Hindawi

Case Reports in Psychiatry

Volume 2020, Article ID 8899615, 3 pages
https://doi.org/10.1155/2020/8899615

Case Report
Bipolar Disorder as Comorbidity with Sjögren’s Syndrome: What
Can We Do?

Selima Chebli, Yosra Zgueb, Uta Ouali, Sana Taleb , and Fethi Nacef
Psychiatry Department A, Razi Hospital, Faculty of Medicine of Tunis, El Manar University of Tunis, Tunisia

Correspondence should be addressed to Sana Taleb; sanataleb14805@gmail.com

Received 4 April 2020; Revised 18 August 2020; Accepted 26 August 2020; Published 11 September 2020

Academic Editor: Jeronimo Saiz Ruiz

Copyright © 2020 Selima Chebli et al. This is an open access article distributed under the Creative Commons Attribution License,
which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

Neuropsychiatric manifestations in Sjögren’s syndrome are common and can occur not only during its course, but also at the onset
of the disease. Depression and anxiety were the most frequently described symptoms. However, the association with bipolar
disorder seems to be rare and not well documented. This case report presents a patient with bipolar disorder as comorbidity
with Sjögren’s syndrome, suggesting that bipolar disorder could be associated with this autoimmune disease, which could lead to
delaying diagnosis and treatment. A better analysis of the clinical background should be done by psychiatrists so to help early
diagnosis and adapting prescription. Corticosteroids indicated in Sjögren’s syndrome should be prescribed with caution in
bipolar disorder.

1. Introduction vous system involvement in Sjögren’s syndrome (CNS-SS)

which includes psychiatric disorder. Cox and Hales [4] have
Bipolar disorder is a disabling chronic mental disease charac- suggested that the incidence of mild to moderate psychiatric
terized by recurrent manic, hypomanic, and depressive epi- and/or cognitive impairment may be as high as 80% in patients
sodes. Its prevalence has been estimated to be around 1 to with CNS-SS. Atypical depression (characterized by irritability,
2%. The etiology of BD remains not entirely known and dif- agitation, and somatic symptoms), anxiety disorder, and psy-
ferent factors have been implicated through time (genetic, chotic features have been most commonly described [5].
biological, and psychosocial). The aim of this article is to highlight that bipolar disorder
Recently, multiple evidences have suggested that the could be associated with Sjögren’s syndrome and that this
immune system, central nervous system, and the endocrine association has a major therapeutic impact. Moreover, we
system are involved in the pathophysiology of BD. want to emphasize that psychiatric manifestations are com-
In fact, the association of systemic autoimmune diseases mon in patients with SS and require early diagnosis and
(SADs) with BD has been reported in several epidemiological appropriate psychotropic therapy.
studies. Crohn’s disease, autoimmune hepatitis, rheumatoid Consent was obtained from the patient for publication of
arthritis, systemic lupus erythematosus (SLE), psoriasis, and this paper.
finally autoimmune thyroiditis were found to increase the
relative risk of BD [1]. However, the number of published 2. Case Presentation
studies about Sjögren’s syndrome (SS) remains limite
Sjögren’s syndrome (SS) is a chronic systemic autoim- Mrs. G was a 54-year-old diabetic patient who was first
mune disorder characterized by the inflammation and the admitted in 2013 into an internal medicine ward for brief epi-
lymphocytic infiltration of exocrine glands. Its hallmark sodes of agitation, boxing movements in the upper limbs, and
symptoms are xerostomia and keratoconjunctivitis sicca [2]. pedaling movements in the lower ones. These episodes have
In addition to the local manifestation of SS (glandular), a evolved over the previous 3 years (2 to 3 times a week) and
neuropsychiatric involvement has been described. In fact, some have been accompanied by occipital migraines, horizontal
studies [3] have recently reported higher rates of central ner- diplopia, and distal sensory loss.
2 Case Reports in Psychiatry

Figure 1: Periventricular T2/flair hypersignals associated with subcortical cortical atrophy.

Neurological examination revealed both pyramidal and ment. They can also be an early manifestation of SS, in which
posterior cordonal syndrome as well as clinical sensory abnor- case they would precede the somatic symptoms [5].
malities involving the left limbs and diplopia. At first, epilepsy According to Ampelas et al. [6], mental disorders in SS
was suspected but EEG was normal. The MRI showed peri- could be explained by secondary psychological distress.
ventricular white matter lesions on T2-weighted images They have suggested that the slow progress and fluctuation
(Figure 1). The cerebrospinal fluid analysis was normal. HIV course of SS created constant discomfort inducing a depres-
and hepatitis C virus serologies and VDRL/TPHA were all sive or anxious reaction to a chronic illness. However, in
negative. The anti-DNA test, antinuclear antibodies, and line with other reports, psychiatric presentation of SS also
anti-SSA/Ro and anti-SSB/La tests were negative. Rheumatoid suggests that mental disorders do not occur only as a
factor and total complement were normal. response to a psychological distress or a reaction to a
Questioning the patient revealed sicca syndrome with chronic disease but may be an early manifestation of the
xerophthalmia and xerostomia during the previous 6 same autoimmune process, which presumes the direct
months. A Schirmer’s test was positive and salivary gland immunological activity of SS on the central nervous system
biopsies showed a lymphocytic infiltration, scored 4 using (by T cells, autoantibodies, cytokines, or the programmed
Chisholm’s criteria (focus score > 1). cell death (apoptosis) [7]. It has been recently suggested
Mrs. G was then diagnosed as having a primary Sjögren’s that autoantibodies (autoAbs) reactive with adrenocortico-
syndrome. Corticosteroid therapy followed by immunosup- tropic hormone (ACTH) and a-melanocyte-stimulating
pressive treatment has been administered with no improve- hormone (a-MSH) might be implicated in the pathogenesis
ment. Because of persisting episodes of agitation, Mrs. G of psychiatric symptomatology [8].
was then referred to our psychiatric ward for evaluation. At Several studies have reported psychiatric involvement in
admission, she complained of irritability, agitation, difficulty SS. The commonest psychiatric manifestations were depres-
in sleeping, poor concentration, and demoralization. She also sion, anxiety, and some personality traits (especially hypo-
reported depressed mood, anhedonia, hopelessness, feeling chondriac and histrionic features) [9]. Some investigators
of guilt toward her children, and abulia. such as Stevenson et al. [10] and more recently, Shen et al.
Focused questioning revealed that Mrs. G had attempted [3] have confirmed that depression is the most frequent
suicide in her twenties and had suffered later from two comorbidity in patient with SS. However, based on the role
depressive episodes with postpartum onset. An interview played by autoimmunity in the etiology of bipolar disorder
with Mrs. G’s family revealed that she previously had an epi- (BD), epidemiological studies have also reported the associa-
sode of antidepressant-induced mania. She also had shown tion of SADs with BD [11]. Wang et al. [1], in a recent study,
periods of elevated mood, decreased need of sleep, and have provided further evidence that SADs including Sjög-
increase energy suggesting hypomanic episodes. ren’s syndrome are associated with higher incidence of BD.
According to the Diagnostic and Statistical Manual of Our case study seems to support the hypothesis that an
Mental Disorders, fifth edition (DSM5) criteria, “bipolar dis- abnormal autoimmune process is associated with increased
order” was diagnosed. We also diagnosed histrionic person- expression of psychiatric symptoms.
ality disorder. Further studies on patients with Sjögren’s syndrome
A mood stabilizer was initiated (carbamazepine might be needed for a better comprehension of this associa-
600 mg/day) in association with prednisone (20 mg/day), tion as treatment and prognosis vary. Indeed, clinicians
and the patient showed a significant improvement within 4 should remain vigilant for psychiatric symptoms as affective
weeks. The instability disappeared, and no more episodes of disorders are manageable if adequately treated.
agitation or altered mood were reported. Our patient showed at first no improvement under corti-
costeroid therapy and seemed to have more episodes of agita-
3. Discussion tion. However, starting her on mood stabilizer proved to be
efficient. In fact, corticosteroids, largely used in the treatment
Psychiatric disorders associated with Sjögren’s syndrome are of autoimmune diseases, could induce psychiatric manifesta-
a clinical reality. Indeed, these disorders might constitute a tions like manic syndrome and should be used with caution
complication of SS due to central nervous system involve- in case of association with BD [12].
Case Reports in Psychiatry 3

Therefore, a rigorous analysis of clinical background and [7] T. R. Esch, “Pathogenetic factors in Sjögren’s syndrome: recent
psychiatrist assessment should be done systematically in developments,” Critical Reviews in Oral Biology and Medicine,
patient with autoimmune disease in order to avoid delays of vol. 12, no. 3, pp. 244–251, 2016.
diagnosis and to better adapt the prescription of corticoster- [8] S. O. Fetissov, J. Hallman, I. Nilsson, A. K. Lefvert, L. Oreland,
oid/immunosuppressive agent. Appropriate additional psy- and T. Hokfelt, “Aggressive behavior linked to corticotropin-
chiatric treatment could then lead to remarkably rapid reactive autoantibodies,” Biological Psychiatry, vol. 60, no. 8,
relief as we reported in our case after initiating a mood pp. 799–802, 2006.
stabilizer. [9] E. O. Johnson, M. Kostandi, and H. M. Moutsopoulos, “Hypo-
thalamic-pituitary-adrenal axis function in Sjogren’s syn-
drome: mechanisms of neuroendocrine and immune system
4. Conclusion homeostasis,” Annals of the New York Academy of Sciences,
vol. 1088, no. 1, pp. 41–51, 2006.
Neuropsychiatric manifestations in Sjögren’s syndrome are [10] H. A. Stevenson, M. E. Jones, J. L. Rostron, L. P. Longman, and
common and can occur not only during its course but also E. A. Field, “UK patients with primary Sjogren’s syndrome are
at the onset of the disease. This evidence leads to important at increased risk from clinical depression,” Gerodontology,
clinical considerations. vol. 21, no. 3, pp. 141–145, 2004.
Clinicians (rheumatologists, neurologists, ophthalmolo- [11] J. D. Rosenblat and R. S. McIntyre, “Are medical comorbid
gists, and psychiatrists) should be aware that psychiatric dis- conditions of bipolar disorder due to immune dysfunction?,”
orders are possible in SS patients (both at the onset and Acta Psychiatrica Scandinavica, vol. 132, no. 3, pp. 180–191,
during the course of the autoimmune syndrome) and then 2015.
may need psychiatric help and additional appropriate psy- [12] A. A. Wyszynski and B. Wyszynski, “Treatment of depression
chotropic therapy. with fluoxetine in corticosteroid-dependent central nervous
Thus, first, we suggest that the diagnosis of Sjogren’s syn- system Sjögren’s syndrome,” Psychosomatics, vol. 34, no. 2,
drome should be considered in patients who have neuropsy- pp. 173–177, 1993.
chiatric disturbance of unexplained cause and in patients
with a characteristic psychiatric profile of hysteria, somatiza-
tion, and atypical mood disorder. Second, it is important to
appropriately screen for mental disorders including bipolar
disorder starting at disease onset, because affective disorders
in SS patients are treatable and can considerably improve
quality of life.

Conflicts of Interest
We have no conflict of interest.

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