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Journal of Psychosomatic Research 115 (2018) 58–65

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Journal of Psychosomatic Research


journal homepage: www.elsevier.com/locate/jpsychores

Review article

Death anxiety and its association with hypochondriasis and medically T


unexplained symptoms: A systematic review

Birgit M. aan de Steggea, , Lineke M. Takb,c, Judith G.M. Rosmalenb,c, Richard C. Oude Voshaarc,d
a
Department of Old-Age Psychiatry, Dimence, Institute for Mental Health, Overijssel, the Netherlands
b
Specialistic Center for Complex Medically Unexplained Symptoms and Somatic Symptom Disorders, Dimence, Institute for Mental Health, Overijssel, the Netherlands
c
University of Groningen, University Medical Center Groningen, Interdisciplinary Center Psychopathology and Emotion Regulation, the Netherlands
d
University of Groningen, University Medical Center Groningen, Department of Old-Age Psychiatry, the Netherlands

A R T I C LE I N FO A B S T R A C T

Keywords: Objective: To systematically review of the available literature to (1) examine the association between death
Death anxiety anxiety and hypochondriasis and (2) examine the association between death anxiety and medically unexplained
Hypochondriasis symptoms (MUS).
Medically unexplained symptoms Methods: A systematic literature search was conducted in Embase, PsycINFO, Pubmed and Ovid databases and
reference lists of selected articles. Articles were included when the research population concerned people with
hypochondriasis and/or MUS in who death anxiety was assessed by a validated research method. Two in-
dependent reviewers verified that the studies met the inclusion criteria, assessed the quality of the studies and
extracted relevant characteristics and data. The data were descriptively analysed.
Results: Of the 1087 references identified in the search, six studies on the association between death anxiety and
hypochondriasis and three studies on the association between death anxiety and MUS met inclusion criteria. All
studies found a positive association of death anxiety with hypochondriasis and/or MUS. The design of all studies
was cross-sectional and the overall quality of the studies was low. The influence of age or sex on these asso-
ciations was not analysed in any of the studies. Given the diversity in setting, population, study design, and
methods used, a meta-analysis was not possible.
Conclusion: All studies found a positive association of death anxiety with hypochondriasis and/or MUS.
Acknowledging that death anxiety may play a prominent role in hypochondriasis/MUS populations, future re-
search should address (potentially modifiable) determinants of death anxiety in these populations.

1. Introduction DSM-IV, can now fulfil the criteria for SSD or illness anxiety disorder
(IAD) in the DSM-5. This depends on the impact of the physical
Hypochondriasis is an excessive fear of or preoccupation with a symptoms on patient's functioning [2]. In IAD, physical symptoms are
serious illness. Medically unexplained symptoms (MUS) are persistent of minor relevance. The main characteristics of the disorder are anxiety
bodily complaints for which adequate examination does not reveal or being convinced about suffering from a serious disease, due to mis-
sufficient explanatory structural or other specified pathology [1]. Both, interpretations of bodily symptoms. According to DSM-5, 75% of pa-
hypochondriasis and MUS, are common conditions in the general po- tients previously diagnosed with hypochondriasis would fit the new
pulation, but only in case of significant distress or impairment in category of SSD and 25% would fit the new IAD.
functioning, these conditions are considered psychiatric disorders. In Hypochondriasis is estimated to be equally prevalent in men and
DSM-IV these conditions were classified within the section of somato- women, whereas MUS predominantly affect women [3]. A study ex-
form disorders, emphasizing the presence of unexplained physical amining the relationship of hypochondriasis with age found no asso-
symptoms. In DSM-5 these conditions are classified as somatic symptom ciation [4]. However a recent review suggests that prevalence rates of
disorder (SSD), emphasizing dysfunctional cognitions, emotions or be- MUS and hypochondriasis decline after the age of 65 years [5].
haviour in relation to physical symptoms [2]. Patients previously di- Given the debate on classification, we choose to use the terms hy-
agnosed with hypochondriasis (section somatoform disorder) in the pochondriasis and MUS in their broadest sense, as until recently these


Correspondending author at: Dimence, Department of Old-Age Psychiatry, Pikeursbaan 3, 7411 GT Deventer, the Netherlands.
E-mail addresses: b.aandestegge@dimence.nl (B.M. aan de Stegge), l.tak@dimence.nl (L.M. Tak), j.g.m.rosmalen@umcg.nl (J.G.M. Rosmalen),
r.c.oude.voshaar@umcg.nl (R.C. Oude Voshaar).

https://doi.org/10.1016/j.jpsychores.2018.10.002
Received 20 June 2018; Received in revised form 8 October 2018; Accepted 9 October 2018
0022-3999/ © 2018 Elsevier Inc. All rights reserved.
B.M. aan de Stegge et al. Journal of Psychosomatic Research 115 (2018) 58–65

terms have most often been used, and they also apply to subjects in- of themes in drawings of death concepts) also offer a non-obtrusive
cluded in studies in non-clinical populations. entry into the concerns of a person who might be reluctant to ac-
Death anxiety can be defined as conscious or unconscious fear of knowledge the fearfulness of death in response to direct questions [22].
death, due to the awareness that death can occur at any time. This In this systematic review we will: (1) examine the association be-
awareness of our imminent death makes us vulnerable, because it tween death anxiety and hypochondriasis and (2) examine the asso-
threatens our evolutionary predestination to survive [6,7]. ciation between death anxiety and MUS. Additionally we will explore
Although people suffering from hypochondriasis or MUS may be whether these associations differ for age and sex.
afraid of death, little scientific research is done on death anxiety as a
possible contributing factor for developing hypochondriasis or MUS.
This gap in the medical literature may be explained in several ways. 2. Methods
Firstly, the idea that fear of death is irrelevant for the development of
psychopathology, since it is a normal aspect of everyday life. Secondly, 2.1. Protocol
reluctance on the side of psychiatrists and psychotherapists to deal with
this subject, for example because of their own fear of death. Thirdly, A review protocol for this study was developed in February 2017,
continuing influence of Freud's postulation that fear of death rarely, if and registered in the Prospero register (CRD42017058198).
ever, can be a part of unconscious neurotic conflicts [8].
In contrast to the medical literature, philosophers and psychologists
2.2. Search strategy
have investigated death anxiety for many years, resulting in many pa-
pers on death anxiety in nonclinical populations. These studies have
The literature search was conducted in accordance with the
collectively contributed to the idea of death anxiety as a transdiagnostic
Preferred Reporting Items for Systematic Reviews and Meta-Analyses
construct. Higher levels of death anxiety are generally accompanied by
(PRISMA) statement for reporting meta-analysis [23].
elevated levels of neuroticism, as is shown in numerous population-
In November 2017 a systematic literature search was conducted in
based studies [9]. In a review study, death anxiety has been postulated
Embase, PsycINFO, Pubmed and Ovid databases with the following
to play a significant role in the development and severity of depressive-,
keywords: functional somatic disorder*, medically unexplained
eating-, obsessive-compulsive- and several anxiety disorders [10]. In
symptom*, nonorganic somatic symptom disorder*, psychosomatic*,
this context these disorders are regarded as a consequence of an in-
psychogenic*, hysteri*, conversion disorder*, functional disorder*, so-
efficient death anxiety-buffering system [11–13]. Death anxiety can
matization, somatoform disorder*, psychophysiologic disorder*, so-
also be hypothesized to be a basic fear underlying the development,
matoform*, hypochondri*, illness anxiety disorder*, death anxiety, fear
course, and maintenance of hypochondriasis and/or MUS [12,14,15].
of death, mortality salience, terror management theory, attitude to
With respect to the association between death anxiety and hypochon-
death, and existential anxiety. There was no restriction to publication
driasis or MUS, a sense of bodily threat is thought to be related to pa-
year. Titles and abstracts were screened by two independent reviewers
thological fear of death [14,16,17]. Because death is typically preceded
(BMS and LMT) and full text articles were retrieved for potential re-
by cessation of vital bodily functions, death anxiety experienced by
levant articles. These full text articles were assessed for eligibility.
patients with MUS is thought to involve fears of bodily failure, pain,
Reference lists from the selected studies and relevant articles on the
separation, and loss of control and power [17,18].
subject were searched for additional relevant papers. In case of dis-
Although there are a number of theories on the association between
agreement RCOV was consulted for a final decision. Post-hoc, at the
death anxiety and/or MUS, few studies have investigated this associa-
request of a reviewer, we performed an additional search with the
tion empirically. Therefore a systematic review on this topic is war-
keywords ‘illness phobia’ and ‘thanatophobia’. The search with illness
ranted. More knowledge is needed about how death anxiety cuts across
phobia yielded no extra relevant articles. The results for thanatophobia
diagnostic categories and whether or not it should be considered a
included one potentially relevant article. After reading this article it
determinant for developing hypochondriasis or MUS. If so, death an-
turned out to be based on the same sample as our included study by
xiety deserves a more prominent place in the assessment and treatment
Barsky et al. (1989).
of hypochondriasis and MUS.
For proper interpretation of individual studies included in this re-
view, knowledge of different concepts of death anxiety and their po- 2.3. Inclusion and exclusion criteria
tential impact for the association between death anxiety and hy-
pochondriasis and/or MUS are deemed necessary. Appendix A presents The subject of research had to concern people with hypochondriasis
a list of these concepts. and/or MUS, or patients who met criteria from the DSM-IV or DSM-5
such as illness anxiety disorder, somatoform disorders or somatic
1.1. Tools to measure death anxiety symptom disorder. Furthermore, death anxiety should be assessed by
validated instruments or research methods.
A variety of instruments have been developed to assess death an- Due to the expected limited number of original articles, relevant
xiety in both clinical and non-clinical populations. Many scales consider observational research was also included, except for case studies or
death anxiety to be a unidimensional construct. An example of a mul- series with less than ten participants. Demographic information about
tidimensional scale is the Multidimensional Fear of Death Scale age and sex had to be described. Only articles in English, French,
(MFODS), which measures eight distinct apprehensions about death German and Spanish were included.
and dying [19]. When fear of death is suppressed from conscious
awareness, a questionnaire with a choice response format may not be
the most appropriate tool and indirect testing may be needed. One 2.4. Data extraction
example of measuring death anxiety indirectly is through the Gott-
schalk-Gleser verbal content analysis. Verbatim transcripts of five Reviewers BMS and LMT independently scored the included studies.
minutes of free speech are submitted to a content analysis with a The following variables were extracted from all studies: author, country
scoring scheme. Whenever death anxiety is mentioned, a weight is as- and year of publication, study design, death anxiety concept, mea-
signed according to the degree of (psychodynamic) association with the surement tools for death anxiety and hypochondriasis/MUS, setting/
self [20]. A computerized language analysis tool is Linguistic Inquiry characteristics, sample size, age and sex of participants, and conclu-
and Word Count (LIWC) [21]. Phenomenographic approaches (coding sions.

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B.M. aan de Stegge et al. Journal of Psychosomatic Research 115 (2018) 58–65

2.5. Study quality area and methodology, we will first describe each study separately,
followed by brief integrative analysis. Tables 1 and 2 provide a sum-
BMS and LMT assessed the methodological quality of the selected mary of study characteristics.
studies. For this review we made an adaptation of the Newcastle-
Ottawa Quality Assessment Scale (NOS) for cohort studies, available on 3.2.1. Included studies about the association between death anxiety and
request [24]. Such an adaptation of the NOS for the use in a systematic hypochondriasis
review of cross- sectional research has been done before [25]. Dis- 3.2.1.1. Kellner 1987 [26]. Matched groups of psychiatric outpatients
agreements were resolved by consensus or RCOV was consulted for a with hypochondriasis, nonpsychotic psychiatric disorders, patients in a
final decision. family medicine clinic and workers at local companies (every group
consisting of 21 subjects) were compared on all eight subscales of the
3. Results Illness Attitude Scales (IAS), including a subscale measuring death
anxiety. Patients with hypochondriasis had significantly different scores
3.1. Study selection on all scales of the IAS and reported substantially more death anxiety
compared to the other three groups who did not have significantly
A search in four databases yielded a total of 1795 citations. After different scores from each other.
removal of duplicates and screening titles and abstracts for subject re-
levance and language restrictions, full text articles of 41 citations were 3.2.1.2. Barsky 1989 [27]. Hypochondriasis, the tendency to amplify
retrieved. Of these, 32 did not meet the inclusion criteria (no original somatic sensations, and health-related attitudes and concerns
research, two papers on the same study sample, case reports) and were (including death anxiety) were studied in a random sample of 177
excluded. The language restriction did not lead to exclusion of a po- outpatients at a general medicine clinic. Hypochondriasis was highly
tentially relevant articles. After cross-referencing the 40 articles, two correlated with fear of ageing and death. Somatosensory amplification
more articles were included. No general population studies meeting our was the most powerful predictor of hypochondriasis (more strongly in
criteria were found. women than in men) and was also significantly correlated with fear of
In total, nine studies were included: six studies concerning the as- death and bodily vulnerability.
sociation death anxiety and hypochondriasis and three studies con-
cerning the association death anxiety and MUS. None of the included 3.2.1.3. James 2002 [28]. The association between beliefs about death,
studies used DSM-5 criteria. Fig. 1 depicts the study selection process superstitious beliefs, and hypochondriasis was explored in 106 Roman
and results of each review step. Catholics and 197 atheists. Hypochondriasis was associated with death
anxiety in both groups. The expected negative association between
3.2. Study characteristics positive beliefs about death and hypochondriasis was not supported in
either sample. Indifference toward death was significantly negatively
Because of the heterogeneity of the included studies in both content correlated with somatosensory amplification among atheists.
Identification

Records identified through


database searching
N=1795

Records screened Records excluded


after removing duplicates N=1047
N=1087 Subject not relevant N=1043
Screening

Language restriction N=4

Full-text articles assessed for Full-text articles excluded:


eligibility N=40 No original research article N=31
Case reports <10 persons: N=2
Eligibility

Eligible articles
from citation search
N=2

Articles included for the review


N= 9
Included

Studies included for Studies included for death


death anxiety and MUS anxiety and hypochondriasis
N=3 N=6

Fig. 1. Study Selection.

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B.M. aan de Stegge et al.

Table 1
Studies on death anxiety and hypochondriasis.
First Coun-try Design Concept death Death anxiety Hypochondria-sis Setting/Characteristics Sample size F (%) Age Conclusions
author anxiety
Year

Kellner USA Cross-sectional, Cognitive IAS DSM-III criteria Psychiatric outpatients with 84 62 Mean 40 Death anxiety was higher in patients
1987 case-control approach hypochondriasis matched (range with hypochondriasis (thanatophobia
nonhypochondriacal psychiatric patients, 24–60) scale of the IAS, X = 7.6 (SD 3.7))
family practice patients and employees. compared to psychiatric patients
X = 2.3 (SD 2.0), family practice
patients X = 1.3 (SD 1.8) and
employees X = 1.1 (SD 1.5), all
p < .001.
Barsky USA Cross-sectional, Cognitive Self-developed WI Medical outpatients primary care. 177 62 Mean 54.2 Death anxiety was correlated with
1989 correlational approach questionnaire (range hypochondriasis score (r = 0.51,
18–86) p < .001).
James UK Cross-sectional, Cognitive DPS, DBQ, HAQ, SIQ, SSAS Sample of Roman Catholics of a religious 303 48 Mean 31.6 Death anxiety was correlated with
2002 correlational approach community. (range hypochondriasis.
Atheists of an Atheist/agnostic organization 18–79)
and a university
Noyes USA Cross-sectional, Cognitive Self-developed WI, HAQ, SSI, Academic general medicine outpatients: 49 162 72 Mean 51.1 Death anxiety was higher in patients

61
2002 case-control approach questionnaire Structured Diagnostic with hypochondriasis, 113 without with hypochondriasis (53.3 (SD 20.8))
Existential Interview for hypochondriasis. compared to patients without
approach Hypochondria-sis hypochondriasis (29.4 (SD 18.9)),
Psychoana- (DSM-IV) p < 0.001. This association was
lytic approach independent of age.
Hiebert Canada Cross-sectional, Cognitive Thanato-phobia IAS, (without Patients with DSM-IV hypochondriasis, 39 Not provided Not Death anxiety was correlated with the
2005 Correla-tional approach subscale of IAS, thanatophobia setting unknown. provided extent of hypochondriacal symptoms
TDAS, MFODS subscale), WI (IAS Thanatophobia subscale: r = 0.62;
TDAS: r = 0.51, MFODS r = −0.48).
Schütte Germa-ny Cross-sectional, Cognitive IAS, MODDI IAS, WI Self-help groups with patients with 120 73 45 (range Death anxiety in patients with
2016 case-control approach hypochondriasis, panic disorder, depression 18–65) hypochondriasis (71.17 (SD 12.30)) is
(all DSM-IV-TR criteria) and healthy comparable to patients with panic
controls. Inclusion via health care disorder (69.97 (SD 11.58)) and higher
professionals and local newspaper compared to patients with depression
announcements. (57.20 (SD 9.03)) and healthy controls
(54.20 (SD 12.18), F = 15.680, df = 3,
p < 001).

DBQ: Death Beliefs Questionnaire (A. Wells & A. Hackman, unpublished), DPS: Death Perspective Scale (Spilka, 1977), HAQ: Health Anxiety Questionnaire (Lucock & Morley 1996), IAS: Illness Anxiety Scale (Kellner,
1986), MFODS: Multidimensional Fear of Death Scale (Hoelner, 1979), MODDI: Multidimensional Orientation toward Death and Dying Inventory (Wittkowski, 1996), SSAS: Somatosensory Amplification Scale (Barksy,
Wyshak, & Klerman, 1991), SIQ: Symptom Interpretation Questionnaire (Robbins & Kirmayer, 1991), SSI: Somatic Symptom Inventory (Barsky, 1992), TDAS: Templer Death Anxiety Scale (Templer, 1970), WI: Whiteley
Index (Pilowsky, 1967).
Journal of Psychosomatic Research 115 (2018) 58–65
B.M. aan de Stegge et al. Journal of Psychosomatic Research 115 (2018) 58–65

ACR: American College of Rheumatology, ASDA: Arabic Scale of Death Anxiety (Abdel-Khalek, 2003), CLFODS: Collett-Lester Fear of Death Scale (Lester & Abdel-Khalek, 2004), FD: functional dyspepsia, DU: duodenal
ulcer, FM: Fibromyalgia, GAD: generalized anxiety disorder, Gottschalk-Gleser content analysis of verbal behavior (Gottschalk & Gieser, 1969), GBB: Giessen Subjectiv Complaints Checklist (Brahler & Scheer, 1995), IBS:
3.2.1.4. Noyes 2002 [18]. A self-developed fear of death scale and

patients with FM (0.72 SD 0.36) (not


rs 0.29 and 0.24; CLFODS Pearson rs

Death anxiety was higher in patients

Death anxiety was higher in patients


with FD (0.87 SD 0.54) compared to
associated with MUS (ASDA Pearson

statistically significant) and healthy


several measurement tools for hypochondriasis were administered to

0.23 and 0.22, both p < 0.01, for

controls (0.57 SD 0.25), F = 4.9,


with IBS compared to the other
162 general medical outpatients. Patients who met the diagnostic
Death anxiety was positively

groups. F = 3.39, p < 0.05.


Kuwait and US participants
criteria for hypochondriasis (n = 49) scored considerably higher on
the fear of death scale than patients who did not meet these criteria
(n = 113). Age was negatively correlated with both hypochondriasis
and fear of death. Adjusting for age, there was a high correlation
respectively). between scores on the fear of death scale and both hypochondriasis and
Conclusions

p < 0.01.
MUS. A factor analysis of the fear of death scale yielded three
dimensions: fear of dying, loss of meaning, and fear of separation.
These three dimensions were highly correlated with hypochondriasis.

29–67), FD mean 38
Mean 21,2 (Kuwait)

FM mean 45 (range 3.2.1.5. Hiebert 2005 [29]. Thirty-nine adults with hypochondriasis

Controls mean 46
GAD mean 34.4
and 22,4 (USA)

(range 20–60),
completed questionnaires regarding illness and death concerns. The
IBS mean 31.3
DU mean 32.8

(range 23–68)
results revealed that 93% of the respondents was very much afraid to
Age (yrs)

die, 87% was afraid of dying a painful death, 84% often thought about
how short life really is, and 75% was afraid of news that reminded them
of death. Mean scores on the death anxiety measures were compared to
mean scores reported in the literature. Individuals with
USA 75.9

IBS: 100,

GAD 69
Kuwait

DU 40,

hypochondriasis had higher scores on death anxiety than samples


F (%)

75,1

100

with panic disorder, healthy control groups, and human


immunodeficiency virus (HIV)-positive males.
17, Controls
10, GAD 13
Sample size

IBS 14, DU

FM 42, FD
N = 209

N = 191
Kuwait

3.2.1.6. Schütte 2016 [30]. Health behavior, fear and acceptance of


USA

death and dying, and coping with a hypothetical bodily disease were
48

assessed in patients with hypochondriasis, panic disorder, depression,


departments for rheumatology

and healthy controls (all groups n = 30). The hypochondriasis and


department general hospital
or psychiatric hospital with
Patients from an outpatient

Matched healthy controls

panic disorder groups showed more fear and less acceptance of death
Undergraduate students
Setting/Characteristics

and dying than patients with depression and healthy controls. Patients
and gastroenterology
Outpatients clinical

with hypochondriasis ruminated more when confronted with their most


DU, IBS or GAD.

feared disease than when confronted with other diseases.


3.2.1.6.1. Analysis of the association between death anxiety and
hypochondriasis. In total six studies were performed in a
heterogeneous group of 885 primary care patients, patients with
hypochondriasis or other psychiatric disorders and matched healthy
pain, suggestive of spastic

controls. All studies found a positive association between death anxiety


gastroscopy or abdominal
IBS (recurrent abdominal

epigastric complaints, no
FM: ACR criteria for FM,
endoscopic examination)

FD: > three months of

somatic explanation on

and hypochondriasis.
form, no lesions on

3.2.1.6.2. Influence of age and sex on the association death anxiety and
hypochondriasis. Mean age of study participants was between 31.6 and
54.2 years, ranging from 18 till 86 years. The percentage of females
ultrasound.

ranged between 48 and 73%. None of the studies analysed the influence
Irritable bowel syndrome, SSI: Somatic Symptom Inventory (Abdel-Khalek, 2003).
MUS

GBB

of age or sex on the association between death anxiety and


SSI

hypochondriasis.
speech content analysis

speech content analysis


Gleser verbal behavior

Gleser verbal behavior


Studies on death anxiety and medically unexplained symptoms (MUS).

3.2.2. Included studies on the association between death anxiety and MUS
Five-minutes free

Five-minutes free
using Gottschalk-

using Gottschalk-
ASDA, CLFODS

3.2.2.1. Abdel-Khalek 2009 [31]. Two undergraduate samples from


Death anxiety

Kuwait (209 students) and the USA (191 students) filled in a variety
of death anxiety scales. The Kuwaiti sample had significantly higher
scales

scales

scores on all scales than the American sample. Scores on the death
anxiety scales were positively correlated with the Somatic Symptoms
Inventory. Whether the somatic symptoms were medically (un)
Psycho-analytic
Concept Death

aspects in the

explained was not reported.


discussion
Cognitive

approach
anxiety

section

3.2.2.2. Lolas 1986 [32]. This study used the Gottschalk-Gleser verbal

content analysis of free speech to measure anxiety and hostility in 10


patients with duodenal ulcer, 14 patients with irritable bowel syndrome
sectional

sectional

sectional

(IBS) and 13 patients with generalized anxiety disorder. Patients with


Design

Cross-

Cross-

Cross-

IBS expressed significantly more death anxiety than the other two
groups. Patients with IBS were closer to patients with a generalized
Country

Norway
Kuwait

anxiety disorder than to duodenal ulcer patients in terms of intensity


Chile

and pattern of verbal affective expression.


Abdel-Khalek

3.2.2.3. Malt 2003 [33]. Gottschalk-Gleser verbal content analysis of


2009

1986

2003

free speech and the Giessen Subjective Complaints Checklist were used
Author
Table 2

Lolas

Malt

to assess psychological states and somatic symptoms in 42 females with


fibromyalgia, 17 females with functional dyspepsia and 48 population-

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B.M. aan de Stegge et al. Journal of Psychosomatic Research 115 (2018) 58–65

based randomly selected control subjects. Patients with functional MUS studies. Reasons for a low score were mostly not substantiating the
dyspepsia and fibromyalgia scored significantly higher on death sample size of the studied population or absence of a description of non-
anxiety than the control group. In functional dyspepsia patients, response. Only four articles adjusted for age or sex. Almost all studies
death anxiety explained 59% of the variance in gastrointestinal scored the maximum score on the assessment of risk factor (death an-
symptoms, suggesting an etiological association between death xiety) and outcome (hypochondriasis/MUS).
anxiety and somatic symptoms.
3.2.2.3.1. Analysis of the association between death anxiety and 4. Discussion
MUS. In total three studies were performed in a heterogeneous group
of 534 students, patients with MUS or a generalized anxiety disorder, The purpose of this systematic review was to examine the associa-
and healthy controls. All studies found a positive association between tions between death anxiety and hypochondriasis and death anxiety
death anxiety and MUS. and MUS. The results of the included nine studies were robust: all found
3.2.2.3.2. Influence of age and sex on the association between death a positive association of death anxiety with hypochondriasis and/or
anxiety and MUS. Mean age of participants in the three studies was MUS. The methodological quality of all individual studies, however,
between 21.2 and 45.0 years; the oldest participant was 68 years old. was low.
None of the studies analysed the influence of age or sex on the In addition, we aimed to explore whether these associations differed
association between death anxiety and MUS. for age and sex.
In only three studies, patients aged 65 years or above were included
3.2.3. Used death anxiety concepts [27,28,33].
The majority of the included studies for both groups used death In all studies, the proportion of female study participants is higher
anxiety concepts with a cognitive approach, focussing on somatic am- than of male participants. No conclusions could be drawn on the in-
plification, learned illness behavior, and death attributions [26–30,32]. fluence of age and sex on the association between death anxiety and
One study started with a theoretical background with cognitive-, ex- either hypochondriasis or MUS, because none of the studies analysed
istential- and psychoanalytical aspects [18] and another study referred this association as a function of age and sex. Some studies made com-
to underlying psychoanalytical aspects [33]. One study did not mention ments on the influence of age or sex on death anxiety, hypochondriasis,
any death anxiety concept at all [31]. There were no studies based on or MUS separately, but conclusions based on these results would bear
the TMT concept. the risk of bias, because our search was not focused on these associa-
tions separately.
3.3. Measurement instruments used for death anxiety Hypochondriasis and MUS both can occur on a continuum ranging
from a trait in healthy persons to pathological disorders. Definitions of
Three studies used the thanatophobia subscale of the IAS hypochondriasis and MUS have changed over time and possibly will
[26,29,30]. The IAS is devised to asses hypochondriasis and abnormal change in the next future. We therefore decided to include as many
illness behaviour, with the thanatophobia subscale containing only studies as possible on patient as well as non-patient populations. A
three items: “Are you afraid of news which reminds you of death”, limitation of this choice is that this wide range may reduce compar-
“Does the thought of death scares you?” and “Are you afraid that you ability of results.
may die soon?” Three studies used a self-developed questionnaire Although terminology and criteria have changed, none of the in-
[18,27,28]. Two studies used the Gottschalk-Gleser verbal content cluded studies used criteria from the DSM-5. This is important to know
analysis to measure death anxiety [32,33]. The remaining studies all while interpreting our data, because of the impact of the changes from
used a different instrument, see Tables 1 and 2. DSM-IV to DSM-5 on the prevalence of psychiatric diagnoses in this
field. In DSM-5, 75% of former hypochondriasis patients would fit in
3.4. Analysis of study quality the SSD population. We hypothesize that death anxiety may play a
more prominent role in the SSD group from DSM-5 than for example in
The methodological quality of the studies was assessed with an the undifferentiated somatoform disorder group from DSM-IV.
adapted version of the NOS for cohort studies (available on request). Although we tried to be as comprehensive as possible in our lit-
Results of the quality assessment are shown in Tables 3 and 4. None of erature search, we found only nine relevant studies. The quality of the
the articles reached the maximum score of 11 stars. The average score studies, assessed with an adapted NOS-scale, was generally low. In
was six stars for the hypochondriasis studies and seven stars for the addition, the included populations and settings were heterogeneous.

Table 3
Quality appraisal included studies hypochondriasis1.
Study (first Selection Comparability Outcome
author)

Representa- Sample size Non- Selection non- Ascertainment of Based on design and Assessment on Statistical test
tiveness sample respondents exposed sample suspected risk factor analysis outcome

Kellner – – – – ** ** ** *
1987
Barsky * – – * correlational * not applicable * *
1989
James – – – * correlational ** ** * *
2002
Noyes * – – * correlational * * ** –
2002
Hiebert – – – * correlational ** not applicable ** *
2005
Schutte * – – * ** ** ** *
2016

1
Based on an adapted version of the Newcastle-Ottawa Quality Assessment Scale for cohort studies (available on request).

63
B.M. aan de Stegge et al. Journal of Psychosomatic Research 115 (2018) 58–65

Table 4
quality appraisal included studies MUS1.
Study (first Selection Comparability Outcome
author)

Representa- Sample size Non- Selection non- Ascertainment of risk Based on design and Assessment on Statistical test
tiveness sample respondents exposed sample factor analysis outcome

Abdel-Khalek – – – * correlational ** – * *
2009
Lolas * – – * correlational ** – ** *
1986
Malt * * – – ** * ** *
2003

1
Based on an adapted version of the Newcastle-Ottawa Quality Assessment Scale for cohort studies (available on request).

This has prevented integrating and comparing data from the different verbal content analysis [33]. One study referred to cognitive mechan-
studies in a meaningful meta-analysis. On the other hand, the robust isms in the discussion section, but used the Gottschalk-Gleser verbal
positive association between death anxiety and both hypochondriasis content analysis to examine death anxiety, an instrument more suitable
and MUS, even in such heterogeneous populations, could also be seen to measure subconscious mechanisms [32]. The other studies did not
as underscoring the perspective of death anxiety as a transdiagnostic explain their choice for a specific death anxiety measurement instru-
construct. ments.
To the best of our knowledge, we provided the first systematic re- For future research we would recommend to focus on the associa-
view on the association between death anxiety and hypochondriasis tion death anxiety with IAS and/or SSD, considering the changes from
and/or MUS. A previous narrative review focussed on the association of DSM-IV to DSM-5. Statistical techniques that can test conceptual
death anxiety with a wide range of psychiatric disorders, including a models are required, as are designs that move beyond correlations to
paragraph on death anxiety and hypochondriasis and MUS. Results experimental procedures. Studies that are based on clear theoretical
from three out of our nine included studies were included in this pre- perspectives are more likely to have real-world applications than iso-
vious review [18,26,29]. Neither the selection procedure nor a quality lated studies that relate death anxiety to some other construct with little
assessment of the included studies was reported. Similar to our review, justification except that both can be measured [14].
these authors concluded that death anxiety is associated with both For measuring death anxiety we would like to advice using a vali-
hypochondriasis and MUS [10]. The strength of our review is the sys- dated multidimensional measurement instrument, considering the
tematic methodology, inclusion of a wider range of studies on this multifaceted nature of death anxiety. Additional indirect testing of
specific topic and assessment of the study quality. death anxiety with the Gottschalk-Gleser method, LIWC or phenom-
Looking at the death anxiety concepts used in our included studies, enographic approaches can be useful to detect (partly) subconscious
several mechanisms can be considered about how death anxiety might death anxiety. Future research on the reliability of these indirect
contribute to developing or maintaining hypochondriasis and or MUS. measurement instruments and how to interpret them, especially in re-
This ranges from fear of bodily failure with consequently somatic am- lation to self-report questionnaires is needed. More generally, we re-
plification to death anxiety as an expression of separation fear. commend studies to fulfil the quality criteria we used in our appraisal,
Additional information about this relationship may provide a more paying more attention to representativeness, sample size, non-response
complete clinical picture. and appropriate statistical methods.
Because of the cross-sectional study design of all included studies, Our study suggests an association between death anxiety and hy-
and a lack of longitudinal studies, no conclusions about causality can be pochondriasis and/or MUS, but the cross-sectional design prevents
made. Death anxiety may be an underlying risk factor for developing conclusions on causality. Our findings justify further research on death
hypochondriasis and MUS, however, suffering from (medically un- anxiety and its role in the development or maintenance of hypochon-
explained) somatic complaints can also elicit death anxiety. In this driasis and MUS. Considering changes from DSM IV to DSM-5 this could
described mechanism, fear of death is separated from fear of illness. be replaced with IAS and SSD. Additional analysis of modifying effects
Furthermore a third variable could be present, such as anxiety, negative of age and sex in the association between death anxiety and IAS and/or
affect or past or present significant physical illness, which can lead to SSD is recommended.
hypochondriasis and/or MUS and to death anxiety. When we know more about specific subgroups in the hypochon-
In the included studies, a variety of measurement instruments have driasis/MUS populations in which death anxiety might play a con-
been used to assess death anxiety. Most studies used questionnaires, tributing role, further research could develop specific treatments tar-
which consider death anxiety as a unidimensional construct. Some geting this factor.
studies used a multidimensional scale, such as the Death Perspective In conclusion, we have found robust evidence for a positive asso-
Scale (DPS), the Multidimensional Fear of Death Scale (MFODS) and the ciation between death anxiety and hypochondriasis and/or MUS,
Multidimensional Orientation toward Death and Dying Inventory however, the quality of the studies on which this review is based was
(MODDI) [28–30]. Two studies used the Gottschalk-Gleser verbal con- low. More research is needed to gain additional information on this
tent analysis [32,33]. The LIWC method or phenomenographic ap- association, such as the role of age and sex, underlying mechanisms and
proaches were not used in the included studies. causality. This might lead to further research on treatment options for
The validity of the used death anxiety measurement instruments can specific subgroups in hypochondriasis/MUS populations, in which
be questioned. Three studies used self-developed questionnaires death anxiety might play a prominent role.
[18,27,28]. Only two studies combined hypothesized underlying con-
cepts/mechanisms with a specific death anxiety measurement instru-
ment. The study with a cognitive-, existential- and psychoanalytical Funding
background used a self-developed questionnaire (not validated), with
questions about fear of dying, loss of meaning and fear of separation This research did not receive any specific grant from funding
[18]. The study with a psychoanalytic basis used the Gottschalk-Gleser agencies in the public, commercial, or not-for-profit sectors.

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