Home About us Editorial board Ahead of print Current issue Archives Submit article Guidelines Contacts Login 
ISSN: Print -2349-0977, Online - 2349-4387


 
 Table of Contents  
CASE IN POINT: NEUROLOGY
Year : 2015  |  Volume : 2  |  Issue : 1  |  Page : 52-53

Hyper somnolence in Kleine-Levin syndrome secondary to tuberculous meningitis


Metro Center for Respiratory Diseases, Metro Multispeciality Hospital, Sector 11, Noida, Uttar Pradesh, India

Date of Web Publication26-Oct-2015

Correspondence Address:
Dr. Arjun Khanna
Metro Center for Respiratory Diseases, Metro Multispeciality Hospital, Sector 11, Noida, Uttar Pradesh
India
Login to access the Email id

Source of Support: None, Conflict of Interest: None


DOI: 10.4103/2349-0977.168240

Rights and Permissions

How to cite this article:
Khanna A, Sinha AK, Periwal P, Talwar D. Hyper somnolence in Kleine-Levin syndrome secondary to tuberculous meningitis. Astrocyte 2015;2:52-3

How to cite this URL:
Khanna A, Sinha AK, Periwal P, Talwar D. Hyper somnolence in Kleine-Levin syndrome secondary to tuberculous meningitis. Astrocyte [serial online] 2015 [cited 2022 Jul 3];2:52-3. Available from: http://www.astrocyte.in/text.asp?2015/2/1/52/168240

Kleine–Levin syndrome (KLS) has been described in literature as a rare cause of recurrent hypersomnia, hyperphagia, hypersexuality, and behavior abnormalities, usually seen in adolescents and teenagers, without any definite etiology or risk factors. Around 10 cases of KLS have been reported from India, most of which have been in the pediatric population and were idiopathic. We present a case of KLS secondary to tuberculous meningitis (TBM). This association has been rarely described earlier.[1]

An 18-year-old female presented with fever and headache for the past 3 weeks. She was admitted to the emergency ward with seizures and altered sensorium. After stabilization, neuroimaging and cerebrospinal fluid (CSF) examination was done. This was suggestive of TBM. The CSF protein level was raised (64 mg/dl), sugar (58 mg/dl). CSF cell count was 68 cells/cumm, mainly lymphocytes (95%), CSF adenosine deaminase was raised 22 IU/L, and CSF polymerase chain reaction (PCR) for Mycobacterium tuberculosis was positive. The patient was started on antituberculous therapy with Isoniazid, Rifampicin, Pyrizinamide, Ethambutol, and Streptomycin along with oral prednisolone. She improved on therapy and was discharged in 2 weeks time without any residual neuro deficit.

Around 1 month postdischarge, the patient complained of lethargy, apathy, and increased somnolence. There was history of using foul abusive language and masturbation in front of family members. The patient underwent repeat magnetic resonance imaging (MRI) brain, which was normal. The treating physician made a diagnosis of Isoniazid psychosis and the drug was withheld. Over the next week, the lethargy increased and the patient was sleeping over 18 h every day.

On presentation to our sleep clinic, we saw a thin female with mild pallor. She was drowsy, but arousable. Rest of the clinical examination was normal. She would spend most of her time in the ward sleeping. When the patient was not sleeping, she would be agitated and use offensive language and masturbate in front of others. There was no hyperphagia, seizures, weight gain, or history of intoxication.

A provisional diagnosis of KLS was made on the basis of these clinical features. Routine blood parameters were normal. Repeat Spinal tap revealed a normal CSF report. Electroencephalograph was normal and did not show any evidence of seizures. Polysomnography was done and it was essentially normal. The patient was treated with intravenous fluids, deep venous thrombosis prophylaxis, and intravenous parenteral nutrition. Over the next week, the hypersomnolence decreased and the total sleep period went down. Around 3 weeks after presentation, the sleep–wake cycle was normal and hypersexuality also subsided. The patient had partial amnesia about the whole event. However, she continued to have a labile effect and was given lithium by a psychiatrist for the same. Three months after this episode, the patient was readmitted with similar complaints, but this time the hypersomnolence–hypersexuality cycle lasted only for 5 days, after which the patient made near complete recovery and was discharged. Presently, the patient is doing well and continues to be on oral lithium for the mood disturbance. She has completed one year of anti-TB therapy and has no major neuro-cognitive dysfunction.

KLS was first described by Kleine in 1925 and elaborated on by Levin in 1936.[2] In 1990, KLS was classified by the International Classification of Sleep Disorders (ICSD). It was revised to its current state in 2005 with the release of the ICSD-revision 2 (ICSD-2).[3] KLS is rare, affecting an estimated 1–5 per million individuals.[4] The exact etiology of KLS is not known. Some report a prodromal flu-like illness or coryza prior to the onset of KLS.[5]

The symptoms of KLS are intermittent and periodic. The essential diagnostic criteria includes the presence of recurrent hypersomnolence with any of these features: Hyperphagia, hypersexuality, abnormal behavior, cognitive dysfunction.[3] Our patient had hypersomnolence with hypersexuality and behavior disturbance. During episodes, patients have excessive day-time sleepiness, despite sleeping anywhere from 12 to 21 h per day. The classic triad of hypersomnia, hyperphagia, and hypersexuality are present in only up to 45% of patients presented.[6] The differential diagnosis of KLS can include drug or alcohol use, temporal lobe epilepsy, Klüver–Bucy syndrome secondary to bilateral temporal lobe lesions, mass lesions, metabolic encephalopathies, such as mitochondrial disease or urea cycle defects, Lyme disease, and acute intermittent porphyria.[7] No medication has been convincingly demonstrated to be efficacious in the treatment of KLS.[8] Apart from supportive management, multiple pharmacological agents such as modafinil, lithium, valproicacid have been tried with varying success. Educating attendants and refraining from activities that can endanger the life of the patient or others around them seems prudent.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
  References Top

1.
Thacker AK, Aeron A, Haider J, Rao KM. Kleine-Levin syndrome in tubercular meningitis. Ann Indian Acad Neurol 2007;10:270-1.  Back to cited text no. 1
  Medknow Journal  
2.
Critchley M, Hoffman HL. The syndrome of periodic somnolence and morbid hunger (Kleine-Levinsyndrome). Br MedJ 1942;1:137-9.  Back to cited text no. 2
    
3.
American Acade my of Sleep Medicine. International Classification of Sleep Disorders: Diagnostic and Coding Manual. 2nd ed. Westchester, IL: American Academy of Sleep Medicine;2005.  Back to cited text no. 3
    
4.
Frenette E, Kushida CA. Primaryhy persomnias of centralorigin. Semin Neurol 2009;29:354-67.  Back to cited text no. 4
    
5.
Huang YS, Guilleminault C, Lin KL, Hwang FM, Liu FY, Kung YP. Relationship between Kleine-Levinsyndrome and upperre spiratory infectionin Taiwan. Sleep 2012;35:123-9.  Back to cited text no. 5
    
6.
Arnul fI, Lin L, Gadoth N, File J, Lecendreux M, Franco P, et al. Kleine-Levinsyndrome: a systematic study of 108 patients. Ann Neurol 2008;63:482-93.  Back to cited text no. 6
    
7.
Miglis MG, Guilleminault C. Kleine-Levinsyndrome: areview. NatSci Sleep 014;6:19-26.  Back to cited text no. 7
    
8.
Huang YS, Lakkis C, Guilleminault C. Kleine-Levinsyndrome: current status. Med Clin North Am 2010;94:557-62.  Back to cited text no. 8
    



This article has been cited by
1 Periodic hypersomnolence in a young female patient: a case report and literature review
Zahra Banafsheh Alemohammad, Sajedeh Jamshidi, Arezu Najafi
The Egyptian Journal of Neurology, Psychiatry and Neurosurgery. 2022; 58(1)
[Pubmed] | [DOI]



 

Top
 
 
  Search
 
Similar in PUBMED
   Search Pubmed for
   Search in Google Scholar for
Access Statistics
Email Alert *
Add to My List *
* Registration required (free)

 
  In this article
References

 Article Access Statistics
    Viewed2710    
    Printed58    
    Emailed0    
    PDF Downloaded163    
    Comments [Add]    
    Cited by others 1    

Recommend this journal


[TAG2]
[TAG3]
[TAG4]