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"Neuro-psycho- BS": A Case Report of Rare Association with Bipolar Disorder.

1. Introduction

Bechet's syndrome (BS) is a systemic, chronic, relapsing vasculitis, typically characterized by recurrent orogenital ulcers, ocular inflammation and skin manifestations; articular, vascular, gastroenteric and neurological involvement may also occur.[1] Besides the other clinical features of BS, it seems relatively frequent that patients with BS develop a neurobehavioral syndrome, characterized by euphoria, bipolar disorders and paranoid attitudes, loss of insight/disinhibition, and indifference to their disease, defined as 'neuro-psycho-BS'.[2]

Bechet's disease and bipolar disorder share features like relapsing and remitting course and viral triggers have been reported for both.[3] In autoimmune diseases affective symptoms have been noted and autoantibodies are reported to accompany affective syndromes.[4] An autoimmune pathogenesis as one of the possible pathogenesis was proposed for unipolar or bipolar disorders.[3] Another possible mechanism could be due to NBD parenchymal damage in the brain.

2. Case report

A 49 year old gentleman developed Bechet's syndrome at the age of 38 years.The illness was episodic in nature with intermittent remission and exacerbations. The symptoms were characterized initially by slurring of speech, gait disturbance, visual disturbance, right side drooping of the eyelid, swelling and redness of periorbital region, recurrent aphthous lesion in the mouth along with weakness of the lower limbs and unsteady gait. He was found to be HLA-B51 leucocyte antigen positive.

For the past 4 years along with the above said symptoms which were also episodic in nature, the patient presented with episodic illness with symptoms characterized by euphoric mood, over familiarity, increased goal-oriented behavior, increased self-esteem, grandiose delusions, overspending, increased libido and exhibited disinvited behavior. Currently he presented with 2 months of relapse of similar symptoms such as euphoric mood, reduced need for sleep, over activity, increased libido, over spending, increased energy level and over religiosity affecting bio-socio-occupational function followed by exaggeration of Bechet's syndrome even with good medical compliance. Patient was not on any mania inducing drug to suspect iatrogenic causes. Basic laboratory profile is included in table 1. Likewise, he had three similar relapses and all were followed by exaggeration of Bechet's disease. His Young mania rating scale (YMRS) point was recorded as 35 at the time of admission. The patient was started on valproate 1000mg/d and quetiapine 400mg/d. The patient showed improvement in both his manic and psychotic symptoms in the next 3-4 weeks. YMRS was again assessed at this point of time and was recorded as 4. The patient was also initiated on methylprednisolone 50mg/day which was gradually tapered and stopped over the next 7 weeks and azathioprine 75 mg/day which the patient continued to take post-discharge as maintenance treatment for the primary illness which was necessary to prevent both medical as well as psychiatric relapses.

3. Discussion

Behcet's disease usually starts around the third or fourth decade of life. The characteristic inflammatory lesion in BD is secondary to involvement of large and small arteries as well as venous vessels. Around 5-20% of the population with BD also exhibit neurological complications, and are defined as neuro-Bechet's disease (NBD).[5]

Neuro-Bechet's disease underlying pathology is divided into vascular and parenchymal lesions. Patients with vascular lesions display intracranial hypertension with papilledema, headaches, focal neurological deficits and occasional pyramidal symptoms. In contrast, parenchymal lesions cause pyramidal symptoms in 96% of patients and behavioral alterations in half. A minority also suffers from hypersomnia, sensory and gait aberrations and hearing and sight problems. Only 2% of cases exhibit psychiatric symptoms, such as acute psychosis.[6]

Bipolar disorders have been described in rare cases. In 2002, Aydin et al. reported a case of neuro-BS in which the initial onset was hypomania.[7] To date, there have been no guidelines for treatment of these disorders in BS, probably because bipolar disorders have been studied less frequently than other types of disorder in BS patients. Indeed, these symptoms may be related to a neurological substrate of the syndrome, and we can hypothesize that they develop in the middle stage of the disease. A combination of mood-stabilizing drugs, such as sodium valproate, carbamazepine and olanzapine, may produce some improvement in the disease. [1]

In our patient there were both neurological and psychiatric symptoms with parenchymal involvement that matches with the imaging findings and mood episodes exaggeration of Bechet's symptoms. It's unclear whether the psychiatric symptoms are due to the underlying NBD or independent, as evidence from literature on the causal relationship between psychiatric symptoms and NBD is limited.

Only a few case reports have linked NBD with bipolar disorder, one reported manifestation of mood disorder during an NBD exacerbation.[8] Deniz O et al. reported a psychotic patient with an NBD exacerbation and a patient whose affective symptoms preceded BD.[9] Due to the lack of literature we might assume that the psychiatric disorder in our patient could be caused by NBD parenchymal damages in the brain. Also, there is an association between bipolar disorder and inflammatory cytokines. Studies have proved that increased CRP level may predict manic episode in a depressive patient. Also, there were multiple active inflammatory markers in men with bipolar illness.[10] Organ specific autoimmunity like thyro-peroxidase antibodies (TPO-Abs), antibodies to H/KAT-Pase GAD65A, antibodies to gliadin have been linked with Bipolar disorder as well as unaffected relatives of bipolar disorders.[11] Then there is also a lack of literature about the causal relationship between the two and little research has been done so far with respect to treatment of NBD with mood disorders.

Combination of glucocorticoids and azathioprine along with mood stabilizers and anti-psychotics appears to be effective as it showed good results in our patients although specific guidelines are not available.

Further research is required to understand the casual relationships, and is needed from a treatment prospective as well. This case report is just an eye opener in this sparsely researched area.

Funding statement

No funding was provided for this article.

Conflicts of interest statement

Authors report no conflict of interest related to this manuscript

Informed consent

Patient provided written informed consent

Authors' contributions

All authors contributed to the drafting of this manuscript

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References

[1.] Talarico R, Palagini L, d'Ascanio A, Elefante E, Ferrari C, Stagnaro C, et al. Epidemiology and Management of Neuropsychiatric Disorders in Behcet's Syndrome. CNS Drugs. 2015; 29(3): 189-196

[2.] Siva A, Hirohata S. Behcet's syndrome and the nervous system. In: Yazici Y, Yazici H (eds). Behcet's Syndrome. New York, NY: Springer; 2010. pp: 95-113

[3.] Yolken RH, Torrey EF. Viruses, schizophrenia, and bipolar disorder. Clin Microbiol Rev. 1995; 8(1): 131-145

[4.] Maes M, Meltzer H, Jacobs J, Suy E, Calabrese J, Minner B, et al. Autoimmunity in depression: increased antiphospholipid autoantibodies. Acta Psychiatrica Scandinavica. 1993; 87(3): 160-166

[5.] Akman-Demir G, Serdaroglu P, Tasci B. Clinical patterns of neurological involvement in Behcet's disease: evaluation of 200 patients. The Neuro-Behcet Study Group. Brain. 1999; 122 ( Pt 11): 2171-2182

[6.] van Ham C, Schrijvers D, De Picker L, Vandendriessche F, Sabbe B. Neuropsychiatric features in Behcet's disease: A case report. Clin Neurol Neurosurg. 2014; 127:13-4

[7.] Aydin N, Aydin MD, Deniz O, Kirpinar I. Neuro-Behcet's disease involving the pons with initial onset of affective symptoms. Eur Arch Psychiatry Clin Neurosci. 2002; 252(1): 44-46.

[8.] Alevizos B, Anagnostara C, Christodoulou G. Resistant bipolar disorder precipitated by Behcet's syndrome. Bipolar Disord. 2004; 6(3): 260-263. doi: http://dx.doi.org/10.1111/j.1399-5618.2004.00114.x

[9.] Deniz O, Qaykoylii A, Vural G, Albayrak Y, Temel S, Aydm i, et al. A case study of Neuro-psycho-Behcet's Syndrome presenting with psychotic attack. Clin Neurol Neurosurg. 2009; 111(10): 877-879. doi: http://dx.doi.org/10.1016/j.clineuro.2009.07.009

[10.] Leboyer M, Soreca I, Scott J, Frye M, Henry C, Tamouza R, et al. Can bipolar disorder be viewed as a multi-system inflammatory disease? J Affect Disord. 2012; 141(1): 1-10. doi: http://dx.doi.org/10.1016/j.jad.2011.12.049

[11.] Becking K, Boschloo L, Vogelzangs N, Haarman BCM, Riemersma-van der Lek R, Penninx BWJH, et al. The association between immune activation and manic symptoms in patients with a depressive disorder. Transl Psychiatry. 2013; 3(10): e314-e314. doi: http://dx.doi.org/10.1038/tp.2013.87

Mustafa ALI, Soumitra DAS (*)

(1) Dept. of Psychiatry, National Institute of Mental Health and Neurosciences, Bangalore, India

(*) correspondence: Soumitra Das; Mailing address: New Kabini Hostel, NIMHANS, Bangalore, India; Postcode: 560029; E-Mail: soumitratdmc@gmail.com

Mustafa ali obtained a bachelor's degree in clinical medicine in 2012 from Government Medical College, Srinagar, Jammu and Kashmir, India. He started to work at National Institute of Mental Health and Neurosciences, Bangalore, India in 2015, and now is working as a resident doctor in the department of psychiatry. His research interests are schizophrenia, bipolar, and addiction.

doi: http://dx.doi.org/10.11919/j.issn.1002-0829.217126]
Table 1. Assessment and results

ASSESMENT                              RESULT

CNS EXAMINATION                        B/L PERIORBITAL SWELLING. LEFT
                                       EYE-PTOSIS, IMPAIRED
                                       TAMDEM GAIT
MMSE                                   30/30
NEURO-PSYCHOLOGICAL ASSESMENT(NIMHANS  INVOLVEMENT OF DLPFC AND RIGHT
                                       TEMPORAL LOBE
Neuropsychology Battery-2004)
YMRS                                   35 at admission and 4 after four
                                       weeks of admission
HLA-B51 LEUCOCYTE ANTIGEN              POSITIVE
BLOOD INVESTIGATIONS                   NEGATIVE FOR ANA PROFILE, RA
                                       FACTOR, VDRL, SERUM
                                       NMO,HBS,HCV
                                       Multiple focal lesions in basal
                                       ganglia, internal capsule,
                                       thalamus and deep white matter of
                                       frontal lobe.
MRI BRAIN                              Round lesion in right temporal
                                       lesion
                                       Multiple well defined
                                       demyelinating lesions.

MMSE: Mini-Mental State Examination; YMRS: Young Mania Rating Scale


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Title Annotation:CASE REPORT
Author:Ali, Mustafa; Das, Soumitra
Publication:Shanghai Archives of Psychiatry
Article Type:Case study
Date:Feb 1, 2018
Words:1573
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