Retrospective Study Open Access
Copyright ©The Author(s) 2025. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Methodol. Mar 20, 2025; 15(1): 97171
Published online Mar 20, 2025. doi: 10.5662/wjm.v15.i1.97171
Restless head syndrome: A retrospective study
Sanjay Prakash, Varoon Vadodaria, Chetsi S Shah, Department of Neurology, Smt. B. K. Shah Medical Institute and Research Centre, Sumandeep Vidyapeeth, Vadodara 391760, Gujarāt, India
Niraj Chawda, Department of Medicine, Smt. B. K. Shah Medical Institute and Research Centre, Sumandeep Vidyapeeth, Vadodara 391760, Gujarāt, India
Anurag Prakash, Department of Medicine, Parul Institute of Medical Sciences and Research Centre, Parul University Waghodia, Vadodara 391760, India
ORCID number: Sanjay Prakash (0000-0001-7322-904X); Varoon Vadodaria (0000-0003-3959-9367); Niraj Chawda (0000-0002-8624-000X); Chetsi S Shah (0009-0001-9727-4440); Anurag Prakash (0009-0000-7611-5080).
Author contributions: Prakash S and Vadodaria V were involved in the conceptualization of the project, literature review, statistical analysis, and writing the original draft; Chawda N, Shah C, and Prakash A were involved in the literature review, analysis and interpretation of data, and revising the manuscript for intellectual content; all authors read and approved the final version of the manuscript.
Institutional review board statement: The Institutional Ethics Committee approved the study (SVIEC/ON/MEDI/RP/MARCH/24135).
Informed consent statement: Written informed consent was obtained from all patients to publish this observation.
Conflict-of-interest statement: All the authors declare no conflict of interest for this article.
Data sharing statement: Anonymized data not presented herein is available upon reasonable request from the corresponding author on rational request by any qualified researcher.
Open-Access: This article is an open-access article that was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution NonCommercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See: https://creativecommons.org/Licenses/by-nc/4.0/
Corresponding author: Sanjay Prakash, DM, Professor, Department of Neurology, Smt. B. K. Shah Medical Institute and Research Centre, Sumandeep Vidyapeeth, Piparia, Waghodia, Vadodara 391760, Gujarāt, India. drprakashs@yahoo.co.in
Received: May 24, 2024
Revised: July 7, 2024
Accepted: July 15, 2024
Published online: March 20, 2025
Processing time: 127 Days and 18.9 Hours

Abstract
BACKGROUND

Restless legs syndrome (RLS) is characterized by an urge to move with an unpleasant sensation in the lower limbs. RLS typically affects the legs. However, it can also affect several other body regions, such as the arms, abdomen, face, neck, head, and genital area. There are only a few reports of the RLS variant affecting the head.

AIM

To assess the epidemiological, clinical, and other aspects of the RLS variant affecting the head.

METHODS

We conducted a retrospective study of 17 adult patients (> 18 years) who met the RLS criteria and simultaneously experienced RLS-like symptoms in the head.

RESULTS

The median age at which symptoms appeared was 41.6 years. Males and females were equally affected (1.1:1). All 17 patients had uncomfortable sensations in the lower legs. Insomnia or disturbed sleep was the most common comorbidity (n = 16, 88.2%). However, headache was the most common presenting or primary symptom (n = 10, 70.5%). Dizziness or an abnormal sensation in the head was the second most common presenting symptom (5 patients, 29.4%). Other presenting features were leg pain, backache, and generalized body pain. All patients responded favorably to dopaminergic medications.

CONCLUSION

If RLS-related unpleasant sensations and pain are felt in the head, they may be misinterpreted as headache, dizziness, or psychosomatic symptoms. RLS and headaches in a subset of patients may be two phenotypic manifestations of the same disorder.

Key Words: Restless legs syndrome; Restless head syndrome; Dopamine; Headache; Migraine; Tension-type headache

Core Tip: Restless head syndrome is an evolving concept. If restless legs syndrome (RLS)-like symptoms are experienced in the head, they may be misinterpreted as headache, dizziness, or psychosomatic symptoms. We suggest that headaches in a subgroup of RLS patients might be part of the RLS spectrum. Such patients may respond to dopaminergic medications.



INTRODUCTION

Restless legs syndrome (RLS) is a common but often misdiagnosed sensorimotor disorder. It is characterized by an irrepressible urge to move the legs, typically accompanied by unpleasant and uncomfortable sensations in the legs. RLS typically affects the legs, but it can also affect various other body regions, including the arms, abdomen, face, head, neck, and genital area[1,2]. The unpleasant sensations include twitchy, itchy, burning, current-like, ants crawling, creeping, throbbing, tight feeling, prickling, soda-bubbling, restlessness, and pain. About 30% to 50% of RLS patients described pain as a primary component[2]. If RLS-related unpleasant sensations and pain are felt in the head, they may be misinterpreted as dizziness, headache, or psychosomatic symptoms. A few authors have shown that dopamine agonists can relieve headaches and dizziness in RLS patients[3,4]. It is termed restless head syndrome[3,5]. This study focused on 17 patients who not only experienced classic RLS symptoms but also reported pain and other unspecified symptoms in the head. Both RLS and head-related symptoms improved after treatment with dopamine agonists.

MATERIALS AND METHODS

We present data on patients who, in addition to typical RLS symptoms, had pain or other non-specific head symptoms and were given a dopamine agonist. The inclusion criteria were as follows: (1) Age > 18 years; (2) patients who met the International Restless Legs Syndrome Study Group (IRLSSG) criteria for RLS; (3) RLS-like symptoms in the head; (4) patients who received dopamine agonists; and (5) a minimum follow-up of more than 4 wk. Our team has been working on RLS for several years. In clinical practice, we noticed that medications started for RLS elicited a response with regard to headache, dizziness, and other nonspecific symptoms of the head. As a departmental policy, we routinely subject every patient with RLS and other head-related symptoms to a thorough clinical history and physical examination. So, all of the data reported here was collected prospectively. All such patients are initially treated with a dopamine agonist alone. The participants were seen in the neurology department of a tertiary hospital between January 2021 and February 2024. The exclusion criteria included: (1) Age < 18 years; (2) patients who did not receive any dopamine agonist; (3) a follow-up of less than 4 wk; (4) patients who did not give consent; and (5) patients having a history of any comorbid conditions, including diabetes mellitus, hypertension, and thyroid disorders. We also excluded the patients who met the migraine criteria. The Institutional Ethics Committee approved the study (SVIEC/ON/MEDI/RP/MARCH/24135). The study was conducted according to the Helsinki Declaration principles.

Statistical analysis

Continuous data are expressed as the mean ± SD, and dichotomous variables are expressed as frequencies and percentages. Anonymized data not presented herein is available upon reasonable request from the corresponding author on rational request by any qualified researcher.

RESULTS

We found 24 patients who met the inclusion criteria. Seven patients were excluded for the following reasons: Follow-up less than 4 wk (n = 2), those who did not give consent (n = 1), and a history of associated diseases (n = 4; diabetes mellitus-2 patients, hypertension-1, and hypothyroidism-1). Because cases of restless head syndrome are not well documented in the literature, we are providing details about three patients. The clinical characteristics of the remaining 14 patients are summarised in Table 1. Table 2 summarises the clinical, epidemiological, and other features of all 17 cases.

Table 1 Details of patients with restless head syndrome.
Age/sex
Presenting or chief complain
Duration of illness
Head-related symptoms
Symptoms/sensations of RLS in legs
Arms-back involvement
Insomnia
Lab abnormalities
Treatment
Symptoms
Severe in night
Relief by massage
Dopaminergic drugs
Response
56; FHeadache15 moTight feeling, stretchingYesYesBurning, stretching feelingArmsYesLow ferritin levelPramipexole 0.250 mg ODMarked response in 5-6 d
45; MDizziness3 yearsWorms moving, crawlingYesYesPain, crawlingArms, backYesRopinirole 0.50 mg ODComplete response in 2 wk
34; FHeadache2-3 yearsTight feeling, stretchingYesYesTight feeling, stretchingYesPramipexole 0.125 mg ODMarked response in 2 wk
52; FLeg pain5-6 yearsTight feeling, tearingYesYesTearingArms, BackYesRopinirole 0.50 mg ODMarked response in 10 d
45; MDizziness2 yearsCreeping, floatingYesYesThrobbing, crawlingArmsYesPramipexole 0.250 mg ODMarked response in 7-8 d
34; FHeadache2-3 yearsTight feelingYesYesCreeping, painYesRopinirole 0.25 mg ODComplete response in 2 wk
34; FHeadache, leg pain6-7 moTight feeling, stretchingYesYesPain, stretching, burningArmsYesLow serum ironLevodopa/carbidopa (100/10 mg)Marked response in 10 d
25; MHeadache, backache5-6 moThrobbing, tight feelingYesYesPain, burning SensationArms, backYeslow serum ironPramipexole 0.250 mg ODMarked response in 2 wk
45; FHeadache, backache6 moThrobbing, stretchingYesYesBurning, stretchingYesRopinirole 0.50 mg ODMarked response in 1-2 wk
46; MLeg pain, headache4-5 yearsStretching sensation, tight feelingYesYesPain, stretching, throbbingArms, backYeslow serum ironlevodopa/carbidopa (100/10 mg)Marked response in 2 wk
56; MHeadache, dizziness10 moJittery, twitching, numbnessYesYesTight feeling, pain, itchy sensationArmsYesLevodopa/carbidopa (100/10 mg)Complete response in 1-2 wk
32; FHeadache4-5 yearsTight feeling, stretchingYesYesTearing, ants crawlingArms, backLevodopa/carbidopa (100/10 mg)Marked response in 5-6 d
46; MHeadache, dizziness2-3 yearsCreeping, stretchingYesYesPulling, stretchingArms, backYesLow vitamin DPramipexole 0.250 mg ODMarked response in 10 d
54; FHeadache6-7 yearsTight feeling, stretchingYesYesCreeping, painYesPramipexole 0.250 mg ODComplete response in 2 wk
Table 2 Epidemiological profiles and clinical features of 17 patients with restless head syndrome.
Parameter
N (17) (%)
Age (years) (mean ± SD)41.6 (± 9.6)
Sex (Male:Female)9:8 (1.2:1)
Duration of illness (mo)53.9 ± 26.2
Presenting features or chief complaints
Headache12 (70.5)
Dizziness5 (29.4)
Leg pain3 (17.3)
Backache2 (11.8)
Generalized body pain2 (11.8)
Clinical features
Lower legs discomforts17 (100)
Headache14 (82.3)
Arms discomforts12 (70.5)
Backache6 (35.2)
Dizziness5 (29.4)
Generalized body pain3 (17.3)
Insomnia16 (88.2)
Met international restless legs syndrome study group criteria17 (100)
Treatment (dopaminergic agents)
Pramipexole 0.125 to 0.250 mg daily8 (47.1)
Levodopa/carbidopa (100/10 mg)5 (29.4)
Ropinirole 0.50 mg daily4 (23.5)

The median age at which symptoms appeared was 41.6 years (range: 32-56 years). Males and females were equally affected (9 female and 8 male cases). All 17 patients had uncomfortable sensations in the lower legs and met the IRLSSG criteria for RLS. Insomnia or disturbed sleep was the most common comorbidity, affecting 16 patients (88.2%). Other common symptoms were headache (14 patients, 82.3%), arm discomforts (12 patients, 70.5%), backache (6 patients, 35.2%), dizziness or unpleasant sensations in the head (5 patients, 35.2%), and generalized body pain (3 patients, 17.3%). In this cohort, headaches accounted for the most common presenting or chief symptom (10 patients, 70.5%). Dizziness or an abnormal sensation in the head was the second most common presenting symptom (5 patients, 29.4%). Other presenting features were leg pain, backache, and generalized body pain.

The patients described their headache or head-related symptoms as a tight feeling, stretching sensation, worms moving, crawling, tearing, creeping, floating, throbbing, jittery, twitching, and numbness. No migranous features, such as nausea, vomiting, photophobia, or phonophobia, were noted with the headaches and other head-related symptoms. The character of headaches fulfilled the International Classification of Headache Disorders, third edition (ICHD-3) criteria of chronic tensiontype headache (CTTH). All patients experienced more intense symptoms in the evening and at night. Headaches and dizziness were also worse at night. All patients experienced relief from lower limb discomfort with leg massages or stretches. Similarly, head massage provided comfort to all patients suffering from headaches, dizziness, or abnormal sensations in the head.

Most of the laboratory workup was unremarkable; three patients had low blood iron levels, one had low ferritin levels, and one had a low vitamin D level. Every patient responded well to dopaminergic treatment. Pramipexole was administered to eight individuals, whereas levodopa/carbidopa (100/10 mg) and ropinirole (0.25-0.50 mg daily) were prescribed to five and four patients, respectively. Each patient showed a marked response within 1-2 wk. All patients were followed up for at least 4 wk (as an inclusion criterion). A total of 10 patients had been followed up for more than 6 mo. The response was consistent with the drugs. The symptoms reappeared in 12 individuals when the medication was discontinued or skipped. In all of these individuals, resuming the drug produced relief from the symptoms within a few days to a few weeks.

Case descriptions

Case-1: A 31-year-old man had a 5-year history of episodic headaches. In the early years of the illness, headaches occurred about 2-4 times each month. However, the frequency increased progressively over the years, with headaches occurring virtually every day in the prior year. The headache was holocephalic and felt like a tight band across the head. The headache would sometimes feel like it was burning and tearing. The pain mostly fluctuated from mild to moderate in intensity. The symptoms used to be more intense in the evenings and at night. Physical activity did not worsen the headache. He used to feel less or no headaches while engaging in physical activity. The patient never felt nausea, photophobia, or phonophobia. Massaging the pericranial muscles used to relieve headaches.

When questioned, the patient acknowledged having uncomfortable sensations (especially tearing-type pain) in both legs for 4-5 years. Initially, it happened infrequently (4-5 d each month). However, it progressed to every day during the previous 10 mo. The uncomfortable sensation persisted throughout the day, intensifying in the evening and at night. Moving or stretching his legs helped him feel better. In addition, he had been experiencing worsening insomnia for the previous 15 mo. He needed two to four hours to fall asleep. His sleep was not refreshing. The patient visited several doctors regarding his headache. Various preventive drugs were started over the last 2-3 years for the increasing frequency of headaches. He received amitriptyline, sodium valproate, topiramate, flunarizine, and venlafaxine, but none of these medications significantly improved his headaches. The patient never consulted a doctor about his lower limb issues, believing them to be the result of overwork or excessive physical activity. Physical and neurological examinations did not reveal any abnormalities. Biochemical parameters such as hemoglobin, serum iron, serum ferritin levels, thyroid function, renal function, and serum vitamin D levels were unremarkable. Magnetic resonance imaging (MRI) of the spine and brain did not reveal any abnormalities. Nerve conduction study (NCV) and electromyography (EMG) were reported to be normal.

The lower limbs’ symptoms met the IRLSSG criteria for RLS[1]. The IRLS score was 19, indicating a moderate form of the disease. The clinical characteristics of headaches fulfilled the ICHD-3 criteria for chronic tension-type headache[6]. Pramipexole (0.25 mg) was prescribed at bedtime. RLS began to improve within a few days of initiating medication, and a near-complete response was observed within 10 d. In parallel, there was a noticeable decrease in the frequency of headaches, with just 3-4 mild to moderate headache episodes noted over a 6-wk period. The patient was followed up for 15 mo, showing a consistent and nearly complete response. Whenever the drug was stopped or skipped, the RLS, headaches, and insomnia reappeared within a few days. Restarting the medication always resulted in relief from RLS, insomnia, and headaches.

Case-2: A 39-year-old man had a 4-year history of dizziness. Initially, the dizziness was intermittent and short-lasting. However, it gradually worsened, and now it has been present daily for the past 9 mo. He described his dizziness as ‘an abnormal sensation in the head’. When we showed him the list of abnormal sensations described for RLS, the patient mentioned the following sensations in his head: Buzzing, pulling, tight feeling, fullness, floating, and worms moving sensations. Although the abnormal head sensations persisted throughout the day, they were more intense when resting or relaxing, especially in the evening and at night. He could not sleep properly because of the uncomfortable sensations in his head. It takes him two to three hours to fall asleep. He also often woke up in the middle of the night. Massage of the pericranial muscles was used to relieve headache. When questioned, the patient acknowledged the presence of unpleasant sensations in both legs for several years. It was infrequent at first, occurring only 2-4 d each month. But for the previous two years, symptoms have been experienced around 10 to 15 d a month. Similar to the abnormal sensation of the head, the unpleasant sensation of the legs was felt as a combination of a stretching sensation, a tight feeling, and diffuse pain. Walking, massaging, and stretching the legs all helped to reduce or alleviate leg discomfort. The patient used to do some activities at night to get relief from abnormal sensations in the legs and head.

The patient visited several physicians regarding his dizziness and abnormal sensations in the head. He received several provisional diagnoses over 3-4 years: Psychogenic dizziness, vestibular migraine, persistent postural perceptual dizziness, and somatic symptom disorder. The patient received a variety of medications and strategies over the years. But none of the medications had any noticeable effects on his condition. Physical and neurological examinations did not reveal any abnormalities. All laboratory tests, including complete blood cell count, serum chemistry, thyroid function test, kidney function test, iron studies, liver function test, serum vitamin D, serum vitamin B12 levels, nerve conduction velocity, and magnetic resonance imaging of the brain and spine, were normal. The lower limbs’ symptoms met the IRLSSG criteria for RLS.

We gave a trial of levodopa/carbidopa (100/10 mg) for RLS. The patient noted a marked improvement in both RLS and dizziness, and the abnormal sensations of the head, within 1-2 wk. The patient had never experienced such relief from any medication for dizziness before. Three months later, the medication was withdrawn. However, the symptoms reappeared after just a few days. Restarting the medication provided complete relief from RLS and dizziness. During a 9-mo follow-up period, the patient demonstrated a consistent and complete response.

Case-3: A 42-year-old woman reported diffuse body pain, including headaches, for about 8 years. The pain initially only affected the legs. The pain spread over time and began to affect additional areas of the body. Although the patient was still experiencing more discomfort in her legs, she was also experiencing pain in her head, neck, arms, scapular area, buttocks, and upper and lower back. For the preceding 2 to 3 years, she had pain all over her body, with the exception of the abdomen and chest.

Initially, the pain was infrequent, 4-5 d in a month. The frequency gradually increased over the years. The pain had been present most of the time for 2 to 3 years. It was felt as a combination of a constant dull ache, a stretching feeling, and a burning sensation. The back pain was frequently accompanied by itchy sensations. The pain was deep-seated and restricted to the muscles. She denied pain in the joints. The diffuse body pain and headaches were mild at the beginning of the day, increased towards the end of the afternoon, and became more severe at night. She used to have less discomfort while engaging in activities. The pain prevented her from falling asleep. In addition, she would often wake up in the middle of the night. Because of the pain and poor sleep, she used to experience anxiety during the night. Her spouse attested to her restlessness during sleep, exhibiting frequent tossing, twisting, and excessive turning in bed. Sometimes, to relieve her discomfort at night, she had to get out of bed to stretch her limbs or take a walk. She used to stretch her back to get relief from back pain and itching.

Along with her generalized body pain and abnormal sensations in the body, she also reported experiencing headaches and dizziness. She described headaches and dizziness as part of her generalized body pain. Headaches and dizziness were felt as a combination of a stretching sensation, a tight feeling, a floating sensation, and diffuse pain. Similar to the abnormal sensation of the legs and back, headache and dizziness were more severe towards the end of the afternoon and at night. Massage of pericranial muscles was used to relieve headache.

The patient visited several physicians over eight years, including rheumatologists, neurologists, and psychiatrists. She received several diagnoses, including fibromyalgia, vitamin D deficiency, somatic symptom disorder, sleep disorders, and other psychiatric disorders. The patient got a variety of drug regimens (antidepressants, anxiolytics, and multivitamins) and strategies for her illness, including cognitive behavioral therapy. She received duloxetine, amitriptyline, milnacipran, pregabalin, gabapentin, fluoxetine, venlafaxine, ibuprofen, naproxen, indomethacin, and multivitamins. A few of them, especially pregabalin and gabapentin, had some effects on pain. However, none of the therapies had any persistent effects. Physical, rheumatological, and neurological examinations were essentially normal. The patient had undergone numerous investigations over the years. Hematologic and biochemical investigations were unremarkable except for low serum iron (34 mcg/dL). A non-contrast MRI scan of the head and spine showed no abnormalities. EMG and nerve conduction studies were normal.

The lower limbs’ symptoms met the IRLSSG criteria for RLS. Pramipexole was started at a dose of 0.125 mg at bedtime. We advised the patient to increase the dose by 0.125 mg every 3 d to 0.5 mg daily. The patient noted a response on the first day of treatment. The response was observed not only in the leg pain, but also in backache, neck pain, and headache. Her sleep pattern also improved. The generalized body aches almost completely disappeared after 10 d of therapy. Sleep patterns also returned to normal, and she could fall asleep in 10-20 min. The patient had never experienced this kind of response from a medication before. We attempted to discontinue the medication after 5 mo. However, it resulted in the reappearance of body pain, headache, dizziness, and insomnia. The reintroduction of pramipexole again produced a complete response. We followed the patient for about 3 years. We made several attempts to discontinue the drug. The longest remission period without medication was 6 wk. Resuming the treatment resulted in a complete response on all occasions.

DISCUSSION

RLS typically affects the legs; however, it can affect other regions of the body[7]. About 35%-50% of patients with RLS may have symptoms in the arm[3]. If RLS symptoms get more severe, they may begin to affect other body parts, such as the trunk, abdomen, face, genitals, oral cavity, and head. The gastrointestinal tract and bladder may also be involved. Apart from the arm, isolated involvement in other areas is uncommon, and only a few cases have been reported. Legs are typically affected in such cases and are more severely affected than other body parts. Involvement in the legs occurs several years before involvement in the other body parts[7,8].

The IRLSSG diagnostic criteria (2014) for RLS include four essential criteria: (1) Urge to move with unpleasant sensations in the legs; (2) worse during rest or inactivity; (3) relief by movement or activity; (4) worse in the evening or at night; and (5) not due to another condition[1]. Our patients’ lower limb symptoms met the IRLSSG diagnostic criteria for RLS. However, it is difficult to prove if accompanying headaches, dizziness or other head-related symptoms, back pain, and generalized body pain were part of the RLS spectrum. The urge to move is the central feature of the RLS criteria. The criteria are largely aimed for symptoms in the lower limbs. It is easy to exhibit the urge to move in the legs. However, it is difficult to prove the “urge to move” when RLS affects other body areas, particularly the head, neck, face, and back. A worsening of symptoms in the evening or at night might be an indirect sign of RLS in that body part. Relief by movement or massage of the affected body part may be another indirect sign of RLS in that body area. Headache, dizziness, other head-related symptoms, back pain, and generalized body pain were more intense in the evening and at night. Additionally, symptoms improved following a massage of the head and other affected body regions. Furthermore, administering dopamine agonists relieved these symptoms. We are confident that accompanying headaches, dizziness or other head-related symptoms, back pain, and generalized body pain are all part of the RLS spectrum. Our cases were unique in that head and back symptoms were prominent, with headache, dizziness, back pain, and generalized body pain being the presenting or primary complaints of the patients.

Lower limb symptoms are the primary clinical features of RLS. However, patients frequently seek medical attention primarily because of disturbed sleep[2]. Patients with RLS often delay seeking medical attention for many years. Moreover, even when they seek medical attention, an accurate diagnosis sometimes takes a decade or more. It may be due to patients’ failure to disclose their clinical features to their physicians[9]. In one study, leg symptoms were reported to physicians by just 32% of individuals with moderate-to-severe RLS[10]. Therefore, diagnosing atypical cases affecting areas other than the legs may be exceedingly challenging. Karroum and colleagues conducted a topographic localization of RLS sensations[11]. All individuals reported RLS symptoms in the legs. About 57% of patients felt symptoms in the arms. The trunk (chest, abdomen, and back) and the head were affected in 9% and 5% of patients, respectively. In two patients, the symptoms spread to the whole body. RLS has several variants, depending on the region of involvement: Restless arm syndrome, restless abdomen syndrome, restless mouth syndrome, restless genital syndrome, restless bladder syndrome, restless bowel syndrome, and restless back syndrome[7,8]. These variants may be the primary or only symptom in a subset of patients with RLS.

The symptoms described in RLS patients are twitchy, itchy, burning, current-like, ants crawling, creeping, throbbing, tight feeling, prickling, soda-bubbling, restlessness, pain, and several other non-specific features[2]. If unpleasant sensations and pain are felt in the head, they may be misinterpreted as dizziness, headache, or psychosomatic symptoms. Even a typical case of RLS affecting the legs is misinterpreted as a psychogenic condition[9]. Therefore, physicians are more likely to diagnose RLS variants affecting atypical areas as a psychotic disorder. In 2020, Prakash and Prakash reported two cases of RLS associated with CTTH. Both RLS and CTTH improved after receiving levodopa. The authors hypothesized that headaches may be a part of the RLS symptom complex in a subgroup of patients[3]. They used the term ‘restless head syndrome or cephalic variant of RLS’ to describe such patients. In 2022, Balgetir and colleagues documented 22 such head-related RLS symptoms, which they also termed restless head syndrome[5]. Recently, Prakash and colleagues reported two cases of RLS accompanied with chronic dizziness[4]. Both the dizziness and RLS were resolved with levodopa or dopamine agonist therapy. The authors speculated that dizziness could be a component of the RLS symptom complex.

In our cohort of 17 patients, headache was the chief complaint in 12 (70.5%) patients. Two of them also reported dizziness as one of their chief complaints. Three additional patients reported dizziness as their primary complaint. This subset of patients may represent restless head syndrome. In addition to headaches, backaches were also the primary complaint in two patients. There have also been reports of restless back as the initial symptom of RLS[12,13]. Generalized body pain was the primary complaint of two patients. A few authors have reported generalized body pain in a few patients with RLS[11]. We hypothesize that generalized body pain due to RLS may be termed ‘restless body syndrome’.

Although RLS typically affects the legs, it appears that RLS might affect any body area. RLS is most likely caused by an interaction between impaired dopaminergic neurotransmission in the subcortex of the brain and systemic or brain iron deficiency[14]. The pathophysiology of RLS suggests that it may also affect other body regions, either with or without leg involvement. RLS-related unpleasant sensations include pain. Dopaminergic fibres modulate pain perception and analgesia. Impaired dopaminergic transmission is associated with several painful conditions[15]. RLS is associated with a number of comorbid conditions, including several painful conditions[16]. The associations of RLS have been noted with fibromyalgia, growing pain, chronic back pain, tension-type headache, and migraine. A few studies have shown that individuals with fibromyalgia and growing pain have a higher prevalence of RLS[17]. Baykal Şahin et al[18] recently demonstrated a higher prevalence of RLS in patients with chronic low back pain. Several studies have revealed that people with RLS have a greater lifetime prevalence of migraine than controls[19]. A few open-label studies have shown that dopaminergic treatment can simultaneously alleviate both RLS and migraine[20]. A few studies have also observed a correlation between RLS and TTH. In a nationwide population-based retrospective cohort analysis, Yang et al[21] had shown that patients with TTH had a higher risk of RLS. In another population-based study, Chung et al[22] similarly showed a higher prevalence of RLS in TTH patients.

The link between RLS and various painful disorders raises several questions. Are the diagnoses incorrect? Does having one painful condition increase the likelihood of having another? Are these painful disorders part of the same disease spectrum? Walters and colleagues suggest that RLS and growing pains may be different phenotypic expressions of the same disorder[23]. A similar hypothesis could be suggested for headache disorders, especially for TTH. TTH’s clinical semiology matches the symptoms described for RLS in the legs. Therefore, RLS and TTH in a subset of patients may be two phenotypic manifestations of the same disorder. As mentioned earlier, RLS affects almost all body parts, including visceral organs. Therefore, we hypothesize that RLS could be a generalized disorder, and there may be generalized pain, including headache.

This retrospective study has several limitations. This is a case series, so there is the possibility of unrecognized selection bias. The treatments were not standardized for these patients. We cannot rule out the possibility of secondary causes in these patients, as a comprehensive evaluation for secondary RLS and other associated symptoms was not conducted.

CONCLUSION

Although RLS typically affects the legs, it appears that RLS might affect any body area, including the head. If RLS-related unpleasant sensations and pain are felt in the head, they may be misinterpreted as headache, dizziness, or psychosomatic symptoms. RLS and TTH in a subset of patients may be two phenotypic manifestations of the same disorder. We propose that headaches in a subgroup of RLS patients might be part of the RLS spectrum. A correct diagnosis is critical, as patients will respond to dopaminergic drugs.

Footnotes

Provenance and peer review: Invited article; Externally peer reviewed.

Peer-review model: Single blind

Corresponding Author's Membership in Professional Societies: Indian Academy of Neurology.

Specialty type: Clinical neurology

Country of origin: India

Peer-review report’s classification

Scientific Quality: Grade C

Novelty: Grade B

Creativity or Innovation: Grade B

Scientific Significance: Grade B

P-Reviewer: Tang ZP S-Editor: Liu JH L-Editor: Wang TQ P-Editor: Yuan YY

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