Chapter 5 Restless Legs Syndrome and Nocturnal Myoclonus (Periodic Limb Movements During Sleep)

An Historical Review

In a lecture on the predormitum (or sleepening, as Gowers, cited in Critchley,¹ liked to call it), Critchley¹ mentioned a predormitum restlessness, which, he said, is by no means a rarity, although physicians may not be consulted specifically on this score. He acknowledged that Ekbom^2–4 was responsible for drawing attention to the condition, calling it restless legs syndrome (RLS). Few medical men, Critchley added, knew that the Ekbom syndrome was graphically described in the 17th century by the great clinical neurologist Thomas Willis.⁵ Ekbom^2–4 admitted that Willis had been the first to describe the disorder, but in 1685, not 1695 as Critchley had suggested. In fact, a description of the syndrome seems to date back to a text Willis wrote in 1683, which was in turn a translation from a paper written in Latin in 1672.⁶

In 1861, Wittmaack⁷ named the disorder “anxietas tibiarum,” misconstruing it as a particular form of hysteria. Beard⁸ also considered the condition to be a form of nervous exhaustion, but specified that it was “one of the myriad results of spinal irritation.” Oppenheim⁹ included the RLS among the forms of neurasthenia, pointing out that it may sometimes appear as a family trait.

For these clinicians, the terms “nervous exhaustion,” “hysteria,” and “neurasthenia” did not have the modern meaning of psychogenic disease or syndrome but simply a clinical condition lacking a well-defined anatomical basis. However, semantic confusion has led the authors of more recent reports to mistake RLS for a form of “neurosis” in the modern sense.

Mussio-Fournier and Rawak¹⁰ in 1940 published a full report of a patient with an inherited familial form of RLS. They pointed out that the clinical symptoms worsened during pregnancy and then improved during the puerperium. Their patient also presented with acroparaesthesia and motor agitation associated with pruritis and urticaria.

Allison¹¹—a sufferer himself—wrote, “This is a common ailment, which I have never seen described,” adding “I have observed this condition mainly in myself, relatives, and friends, as patients do not often complain of it unless it occurs frequently enough to cause loss of sleep.” Allison defined the motor disorder as “a combination of voluntary and involuntary jerks,” using the term “leg jitters.”

Without a doubt, Ekbom has the merit of having extensively illustrated the clinical aspects of the syndrome, dedicating an important series of publications to the subject between 1944 and 1960 and beyond.^2–4

Bonduelle¹² and Jolivet¹³ described the first French observations; Tuvo,¹⁴ the first Italian case; and Barraquer-Ferre,¹⁵ the Spanish patient. A major contribution was made by Nordlander,¹⁶ who documented that iron deficiency anemia exacerbated RLS and that intravenous administration of iron compounds “proved very effective, regardless of whether or not the patient had anemia or sideropenia.” The fact that RLS may arise after acute anemia and regress with appropriate treatment was subsequently also reported by Murphy.¹⁷

Heralding more modern pathophysiological theories on the origin of RLS, Menninger-Lerchenthal¹⁸ deemed it a kind of extrapyramidal disease related to an iron metabolism disorder with a secondary dysfunction of the pallidonigral system.

The notion that anemia and iron deficiency underlie the RLS arising in end-stage renal disease and pregnancy has also been proposed by others.^19–22

The treatment of RLS owes much to the work of Walters, Hening, and colleagues²³ on the efficacy of opioids and to Akpinar,^24,25 who paved the way to the use of L-dopa and dopamine agonists.

Related posts:

The Patient’s Perspective Assessment of Restless Legs Syndrome Features With Standardized Methods Restless Legs Syndrome as a Movement Disorder Restless Legs Syndrome in Neurological Disorders Spinal Cord, Dopamine, Opiates, and Restless Legs Syndrome Restless Legs Syndrome Morbidity: Sleep and Quality of Life

Restless Legs Syndrome