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References (5)
- Publisher
- Springer Journals
- Copyright
- Copyright © The Author(s) 2022
- ISSN
- 0271-9142
- eISSN
- 1573-2592
- DOI
- 10.1007/s10875-022-01207-7
- Publisher site
- See Article on Publisher Site
Abstract
Journal of Clinical Immunology (2022) 42:695–698 https://doi.org/10.1007/s10875-022-01207-7 LE T TER TO EDITOR Psychological Symptoms in Primary Immunodeficiencies: a Common Comorbidity? 1 2,3,4 1,5,6 1 Olivia R. Manusama · Nico J. M. van Beveren · P. Martin van Hagen · Hemmo A. Drexhage · 1,5,6 Virgil A. S. H. Dalm Received: 18 August 2021 / Accepted: 13 December 2021 / Published online: 19 January 2022 © The Author(s) 2022 To the Editor, according to the International Union of Immunological Soci- eties (IUIS) [1]. The study was conducted between April Primary immunodeficiency diseases (PIDs) encompass a and June 2018 using the 4-Dimensional Symptom Question- heterogeneous group of rare disorders, characterized by an naire (4-DSQ), a self-reported questionnaire which measures increased susceptibility to infections, autoimmune compli- four symptom dimensions (distress, depression, anxiety, and cations, autoinflammatory diseases, and malignancies [1 ]. somatization) over 50 subitems. For each item, participants Apart from these well-recognized disease manifestations, informed researchers whether they suffered from a spe- PID patients also suffer from psychological distress [2 ], cific symptom in the past week, and if yes, how often. This significantly impacting their quality of life. Psychological resulted in a score for each of the four dimensions, with well-being is currently under-researched in patients with different score ranges per dimension. The complete ques- PIDs. Moreover, the prevalence of psychological symptoms tionnaire has been included in the supplementary material in these patients remains unclear. Therefore, we aimed to (Suppl. doc. 1). gain more insight into the disease burden and the nature of We compared the prevalence of psychological symptoms psychological complaints in a cohort of adult patients with in patients with PID to the prevalence in the Dutch general PIDs. population, using open-access data from the Longitudinal We performed a cross-sectional study in 293 adult Internet Studies for the Social Sciences (LISS) panel, avail- patients with PIDs, who were under treatment at the Pri- able online (https:// www. dataa rchive. lissd ata. nl). We used mary Immunodeficiency Center of the Department of Inter - one of the available samples (n = 4874) that was previously nal Medicine, Erasmus University Medical Center Rotter- used to construct the normative 4DSQ scores, from which dam, the Netherlands, for an established diagnosis of PID randomly two sex- and age-matched controls per patient were selected. To evaluate whether patient-related factors are asso- ciated with these psychological symptoms, we retrieved * Virgil A. S. H. Dalm v.dalm@erasmusmc.nl clinical data from the electronic medical records, includ- ing immunological diagnosis, the presence of autoimmune Department of Immunology, Erasmus University Medical manifestations, and laboratory results of immunoglobulin G Center, Rotterdam, The Netherlands (IgG) levels before the start of immunoglobulin replacement Department of Psychiatry, Erasmus University Medical therapy. Moreover, self-reported data on life-time mental Center, Rotterdam, The Netherlands health treatment, psychiatric diagnosis, prior admission to Department of Neuroscience, Erasmus University Medical a psychiatric ward, use of psychiatric and recreational drugs Center, Rotterdam, The Netherlands and alcoholic beverages, and family history of psychiatric Parnassia Group for Mental Health Care, disorders were collected. Rotterdam/The Hague, The Netherlands Chi-squared tests were used for the individual 4DSQ Division of Allergy & Clinical Immunology, Department items. The Mann–Whitney U test was used for aggregate of Internal Medicine, Erasmus University Medical Center, scores. We performed four hierarchical multiple regression Rotterdam, The Netherlands 6 analyses to study associations between clinical factors and Academic Center for Rare Immunological Diseases psychological symptoms. (RIDC), Erasmus University Medical Center, Rotterdam, The Netherlands Vol.:(0123456789) 1 3 696 Journal of Clinical Immunology (2022) 42:695–698 The significance level was set at P < 0.05 for all statistical mental health disorder and current use of psychiatric medi- tests. SPSS Statistics version 24 and 25 (IBM, NY, USA) cation) and the presence of distress, depression, anxiety, and were used for statistical analyses. somatization. Overall, this study indicates the presence of Of 293 patients, 176 patients (60%) responded. Table S1 a broad range of psychological symptoms in PID patients shows the characteristics of the respondents, hereinafter rather than specific symptoms or a certain symptom dimen- referred to as the study population. Primary antibody defi- sion. We did not find a higher prevalence of three of the 50 ciency disorders were the most prevalent diagnoses in the items scored: the occurrence of flashbacks, perception of study population (see Table S2). Almost half of the study indistinct threat, and the occurrence of repetitive behavior. population had received consultation or treatment for mental The first two items are associated with anxiety and trau- health issues (n = 80; 45.5%), of which 48 patients had been matic stress, whereas repetitive behavior is associated with formally diagnosed with a psychiatric disorder. Table S3 obsessive–compulsive disorder. Our findings are in line with summarizes the comparison of the 4DSQ scores of patients previous reports showing that mental health is compromised with PIDs with the Dutch control population. Cronbach’s in patients with PIDs [2–4]. alpha for internal consistency per dimension of the 4DSQ There has been a growing interest in the relationship ranged between 0.89 and 0.95. For 47 out of 50 items, a sig- between psychiatric disorders and immune disturbances in nificant difference in scores was observed between patients psychiatric research [5]. These relationships have usually with PIDs and the general population. Moreover, patients been investigated by starting with phenotypically defined specifically suffered from a “regular or constant” psycho- psychiatric disorders and subsequently investigating the logical symptom more often than controls, whereas the fre- presence or absence of immune disturbances in patients quency of reporting “sometimes” was comparable between compared to healthy controls. Here, we approached the patients and controls. On all four dimensions, PID patients immune-psychiatry relationship from the opposite direction scored significantly higher, as shown in Table 1 and Fig- by investigating the presence or absence of mental distur- ure S1. Evaluating the frequencies of “moderate” and “high” bances in patients with established immune deficits. scores together — which are both considered to be aberrant Patients with PIDs experience somatic health impedi- — results in prevalence of symptom dimensions of patients ments, such as increased susceptibility to infections, auto- with PID (33.9% distress, 18.9% depression, 22.4% anxi- immunity, allergy, malignancy [1], and long-term treatment ety, and 36.2% somatization) and controls (16.3% distress, from early life, which most likely impact their mental well- 5.7% depression, 8.0% anxiety, and 11.2% somatization). being. There are thus several psychological mechanisms The hierarchical regression models for the 4DSQ dimen- through which patients with PIDs are at risk for mental sions are summarized in Table S4. We found a significant health difficulties, including fear of infections, social iso- association between two covariates (lifetime treatment for a lation, fatigue, maladaptation to illness, concerns over the Table 1 4DSQ dimension 2 4DSQ scale Score range PID patients Controls χ p frequencies of PID patients and (n = 174) (n = 348) controls by conventional cutoffs n % n % Distress 24.6 < .001 Low 0–10 115 66.1 291 83.6 Moderate 11–20 36 20.7 44 12.6 High 21–32 23 13.2 13 3.7 Depression 22.5 < .001 Low 0–2 140 81.4 328 94.3 Moderate 3–5 14 8.1 12 3.4 High 6–12 18 10.5 8 2.3 Anxiety 25.9 < .001 Low 0–3 135 77.6 320 92.0 Moderate 4–9 23 13.2 23 6.6 High 10–24 16 9.2 5 1.4 Somatization 54.6 < .001 Low 0–10 111 63.8 309 88.8 Moderate 11–20 43 24.7 36 10.3 High 21–32 20 11.5 3 0.9 Higher scores indicate higher levels of symptoms. Both “moderate” and “high” scores are considered to be aberrant 1 3 Journal of Clinical Immunology (2022) 42:695–698 697 future impact of illness, and unavailability of good coping neuropsychological manifestations of immune dysregula- strategies. These mechanisms fit within the psychosocial tion, psychological symptoms should be recognized and paradigm of mental health problems. On the other hand, the attended to, in order to improve PID patients’ health-related biological approach to mental health focuses on the direct quality of life. relationship between biological abnormalities and disease Supplementary Information The online version contains supplemen- processes in the brain. With regard to PID, in this frame- tary material available at https://doi. or g/10. 1007/ s10875- 022- 01207-7 . work, the biological aberrancies within the immune system may cause altered brain functioning, ultimately manifesting Acknowledgements We are grateful to all the patients that participated itself as mental dysfunction. This hypothesis is supported in the study. We thank Wiley Editing Services for English language editing. Finally, we thank Prof. Dr. Peter J. van der Spek for his con- by the role of the immune system in brain development and tribution to this project. plasticity and various immune implications in the patho- genesis of psychiatric disorders [5]. In any case, a complex, Author Contribution All authors contributed to the study conception possibly bidirectional interplay between psychological, and design. Material preparation, data collection, and analysis were immunological, and neurological mechanisms exists. Since performed by Olivia Manusama. The first draft of the manuscript was our study is cross-sectional, it was not designed to solve written by Olivia Manusama and all authors commented on previous versions of the manuscript. All authors read and approved the final the cause and effect and dissect the psychological factors manuscript. from biologically mediated processes. Further translational research is warranted to elucidate potential immunologic Funding This study was funded by the H2020 EU MOODSTRATIFI- contributions to the development of psychopathology. CATION project (grant agreement number 754740). From a clinical point of view, PID patients suffer from substantial psychological symptoms, and patient-centered, Availability of Data and Material The datasets generated during and/or analyzed during the current study are available from the corresponding personalized care should be modified in order to address author on reasonable request. this issue. We recommend active screening for psychologi- cal complaints after a diagnosis of PID is established and at Code Availability Not applicable. regular time points during treatment. An important predic- tive factor, as supported by our results, is the previous men- Declarations tal health treatment, which should be consistently taken into account when taking the patient’s history. We also suggest Ethics Approval The protocol was approved by the Medical Ethics involving a psychologist in the multidisciplinary care team Committee of the Erasmus University Medical Center Rotterdam (MEC 2013–026). for PID patients. To the best of our knowledge, this study presents the first Consent to Participate All subjects gave written informed consent in elaborate assessment of psychological complaints in a large accordance with the Declaration of Helsinki. cohort of patients with PID. We consider the use of age- Consent for Publication Not applicable. and sex-matched controls as an additional improvement. However, this study has several limitations. First, a possible Conflict of Interest PMH has received grants and research support non-response bias might play a role, which could assert an from Takeda, CSL Behring, Abbvie, Lamepro, Novartis Nederland, effect in multiple directions. On the one hand, patients who and honoraria or consultation fees from UCB Pharma. The other au- suffer from psychological symptoms may be more likely thors declare no competing interests. to participate in the study as compared to patients who do Open Access This article is licensed under a Creative Commons Attri- not. On the other hand, patients who experience more psy- bution 4.0 International License, which permits use, sharing, adapta- chological symptoms may be impeded from participation. tion, distribution and reproduction in any medium or format, as long Second, considering that most patients suffer from definite as you give appropriate credit to the original author(s) and the source, somatic morbidity, it is difficult to evaluate the presence provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are of somatization symptoms precisely. However, we wish to included in the article’s Creative Commons licence, unless indicated emphasize that clinicians should be aware of the possibility otherwise in a credit line to the material. If material is not included in that physical complaints arise from underlying psychological the article’s Creative Commons licence and your intended use is not mechanisms in patients with PID. Finally, considering the permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a exploratory nature of this study, no multiple test corrections copy of this licence, visit http://creativecommons.org/licenses/by/4.0/. were performed. This study adds to the limited body of empirical evi- dence that a high level of psychological symptoms exists among adult PID patients. Regardless of whether these are emotional sequelae of chronic, multisystem illness or 1 3 698 Journal of Clinical Immunology (2022) 42:695–698 of life in a cohort of 96 patients with common variable immune References deficiencies. Front Immunol. 2014;5:605. 4. Hajjar J, Guffe y D, Minard CG, Orange JS. Increased incidence 1. 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Investigating the effectiveness, acceptability and impact on healthcare usage of providing a cognitive-behavioural based Publisher's Note Springer Nature remains neutral with regard to psychological therapy service for patients with primary antibody jurisdictional claims in published maps and institutional affiliations. deficiency. J Clin Immunol. 2018;38(2):214–20. https:// doi. org/ 10. 1007/ s10875- 018- 0481-3. 3. Tabolli S, Giannantoni P, Pulvirenti F, La Marra F, Granata G, Milito C, Quinti I. Longitudinal study on health-related quality 1 3
Journal
Journal of Clinical Immunology – Springer Journals
Published: Apr 1, 2022