’ or ‘maladi moun’ or ‘shenjing shuairou’ or ‘susto’ or ‘taijin kyofusho’). We limited psychiatric outcomes to common mental disorders (operationalized here as depression, anxiety-related conditions including posttraumatic stress disorder (PTSD) and panic disorder, and somatization-related conditions) because of their significant burden of disease, the breadth of research on CCD and common mental disorders, and feasibility of assessing common mental disorders through self-report. In contrast, psychosis-related conditions have shown poor reliability and low detection through self-report cross-culturally.39,40 In our preliminary searches for substance use disorders, eating disorders and developmental disorders, we identified a limited number of studies precluding synthesis of findings. The psychiatric disorder search terms thus included the following: ‘depression’ or ‘depression, postpartum’ or ‘PTSD’ or ‘stress disorders, post-traumatic’ or ‘fatigue syndrome, chronic’ or ‘fatigue’ or ‘anxiety disorders’ or ‘anxiety’ or ‘panic disorder’ or ‘panic attack’ or ‘somatoform disorders’ or ‘somatic complaints’. Searches were limited to English-language peer-reviewed journal publications. In addition, reference sections of previous reviews on culture-bound syndromes were searched,41–48 and reference sections of articles identified in the search were used to locate additional articles. The initial searches was performed in November 2012 and repeated for new references in March 2013 and September 2013. Data collectionTo extract relevant data, all studies identified through searches were read and evaluated for inclusion by the first author. Inclusion criteria comprised English language, prevalence data for a psychiatric category, prevalence data for a CCD, odds ratios with 95% confidence intervals for association of CCD and psychiatric category or data presented in a manner enabling construction of a two-by-two comparison of psychiatric classification and CCD. Exclusion criteria were case studies and articles lacking original quantitative data. Extracted data included world region, country, study population (including current country of residence for refugee and immigrant populations), researcher label for CCD (e.g. idiom of distress, culture-bound syndrome, cultural syndrome, cultural somatic symptom), language of term, English translation of term, research objective of the study, sample size, sample description, sample origin (clinical, community or school), age group of sample, representative vs convenience or other sample, inclusion and description of control or comparison group, symptom/syndrome description, assessment method for CCD (self-labelling with single-item term, labelling based on a multi-item self-report instrument score, labelling by healthcare provider including traditional healers and clinical providers, labelling from key informant in community), symptom severity assessment, type of symptoms (subjective self-report, externally observable or mixed), CCD prevalence (lifetime, current or unclear), age of onset, duration of current episode, psychiatric diagnostic instrument, administration format of psychiatric instrument (e.g. clinician administered, researcher administered, self-report), validation of instrument in study population, assessment of functioning and impairment, aetiology/perceived cause of CCD, vulnerability factors and risk group for CCD, protective factors against CCD, inclusion of follow-up assessment, percentage lost to follow-up, reasons lost to follow-up, current or prior treatment status, description of study treatment, assessment of psychiatric comorbidities, assessment of biological comorbidities and potential confounds. Quality assessmentTo assess quality, we chose the Systematic Assessment of Quality in Observational Research (SAQOR), which has been developed for assessing quality in observational studies49 and has been used to rate global mental health research conducted across cultural settings.50 SAQOR includes six domains: Sample, Control/Comparison Group, Quality of Exposure/Outcome Measurements, Follow-Up, Distorting Influences and Reporting Data. Each domain contains multiple criteria. For this study, the results section describes modification of SAQOR to develop a version for Cultural Psychiatry Epidemiology (SAQOR-CPE). Meta-analysesOdds ratios were extracted or calculated from quantitative studies to determine the likelihood of a specific psychiatric category given the presence of a specific CCD. Two-by-two tables were constructed for all quantitative papers that included data for categorical outcomes of CCD (yes vs no) and psychiatric categories (yes vs no). If studies only included mean scores on symptom scales without providing information on categorical cut-offs, these studies were not included in the meta-analysis. In the two-by-two tables, CCD were considered the independent variable and psychiatric categories were considered the dependent variable. Odds ratios (OR), 95% confidence intervals, sensitivity, specificity, positive predictive value (PPV) and negative predictive value (NPV) were calculated for all studies in the meta-analysis. If a study contained an empty field in the two-by-two table, then individual study outcomes (OR, sensitivity, specificity, PPV and NPV) were not calculated; however, the participants were included in the meta-analysis summary calculations. Sensitivity was calculated as the proportion of persons positive for both the CCD and the psychiatric category, among all persons with CCD. Specificity was calculated as the proportion of persons negative for the CCD and negative for the psychiatric category, among all persons negative for the CCD. Positive predictive value was calculated as the proportion of participants positive for both the CCD and psychiatric category, among all participants positive for the psychiatric category. Negative predictive value was calculated as the proportion of participants negative for both the CCD and the psychiatric category, among all persons negative for the psychiatric category. Heterogeneity for summary effect sizes was calculated with the Q statistic. The statistic was calculated by summing the squared deviations of each study’s effect estimated from the overall effect estimate; each study was weighted by its inverse variance.51,I2 is another measure of heterogeneity calculated by dividing the difference of the Q statistic and its degrees of freedom by the Q statistic and multiplying this by 100.51 Low values (e.g. <25%) suggest low heterogeneity whereas I2 >75% suggests high heterogeneity with study characteristics and methods influencing the associations. Generalized estimating equations (GEE) were used to assess the influence of study design on effect sizes. GEE is one method that can account for the clustering of multiple comparisons within a single study.52 The odds ratio for each study was used as the dependent variable. Independent variables included world region (Americas, Africa, Asia), researcher label (‘culture-bound …’, ‘idiom …’, ‘popular …’, other ‘… syndrome’ and other label), study objective (compare CCD and psychiatric disorder, instrument validation study, assessment of risk factors for psychological distress, and other), sample size (<100, 100–499 and ≥500 participants) recruitment site (clinical, community or school-based settings), representativeness of sample (representative sample vs all other recruitment forms), CCD type (four groups were created based on greatest number of participants: nervios-related studies, 10 820 participants; dhat studies, 863 participants; hwa-byung studies, 3087 participants; and all other cultural concepts of distress, 4012 participants), CCD-self report (participant endorsed CCD vs studies in which the CCD was attributed to the participant by the researcher, a clinician, or a key informant), assessment method for CCD [categorized into four groups: (i) self-report single item binary categorical endorsement (e.g. yes vs no for ‘Have you ever had an ataque de nervios?’); (ii) self-report multi-symptom instrument score (e.g. mean scale above a cut-off for number of symptoms to meet criteria as a proxy for ataques de nervios, such as symptoms of blinding, fainting and paralysis with symptoms beginning after a troubling experience53); (iii) clinical diagnosis (e.g. clinician making a diagnosis of dhat or hwa-byung based on specific clinical guidelines); or (iv) other third party labelling (e.g. binary categorical label of CCD provided by someone other than participant or clinician; this was usually done by key informants in the community or parents)], prevalence of CCD (lifetime, current/point or unclear), psychiatric categories (classified in five groups: general psychological distress, all anxiety disorders, mood disorders, somatoform disorders and other disorders), controlling for comorbidity (control through inclusion/exclusion criteria or through statistical analysis vs no control for comorbidity) and SAQOR-CPE overall ranking score (very low quality, low quality, medium quality, or high quality). Only analyses with OR outcomes were entered into the GEE. This led to inclusion of 79 comparisons drawn from 26 studies because some studies had multiple comparisons. ResultsStudy characteristicsThrough the search terms, 211 citations were identified; 12 studies were added from reviews and references lists. Of the total of 223 studies evaluated, 4553–97 included quantitative data on both cultural concepts of distress and psychiatric categories (see Figure 1). Ten studies were conducted in Africa, 18 in the Americas and 17 in Asia (see Table 1a, b, c). The most common CCD were nervios-related conditions, comprising 30% of studies. Nine studies (20%) included children, and the remainder only had adult participants. Studies with participants under 18 years of age were predominantly nervios-related conditions, as well as dhat among adolescent boys. Sixteen (35%) of the studies used the label ‘culture-bound’; nine studies (20%) used ‘idiom of distress’; and 23 studies had comparison of CCD with psychiatric disorders as a primary objective. For eight studies, the primary goal was to evaluate association with a risk factor or vulnerable group. Seven studies had instrument adaptation and validation as the primary goal. Figure 1  PRISMA diagram showing selection of studies for inclusion in systematic review of cultural concepts of distress (CCD) and psychiatric disorders Table 1a Studies conducted in Africa, meeting inclusion criteria for comparison of cultural concepts of distress and psychiatric categories
Table 1a Studies conducted in Africa, meeting inclusion criteria for comparison of cultural concepts of distress and psychiatric categories
Table 1b Studies conducted in the Americas, meeting inclusion criteria for comparison of cultural concepts of distress and psychiatric categories
Table 1b Studies conducted in the Americas, meeting inclusion criteria for comparison of cultural concepts of distress and psychiatric categories
Table 1c Studies conducted in Asia, meeting inclusion criteria for comparison of cultural concepts of distress and psychiatric categories
ADI, Amritsar Depressive Inventory; APAI, Acholi Psychosocial Assessment Inventory; BAI, Beck Anxiety Inventory; BDI, Beck Depression Inventory; BFSS, Brain Fag Symptom Scale; BSI, Brief Symptom Inventory; BYI, Beck Youth Inventory; CBT, Cognitive Behavioural Therapy; CDI, Chinese Depression Inventory; CFS, Chalder Fatigue Scale; CIDI, Composite International Diagnostic Inventory; CISR, Clinical Interview Schedule-Revised; CSSI, Cambodian Somatic Symptom and Syndrome Inventory; DIS, Diagnostic Interview Schedule; DISC, Diagnostic Interview Schedule for Children; DSM, Diagnostic and Statistical Manual of Mental Disorders; EPDS, Edinburgh Postnatal Depression Screen; GAD, Generalized Anxiety Disorder; GHQ, General Health Questionnaire; HAM-D, Hamilton Depression Rating Scale); HSCL, Hopkins Symptom Checklist; HTQ, Harvard Trauma Questionnaire; KI, Key Informant; MDD, Major Depressive Disorder; NLAAS, National Latino Asian American Study; OCD, Obsessive Compulsive Disorder);OPD, Outpatient Department; PAQ-R, Panic Attack Questionnaire-Revised; PCL-C, Posttraumatic Stress Checklist; PDS, Posttraumatic Diagnostic Scale; PHQ-9, Patient Health Questionnaire; PSE, Present State Examination; PRIME-MD, Primary Care Evaluation of Mental Disorders; PVPS, Phan Vietnamese Psychiatric Scale; SAD, Social Anxiety Disorder; SCID, Structured Clinical Interview for DSM; SCL-90, Somatic Checklist-90 item; SPS, Spirit Possession Scale; SRQ, Self-Reporting Questionnaire; SSI, Somatization Screening Index; SSQ, Shona Symptom Questionnaire; STAI, State Trait Anxiety Inventory; TKS, Taijin Kyofu Sho. Table 1c Studies conducted in Asia, meeting inclusion criteria for comparison of cultural concepts of distress and psychiatric categories
ADI, Amritsar Depressive Inventory; APAI, Acholi Psychosocial Assessment Inventory; BAI, Beck Anxiety Inventory; BDI, Beck Depression Inventory; BFSS, Brain Fag Symptom Scale; BSI, Brief Symptom Inventory; BYI, Beck Youth Inventory; CBT, Cognitive Behavioural Therapy; CDI, Chinese Depression Inventory; CFS, Chalder Fatigue Scale; CIDI, Composite International Diagnostic Inventory; CISR, Clinical Interview Schedule-Revised; CSSI, Cambodian Somatic Symptom and Syndrome Inventory; DIS, Diagnostic Interview Schedule; DISC, Diagnostic Interview Schedule for Children; DSM, Diagnostic and Statistical Manual of Mental Disorders; EPDS, Edinburgh Postnatal Depression Screen; GAD, Generalized Anxiety Disorder; GHQ, General Health Questionnaire; HAM-D, Hamilton Depression Rating Scale); HSCL, Hopkins Symptom Checklist; HTQ, Harvard Trauma Questionnaire; KI, Key Informant; MDD, Major Depressive Disorder; NLAAS, National Latino Asian American Study; OCD, Obsessive Compulsive Disorder);OPD, Outpatient Department; PAQ-R, Panic Attack Questionnaire-Revised; PCL-C, Posttraumatic Stress Checklist; PDS, Posttraumatic Diagnostic Scale; PHQ-9, Patient Health Questionnaire; PSE, Present State Examination; PRIME-MD, Primary Care Evaluation of Mental Disorders; PVPS, Phan Vietnamese Psychiatric Scale; SAD, Social Anxiety Disorder; SCID, Structured Clinical Interview for DSM; SCL-90, Somatic Checklist-90 item; SPS, Spirit Possession Scale; SRQ, Self-Reporting Questionnaire; SSI, Somatization Screening Index; SSQ, Shona Symptom Questionnaire; STAI, State Trait Anxiety Inventory; TKS, Taijin Kyofu Sho. Quality ratings: SAQOR-CPEWe reviewed the studies to identify types of data commonly reported, and we drew upon broader CCD literature to consider key aspects of CCD relevant to quantitative studies that could influence or confound associations between CCD and psychiatric disorder. These issues were incorporated into the Systematic Assessment of Quality in Observational Research (SAQOR)98 to develop a modified version for Cultural Psychiatry Epidemiology (CPE): the SAQOR-CPE. Table 2 lists the seven categories and their criteria. Table 3 includes the quality scoring for individual studies in the review. Below we describe each category and criterion. Table 2 Systematic Assessment of Quality in Observational Research—Cultural Psychiatry Epidemiology (SAQOR-CPE) adaptation and scoring criteria
Table 2 Systematic Assessment of Quality in Observational Research—Cultural Psychiatry Epidemiology (SAQOR-CPE) adaptation and scoring criteria
Table 3 Systematic Assessment of Quality in Observational Research-Cultural Psychiatry Epidemiology (SAQOR-CPE) ratings
SAQOR-CPE (Cultural Psychiatry Epidemiology) ratings adapted from SAQOR developed by Ross et al. 2011. Adapted scoring criteria: Sample: ‘Adequate’, if ≥3 ‘Yes’; control/comparison group: ‘Adequate’, if ≥3 ‘Yes’; Cultural Concept of Distress: ‘Adequate’, if ≥3 ‘Yes’; Quality of measures: ‘Adequate’, if ≥2‘Yes’; follow-up: ‘Adequate’ if change in CCD reported; distorting influences: ‘Adequate’, if ≥2‘Yes’; data reporting: ‘Adequate’, if both ‘Yes’; SAQOR Quality: High (H) if ‘Adequate’ for all 7 categories, Moderate (M) if ‘Adequate’ for 5-6 categories, Low (L) if ‘Adequate’ for 3-4categories;,Very Low (VL) if ‘Adequate’ for 0-2 categories. Abbreviations: A, Adequate; I, Inadequate; N, no; N/A, not applicable); U, Unclear; Y, Yes. Table 3 Systematic Assessment of Quality in Observational Research-Cultural Psychiatry Epidemiology (SAQOR-CPE) ratings
SAQOR-CPE (Cultural Psychiatry Epidemiology) ratings adapted from SAQOR developed by Ross et al. 2011. Adapted scoring criteria: Sample: ‘Adequate’, if ≥3 ‘Yes’; control/comparison group: ‘Adequate’, if ≥3 ‘Yes’; Cultural Concept of Distress: ‘Adequate’, if ≥3 ‘Yes’; Quality of measures: ‘Adequate’, if ≥2‘Yes’; follow-up: ‘Adequate’ if change in CCD reported; distorting influences: ‘Adequate’, if ≥2‘Yes’; data reporting: ‘Adequate’, if both ‘Yes’; SAQOR Quality: High (H) if ‘Adequate’ for all 7 categories, Moderate (M) if ‘Adequate’ for 5-6 categories, Low (L) if ‘Adequate’ for 3-4categories;,Very Low (VL) if ‘Adequate’ for 0-2 categories. Abbreviations: A, Adequate; I, Inadequate; N, no; N/A, not applicable); U, Unclear; Y, Yes. SampleThe Sample category and each of its original five criteria were retained. Representative refers to studies that can be generalized to a population of interest. Cultural Psychiatry Epidemiology (CPE) studies should use the same epidemiological principles as standard health studies. It is especially crucial that CPE studies have a clear definition of the cultural group of interest to which findings can be applied. For example, ataque de nervios findings from Puerto Rico may not be generalizable to Guatemalans, Cubans or Bolivians in their home countries or after immigration. An exemplar study is the investigation of ataques de nervios in the National Latino and Asian American Study, in which outcomes are presented separately for Puerto Ricans, Cubans, Mexicans and other Latinos, revealing group differences.71 Less than one-third of the studies (29%) in this review used culturally appropriate representative sampling. Source refers to how cases are identified. Authors should clearly state if self-labelling, clinician diagnosis or other key informant identification was used to enroll a participant as either CCD or non-CCD. If an individual other than the participant assigned the label, then the degree of concordance between the external label and the self-label should be reported. A study to validate a postpartum depression measure in the Democratic Republic of Congo provides a good example of this: of 91 women identified with a CCD by key informants, only 41 (45%) self-endorsed the CCD label; of 42 women identified by key informants as not having the CCD, only 20 (48%) self-endorsed not having the CCD.56 This illustrates that using key informants in this setting to identify cases and make generalizations is no better than randomly assigning a group of women to CCD vs not-CCD status. In all, 44 studies reported CCD source. Method refers to the process of recruiting participants. In cross-cultural research, recruitment method may bias prevalence rates and association with risk factors. For example, if key informants are used, they may be less likely to identify high-status individuals in the community who have CCD. Stigma may lead to CCD non-disclosure despite experiencing suffering. A study of Darfuri refugees in Chad used United Nations High Commissioner for Refugees (UNHCR) registration to randomly select participants; because caseness (ie CCD status) was not a criteria in the sampling frame, potential participants were approached without prior knowledge of CCD status, thus reducing potential bias in endorsement.93 A total of 43 studies provided some information on recruitment methods. Sample size and power calculation: studies need to be adequately powered to detect differences between groups in exposures, psychiatric disorders or other factors. Prior studies done with similar populations can be used to estimate prevalence of a CCD. If novel CCD are investigated, primary care workers, traditional healers or other key informants could be used to grossly judge whether a CCD is common or infrequent. Of note, if multiple sites are used, controlling for clustering needs to be considered in calculations; local variation in terminology and social composition may affect CCD endorsement. Only one study used a power calculation, which was based on estimates of receiving quality care and not based on CCD prevalence.69 Inclusion/exclusion criteria are crucial for any epidemiological study, to minimize confounds and to have internally comparable participants. For cultural psychiatry, three domains of inclusion/exclusion criteria are important: (i) cultural group, (ii) psychiatric conditions and (iii) CCD. Defining a cultural group could be based on self-labelled ethnicity, linguistic proficiency, years living in particular region or other culturally salient group identifiers. Common psychiatric exclusion criteria are substance use disorders, psychotic disorders or cognitive impairment. One study of nervios in Mexico used the Composite International Diagnostic Interview (CIDI) to exclude participants with substance use disorders and physical injuries that produce nervios.94 A total of 42 studies reported some form of inclusion/exclusion criteria. Control/Comparison GroupAll criteria for this category were retained for the SAQOR-CPE. Inclusion refers to the presence of a comparison group. For the majority of research objectives in cultural epidemiology studies, a comparison group is needed to test inferences. For example, rate of depression among persons without a CCD is important to determine whether a CCD increases depression risk. In a study of a somatic CCD in Nepal, auditory hallucinations were reported by one-fifth of CCD sufferers; however, the control group reported an equally high prevalence of auditory hallucinations.99 In a validation study of the Hopkins Symptom Checklist (HSCL) in Rwanda, a CCD did not differentiate between persons with and without anhedonia,100 thus demonstrating that this CCD was not appropriate to selectively identify persons with that feature of depression. A total of 36 studies included non-CCD participants. Identifiable refers to use of a strategy to clearly differentiate cases from controls. This is generally straightforward when lifetime prevalence is assessed through self-labelling. However, when assessing current episodes, there should be a clear time period to identify cases and controls. For example, if 2-week prevalence is used, is a control with no lifetime episodes comparable to a control with an episode that ended 3 weeks ago? All but 1 study including a comparison provided information regarding how the non-CCD group was identified. Source refers to cases and controls drawn from similar populations. If cases are selected from a psychiatric clinic and controls are drawn from other medical clinics, this biases the CCD group to have greater prevalence of psychiatric conditions. Community representative samples are ideal to assure the same source.71,72,79,93,101. Only one study lacked information on source of control participants. Matching and randomization may be used in some studies to optimize similarities between groups. For example, if a researcher is trying to identify family-related protective factors against ataque de nervios, then matching based on economic status, educational status and residential region in recruitment or statistical techniques such as propensity score matching would be helpful. Matching could be used to control for issues related to language proficiency55 or years of residence in a new country that may confound endorsement of a CCD. One study employed a matching process. Statistical control refers to using multivariable models to control for issues that may confound relationships between CCD and psychiatric disorders such as socioeconomic status, other psychiatric comorbidities and stressful exposures. A study of dhat found an association with psychological distress (General Health Questionnaire caseness) when statistically controlling for age, district of residence and marital status, all of which were independently associated with dhat.69 In the multivariable analysis, only psychological caseness and region of residence independently associated with dhat. A total of 22 studies included some form of multivariable analysis. Cultural Concept of DistressCCD is a category added to the SAQOR for cultural psychiatry studies. CCD categorical classification refers to the presentation of data on who does and does not endorse a CCD as a dichotomous variable, ideally through self-endorsement by participants; 27 (60%) of the studies included a categorical response by participants regarding whether they did or did not endorse having the CCD. The remainder used either clinically-assigned labels of a CCD or a proxy measure, such as having somatic complaints. CCD prevalence refers to obtaining data on lifetime and/or current prevalence: if current prevalence, then the time period should be specified; 10 (22%) studies assessed lifetime prevalence and 19 (41%) assessed current CCD, such as in the past 2 weeks or past month and 17 (37%) were unclear regarding time frame and whether the time range matched with the timing of the psychiatric disorder. For example, some studies did not include assessment period and others used vague language, such as ‘recent' episodes. CCD label type should describe whether the CCD is attributed according to a single symptom, a constellation of symptoms, a certain type of exposure or being part of a vulnerable group. In many cases, CCD may reflect a combination of the above. A study in Mongolia78 used the Explanatory Model Interview Catalogue (EMIC)31,32 to collect this information. In a study of women in Zimbabwe, explanatory models were collected and revealed that kufungisisa was both a symptom of distress and a cause of health problems.54 A total of 37 studies included information on label type. CCD severity refers to measurement of the frequency, number of associated symptoms or degree of impairment associated with a CCD. For example, two individuals may both endorse lifetime ataques de nervios but one individual may have weekly episodes whereas the other has them every few years. This would impact the association with psychiatric categories; 27 studies included severity information. CCD course refers to the age of onset, duration of episodes, timing of episodes and chronicity of experience, with special attention to overlapping periods with psychiatric symptoms. For example, the mean age of onset of dhat in one study was 21.6 years among men.59 Knowing this could help readers of the study consider potential psychosocial erectile dysfunction versus age- or diabetes-related dysfunction. Studying onset of ataque de nervios revealed that the episodes typically preceded depression and anxiety symptoms,70 which is helpful information for screening and prevention. Only 14 studies included course information. Measurement QualityThe Measurement Quality category of the SAQOR includes exposure and outcome measures. An additional category for functioning was based on the CCD literature. Exposure is important for CCD because explanatory models typically associate certain types of experiences with invoking CCD. For example, family, financial, health and political stressors are strongly associated with jham-jham paresthesia in rural Nepal.79 However, work and academic stressors are not risk factors for jham-jham. This contrasts with brain fag in Nigeria, in which academic stress is assumed to be one of the main precipitants.86,102 Orthostatic hypotension is not assumed by the lay American public to be a trigger for PTSD, but Hinton and colleagues have shown that this sensation mediates post-traumatic psychiatric sequelae among Cambodians.103-106 Therefore, assessing dizziness and orthostatic hypotension triggers is crucial to a culturally salient study of khyal attacks. A total of 32 studies included information on exposures. Psychiatric outcome measures require special attention in cross-cultural research. If an instrument has not been validated in the local context, results are difficult to interpret.107 Lack of association between CCD and the psychiatric measure may be due to using a non-validated instrument rather than cultural-exclusivity of the distress; 24 studies used instruments validated for the cultural group, and some provided psychometric properties for the instrument in that population.54,61,66,79,92 When instruments have not been validated, then significant detail should be provided on how instruments underwent transcultural translation to achieve cross-cultural equivalence.108,109 Functional outcome was added as an additional criterion for measurement quality. Early debates in culture-bound syndrome research raised questions about distinguishing between abnormal behaviours related to cultural performance vs abnormal behaviours associated with impairment in multiple domains of life.110–112 Bolton and colleagues have developed rapid, feasible approaches to create functional impairment measures.113 Their approach makes it easy to assess whether persons with CCD are more likely to have impaired role fulfilment. Other studies in our review used standard functioning measures such as the WHO Disability Assessment Schedule (WHODAS) and the Sheehan Disability Scale. A total of 20 studies reported some form of functioning assessment. Follow-UpThe Follow-Up category includes percentage lost to follow-up and reasons lost to follow-up. We added change in CCD prevalence. Four studies included a follow-up assessment. Percentage lost to follow-up is standard reporting for longitudinal studies. In a Nigerian study, 57% of patients with ode ori participated in a 1-year follow-up evaluation.84 Wherever possible,Reasons for loss to follow-up should be elicited and reported to inform interpretation of results, highlight potential biases, and help shape future longitudinal studies of CCD. In a study of dhat, follow-up rates were much lower among patients receiving counselling compared with patients receiving medication; this suggested that participants were dissatisfied with psychotherapeutic interventions and dropped out.59 We added CCD change at follow-up as an additional criterion. In the CCD literature we reviewed, a major shortcoming was lack of CCD documentation at follow-up. In treatment studies of dhat and hwa-byung, CCD was not evaluated post treatment.59,114,115 Among Cambodian refugees with ‘thinking too much,’ 58% of patients in California received sedatives whereas 20% did in Massachusetts; however, no information was provided regarding which group showed better outcomes.116 The absence of information on resolution of CCD during mental health treatment is a major gap in the existing literature. In studies in Nigeria and China, CCD did not resolve after psychotropic medication despite improvement in psychiatric disorders.77,84 In contrast, studies of culturally adapted psychotherapy treatments showed improvement for ataque de nervios among Latinas and khyal attacks among Cambodian refugees.75,106 A study with pharmacotherapy showed improvement of khyal attacks, ‘thinking a lot' and several other idioms among Cambodian refugees as measured by effect sizes.135 Measuring CCD in longitudinal observational and treatment studies is crucial to determine whether CCD worsen, resolve with or are independent of psychiatric symptoms trajectories. Distorting InfluencesDistorting Influences in the original SAQOR focused on distorting influences related to maternal depression.98 We modified the distorting influences category for factors that commonly confound cultural psychiatry epidemiology studies. Psychiatric comorbidities: because of the high rate of comorbidity among psychiatric disorders, it is possible that associations between CCD and a target psychiatric disorder are the result of another condition. For example, PTSD and depression are often comorbid. If CCD have significant associations with PTSD, it may be that the associations are better explained by associations with depression. Controlling for comorbidities through selection criteria and analysis is crucial. In a study of social phobia and taijin-kyofu-sho (TKS), a CCD in Japan and Korea, the researchers excluded persons with major depressive disorders, bipolar affective disorder, psychosis and substance misuse to assure that associations between TKS and social phobia were not the result of mutual associations with other disorders.65 In a study of a fatigue CCD in Mongolia, yadargaa associated significantly with a scale for chronic fatigue syndrome in bivariate analysis. However, when other psychiatric conditions were entered into the analysis, yadargaa associated significantly with depression but the association with chronic fatigue syndrome was no longer significant.78 A study in Uganda among war-affected youth stands out in the CCD literature because multiple CCD were addressed in the same population.57 This allowed for testing CCD comorbidities in addition to psychiatric comorbidities. Half of the studies include psychiatric comorbidity information. Physical health comorbidities also impact associations between CCD and psychiatric diagnoses. Investigators of dhat often account for physical comorbidities, especially sexually transmitted infections (STIs), in their analyses.59,64,69 Including STIs among both dhat cases and controls revealed that STIs were not associated with dhat; instead, psychological distress differed between the groups.69 Controlling for possible physical pathologies led to the finding in Nepal that a somatic complaint of paraesthesia, which was strongly associated with depression, was not the result of psychosomatization but a consequence of physical health problems, commonly B12 deficiency.79 Six studies included information on physical comorbidities. Treatment status is a potential confound. If participants are receiving biomedical or traditional treatments, this may influence psychiatric disorders, CCD or both. Seven studies included information on treatment status. Other confounds include linguistic proficiency differences which may influence endorsement of CCD. One study reported that missing data were significantly more common among persons with low English proficiency.55 A total of 22 studies included other potential confounds. Reporting of DataIn the Reporting of Data category, the SAQOR requires that all studies include information on missing data. Missing data were repoted by only one study.55 Clarity and accuracy of data refers to use of confidence intervals, multivariable analyses, and tables and figures that are easily interpreted. A total of 37 studies presented data clearly. Other studies inconsistently identified CCD vs non-CCD groups; for example, they did not clarify which participants were included in analyses or included figures that did not clarify CCD association with psychiatric measures in quantitative comparisons. Meta-analyses for likelihood of a psychiatric classification given presence of a cultural concept of distressMeta-analyses were conducted with psychiatric conditions as the outcome (see Table 4 and Figures 2–6). The results should be interpreted as the odds that an individual has a given psychiatric disorder given endorsement of a CCD. For example, among persons who endorse dhat, ataque de nervios, susto or other CCD, there is an 8-fold greater odds of experiencing bivariate depression compared with persons who do not endorse a CCD. The level of heterogeneity, not surprisingly, was significant for most of the outcomes: all anxiety disorders (Q = 13.75, df = 28, P < .05), panic (Q = 2.43, df = 8, P < .05), PTSD (Q = 0.10, df = 2, P < .05), depression (Q = 6.15, df = 19, P < .05), somatoform disorders (Q = 0.67, df = 6, P < .05), and general anxiety (Q = 8.70, df = 16, P < .10). Converting Q statistics to I2 to account for small numbers of studies, all summary effects had heterogeneity percentages greater than 75%. Only general psychological distress had a non-significant test of heterogeneity, (Q = 7.41, df = 8, P = 0.5), with I2 = 8% suggesting that associations of general psychological distress with CCD are generally homogeneous with limited variance attributable to between-study characteristics. Figure 2 Meta-analysis with forest plot for odds of having depression given presence of cultural concepts of distress (CCD); n = 9032, odds ratio = 7.55 (95% confidence interval, 6.69–8.52) Figure 3 Meta-analysis with forest plot for odds of having general anxiety given presence of cultural concepts of distress (CCD); n = 8211, odds ratio = 5.06 (95% confidence interval, 4.48–5.70) Figure 4 Meta-analysis with forest plot for odds of having panic attacks/disorder or PTSD given presence of cultural concepts of distress (CCD); panic attacks/disorder, n = 6158, odds ratio = 4.48 (95% confidence interval, 3.77–5.32); posttraumatic stress disorder (PTSD), n = 1246, odds ratio = 10.10 (95% confidence interval, 7.51–13.57) Figure 5 Meta-analysis with forest plot for odds of having general psychological distress given presence of cultural concepts of distress (CCD); n = 6658, odds ratio = 5.39 (95% confidence interval, 4.71–6.17) Figure 6 Meta-analysis with forest plot for odds of having somatoform disorders given presence of cultural concepts of distress (CCD); n = 3268, odds ratio = 2.68 (95% confidence interval, 2.18–3.28) Table 4 Meta-analysis for odds of meeting criteria for a psychiatric category among persons endorsing a cultural concept of distress
Table 4 Meta-analysis for odds of meeting criteria for a psychiatric category among persons endorsing a cultural concept of distress
Potential sources of between-study variation in association of cultural concepts of distress with psychiatric categoriesGiven the high heterogeneity among the studies, we used generalized estimating equations (GEE) to determine the association of study design with strength of odd ratios between CCD and psychiatric categories (see Table 5). We conducted 13 bivariate analyses of study characteristics with strengths of odds ratios between CCD and psychiatric disorders. Variables significant in bivariate analyses were entered into the multivariable analysis. In the multivariable analysis, studies conducted in the Americas had greater ORs than those conducted in Africa or Asia; studies labelled as ‘culture-bound’ had greater ORs than any of the other labels; validation studies had ORs 16 points greater than studies in which the objective was to compare CCD with psychiatric disorders; greater sample sizes were associated with greater ORs; self-report multi-item checklists had 6 points greater ORs than dichotomous categorical self-report scores, and medium quality SAQOR-CPE rankings were 7 points below ORs of very low quality studies. Table 5 Generalized estimating equation for association of study design variables with magnitude of odds ratio between psychiatric category and cultural concept of distress
N = 79 comparisons; number of studies 26. Only items with significant bivariate associations were included in the multivariable model. *P < .05; **P < .01; ***P < .001. Table 5 Generalized estimating equation for association of study design variables with magnitude of odds ratio between psychiatric category and cultural concept of distress
N = 79 comparisons; number of studies 26. Only items with significant bivariate associations were included in the multivariable model. *P < .05; **P < .01; ***P < .001. DiscussionWithin the growing body of literature comparing cultural concepts of distress (CCD) and psychiatric disorders, there is a wide range of quality and epidemiological rigor. Twelve (27%) of the studies had medium quality based on the Systematic Assessment of Quality in Observational Research–Cultural Psychiatry Epidemiology (SAQOR-CPE) ranking system. The remainder were of low or very low quality. Studies lack both basic criteria for epidemiological reporting (e.g. representative samples, prevalence parameters, missing data frequency and management, and controlling for potential confounds) and key aspects of CCD reporting (e.g. differentiation among symptoms, syndromes, and aetiological models; operationalization of cultural and ethnic groups to generalize findings; assessment of confounders; and severity and course of distress). Making generalizations based on summary effects from meta-analysis is impeded by the high degree of heterogeneity in all but one of the analyses. The high degree of heterogeneity is not surprising given the wide range of quality and methodological approaches among the studies. Studies conducted in the Americas were more likely to show an association of CCD with psychiatric disorders. This may represent acculturation issues among the populations studied because most of the participants were immigrants in the USA. Phan and colleagues suggested that CCD were strongly associated with psychiatric disorders among Vietnamese immigrants in Australia because of acculturation effects that reframe understandings of mental health and disorder.92 We were surprised to find that studies in which the researcher referred to the CCD as ‘culture-bound’ had stronger associations between the CCD and psychiatric disorders than all other labels. This was counter-intuitive given that ‘culture-bound’ implies a distinction from psychiatric nosology. However, we found that labels such as ‘culture-bound’ or ‘idioms of distress’ were not applied systematically. The same CCD, e.g. ataques de nervios, was described as a culture-bound syndrome, idiom of distress, and popular category by different researchers. Moreover, the category labels for CCD change between studies even within single research teams. Therefore, we do not suggest that comparing studies based on the label used is an informative lens and may lead to potentially spurious associations. The finding that validation studies were most likely to show an association between CCD and psychiatric disorders is expected, given that in validation studies researchers likely try to identify the CCD that are most similar to a psychiatric category. Furthermore, there is high likelihood of a publication bias in validation studies with negative findings less likely to be published (it is rare to read a published negative validation study). The same publication bias may not hold for studies comparing CCD and psychiatric disorders that have negative findings, as this would still be theoretically significant for culture-bound suppositions. Multi-item checklists for CCD assessment were associated with stronger associations between CCD and psychiatric disorders. This is consistent with checklists operating more similarly to psychiatric diagnostic criteria. Studies in which single items are used for CCD endorsement likely enable greater diversity of manifestations and framings. The final noteworthy finding of our review is that medium quality studies had weaker associations between CCD and psychiatric disorders than very low quality studies (no high quality studies were identified in this review). This raises a crucial issue: we do not hypothesize that greater epidemiological rigor will foster stronger associations between CCD and psychiatric disorders. The converse is equally likely: more rigorous and culturally appropriate studies (as recognized by higher SAQOR-CPE rankings) may represent studies that describe CCD more accurately and thus capture the uniqueness from psychiatric categories. For example, studies than controlled for psychiatric and physical health comorbidities had weaker associations than those not controlling for comorbidity. One of the most important quality issues was better documentation of CCD course and timing in association with psychiatric disorders. Future studies that closely document course and use longitudinal designs in well-contextualized community settings will shed new light on the experience and meaning of CCD and their association psychiatric pathological categories. Emulating the work of pioneers in psychiatric epidemiology, such as Alexander Leighton who followed a rural population in Canada over decades to understand life trajectories of mental illness, can help inform future studies.28,117,118 LimitationsThe objective of this review was to provide an overview of the quality of epidemiological studies comparing CCD and psychiatric disorders. Whereas the issues highlighted here and the recommendations provided can be used to strengthen the epidemiological rigor of CCD studies, we caution against generalizing the findings beyond the literature identified here. We limited our initial search of the literature to PubMed/MEDLINE and English-language publications. All of studies were coded by the first author; future reviews should include multiple coders with inter-rater reliability metrics. Future research also should incorporate databases such as PsycInfo, which may include more rigorous psychological studies, and Web of Science, which will capture social science and medical anthropological journals not indexed in PubMed. Inclusion of books and book chapters would also bolster the social science representation. Ultimately, to make broad claims about the association of CCD and psychiatric disorders, access to investigators’ original data would be most helpful because many of the shortcomings reported here may reflect what is reported rather than what is collected. We hope that the adaptation of the SAQOR-CPE can be applied to broader searches and to the design of future cultural psychiatric epidemiology studies. Applications to global mental healthThe DSM-5 and other publications have provided recommendations for the application of CCD to improve clinical care.15,119 CCD also can be applied to improve research and public health interventions in global mental health, with special attention to low resource settings:
ConclusionsDespite claims that cultural concepts of distress are not amenable to epidemiological study, our literature review demonstrated a range of important contributions of CCD epidemiological studies to detection of mental health problems, evaluation of interventions, identification of vulnerable groups, and identification of cultural biases in psychiatric diagnostic criteria. The literature, however, suffers from a lack of epidemiological rigor and lack of comprehensive data collection about key issues in CCD. Tools such as the SAQOR-CPE are needed to systematically evaluate this literature and establish guidelines for research design and reporting for global mental health studies. Ultimately, combining the strengths of psychiatric epidemiology and cultural psychiatry will foster equitable, feasible, and effective global mental health services. FundingThis work was supported by the National Institute of Mental Health [U19 MH095687-01S1, South Asian Hub for Advocacy, Research & Education on Mental Health (SHARE), Principal Investigators: Vikram Patel and Atif Rahman] supplement for continuity of research experience during clinical training provided to the first author (BAK). Author BNK is supported by the National Science Foundation Graduate Research Fellowship [Grant No. 0234618]. Conflict of interest: None declared.
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What term describes a cluster of symptoms that are considered recognizable diseases only within specific cultures?In medicine and medical anthropology, a culture-bound syndrome, culture-specific syndrome, or folk illness is a combination of psychiatric and somatic symptoms that are considered to be a recognizable disease only within a specific society or culture.
What type of mental illness symptoms is culture specific?Culture-bound syndromes include, among others, amok, amurakh, bangungut, hsieh-ping, imu, jumping Frenchmen of Maine syndrome, koro, latah, mal de pelea, myriachit, piblokto, susto, voodoo death, and windigo psychosis.
What are the symptoms of cultureAtaque de nervios, seen in Latin American and Latin Mediterranean cultures, is associated with a sense of being out of control, uncontrollable shouting, trembling, crying, heat in the chest rising to the head, and fainting or seizure-like episodes, and somewhat resembles panic disorder.
What is a cultural symptom?Improving Cultural Competency for Behavioral Health Professionals. Cultural syndromes are clusters of symptoms and attributions that tend to co-occur among individuals in specific cultural groups, communities, or contexts. They are recognized locally as coherent patterns of experience.
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What are clusters of symptoms that are considered recognizable diseases only within specific cultures?
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