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Narcolepsy-Cataplexy Syndrome Should Be Ex- 1 White Blood Cells

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Narcolepsy-Cataplexy Syndrome Should Be Ex- 1 White Blood Cells 592 CASE REPORTS: NARCOLEPSY-CATAPLEXY SYNDROME Canad5 . vA.J. The cephalic vein was dilated and tortuous and A CASE OF THE NARCOLEPSY- could be traced from the dorsum of the hand to the SYNDROME* infraclavicular fossa. There was a fine, soft nodular- CATAPLEXY ity in the subcutaneous tissue of the flexor surface of the forearm, overlain by several dilated venules. J. TODD, M.B., B.S., D.P.M., In addition, extending from the wrist flexion crease Menston, Yorks., England proximally two inches and from the mid-dorsal line to the mid-ventral line on the ulnar side, there was IN 1877, Westphal"9 published the first case of a soft-tissue mass appearing as an area of localized a troubled by brief but irresistible at- chronic lymphoedema. About the elbow, several bony patient hard nodules could be palpated, movable in the sub- tacks of diurnal sleep. Soon afterwards, cutaneous tissue. These ranged in size from one-half Gelineau12 described a similar case, and applied to three cm. in diameter and were not tender. The the term "narcolepsy" to this particular form of radial pulse at the right wrist could be palpated sleep disorder. With wider experience, it became only poorly, the ulnar pulse not at all. evident that a tendency to narcolepsy was com- monly associated with a proclivity to attacks of LABORATORY INVESTIGATIONS emotionally induced muscular weakness; thus, Hemoglobin level 12.4 g. % (86%); E.S.R. 12 many narcoleptic subjects are apt to buckle at mm. in one hour; white cell count 11,050; differ- the knees, or even fall to the ground, when ential count: polymorphonuclears 64%, staff cells 2%, lymphocytes 34%; Kahn test negative. Total protein amused, frightened, or angered by some environ- 6.3 g. %; albumin 4.6 g. %; globulin 1.7 g. %. mental situation. Adie1 was the first to apply Calcium 5.1 mEq/l. (10.2 mg. %); phosphorus 1.2 the term "cataplexy" to these emotionally in- mEq/l. (2.2 mg. %); alkaline phosphatase 7 King- duced attacks of muscular hypotonia. The re- Armstrong units; acid phosphatase 1.8 King- searches of Wilson,22 Levin,13 Daniels,7 and Armstrong units. Brain5 have shown that the purview of the Urinalysis: pH 4.5; protein, sugar and acetone narcolepsy-cataplexy syndrome should be ex- 1 white blood cells. negative; plus, tended to include a group of ancillary symptoms: catalepsy, amnesic states, diurnal somnambulism, DISCUSSION sleep-paralysis, hallucinatory episodes, vivid The diagnosis of melorheostosis is a radiologi- dreams, and adiposo-genital dystrophy. cal one. Characteristic is a dense, structureless The two major symptoms-narcolepsy and sclerosis "flowing like candle-grease" and often cataplexy-are not "cardinal" in the strict sense appearing to follow the distribution of a main of the word, since either may appear alone vessel or nerve. In this case, such a distribution, without (in the writer's opinion) invalidating apparently following the ulnar nerve, is seen a diagnosis of "narcolepsy-cataplexy syndrome". (Fig. 1). The involvement is patchy in contrast to In this connection, it is noteworthy that narco- osteopetrosis and may be endosteal or cortical. lepsy may antedate by several years the appear- Deposits of bone in periarticular soft tissues are ance of cataplexy, or vice versa.7 20 Moreover, seen, as in this case (Figs. 1 and 2), and areas the two main symptoms are subject to substitu- of decreased bone density are also common. tions and transitions of various kinds:20 sleep Pain, described as the most frequent symptom by attacks may be induced by emotion, cataplectic Fairbank,' was absent in this case; limitation of attacks may occur spontaneously, cataplectic movement was present though not a complaint. attacks may terminate in sleep, and an attack of The deformity of the ulnar two fingers and the cataplexy may replace narcoleptic sleep if the venous abnormality were notable in this case. latter is prevented by deliberate interference. The ancillary symptoms are often encountered REFERENCES apart from the narcolepsy-cataplexy combina- 1. FAIRBANK, SIR THOMAS: An atlas of general affec- tions of the skeleton, E. & S. Livingstone, Ltd., tion, but their incidence is significantly high in Edinburgh, 1951, p. 49. 2. Idem: Personal communication. conjunction with it. All writers on the subject agree that narcolepsy occurs more frequently in the male; in a group of 66 cases studied by Levin,13 the ratio of male to female cases was 5:1. *Menston Hospital, Menston, Yorks., England. Canad. M. A. J. Sept. 15, 1957, vol. 77 CASE REPORTS: NARCOLEPSY-CATAPLEXY SYNDROME 593 The following case of the narcolepsy-cata- the right side of his body more severely than the plexy syndrome has some interesting features. left. His description of the emotional stimuli liable to provoke an attack was as follows: "With regard to the attacks of limb-weakness and twitching, I An intelligent man, aged 42 years, was referred find these would occur many times each day, under to an out-patient clinic because of a group of symp- any amount of circumstances, and I would always toms which had troubled him for three years. These be aware what caused it. To cite a few causes, how- symptoms consisted in attacks of diurnal sleep and ever: if I were contradicted or opposed; if I were emotionally induced muscular weakness; moreover, saying something I thought clever or witty; if I had he had developed a proclivity to nocturnal hallucin- a ready answer to someone's problems; if I were the atory experiences and vivid dreams. He had gained recipient of praise for anything I had done. All these 28 lb. in weight since the onset of his narcolepsy, happenings and many others, have brought on at- although he had not become noticeably obese. tacks." His worst fit of cataplexy was induced by There had been no loss of libido, but his sexual the wave of pleasurable emotion which swept over appetite had never been strong. him on finding himself the possessor of a "grand slam" hand at bridge. DIURNAL SYMPTOMS Nocturnal symptoms. - Since the onset of his Narcoleptic attacks.-The attacks of sleep were a narcolepsy, he has been troubled during the night source of embarrassment to him as they were irresis- by hallucinatory experiences and bizarre dreams. A tible and liable to supervene at any time. On one curious feature of these nocturnal visitations is that occasion, he fell asleep in the act of filling in the a certain theme or pattern would recur again and passport of a business client, his pen jerking across again. On several occasions, he has woken from a the page as he slumped over his desk; on another, sound sleep with the impression that the bedclothes he was overcome by sleep while eating a meal, his were being moved by some invisible person or in- fork coming to rest at a point half way between his tangible agency. When this happened, his wife plate and mouth. He gave the following general (who occupied a separate bed) would be awakend account of his narcoleptic naps: "The sleepiness by his restless movements and loud cries. Further- will occur at any time of day, maybe as little as half more, he has repeatedly awakened during the night an hour after rising from a good night's sleep; the with the feeling that another body "lighter than a sleep is not very deep, perhaps of 20 minutes' real one" is lying beside him in the bed, and is duration if I were undisturbed. Prior to an attack pressing down upon him. This "presence" seems to there is no feeling of tiredness. I have, on occasion, be breathing in unison with him. Sometimes, he has dozed off in the middle of a conversation, talking vaguely identified the second body with his wife nonsense. When this happens, the sound of my own or mother. When experiencing these illusory phe- voice arouses me to wakefulness immediately, and nomena, he is convinced that he is fully awake. I am aware that what I have said was foolish, and Nevertheless, he realizes in retrospect that his ex- had nothing to do with the subject under discussion. periences are akin to dreams. In addition to these While attacks will occur at any time, I find they are hallucinatory episodes, his sleep is disturbed by certain to occur under certain circumstances. For vivid and bizarre nightmares. instance, I find it quite impossible to read at any Background history.-There is no history of head time of day for more than five minutes ." His injury, encephalitis lethargica, influenza, or epilepsy. attempts to ward off attacks of sleep by an effort He has not been troubled by recurrent headaches, of will had precipitated double vision and a curious excessive thirst, or nocturia; nor is there a history form of metamorphopsia. He described the latter as of intellectual or personality deterioration. When he follows: "Two figures would, without much warning, was 12 years old, he contracted "septicaemia" from change position, that is, 32 would become 23 by septic abrasions on his knees. At that time, he had means of a very graceful leap, each to come to rest a high temperature, moderate headache, lymphan- in the other's position. Then (in other instances), gitis of his thighs, and a very sore tongue-"it felt the whole number would turn a full circle as though as though it had been burnt". The family history on a central pivot." He sometimes dreamed during is non-contributory. the spells of narcoleptic sleep. Some of these dreams Physical examination.-The patient was of healthy were so life-like and realistic that, as he put it, "they appearance, but had a sleepy facial expression.
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