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Year : 2022  |  Volume : 31  |  Issue : 2  |  Page : 382-383  Table of Contents     

A case of restless legs Syndrome – Ill-ness or ill-spirit?

Department of Psychiatry, INHS Dhanvantari, Port Blair, Andaman and Nicobar Islands

Date of Submission30-Oct-2020
Date of Decision08-Jan-2021
Date of Acceptance24-Jul-2021
Date of Web Publication05-Jan-2022

Correspondence Address:
Dr. Neha Sharma
Department of Psychiatry, INHS Dhanvantari, Port Blair

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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/ipj.ipj_213_20

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How to cite this article:
Sharma N. A case of restless legs Syndrome – Ill-ness or ill-spirit?. Ind Psychiatry J 2022;31:382-3

How to cite this URL:
Sharma N. A case of restless legs Syndrome – Ill-ness or ill-spirit?. Ind Psychiatry J [serial online] 2022 [cited 2023 Feb 4];31:382-3. Available from: https://www.industrialpsychiatry.org/text.asp?2022/31/2/382/335029

Restless legs syndrome (RLS) is a neurological sensory - motor disorder, first described in 1672 by Sir Thomas Willis, an English physician.[1],[2] The term RLS was first introduced by Karl A Ekbom, a Swedish neurologist and surgeon, in 1945.[2] It is characterized by intense restlessness and unpleasant creeping sensations deep inside the legs, appearing when the legs are at rest and worst at night. They force patients to keep moving their legs, to get out of bed and wander about.[3] Periodic limb movements (PLMS) are also common during sleep among those suffering from RLS, as PLMS and dopaminergic spinal flexor reflexes share a common spinal origin, and disinhibition of reticulospinal excitatory responses may lead to pathological recruitment of spinal motor neurons.[2] Sleep efficiency is severely reduced.

The prevalence of RLS in general population is 2.5%–15%. Women have higher prevalence.[4] There are idiopathic as well as symptomatic forms of RLS, the latter being associated with pregnancy, iron deficiency, hypothyroidism, diabetes mellitus, Rheumatoid arthritis (RA), Sjogren's Syndrome, and chronic renal failure. The issue of coexistence of RLS and Parkinson's disease is controversial and currently being investigated. Nonfamilial cases are usually associated with neuropathy.

A family history of RLS is very common, with AD transmission, high penetrance, onset before 40 years of age, loci on chromosomes 12q, 14q, 9p, 2q, 20p and 16p and comorbidity with spinocerebellar ataxia type 3 (assuming a possible role of CAG repeat sequences in the SCA 3 gene).[2]

Dopamine dysfunction within the nigrostriatal system has been implicated by findings such as reduced striatal D2 receptor binding suggesting postsynaptic dopaminergic dysfunction during sleep, significant decrease in striatal 18F-DOPA uptake, state dependent decrease in cerebral blood flow in the caudate nuclei and increase in the anterior cingulate gyrus during increasing pain level, possible cerebral generators underlying sensory leg discomfort and PLMS, bilateral cerebellar and contralateral thalamic activation during sensory leg discomfort, and additional activation of the red nucleus and brain stem during combined sensory leg discomfort and PLM.[2]

Differential diagnosis includes akathisia, panic attacks, painful legs, and moving toes syndrome (rare, not relieved by movement), “Vesper's curse,” polyneuropathies, meralgia paresthetica, sleep onset myoclonus, and nocturnal myoclonus. Investigations include polysomnography to record frequency of PLMs, Actigraphy and immobilization tests.[2]

Dopaminergic therapy is the treatment of choice. Anticonvulsants may be used as second-line treatment. Levodopa should be reserved for those patients who fail to respond to alternative medications because of the high risk of inducing augmentation. Hypnosedatives also have a role in RLS management. Patients with intractable RLS may require combination treatment.[1] Nonpharmacological measures include sleep hygiene and avoidance of stimulants or aggravating drugs (for example, caffeine, alcohol, antihistamines, and certain antidepressants). In physiological conditions such as pregnancy, symptoms may resolve after delivery. In iron deficiency anemia, iron supplementation should be given first.[2]

A 46-year-old married woman from rural Haryana, mother of two, homemaker by occupation, with no known physical or psychiatric illness, no family history of psychiatric illness or substance abuse, premorbidly well-adjusted, was brought by her son, with complaints of long-standing (18 years) history of sleep disturbance. During the last trimester of her second pregnancy, she had noticed a strange, creeping sensation in both her legs while lying in bed at night, described as if something was gnawing her flesh. This would be temporarily relieved on moving, stretching, or massaging the legs. She found it difficult to sleep for more than 4–5 h a night and had to keep stretching or massaging her calves, feeling tired the whole day. One night, she heard her name being called by a female voice, and started believing that there was an evil power trying to harm her and her unborn child. She blamed a female acquaintance in her village, whom she suspected to be jealous of her and to have cast a black magic spell on her. Fearfulness further aggravated sleep disturbance. She approached the local faith-healer in her village, who gave her a black ribbon to tie around her arm, and reassured her. Within a few days of this, she delivered a healthy male child, and her abnormal sensations also subsided, which reaffirmed her faith in the existence of evil spirits, black magic and their remediation.

After a year, the sensations returned, this time affecting her during daytime napping as well. She approached the same faith-healer, to no avail, tried another Guru, was advised a series of religious ceremonies to ward off the evil spirit, tried yet another faith-healer in a distant town, and even stayed in an ashram for 2 months to undergo a “purification process.” None of these proved to be of any sustained benefit to her, despite her spending large amounts of money and 10 years of time on these practices. Her symptoms would wax and wane, but would never completely subside. Her son finally took her to a general practitioner, who then referred her for psychiatric treatment as a possible case of Psychosis.

On presentation, her general and systemic examination was within normal limits. Mental status examination revealed an anxious woman with psychomotor agitation, animated speech, anxious mood and affect, thinking dominated by superstitious explanation for her abnormal experiences, hypnagogic hallucinations in a clear sensorium with preserved insight and deranged biodrives in the form of initial insomnia and reduced energy. Investigations revealed no features of RA, uremia, diabetes, thyroid abnormalities, anemia, or any other metabolic abnormality.

She was managed as a case of RLS with pramipexole, initially started at 0.25 mg HS and after 2 weeks, increased to 0.5 mg HS, with a short course (2 weeks) of zolpidem 10 mg HS, to which she responded quickly and satisfactorily. Her culture-based superstitious beliefs and associated distress was addressed in a nonconfrontational manner, gradually integrating her beliefs with psychoeducation about the illness. Family support was solicited to facilitate the implementation of sleep hygiene measures and to ensure continuity in treatment and rehabilitation. At 6 months' follow-up, she was in remission, motivated for adherence with treatment and fully functional in socio-occupational spheres.

In this case, the onset of symptoms was during pregnancy with spontaneous remission after delivery, which is a known occurrence in RLS, as is female gender. However, relapse after a year and persistent symptoms for more than a decade, along with the absence of family history of RLS makes it an atypical presentation.

Pramipexole has been reported in literature to be effective in the dose range of 0.25–0.75 mg per day,[5] which was successfully used in this case.

Being a neurological illness rarely treated by psychiatrists, the role of psychotherapy in the management of RLS has not been given due importance. In this case, since the cultural beliefs and attitude of the patient were so closely intertwined with her symptoms, it was of utmost necessity to address these issues in therapy in a nonconfrontational manner, so as to psycho-educate her about her illness and to enhance acceptance towards medical treatment, which was a crucial factor in ensuring adherence in the long-term.

In conclusion, cultural factors play a role in determining patient's attitude toward and explanation of their illness, as well as time lapse before seeking treatment and subsequent adherence with it. Hence, cultural factors should invariably be addressed in therapy even for disorders that are seemingly purely neurological.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient has given her consent for her clinical information to be reported in the journal. The patient understands that name and initials will not be published and due efforts will be made to conceal identity, but anonymity cannot be guaranteed.

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Conflicts of interest

There are no conflicts of interest.

   References Top

Schapira AH. Restless legs syndrome: An update on treatment options. Drugs 2004;64:149-58.  Back to cited text no. 1
Chaudhuri KR, Appiah-Kubi LS, Trenkwalder C. Restless legs syndrome. J Neurol Neurosurg Psychiatry 2001;71:143-6.  Back to cited text no. 2
Ekbom K, Ulfberg J. Restless legs syndrome. J Intern Med 2009;266:419-31.  Back to cited text no. 3
Earley CJ. Restless legs syndrome. N Engl J Med 2003;348:2103-9.  Back to cited text no. 4
Montplaisir J, Denesle R, Petit D. Pramipexole in the treatment of restless legs syndrome: A follow-up study. Eur J Neurol 2000;7:27-31.  Back to cited text no. 5


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