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A rare case of partially empty sella with epileptiform discharges presenting with first episode of mania


 Department of Psychiatry, D. Y. Patil Medical College, D Y Patil Education Society (Deemed University), Kolhapur, Maharashtra, India

Date of Submission13-Jul-2022
Date of Acceptance22-Aug-2022
Date of Web Publication22-Sep-2022

Correspondence Address:
Ankit Halder,
Department of Psychiatry, D. Y. Patil Medical College D Y Patil Education Society (Deemed University), Kolhapur, Maharashtra
India
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/ipj.ipj_122_22



How to cite this URL:
Halder A, Ravindran NP, Keshri SM, Harshe G. A rare case of partially empty sella with epileptiform discharges presenting with first episode of mania. Ind Psychiatry J [Epub ahead of print] [cited 2022 Nov 29]. Available from: https://www.industrialpsychiatry.org/preprintarticle.asp?id=356694



Sir,

Busch described the condition empty sella syndrome (ESS) in 1951 where the sella turcica is filled with cerebrospinal fluid (CSF) causing displacement or flattening of the normal pituitary gland.[1] It is reported to occur in 5.5%–23.5% of the population[2] which is pathophysiologically characterized by either anatomic abnormalities in the diaphragma sellae (primary ESS) or damage to the pituitary by irradiation/surgery, or autoimmunity leading to the availability of “empty” space in the sella (secondary ESS).[1] The common signs and symptoms includes headache, papilledema due to raised intracranial pressure, leakage of CSF through nose and weight gain.[1] It can also present as decreased libido, galactorrhea, amenorrhea and secondary infertility.[3] Psychiatric manifestations of ESS are rare but can occur in the form of behavioral disturbances and psychotic symptoms.[4],[5]

A 25-year-old male presented to us with violent behavior towards family members, reduced need for sleep, elevated self-esteem and grandiose delusions claiming himself to be the prime minister of India. It was associated with one episode of tonic–clonic movements two nights back, with urinary incontinence. Patient also complained of jerky movements in his left arm occasionally about two to three times a day following this. All the symptoms started abruptly for past 10 days, this being his first such episode in his lifetime. There was no history of any febrile illness, head injury, substance abuse; blood parameters that included serum electrolytes, serum prolactin, vitamin B12 levels, and serum osmolality were all within normal limit. He was diagnosed to be in mania as per Diagnostic and Statistical Manual of Mental Disorders (DSM 5). Young's Mania Rating Scale (YMRS) score was 24. The patient was subjected to magnetic resonance imaging (MRI) scan which revealed empty sella [Figure 1]. 10–20 electroencephalogram findings revealed generalized periodic epileptiform discharges (GPED). The patient was placed on sodium valproate 1000mg, haloperidol 10 mg and trihexyphenidyl 4 mg in divided dosages. The symptoms reduced in severity and YMRS score came 12 on day 5 of admission and EEG was normal. This was his first episode of mania that coexisted with first lifetime episode of seizure. GPEDs results in kindling of neuronal pathways, leading to psychiatric manifestations such as mania. Lateral hypothalamic kindling can induce manic-like behavior in a rat model.[5]
Figure 1: MRI scan showing empty sella

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There are reports of atypical presentations of ESS in the form of acute psychosis, sinus bradycardia, herniation of anterior cerebral artery.[5],[6],[7] The presentation of empty sella as seizure episode has been reported rarely.[8] Moreover, it is even more rare to be presenting with both seizure episode and mania.[9] In one case report mania was precipitated by hyponatremia in a patient with ESS[10] whereas in our case sodium and other electrolytes level were in normal range. The possible mechanism of such is still a gray area.[9] Even there is also a dilemma of seizure precipitating mania or vice-versa.

More sample based studies is needed to establish the neurochemical events leading to the same for better diagnosis and management.

Ethical concerns

Proper consent was taken from the patient regarding publication of the case without revealing patient's identity

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
Sachdev Y, Evered DC, Appleby A, Hall R. The empty sella syndrome. Postgrad Med J 1976;52:703-5.  Back to cited text no. 1
    
2.
Jordan RM, Kendall JW, Kerber CW. The primary empty sella syndrome: Analysis of the clinical characteristics, radiographic features, pituitary function and cerebrospinal fluid adenohypophysial hormone concentrations. Am J Med 1977;62:569-80.  Back to cited text no. 2
    
3.
Ekhzaimy AA, Mujammami M, Tharkar S, Alansary MA, Al Otaibi D. Clinical presentation, evaluation and case management of primary empty sella syndrome: A retrospective analysis of 10-year single-center patient data. BMC Endocr Disord 2020;20:142.  Back to cited text no. 3
    
4.
Bardoloi PS, Abba-Aji A. A case of empty Sella syndrome presenting with neuropsychiatric symptoms. J Neurol Neurosci 2017;8:197.  Back to cited text no. 4
    
5.
Singhai A, Kapur K, Khadanga S, Paramanandam A. Acute psychoses: An unusual presentation of empty sella syndrome. CHRISMED J Health Res 2019;6:187-90.  Back to cited text no. 5
  [Full text]  
6.
Braiteh N, Breiteh N, Kashou HE. Sinus bradycardia as a rare and unusual presentation of partial empty sella syndrome: A case report. Am J Case Rep 2019;20:1071-4.  Back to cited text no. 6
    
7.
Sivaraju L, Thakar S, Hegde AS. Visual deterioration and herniation of the anterior cerebral artery: Unusual presentation of an empty sella syndrome complicating decompression of a rathke cleft cyst. J Neuro-Ophthalmol 2016;36:156-8.  Back to cited text no. 7
    
8.
Sethuraman VK, Viswanathan S, Aghoram R. Refractory hypoglycemia and seizures as the initial presenting manifestation of empty sella syndrome. Cureus 2018;10:e2803. doi: 10.7759/cureus.2803.  Back to cited text no. 8
    
9.
Das N, Agarwal K, Prashad S. Partially empty sella with generalized epileptiform discharges presented as manic episode in an elderly female: A case with diagnostic dilemma–Causation or coincidence? Indian J Psychiatry 2021;63:298-300.  Back to cited text no. 9
  [Full text]  
10.
Yang CH, Lin YC, Chou PH, Chen HC, Chan CH. A case report of late onset mania caused by hyponatremia in a patient with empty sella syndrome. Medicine 2016;95:e2629. doi: 10.1097/MD.0000000000002629.  Back to cited text no. 10
    


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