Visibility of Neurofibromatosis 1 and Psychiatric Morbidity

Wolkenstein, Pierre; Zeller, J; Revuz, J; Écosse, Emmanuel; Leplège, Alain

doi:10.1001/archderm.139.1.103

Cited by 30 publications

(39 citation statements)

References 3 publications

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“…We found that NF1 patients experienced and appraised their own bodies more negatively than patients with a dermatological or psychiatric disorder. In accordance with previous findings [8,9,10], we confirmed that disease visibility had an impact on a depressive state, distress and quality of life. In NF1 patients, tumor manifestations are unpredictable, show continuous growth in most cases and are not influenced by psychosocial stress.…”

Section: Discussionsupporting

confidence: 93%

“…Disease visibility is one of the main concerns for adult NF1 patients. Wolkenstein et al [8] conclude in their paper on psychiatric morbidity and disease visibility in NF1 that ‘patients’ first priority is cosmetic correction’ and therefore ‘reducing deformity with dermatological and/or plastic surgery should not be neglected by physicians as it could lessen the impact of NF1 on patients’ mental health and improve their well-being’. However, scars and the continuous progression of skin tumours are burdens that surgery cannot eliminate.…”

Section: Discussionmentioning

confidence: 99%

“…In NF1 patients, disease visibility is associated with psychiatric morbidity [8] and has a negative impact on quality of life [9,10,11]. Research on the consequences of disfiguring disease symptoms shows that the patients concerned have a higher risk of developing low self-esteem, self-rejecting cognitions, problems in interpersonal relationships and a negative body image [12] which results in psychological stress.…”

Section: Introductionmentioning

confidence: 99%

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Psychological Burden in Adult Neurofibromatosis Type 1 Patients: Impact of Disease Visibility on Body Image

Granström¹,

Langenbruch²,

Augustin³

et al. 2012

Dermatology

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Aim: To evaluate the impact of disease visibility on psychological stress factors in neurofibromatosis type 1 (NF1) and to explore the body image of NF1 patients. Methods: 228 adult NF1 patients participated in this cross-sectional survey. The questionnaire assessed perceived disease visibility and patients’ body image. Outcome parameters were depression, distress and quality of life. Mediation models were performed to test if body experience mediated the effect of disease visibility on outcome parameters. Results: Adult NF1 patients had a negative body image, expressed by bodily insecurity/uneasiness and fewer feelings of attractiveness and self-confidence. Compared to the body image of patients with other disfiguring diseases, patients with NF1 felt less attractive, displayed less self-confidence (women: p < 0.001; men: p < 0.001) and were more insecure/uneasy and sexually dissatisfied with their bodies (men: p < 0.001; women: p ≤ 0.12, d = 0.28). The found effect of disease visibility on psychological stress was completely mediated by how patients experienced their bodies. Conclusions: Our study shed light on the importance of how NF1 patients experience and appraise their own bodies. We revealed that body image is an important link between disease visibility and psychological well-being. The body image of patients can be improved with psychotherapeutic interventions.

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Section: Discussionsupporting

confidence: 93%

Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

See 1 more Smart Citation

Psychological Burden in Adult Neurofibromatosis Type 1 Patients: Impact of Disease Visibility on Body Image

Granström¹,

Langenbruch²,

Augustin³

et al. 2012

Dermatology

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“…In addition to these clinical features, a high frequency of anxiety and other psychopathological symptoms have been documented in the adult NF1 population [Kayl and Moore, 2000;Wolkenstein et al, 2003;Belzeaux and Lancon, 2006]. Unfortunately, the current available data do not allow a comprehensive explanation of the anxiety symptoms, neither a definition of anxiety disorders in NF1 subjects.…”

Section: Introductionmentioning

confidence: 99%

“…Co-morbid psychiatric disorders in adults with NF1 have been investigated in few studies. In a sample of 128 adult patients with NF1 an unspecified psychiatric morbidity has been diagnosed in 32.8% of cases, and a direct link with the disease's visibility has been found [Wolkenstein et al, 2003]. Only one study reported a prevalence of anxiety disorders in adult NF1 subjects, ranging from 1% to 6%, and the impairment of their quality of life has been related to the development of a psychiatric condition [Belzeaux and Lancon, 2006].…”

Section: Introductionmentioning

confidence: 99%

Detecting anxiety symptoms in children and youths with neurofibromatosis type I

Pasini

Lo-Castro

Carlo

et al. 2012

American J of Med Genetics Pt B

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Children with Neurofibromatosis type 1 (NF1) are known to have cognitive, social, and behavioral deficits. Fifteen NF1-subjects (5 boys, 10 girls, mean age = 13.4), and 15 healthy controls matched for age and sex were assessed on the presence of anxiety symptoms, using the Multidimensional Anxiety Scale for Children (MASC), self-report questionnaire. Significant group differences emerged with regard to MASC total (Z = -2.058, P = 0.041) and anxiety disorder index (ADI; Z = -2.202, P = 0.026), but not with regard to single scales. When the severity and visibility of NF1 were considered, correlation between severity and social anxiety, and severity and MASC total was found. This is the first study assessing anxiety symptoms in NF1 children and youths. A precocious psychological survey and intervention in NF1 subjects, may contribute to reduce the risk of psychiatric disorders in adulthood.

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Impact of neurofibromatosis 1 on Quality of Life: A cross‐sectional study of 176 American cases

Page

Écosse

et al. 2006

American J of Med Genetics Pt A

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133

139

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Neurofibromatosis 1 (NF1), a genetic condition most commonly characterized by the presence of dermal neurofibromas and café au lait macules, has a significant impact on Quality of Life (QoL). There is a wide range of phenotypic variability, so that affected individuals may have either medically devastating or relatively mild manifestations that do not impact their daily lives. In this study, the SF-36 and Skindex questionnaires were used to quantitatively investigate the impact of severity and visibility on QoL in an American population. Participants were recruited primarily through advertisements distributed by The Children's Tumor Foundation (CTF). The majority participated by completing a mailed questionnaire in which they rated themselves using Ablon's visibility and Riccardi's severity scales, and then completed the SF-36 and Skindex questionnaires. Participants with NF1 reported a significant impact in all aspects of skin-disease-specific QoL, but the emotional aspect demonstrated the greatest effect. Participants with more visible signs of NF1 reported significantly greater overall effects on their skin-disease-specific QoL than those whose manifestations were more subtle. All domains of general health QoL were affected as well, especially in participants who reported having severe complications. Interestingly, greater visibility was not found to be associated with a significant decrease in general health QoL. These findings are consistent with those found in a French cohort, and demonstrate the utility of incorporating tools designed for use in both the general population and for patients with skin disease in examining the impact of NF1 on QoL.

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Visibility of Neurofibromatosis 1 and Psychiatric Morbidity

Cited by 30 publications

References 3 publications

Psychological Burden in Adult Neurofibromatosis Type 1 Patients: Impact of Disease Visibility on Body Image

Psychological Burden in Adult Neurofibromatosis Type 1 Patients: Impact of Disease Visibility on Body Image

Detecting anxiety symptoms in children and youths with neurofibromatosis type I

Impact of neurofibromatosis 1 on Quality of Life: A cross‐sectional study of 176 American cases

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