|Year : 2021 | Volume
| Issue : 1 | Page : 79-81
Reversible cerebellar ataxia and bipolar disorder secondary to tuberculous adrenalitis
Mansoor C Abdulla
Department of General Medicine, MES Medical College, Perinthalmanna, Kerala, India
|Date of Submission||02-Nov-2020|
|Date of Acceptance||30-Nov-2020|
|Date of Web Publication||28-Feb-2021|
Mansoor C Abdulla
Department of General Medicine, MES Medical College, Perinthalmanna - 679 338, Kerala
Source of Support: None, Conflict of Interest: None
Addison's disease is primary adrenal insufficiency which is most commonly due to an autoimmune disease. Patients usually presents with fatigue, anorexia, gastrointestinal complaints, salt craving, and orthostatic hypotension. Rare manifestations during presentation can result in diagnostic dilemma. A 53-year-old male presented with cerebellar ataxia and bipolar disorder secondary to Addison's disease (due to tuberculous adrenalitis) which improved following treatment with steroids. We describe a patient with Addison's disease who presented with two rare manifestations which to our knowledge is the first such report.
Keywords: Addison's disease, bipolar disorder, cerebellar ataxia
|How to cite this article:|
Abdulla MC. Reversible cerebellar ataxia and bipolar disorder secondary to tuberculous adrenalitis. Int J Mycobacteriol 2021;10:79-81
|How to cite this URL:|
Abdulla MC. Reversible cerebellar ataxia and bipolar disorder secondary to tuberculous adrenalitis. Int J Mycobacteriol [serial online] 2021 [cited 2021 Apr 22];10:79-81. Available from: https://www.ijmyco.org/text.asp?2021/10/1/79/310504
| Introduction|| |
Addison's disease (AD) is primary adrenal insufficiency which is most commonly (80%) due to an autoimmune disease. In developing countries, tuberculosis still remains a major cause of AD. The most commonly involved endocrine organ in tuberculosis is the adrenal gland. Patients usually presents with fatigue, anorexia, gastrointestinal complaints, salt craving, and orthostatic hypotension. Rare manifestations during presentation can result in diagnostic dilemma.
| Case Report|| |
A 53-year-old male (working as block development officer) presented with imbalance while walking and slurred speech for 1 week and psychiatric symptoms for 1½ months. The problems started as progressive depressive symptoms with sadness, loss of interest in activities, indecisiveness, and recurrent suicidal ideation for the last 1½ months. He had decreased sleep, excessively talking, impulsive and aggressive behavior for 2 weeks prior to admission under our care. His medical history was unremarkable. He was an occasional alcohol consumer for 8 years and had his last drink few months prior to the onset of symptoms. He denied history of high risk behavior and had no sick contacts. On examination, he was afebrile and had normal blood pressure (with no postural hypotension). He had hyperpigmentation over the hands, feet, and oral cavity. Neurological examination showed scanning speech, limb (upper and lower) incoordination, tandem gait was impossible and gait was ataxic. He had no nystagmus, fundus was normal and rest of the examination was unremarkable. Hemoglobin was 10.7 g/dl, total leukocyte count 10,550/ml, platelet count 240,000/ml, and erythrocyte sedimentation rate 34 mm in1 h. Peripheral smear showed normocytic normochromic red blood cells. Biochemical parameters revealed random blood sugar 95 mg%, urea 36 mg/dl, creatinine 1.0 mg/dl, sodium 128 mmol/l, potassium 5.4 mmol/l, calcium 9.1 mg%, magnesium 2.1 mg%, and phosphorus 3.6 mg%. Liver function test and thyroid function test were normal. Magnetic resonance imaging of the brain was normal. HIV, hepatitis B, and hepatitis C serology were negative. Adrenocorticotropic hormone was high and 8.00 a.m. serum cortisol was very low 1.33 mcg/dl (normal range 4.46–22.7 mcg/dl), confirming primary adrenal insufficiency. Contrast-enhanced computed tomography of abdomen showed bilateral bulky adrenal with heterogeneous enhancement [Figure 1]. Histopathological examination of adrenal gland showed extensive areas of caseous necrosis bordered by epithelioid histiocytes forming granuloma with dense lymphocytic infiltrates and multinucleated giant cells (necrotizing granulomatous lesions) [Figure 2]. Mantoux test was strongly positive. A diagnosis of adrenal insufficiency secondary to tuberculosis was made and the patient was started on hydrocortisone 50 mg intravenously four times daily which was changed to oral hydrocortisone later. He was also started on anti-tubercular treatment (isoniazid 300 mg, rifampicin 450 mg, pyrazinamide 1 g, and ethambutol 800 mg daily). His cerebellar symptoms and psychiatric manifestations improved completely at the end of 1 week. Serum sodium and potassium also became normal after treatment.
|Figure 1: Contrast-enhanced computed tomography of abdomen showing bilateral bulky adrenal with heterogeneous enhancement|
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|Figure 2: Histopathological examination of adrenal gland showing extensive areas of caseous necrosis bordered by epithelioid histiocytes forming granuloma with dense lymphocytic infiltrates and multinucleated giant cells|
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| Discussion|| |
Neuropsychiatric manifestations can be seen associated with most endocrinopathies including AD. When patients present with such atypical manifestations diagnosis of AD may be difficult unless properly evaluated. Studies have shown that men are slightly more likely to have psychiatric manifestations of AD than women. Psychiatric symptoms usually start before the diagnosis and treatment of adrenal insufficiency. Depression is the most common disorder associated with adrenal insufficiency, with usually mild mood symptoms, a decrease in motivation, and altered behavior. Psychosis, anxiety, mania, and cognitive impairment are seen less frequently. Bipolar disorder secondary to AD was not previously reported. The exact mechanism of psychiatric manifestations in AD is unknown. Hyponatremia, decrease in cerebral glucocorticoid stimulation and higher endorphin levels (secondary to high proopiomelanocortin levels) are considered to be the possible causes.,
Reversible cerebellar ataxia is usually seen secondary to alcoholism, Wernicke's encephalopathy, acute intermittent porphyria, drugs (carbamazepine and phenytoin), and viral infections. Hyponatremia as a cause for reversible ataxia was reported previously. Cerebellar ataxia has also been reported in the setting of rapid correction of hyponatremia leading to central pontine myelinolysis due to involvement of pontocerebellar fibers and sometimes with extrapontine myelinolysis. Cerebellar syndrome with thyrotoxicosis which completely resolved after normalization of thyroid function with treatment was previously reported. Reversible cerebellar ataxia secondary to AD was not previously reported. Our patient had two atypical manifestations (cerebellar ataxia and bipolar disorder) which improved following treatment for AD with steroids. The mechanisms underlying might be multifactorial as described previously including low serum sodium. Our patient had mild hyponatremia which might have contributed to the neuropsychiatric manifestations apart from the hormone deficiency.
| Conclusion|| |
Atypical manifestations of a disease make the diagnosis difficult. The knowledge of unusual presentations of AD is necessary to the treating physicians in order to make a prompt diagnosis. We describe a patient with AD who presented with two rare manifestations which to our knowledge is the first such report.
Declaration of patient consent
The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.
I would like to thank Dr. Manoj. K Department of psychiatry and Mr. Suni, clinical psychologist whose comments and suggestions were immensely valuable for improving this case report.
Financial support and sponsorship
Conflicts of interest
There are no conflicts of interest.
| References|| |
Anglin RE, Rosebush PI, Mazurek MF. The neuropsychiatric profile of Addison's disease: Revisiting a forgotten phenomenon. J Neuropsychiatry Clin Neurosci 2006;18:450-9.
Thomsen AF, Kvist TK, Andersen PK, Kessing LV. The risk of affective disorders in patients with adrenocortical insufficiency. Psychoneuroendocrinology 2006;31:614-22.
Holtzman CW, Trotman HD, Goulding SM, Ryan AT, Macdonald AN, Shapiro DI, et al
. Stress and neurodevelopmental processes in the emergence of psychosis. Neuroscience 2013;249:172-91.
Mizoguchi K, Ishige A, Takeda S, Aburada M, Tabira T. Endogenous glucocorticoids are essential for maintaining prefrontal cortical cognitive function. J Neurosci 2004;24:5492-9.
Farah Jde L, Lauand CV, Chequi L, Fortunato E, Pasqualino F, Bignotto LH, et al
. Severe psychotic disorder as the main manifestation of adrenal insufficiency. Case Rep Psychiatry 2015;2015:512430.
Asokumar P, Gogate YV, Gangadhar P, Bhansali A. Reversible cerebellar ataxia: A rare presentation of depletional hyponatremia. Neurol India 2011;59:631-2.
] [Full text]
Somarajan A, Paul MB, Kabeer KA, Krishnapriyam SK. Ataxic form of central pontine myelinolysis. Ann Indian Acad Neurol 2007;10:112-4. [Full text]
Elhadd TA, Linton K, McCoy C, Saha S, Holden R. A hitherto undescribed case of cerebellar ataxia as the sole presentation of thyrotoxicosis in a young man: A plausible association. Ann Saudi Med 2014;34:440-3.
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