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Life events and quality of life in female patients with dermatitis artefacta: a comparative study with siblings and controls
Life events and quality of life in female patients with dermatitis artefacta: A comparative study with siblings and controls Yousri El Kissi, Maha Chhoumi, Jaafar Nakhli, Nesrine Kenani, Mohamed Denguezli, Rafiaa Nouira, BechirBen Hadj Ali PII: DOI: Reference:
S0010-440X(14)00171-0 doi: 10.1016/j.comppsych.2014.07.002 YCOMP 51345
To appear in:
Comprehensive Psychiatry
Received date: Revised date: Accepted date:
8 January 2014 1 July 2014 2 July 2014
Please cite this article as: El Kissi Yousri, Chhoumi Maha, Nakhli Jaafar, Kenani Nesrine, Denguezli Mohamed, Nouira Rafiaa, Ali BechirBen Hadj, Life events and quality of life in female patients with dermatitis artefacta: A comparative study with siblings and controls, Comprehensive Psychiatry (2014), doi: 10.1016/j.comppsych.2014.07.002
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ACCEPTED MANUSCRIPT Title: Life events and quality of life in female patients with dermatitis artefacta: A
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comparative study with siblings and controls
Denguezli**, RafiaaNouira**, BechirBen Hadj Ali*.
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Authors: Yousri El Kissi*, Maha Chhoumi*, JaafarNakhli *, NesrineKenani**, Mohamed
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* Department of Psychiatry, FarhatHached hospital, Tunisia
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** Department of Dermatology, FarhatHached hospital, Tunisia
Corresponding author: Yousri El Kissi
Address: FarhatHached Hospital. IbnJazzar Street. Sousse, 4000. Tunisia.
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Phone number: (+216) 73219508 / (+216) 98468626 Fax number: (+216) 73223702
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E-mail:[email protected]
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Manuscript word count: 3316 Table count: 3
Figure count: 0
Running head: Life events and quality of life in dermatitis artefacta
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ACCEPTED MANUSCRIPT Abstract Background: If the severity of Dermatitis Artefacta (DA) is accepted by most authors, few
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published studies have sought to clarify its etiology and impact. It is in this context that this
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work aimed to compare Life Events (LE) and Quality of Life (QoL) scores in patients with DA, in their siblings and in control patients with other chronic dermatological diseases.
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Methods: This is a descriptive and comparative cross-sectional study carried out in the
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dermatology department of FarhatHached hospital in Sousse, Tunisia. Thirty female patients diagnosed with DA according to DSM-IV criteria were retrospectively recruited. For each patient with DA, one of her sisters, the closest in age, was enrolled in the siblings group. The control group consisted of thirty female patients with other chronic dermatological diseases,
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matched with DA patients for age and duration of disease progression. Assessment was based
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on Paykel inventory for LE and on SF-36 for QoL. Results: Compared to both control groups, DA patients reported more LE with more objective
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scoreof QoL.
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negative impact of these events and had a lower score and more often impairedmean global
Conclusions:LE would have a role in the pathogenesis of DA which seems to alter the QoL of patients. These results could help to improve the understanding of DA and incite clinicians to focus not only on the diagnosis and treatment of DA but also on the impact of this disease.
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ACCEPTED MANUSCRIPT Introduction The relation betweenpsychologicalfactors and certain dermatologicaldiseases has been by severalauthors1-4.Factitiousdisorders(FD)ischaracterized
by deliberate
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mentioned
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production or imitation of physical or psychologicalsymptoms in order to adopt the sick rôle2.FD have been a well-knownclinical concept for a longtime1, 5.Even Freud was not
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the first to mention them. However, authors are more or lessunanimous in the opinion
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thatitwasAsher’spublication in 1951 whichinitiatied a more specificinterest in the problem6. Cutaneous Factitious Disorder or Dermatitis Artefacta (DA) is one facet of the spectrum of factitious disease as a whole, which may involve any organ system. Because of lack of stringency in applying diagnostic criteria, it is difficult to determine DA incidence, but its
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feminine predominance is well documented with a sex ratios of 3: l to 20:1. The highest incidence of onset is reported to be in late adolescence to early adult life3, 7.
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Bizarre-appearing lesions may present rather suddenly in regions that are easily accessible to
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the patient and patients are typically unable to describe how the lesions evolved8. Early diagnosis is important since it may prevent unnecessary surgery and chronic morbidity9.It is
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often a source of perplexity and anxiety for dermatologists because they know less about the cause of this self-inflicted condition than the patients themselves3.In fact, the pathogenesis of DA is not clearly understood. Causes include genetic, personal or family history of psychiatric illness and psychosocial factors. It occurs generally in patients with poor coping skills and often represents a maladaptive response to psychological stressor10. Life events (LE) could then have a role in causing DA especially as they are being more and more incriminated in the field of psychosomatic diseases11. In terms of impact,Quality of life (QoL) outcomes are important to DA, because it carries, such as many dermatological diseases, significant psychosocial burdens and morbidity from
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ACCEPTED MANUSCRIPT appearance and symptoms with few cases of mortality12.However, there are to our knowledge no studies that evaluated LE and QoL in DA.
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It is in an attempt to overcome these shortcomings that this study aimed to compare LE and
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QoL scores in patients with DA, in their siblings and in control patients with other chronic
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dermatological diseases. Materials and Methods
2.1. Study population
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2.1.1. The group of patients with DA
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It is a descriptive and comparative cross-sectional study.
The identification of patients with DA was made from records of inpatient and outpatient
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units of dermatology department of FarhatHached Hospital in Sousse over 20 years (from
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January 1986 to December 2006). Medical records were scanned for an explicit diagnosis of DA made by the attending physician. The selected medical records have been then re-
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examined to confirm diagnosis according to the DSM-IV-TR criteria: intentional production or feigning of physical or psychological signs or symptoms (criterion A), in order to assume the sick role (criterion B), with no external incentives -such as economic gain, avoiding legal responsibility, or improving physical well-being, as in Malingering- (criterion C)13. Forty cases of DA have been then identified.Inclusion criteria were: female sex, age greater than or equal to 18 years and minimum of six months of disease duration. Exclusion criteria were: lack of consent to participate in the study, the absence of a sister aged greater than or equal to 18 years and severe intellectual or mental disorder affecting communication. Using these criteria, 35 patients were selected.
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ACCEPTED MANUSCRIPT All selected patients were contacted by mail informing them about the objectives of the study and inviting them to participate within a period of one month. A mail reminder has been sent
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to patients who have not submitted within the specified time. Then, patients who did not
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respond were contacted by phone. After this procedure, 30 patients were recruited.
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2.1.2. The siblings group
One of the sisters of each recruited patient was concomitantly invited to participate in the
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study through the same mail or by phone. As far as possible, the invited sister was the closest in age in order to match the two groups in terms of age. Thirty sisters were then included. 2.1.3. The control group
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Thirty female patients with other chronic dermatological diseases, matched with DA Patients for age and disease duration, were recruited from the same dermatology department. Diseases
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considered were: systemic lupus erythematosus, chronic lupus, vitiligo, acne, lichen,
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pemphigus, chronic eczema, chronic urticaria, scleroderma, Behcet's disease and psoriasis.
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2.2. Variables examined:
2.2.1. Sociodemographic characteristics: age, place of residence, educational level, professional activity and marital status. 2.2.2. Personal and family history: psychiatric disorder, somatic disease, prolonged hospitalization (longer than two weeks) or sustained medical care during childhood (over two years), medical or paramedical profession in the family and personal history of suicide attempt. 2.2.3. Characteristics of lesions: duration of evolution before diagnosis; number of consulted care centers; complications; type of immediate evolution according to the three modalities
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ACCEPTED MANUSCRIPT described by Saez de Ocariz14:no improvement (regression between 0 and 24 % of the initial lesions), improvement (regression between 25 and 89 % of the initial lesions), recovery
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(greater than or equal to 90% of the initial lesions); total number of relapses and number of
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relapses per year.
2.2.4. Life events: they were identified using Paykel inventory which is a scale consisting of
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63 specific types of events divided into 10 categories15. It is intended to cover the 6 months
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prior to the date of onset of symptoms. For each event identified, the objective negative impact was rated.
2.2.5. Quality of Life: It was assessed using theQoL measuring instrument SF-3616.This instrument consists of 36 questions grouped into eight dimensions. The answers are scored
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from 0 to 100. The Mean Global Score (MGS) is obtained by calculating the average of all
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answers. The Lean threshold value admitting that a MGS less than 66.7 means impaired QoL
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was chosen17. 2.3. Data collection
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All the variables were collected in the group of DA patients. For the control group, the lesion characteristics concerned the different dermatological diseases. For the siblings group, data collection concerned sociodemographic characteristics, personal and family history and standardized assessment of LE and QoL. 2.4. Statistical analysis Statistical analysis was performed using SPSS 17.0 software. Comparisons were made between the group of DA patients and the control group on one side and between the group of DA patients and the siblings group on the other side. Pearson’s chi-squared test was used to
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ACCEPTED MANUSCRIPT compare frequencies and Student’s t-test was used for quantitative variables. The Fisher’s exact test was used for the low numbers. The significance level considered was 0.05.
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Results 3.1. DA patients group characteristics
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The average age of patients was 30.23 ± 8.91 years, ranging from 18 to 50 years. Sixty percent of patients resided in urban areas, 40 % had completed primary education, 80% had
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no work and 93.33 % were single. Thirty percent of patients had personal psychiatric history, 60 % had a family history of somatic disease and 36.66 % had prolonged hospitalization or sustained medical care during childhood. There were medical or paramedical professions in
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the family in 16.66 % of cases. The duration of evolution of skin lesions before diagnosis was 34.23 months on average. The mean number of consulted care centers was 2.63 ± 1.9. Among
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the patients, 15 patients (50 %) have a pruritic skin lesions, three (10 %) have sensation of
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burning and 10 (30 %) have polymorphic lesions with variable age. There was no improvement in 60 % of cases, improvement in 33.33% of cases and recovery
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in 6.66% of cases. Complications occurred in 83.33 % of patients (ulceration in 24% of cases, secondary infection in 64 % of cases and necrosis in 12% of cases). Relapses were observed in 83.33 % of patients with averages of 3.17 ± 2.21 and 1.61 ± 1.33 relapses per year. The mean number of LE was 2.33 ± 0.92 and of those with an objective negative impact was 1.57 ± 0.72. The MGS of QoL was 1285 ± 544.71 and was altered in all patients. 3.2. Comparative data: 3.2.1. Sociodemographic characteristics and personal and family history: Table 1 In comparison to both control and siblings groups, patients with DA were less professionally active, more often single and had more personal history of psychiatric disorders, suicide
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ACCEPTED MANUSCRIPT attempts and prolonged hospitalization or medical follow during childhood. Compared to the control group, patients with DA were less educated and had more family history of prolonged
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patients with DA had more personal history of somatic diseases.
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hospitalization or medical follow during childhood. Compared with the siblings group, the
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3.2.2. Characteristics of lesions: Table 2
Patients with DA consulted more care centers than patients in the control group, had more
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complications, more relapses in general and more relapses per year. 3.2.3. LE and QoL: Table 3
Compared to both control groups, patients with DA reported more LE with more objective
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negative impact of these events and had a lower and more often altered MGS of QoL.
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DISCUSSION
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DA is a factitious disorder in which patients create cutaneous lesions in order to satisfy an internal psychologicalneed1, 2. Despite correct treatment, patients with factitious lesions
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have the tendency to become chronic and recurrent patients. However, thereis no universal solution for diagnosing and treating factitious lésions 18. The management of DA patient proved very challenging and treatment was ineffective. An integrated intervention involving appropriate medical specialties, psychiatrists, social workers, social welfare assistance and family support would have been required to provide more effective help to the patient19. Several studies have focused on describing the general characteristics of patients based mostly on clinical observations20, 21.However,few published studies have sought to clarify its etiology and impact on QoL. It is in this context that this work aimed to compare LE and QoL scores in femalpatients with DA, in their siblings and in control patients with other chronic dermatological diseases.
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ACCEPTED MANUSCRIPT The use of Paykel’s inventory to assess LE was motivated by its wide distribution in international studies. Although originally designed for mood disorders22, it was also used in
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several fields of somatic medicine23, 24. It also allows identifying a wide range of events
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specifying the objective negative impact of these events.
To measure QoL, SF-36 allows comparing patients with control groupsand takes into account
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social and self-image aspects in addition to functional aspects which makes it more
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appropriate to the specific problems of chronic skin diseases25. The use of comparison groups aimed to identify the specific characteristics of DA. In fact, many skin conditions are likely to be dependent on the role of LE and to induce an alteration in the QoL11. Only the comparison with a control group with other chronic skin diseases
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can therefore identify the particularities of DA. Similarly, only the comparison with siblings
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allows distinguishing among patients’ characteristics between what is inherent to the illness
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and what could correspond to the family environment. It is generally accepted that in many skin diseases, considered as psychosomatic, several
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psychosocial factors may be involved in the onset or progression of lesions26. These factors are often related to stressful events and involved in a vicious circle with the QoL27. DA for instance is characterized by its dependence of several LE11, 28, 29but no study, to our knowledge, made a standardized evaluation of LE in this disease. The results of the present study indicate that patients with DA had more LE with more objective negative impact of these events compared to both control groups. The higher number of LE in patients with DA than in those with other skin diseases would indicate a greater involvement of LE in DA. On the other hand, although having the same family
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ACCEPTED MANUSCRIPT environment and a comparable lifestyle, patients with DA would be more sensitive to these events than their sisters.
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Someauthors2, 30are of the opinion that patients sufferingfromfactitiousillness have a
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personalhistory in which one findsemotionaldeprivation in earlychildhood and rejection by their parents. This results in important disturbances as regards the body-image, narcissism, theforming
of
relationships.
In
adulthood,
thereis
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and
a
strongtendency
to
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formsymbioticrelationships, accompanied by highly ambivalent feelings. In the event of asubjective or objective threat of abandonmentthese patients willreactwith intenseanxiety and aggression.Mayo and Haggerty30, suggestthatbecause of insufficient separation these aggressive feelings are warded off by means ofprojective identification. It is in this context
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that factitious disorders occur, with thepatient at the same time exacting care, in some cases from a third party. Thepseudologies, which are presumed mostly to have a metaphorical
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significance, alsoserve to keep the person providing the care at a distance. The following
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testablehypothesis can be formulated from this construct: episodes of factitious illness occur
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mainly in situations which are experienced by the patient as abandonment2,5. Indeed, The life event would incite the patient to cause injuries to himself in order to catch the attention of others. Unlike DA, the role of LE in the onset of other skin diseases was investigated by several authors. A recent review focused on the relationship between psoriasis and stress, especially relating to psychosocial, psychological and emotional stress aspects31 .The stressful events would be mainly incriminated in recurrences and extensions of lesions32,33It has also been found that stressful LE can worsen or trigger off a pemphigus34. Concerning QoL, the MGS in patients with DAwas 1285.50 ± 544.71 and was impaired in all cases, which indicates how serious this disease is. Indeed, like most chronic noticeable skin disorders, DA is a disease that is expected to have a significant impact
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ACCEPTED MANUSCRIPT on QoL11. This impact is explained by several factors such as the severity of lesions and evolution by spurts often requiring hospitalization and expensive treatments35. However, to
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our knowledge, no studies have measured the QoL in patients with DA. On the contrary,
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several studies related to other skin diseases are available. In patients with pemphigus34,36and psoriasis37, scores of all SF-36 dimensions were found altered in
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comparison to the general population. In a Malaysian study, patients with psoriasis had a significantly lower QoL score than healthy subjects and comparisons with data for patients
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with other chronic medical conditions demonstrated that psoriasis has a negative effect on health-related QoL similar to the impact of other chronic conditions 38. In another study on psoriasis, patients’ QoL, as measured by the Psoriasis Disability Index,
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was altered, and alteration was more related to depression and anxiety than the severity of the
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disease39.
A study using the Arabic version for Morocco of the Dermatology Life Quality Index found
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that factors associated with poorer QoL in psoriasis were severity, older age and severe
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clinical forms40.Mallon et al. found altered SF-36 dimensions of social functioning and mental health vitality in patients with acne, compared to general population 41. Vitiligo seems to have a less pronounced impact with a moderate impairment of QoL 42,43 but this impairment would be critical in the psychosocial development of children44. In this study, patients with DA had lower and more often altered scores of QoL than patients with other dermatological diseases. This could be explained by the heterogeneity of the control group with skin conditions altering patients’ QoLto more or less important degrees.Anyway, compared to the various dermatological diseases we chose, the impact of DAon QoLseems important.
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ACCEPTED MANUSCRIPT Patients with DA had also a lower and more often altered MGS of QoL than their sisters. These results highlight the impact of DA on the QoL of patients by removing potential
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interference with the home environment and living conditions.
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A limitation of this study is the small sample size, which limits statistical power, but with a very low reported incidence of DA, it has been difficult to recruit a larger number of
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patients.For the same reason, patients were recruited from many years ago. Another limitation
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of the present study is that thecurrent dermatological conditions of patients are not known. Future studies including a control group consisting of healthy subjects, in addition to the control group of patients with other dermatological diseases, would also be beneficial.
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Conclusion
The results of the present study indicate that patients with DA reported more LE with more
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objective negative impact of these events and had a lower and more often altered MGS of QoL.LE would then have a role in the pathogenesis of DA which seems to alter the QoL of
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patients. These results could help to improve the understanding of DA and incite clinicians to
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focus not only on the diagnosis and treatment of DA but also on its impact. References:
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34) Morell-Dubois S, Carpentier O, Cottencin O, Cottencin O, Queyrel V, Hachulla E et al. Stressful life events and pemphigus. Dermatology 2008; 216: 104-108. 35) Anwar W, Murphy N, Powell FC. Learning the cost of dermatitis artefacta. Clin Exp Dermatol 2004; 29: 576-578. 36) Terrab Z, Benchikhi H, Maaroufi A, Hassoune S, Amine M, Lakhdar H. Quality of life and pemphigus. Ann Dermatol Venereol 2005; 132: 321-328. 37) Wahl A, Loge JH, Wiklund I, Hanestad BR. The burden of psoriasis: a study concerning health-related quality of life among Norwegian adult patients with psoriasis compared with general population norms. J Am Acad Dermatol 2000; 43: 803-808.
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Table Legends : *Table 1: Socio-demographic characteristics and medical history of Patients with DA, controls and siblings group p1: Statistical signification between Patients with DA and controls, p2: Statistical signification between Patients with DA and siblings *Table 2: Characteristics of lesions in Patients with DA and in controls
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ACCEPTED MANUSCRIPT p1: Statistical signification between Patients with DA and controls, p2: Statistical signification between Patients with DA and siblings
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*Table 3: Life events and quality of life scores in Patients with DA, controls and siblings p1: Statistical signification between Patients with DA and controls, p2: Statistical
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signification between Patients with DA and siblings
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ACCEPTED MANUSCRIPT Table 1: Socio-demographic characteristics and medical history of Patients with DA, controls and siblings group Controls, n
DA, n (%)
(%)
Siblings, n
p
(%)
Illiterate
11 (36.66)
level
Primary
12 (40)
Secondary
7 (23.33)
14 (46.66)
7 (23.33)
Academic
0 (0)
5 (16.66)
1 (3.33)
24 (80)
16 (53.33)
8 (26.66)
p1 = 0.002
6 (20)
14 (46.66)
22 (73.33)
p2 < 0.001
28 (93.33)
11 (36.33)
10 (33.33)
p1 < 0.001
2 (6.66)
19 (63.33)
20 (66.66)
p2 < 0.001
18 (60)
18 (60)
17 (56.66)
p1= NS
12 (40)
12 (40)
13 (43.33)
p2= NS
Psychiatric disorder
9 (30)
1 (3.33)
1 (3.33)
p1 = 0.006
Present
Marital
Single
Status
Married
Residence
Urban
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activity
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Absent
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Professional
Personal
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Rural
2 (6.66)
11 (36.66)
p1 < 0.001
9 (30)
11 (36.66)
p2 = NS
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Educational
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Patients with
history: n (%)
Suicide attempt
p2 = 0.006 9 (30)
0 (0)
0 (0)
p1 = 0.001 p2 = 0.001
Somatic disease
10 (33.33)
8 (26.66)
0 (0)
p1 = NS p2 < 0.001
Prolonged
8 (26.66)
1 (3.33)
1 (3.33)
p1 = 0.013
hospitalization or
p2 = 0.013
medical follow
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ACCEPTED MANUSCRIPT during childhood Family
Psychiatric disorder
3 (10)
0 (0)
3 (10)
p1 = NS
Somatic disease
18 (60)
11 (36.66)
care during childhood
paramedical
p2= NS 11 (36.66)
p1 = 0.001
5 (16.66)
6 (20)
p2= NS
5 (16.66)
p1 = NS p2= NS
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profession
p1 = NS
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Medical or
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hospitalization or sustained medical
1 (3.33)
18 (60)
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Prolonged
18 (60)
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(%)
p2= NS
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history: n
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p1: Statistical signification between Patients with DA and controls
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p2: Statistical signification between Patients with DA and siblings
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ACCEPTED MANUSCRIPT Table 2: Characteristics of lesions in Patients with DA and in controls
Control
DA
Group
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Patients with
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Item
Duration of evolution before diagnosis
34.23 ± 35.5
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(months) Number of consulted care centers No
p1 = NS
26.02 1.2 ± 1.18
p1 <0.001
18 (60)
12 (40)
Improvement
10 (33.33)
12 (40)
Recovery
2 (6.66)
6 (20)
Yes
25 (83.33)
6 (20)
No
5 (16.66)
24 (80)
3.17 ± 2.21
1.97 ± 2.18
p1 <0.001
1.61 ± 1.33
0.74 ± 0.78
p1 = 0.03
improvement
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Evolution, n (%)
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2.63 ± 1.9
35.2 ±
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Number of relapses
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Complications, n (%)
p1 = NS
p1 <0.001
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Number of relapses/year
p1: Statistical signification between Patients with DA and controls p2: Statistical signification between Patients with DA and siblings
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ACCEPTED MANUSCRIPT Table 3: Life events and quality of life scores in Patients with DA, controls and siblings
Mean global
events
score
with DA
patients
2.33 ± 0.92
0.70 ±
With
1.57 ± 0.72
0.3 ± 0.46
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objective
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1.08
negative impact
Siblings
P
T
Control
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Life
Patients
0.77 ±
p1 <0.001
1.19
p2 <0.001
0.3 ±
p1 <0.001
0.18
p2 <0.001
Mean global
1285.50 ±
2227.67 ±
2546 ±
p1 <0.001
of life
score
544.71
626.62
401.72
p2 <0.001
30 (100)
17 (56.66)
12 (40)
p1 <0.001
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Altered mean
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Quality
global score, n
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(%)
p2 <0.001
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p1: Statistical signification between Patients with DA and controls p2: Statistical signification between Patients with DA and siblings
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ACCEPTED MANUSCRIPT Highlight Compared to both control groups, patients with DA reported more Life Events with more
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objective negative impact of these events and had a lower and more often altered Mean
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Global Score of Quality of life. Life Events would then have a role in the pathogenesis of DA which seems to alter the Quality of Life of patients. These results could help to improve the
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PT
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DA but also on the impact of this disease.
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understanding of DA and incite clinicians to focus not only on the diagnosis and treatment of
22
S0010-440X(14)00171-0 doi: 10.1016/j.comppsych.2014.07.002 YCOMP 51345
To appear in:
Comprehensive Psychiatry
Received date: Revised date: Accepted date:
8 January 2014 1 July 2014 2 July 2014
Please cite this article as: El Kissi Yousri, Chhoumi Maha, Nakhli Jaafar, Kenani Nesrine, Denguezli Mohamed, Nouira Rafiaa, Ali BechirBen Hadj, Life events and quality of life in female patients with dermatitis artefacta: A comparative study with siblings and controls, Comprehensive Psychiatry (2014), doi: 10.1016/j.comppsych.2014.07.002
This is a PDF file of an unedited manuscript that has been accepted for publication. As a service to our customers we are providing this early version of the manuscript. The manuscript will undergo copyediting, typesetting, and review of the resulting proof before it is published in its final form. Please note that during the production process errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain.
ACCEPTED MANUSCRIPT Title: Life events and quality of life in female patients with dermatitis artefacta: A
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comparative study with siblings and controls
Denguezli**, RafiaaNouira**, BechirBen Hadj Ali*.
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Authors: Yousri El Kissi*, Maha Chhoumi*, JaafarNakhli *, NesrineKenani**, Mohamed
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* Department of Psychiatry, FarhatHached hospital, Tunisia
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** Department of Dermatology, FarhatHached hospital, Tunisia
Corresponding author: Yousri El Kissi
Address: FarhatHached Hospital. IbnJazzar Street. Sousse, 4000. Tunisia.
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Phone number: (+216) 73219508 / (+216) 98468626 Fax number: (+216) 73223702
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E-mail:[email protected]
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Manuscript word count: 3316 Table count: 3
Figure count: 0
Running head: Life events and quality of life in dermatitis artefacta
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ACCEPTED MANUSCRIPT Abstract Background: If the severity of Dermatitis Artefacta (DA) is accepted by most authors, few
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published studies have sought to clarify its etiology and impact. It is in this context that this
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work aimed to compare Life Events (LE) and Quality of Life (QoL) scores in patients with DA, in their siblings and in control patients with other chronic dermatological diseases.
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Methods: This is a descriptive and comparative cross-sectional study carried out in the
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dermatology department of FarhatHached hospital in Sousse, Tunisia. Thirty female patients diagnosed with DA according to DSM-IV criteria were retrospectively recruited. For each patient with DA, one of her sisters, the closest in age, was enrolled in the siblings group. The control group consisted of thirty female patients with other chronic dermatological diseases,
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matched with DA patients for age and duration of disease progression. Assessment was based
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on Paykel inventory for LE and on SF-36 for QoL. Results: Compared to both control groups, DA patients reported more LE with more objective
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scoreof QoL.
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negative impact of these events and had a lower score and more often impairedmean global
Conclusions:LE would have a role in the pathogenesis of DA which seems to alter the QoL of patients. These results could help to improve the understanding of DA and incite clinicians to focus not only on the diagnosis and treatment of DA but also on the impact of this disease.
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ACCEPTED MANUSCRIPT Introduction The relation betweenpsychologicalfactors and certain dermatologicaldiseases has been by severalauthors1-4.Factitiousdisorders(FD)ischaracterized
by deliberate
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mentioned
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production or imitation of physical or psychologicalsymptoms in order to adopt the sick rôle2.FD have been a well-knownclinical concept for a longtime1, 5.Even Freud was not
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the first to mention them. However, authors are more or lessunanimous in the opinion
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thatitwasAsher’spublication in 1951 whichinitiatied a more specificinterest in the problem6. Cutaneous Factitious Disorder or Dermatitis Artefacta (DA) is one facet of the spectrum of factitious disease as a whole, which may involve any organ system. Because of lack of stringency in applying diagnostic criteria, it is difficult to determine DA incidence, but its
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feminine predominance is well documented with a sex ratios of 3: l to 20:1. The highest incidence of onset is reported to be in late adolescence to early adult life3, 7.
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Bizarre-appearing lesions may present rather suddenly in regions that are easily accessible to
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the patient and patients are typically unable to describe how the lesions evolved8. Early diagnosis is important since it may prevent unnecessary surgery and chronic morbidity9.It is
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often a source of perplexity and anxiety for dermatologists because they know less about the cause of this self-inflicted condition than the patients themselves3.In fact, the pathogenesis of DA is not clearly understood. Causes include genetic, personal or family history of psychiatric illness and psychosocial factors. It occurs generally in patients with poor coping skills and often represents a maladaptive response to psychological stressor10. Life events (LE) could then have a role in causing DA especially as they are being more and more incriminated in the field of psychosomatic diseases11. In terms of impact,Quality of life (QoL) outcomes are important to DA, because it carries, such as many dermatological diseases, significant psychosocial burdens and morbidity from
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ACCEPTED MANUSCRIPT appearance and symptoms with few cases of mortality12.However, there are to our knowledge no studies that evaluated LE and QoL in DA.
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It is in an attempt to overcome these shortcomings that this study aimed to compare LE and
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QoL scores in patients with DA, in their siblings and in control patients with other chronic
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dermatological diseases. Materials and Methods
2.1. Study population
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2.1.1. The group of patients with DA
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It is a descriptive and comparative cross-sectional study.
The identification of patients with DA was made from records of inpatient and outpatient
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units of dermatology department of FarhatHached Hospital in Sousse over 20 years (from
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January 1986 to December 2006). Medical records were scanned for an explicit diagnosis of DA made by the attending physician. The selected medical records have been then re-
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examined to confirm diagnosis according to the DSM-IV-TR criteria: intentional production or feigning of physical or psychological signs or symptoms (criterion A), in order to assume the sick role (criterion B), with no external incentives -such as economic gain, avoiding legal responsibility, or improving physical well-being, as in Malingering- (criterion C)13. Forty cases of DA have been then identified.Inclusion criteria were: female sex, age greater than or equal to 18 years and minimum of six months of disease duration. Exclusion criteria were: lack of consent to participate in the study, the absence of a sister aged greater than or equal to 18 years and severe intellectual or mental disorder affecting communication. Using these criteria, 35 patients were selected.
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ACCEPTED MANUSCRIPT All selected patients were contacted by mail informing them about the objectives of the study and inviting them to participate within a period of one month. A mail reminder has been sent
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to patients who have not submitted within the specified time. Then, patients who did not
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respond were contacted by phone. After this procedure, 30 patients were recruited.
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2.1.2. The siblings group
One of the sisters of each recruited patient was concomitantly invited to participate in the
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study through the same mail or by phone. As far as possible, the invited sister was the closest in age in order to match the two groups in terms of age. Thirty sisters were then included. 2.1.3. The control group
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Thirty female patients with other chronic dermatological diseases, matched with DA Patients for age and disease duration, were recruited from the same dermatology department. Diseases
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considered were: systemic lupus erythematosus, chronic lupus, vitiligo, acne, lichen,
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pemphigus, chronic eczema, chronic urticaria, scleroderma, Behcet's disease and psoriasis.
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2.2. Variables examined:
2.2.1. Sociodemographic characteristics: age, place of residence, educational level, professional activity and marital status. 2.2.2. Personal and family history: psychiatric disorder, somatic disease, prolonged hospitalization (longer than two weeks) or sustained medical care during childhood (over two years), medical or paramedical profession in the family and personal history of suicide attempt. 2.2.3. Characteristics of lesions: duration of evolution before diagnosis; number of consulted care centers; complications; type of immediate evolution according to the three modalities
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ACCEPTED MANUSCRIPT described by Saez de Ocariz14:no improvement (regression between 0 and 24 % of the initial lesions), improvement (regression between 25 and 89 % of the initial lesions), recovery
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(greater than or equal to 90% of the initial lesions); total number of relapses and number of
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relapses per year.
2.2.4. Life events: they were identified using Paykel inventory which is a scale consisting of
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63 specific types of events divided into 10 categories15. It is intended to cover the 6 months
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prior to the date of onset of symptoms. For each event identified, the objective negative impact was rated.
2.2.5. Quality of Life: It was assessed using theQoL measuring instrument SF-3616.This instrument consists of 36 questions grouped into eight dimensions. The answers are scored
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from 0 to 100. The Mean Global Score (MGS) is obtained by calculating the average of all
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answers. The Lean threshold value admitting that a MGS less than 66.7 means impaired QoL
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was chosen17. 2.3. Data collection
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All the variables were collected in the group of DA patients. For the control group, the lesion characteristics concerned the different dermatological diseases. For the siblings group, data collection concerned sociodemographic characteristics, personal and family history and standardized assessment of LE and QoL. 2.4. Statistical analysis Statistical analysis was performed using SPSS 17.0 software. Comparisons were made between the group of DA patients and the control group on one side and between the group of DA patients and the siblings group on the other side. Pearson’s chi-squared test was used to
6
ACCEPTED MANUSCRIPT compare frequencies and Student’s t-test was used for quantitative variables. The Fisher’s exact test was used for the low numbers. The significance level considered was 0.05.
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Results 3.1. DA patients group characteristics
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The average age of patients was 30.23 ± 8.91 years, ranging from 18 to 50 years. Sixty percent of patients resided in urban areas, 40 % had completed primary education, 80% had
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no work and 93.33 % were single. Thirty percent of patients had personal psychiatric history, 60 % had a family history of somatic disease and 36.66 % had prolonged hospitalization or sustained medical care during childhood. There were medical or paramedical professions in
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the family in 16.66 % of cases. The duration of evolution of skin lesions before diagnosis was 34.23 months on average. The mean number of consulted care centers was 2.63 ± 1.9. Among
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the patients, 15 patients (50 %) have a pruritic skin lesions, three (10 %) have sensation of
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burning and 10 (30 %) have polymorphic lesions with variable age. There was no improvement in 60 % of cases, improvement in 33.33% of cases and recovery
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in 6.66% of cases. Complications occurred in 83.33 % of patients (ulceration in 24% of cases, secondary infection in 64 % of cases and necrosis in 12% of cases). Relapses were observed in 83.33 % of patients with averages of 3.17 ± 2.21 and 1.61 ± 1.33 relapses per year. The mean number of LE was 2.33 ± 0.92 and of those with an objective negative impact was 1.57 ± 0.72. The MGS of QoL was 1285 ± 544.71 and was altered in all patients. 3.2. Comparative data: 3.2.1. Sociodemographic characteristics and personal and family history: Table 1 In comparison to both control and siblings groups, patients with DA were less professionally active, more often single and had more personal history of psychiatric disorders, suicide
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ACCEPTED MANUSCRIPT attempts and prolonged hospitalization or medical follow during childhood. Compared to the control group, patients with DA were less educated and had more family history of prolonged
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patients with DA had more personal history of somatic diseases.
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hospitalization or medical follow during childhood. Compared with the siblings group, the
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3.2.2. Characteristics of lesions: Table 2
Patients with DA consulted more care centers than patients in the control group, had more
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complications, more relapses in general and more relapses per year. 3.2.3. LE and QoL: Table 3
Compared to both control groups, patients with DA reported more LE with more objective
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negative impact of these events and had a lower and more often altered MGS of QoL.
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DISCUSSION
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DA is a factitious disorder in which patients create cutaneous lesions in order to satisfy an internal psychologicalneed1, 2. Despite correct treatment, patients with factitious lesions
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have the tendency to become chronic and recurrent patients. However, thereis no universal solution for diagnosing and treating factitious lésions 18. The management of DA patient proved very challenging and treatment was ineffective. An integrated intervention involving appropriate medical specialties, psychiatrists, social workers, social welfare assistance and family support would have been required to provide more effective help to the patient19. Several studies have focused on describing the general characteristics of patients based mostly on clinical observations20, 21.However,few published studies have sought to clarify its etiology and impact on QoL. It is in this context that this work aimed to compare LE and QoL scores in femalpatients with DA, in their siblings and in control patients with other chronic dermatological diseases.
8
ACCEPTED MANUSCRIPT The use of Paykel’s inventory to assess LE was motivated by its wide distribution in international studies. Although originally designed for mood disorders22, it was also used in
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several fields of somatic medicine23, 24. It also allows identifying a wide range of events
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specifying the objective negative impact of these events.
To measure QoL, SF-36 allows comparing patients with control groupsand takes into account
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social and self-image aspects in addition to functional aspects which makes it more
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appropriate to the specific problems of chronic skin diseases25. The use of comparison groups aimed to identify the specific characteristics of DA. In fact, many skin conditions are likely to be dependent on the role of LE and to induce an alteration in the QoL11. Only the comparison with a control group with other chronic skin diseases
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can therefore identify the particularities of DA. Similarly, only the comparison with siblings
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allows distinguishing among patients’ characteristics between what is inherent to the illness
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and what could correspond to the family environment. It is generally accepted that in many skin diseases, considered as psychosomatic, several
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psychosocial factors may be involved in the onset or progression of lesions26. These factors are often related to stressful events and involved in a vicious circle with the QoL27. DA for instance is characterized by its dependence of several LE11, 28, 29but no study, to our knowledge, made a standardized evaluation of LE in this disease. The results of the present study indicate that patients with DA had more LE with more objective negative impact of these events compared to both control groups. The higher number of LE in patients with DA than in those with other skin diseases would indicate a greater involvement of LE in DA. On the other hand, although having the same family
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ACCEPTED MANUSCRIPT environment and a comparable lifestyle, patients with DA would be more sensitive to these events than their sisters.
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Someauthors2, 30are of the opinion that patients sufferingfromfactitiousillness have a
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personalhistory in which one findsemotionaldeprivation in earlychildhood and rejection by their parents. This results in important disturbances as regards the body-image, narcissism, theforming
of
relationships.
In
adulthood,
thereis
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and
a
strongtendency
to
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formsymbioticrelationships, accompanied by highly ambivalent feelings. In the event of asubjective or objective threat of abandonmentthese patients willreactwith intenseanxiety and aggression.Mayo and Haggerty30, suggestthatbecause of insufficient separation these aggressive feelings are warded off by means ofprojective identification. It is in this context
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that factitious disorders occur, with thepatient at the same time exacting care, in some cases from a third party. Thepseudologies, which are presumed mostly to have a metaphorical
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significance, alsoserve to keep the person providing the care at a distance. The following
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testablehypothesis can be formulated from this construct: episodes of factitious illness occur
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mainly in situations which are experienced by the patient as abandonment2,5. Indeed, The life event would incite the patient to cause injuries to himself in order to catch the attention of others. Unlike DA, the role of LE in the onset of other skin diseases was investigated by several authors. A recent review focused on the relationship between psoriasis and stress, especially relating to psychosocial, psychological and emotional stress aspects31 .The stressful events would be mainly incriminated in recurrences and extensions of lesions32,33It has also been found that stressful LE can worsen or trigger off a pemphigus34. Concerning QoL, the MGS in patients with DAwas 1285.50 ± 544.71 and was impaired in all cases, which indicates how serious this disease is. Indeed, like most chronic noticeable skin disorders, DA is a disease that is expected to have a significant impact
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ACCEPTED MANUSCRIPT on QoL11. This impact is explained by several factors such as the severity of lesions and evolution by spurts often requiring hospitalization and expensive treatments35. However, to
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our knowledge, no studies have measured the QoL in patients with DA. On the contrary,
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several studies related to other skin diseases are available. In patients with pemphigus34,36and psoriasis37, scores of all SF-36 dimensions were found altered in
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comparison to the general population. In a Malaysian study, patients with psoriasis had a significantly lower QoL score than healthy subjects and comparisons with data for patients
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with other chronic medical conditions demonstrated that psoriasis has a negative effect on health-related QoL similar to the impact of other chronic conditions 38. In another study on psoriasis, patients’ QoL, as measured by the Psoriasis Disability Index,
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was altered, and alteration was more related to depression and anxiety than the severity of the
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disease39.
A study using the Arabic version for Morocco of the Dermatology Life Quality Index found
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that factors associated with poorer QoL in psoriasis were severity, older age and severe
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clinical forms40.Mallon et al. found altered SF-36 dimensions of social functioning and mental health vitality in patients with acne, compared to general population 41. Vitiligo seems to have a less pronounced impact with a moderate impairment of QoL 42,43 but this impairment would be critical in the psychosocial development of children44. In this study, patients with DA had lower and more often altered scores of QoL than patients with other dermatological diseases. This could be explained by the heterogeneity of the control group with skin conditions altering patients’ QoLto more or less important degrees.Anyway, compared to the various dermatological diseases we chose, the impact of DAon QoLseems important.
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ACCEPTED MANUSCRIPT Patients with DA had also a lower and more often altered MGS of QoL than their sisters. These results highlight the impact of DA on the QoL of patients by removing potential
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interference with the home environment and living conditions.
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A limitation of this study is the small sample size, which limits statistical power, but with a very low reported incidence of DA, it has been difficult to recruit a larger number of
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patients.For the same reason, patients were recruited from many years ago. Another limitation
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of the present study is that thecurrent dermatological conditions of patients are not known. Future studies including a control group consisting of healthy subjects, in addition to the control group of patients with other dermatological diseases, would also be beneficial.
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Conclusion
The results of the present study indicate that patients with DA reported more LE with more
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objective negative impact of these events and had a lower and more often altered MGS of QoL.LE would then have a role in the pathogenesis of DA which seems to alter the QoL of
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patients. These results could help to improve the understanding of DA and incite clinicians to
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focus not only on the diagnosis and treatment of DA but also on its impact. References:
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41) Mallon E, Newton JN, Klassen A, Stewart-Brown SL, Ryan TJ, Finlay AY. The quality of life in acne: a comparison with general medical conditions using generic questionnaires. Br J Dermatol 1999; 140: 672-676.
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43) Kiprono S, Chaula B, Makwaya C, Naafs B, Masenga J. Quality of life of patients with vitiligo attending the Regional Dermatology Training Center in Northern Tanzania. Int J
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44) Dertlioglu SB, Cicek D, Balci DD, Halisdemir N. Dermatology life quality index scores in children with vitiligo: comparison with atopic dermatitis and healthy control subjects. Int J Dermatol 2013; 52: 96-101.
Table Legends : *Table 1: Socio-demographic characteristics and medical history of Patients with DA, controls and siblings group p1: Statistical signification between Patients with DA and controls, p2: Statistical signification between Patients with DA and siblings *Table 2: Characteristics of lesions in Patients with DA and in controls
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ACCEPTED MANUSCRIPT p1: Statistical signification between Patients with DA and controls, p2: Statistical signification between Patients with DA and siblings
RI P
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*Table 3: Life events and quality of life scores in Patients with DA, controls and siblings p1: Statistical signification between Patients with DA and controls, p2: Statistical
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CE
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signification between Patients with DA and siblings
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ACCEPTED MANUSCRIPT Table 1: Socio-demographic characteristics and medical history of Patients with DA, controls and siblings group Controls, n
DA, n (%)
(%)
Siblings, n
p
(%)
Illiterate
11 (36.66)
level
Primary
12 (40)
Secondary
7 (23.33)
14 (46.66)
7 (23.33)
Academic
0 (0)
5 (16.66)
1 (3.33)
24 (80)
16 (53.33)
8 (26.66)
p1 = 0.002
6 (20)
14 (46.66)
22 (73.33)
p2 < 0.001
28 (93.33)
11 (36.33)
10 (33.33)
p1 < 0.001
2 (6.66)
19 (63.33)
20 (66.66)
p2 < 0.001
18 (60)
18 (60)
17 (56.66)
p1= NS
12 (40)
12 (40)
13 (43.33)
p2= NS
Psychiatric disorder
9 (30)
1 (3.33)
1 (3.33)
p1 = 0.006
Present
Marital
Single
Status
Married
Residence
Urban
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activity
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Absent
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Professional
Personal
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Rural
2 (6.66)
11 (36.66)
p1 < 0.001
9 (30)
11 (36.66)
p2 = NS
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Educational
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Patients with
history: n (%)
Suicide attempt
p2 = 0.006 9 (30)
0 (0)
0 (0)
p1 = 0.001 p2 = 0.001
Somatic disease
10 (33.33)
8 (26.66)
0 (0)
p1 = NS p2 < 0.001
Prolonged
8 (26.66)
1 (3.33)
1 (3.33)
p1 = 0.013
hospitalization or
p2 = 0.013
medical follow
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ACCEPTED MANUSCRIPT during childhood Family
Psychiatric disorder
3 (10)
0 (0)
3 (10)
p1 = NS
Somatic disease
18 (60)
11 (36.66)
care during childhood
paramedical
p2= NS 11 (36.66)
p1 = 0.001
5 (16.66)
6 (20)
p2= NS
5 (16.66)
p1 = NS p2= NS
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profession
p1 = NS
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Medical or
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hospitalization or sustained medical
1 (3.33)
18 (60)
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Prolonged
18 (60)
RI P
(%)
p2= NS
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history: n
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p1: Statistical signification between Patients with DA and controls
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p2: Statistical signification between Patients with DA and siblings
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ACCEPTED MANUSCRIPT Table 2: Characteristics of lesions in Patients with DA and in controls
Control
DA
Group
T
Patients with
RI P
Item
Duration of evolution before diagnosis
34.23 ± 35.5
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(months) Number of consulted care centers No
p1 = NS
26.02 1.2 ± 1.18
p1 <0.001
18 (60)
12 (40)
Improvement
10 (33.33)
12 (40)
Recovery
2 (6.66)
6 (20)
Yes
25 (83.33)
6 (20)
No
5 (16.66)
24 (80)
3.17 ± 2.21
1.97 ± 2.18
p1 <0.001
1.61 ± 1.33
0.74 ± 0.78
p1 = 0.03
improvement
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Evolution, n (%)
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2.63 ± 1.9
35.2 ±
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Number of relapses
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Complications, n (%)
p1 = NS
p1 <0.001
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Number of relapses/year
p1: Statistical signification between Patients with DA and controls p2: Statistical signification between Patients with DA and siblings
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ACCEPTED MANUSCRIPT Table 3: Life events and quality of life scores in Patients with DA, controls and siblings
Mean global
events
score
with DA
patients
2.33 ± 0.92
0.70 ±
With
1.57 ± 0.72
0.3 ± 0.46
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objective
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1.08
negative impact
Siblings
P
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Control
RI P
Life
Patients
0.77 ±
p1 <0.001
1.19
p2 <0.001
0.3 ±
p1 <0.001
0.18
p2 <0.001
Mean global
1285.50 ±
2227.67 ±
2546 ±
p1 <0.001
of life
score
544.71
626.62
401.72
p2 <0.001
30 (100)
17 (56.66)
12 (40)
p1 <0.001
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Altered mean
ED
Quality
global score, n
CE
(%)
p2 <0.001
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p1: Statistical signification between Patients with DA and controls p2: Statistical signification between Patients with DA and siblings
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ACCEPTED MANUSCRIPT Highlight Compared to both control groups, patients with DA reported more Life Events with more
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objective negative impact of these events and had a lower and more often altered Mean
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Global Score of Quality of life. Life Events would then have a role in the pathogenesis of DA which seems to alter the Quality of Life of patients. These results could help to improve the
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DA but also on the impact of this disease.
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understanding of DA and incite clinicians to focus not only on the diagnosis and treatment of
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