1
358 crystalloid inclusions) which are known to develop in non- tumorous livers of women on contraceptive steroids.5 Ii Although these fine structural changes are non-specific and may occur also in various other conditions, they were thought to represent the fine-structural manifesta- tions of the protracted action of contraceptive steroids in our case. Our findings do not clarify whether or not contraceptive steroids are involved in the causation of benign hepatomas. Further critical studies are needed. Department of Pathology, St. Michael’s Hospital, University of Toronto, Toronto, Ontario, Canada. E. HORVATH K. KOVACS R. C. Ross. 1. Baum, J. K., Holtz, F., Bookstein, J. J., Klein, E. W. Lancet, 1973, ii, 926. 2. Contostavlos, D. L. ibid. p. 1200. 3. ibid. p. 1481. 4. Horvath, E., Kovacs, K., Ross, R. C. Digestion, 1972, 7, 74. 5. Perez, V., Gorosdisch, S., De Martire, J., Nicholson, R., Di Paola, G. Science, 1969, 165, 805. PRETENDING TO BE UNCONSCIOUS SIR,-Dr Hopkins’ article 1 prompts me to present the following case-history. A young hospital worker was admitted overnight after complaining of headache following involvement in a road traffic accident in whjtch she was neither injured nor rendered unconscious. She returned to duty next day but was readmitted as an emergency after senior nursing staff had seen her " staggering and bumping into things ". After admission she had a " fit " and became inaccessible. The duty house-physician, thinking her heart had stopped, resuscitated her with a blow on the chest and she was then monitored until next morning. Next morning the duty physician noted that there was a very emotional atmosphere in her side-ward, where it was felt among the nursing staff that neurosurgical aid should have been summoned long before. Others considered she might have tetanus, because of her posture which resembled opisthotonos. The patient resisted attempts at examination, and her attacks of hyperventilation alternated with breath-holding to the point of cyanosis. The physician made a diagnosis of hysteria but decided to cover himself by getting an anses- thetist to attend while she was sedated. A neurosurgeon felt that there was no neurological damage, and the physi- cian then approached a psychiatrist, who agreed that further exclusion of organic disease was called for. Electro- encephalography and lumbar puncture and some further blood-tests were done. No underlying disease was found. These tests occupied two days, during which time she remained " unconscious ", her hydration being assured by a drip. Then she suddenly became accessible and rational, but denied recollection of events since the accident and seemed quite unconcerned. Subsequently it became clear that she had escaped from an intolerable situation caused by the stress of simultaneously coping with her career and supporting her family financially and emotionally while her parents planned divorce. She soon returned to duty. The case illustrates the reluctance to rely on a purely clinical diagnosis, the efforts felt appropriate to exclude organic disease, and the emotional involvement of other staff when a colleague is the patient. Consequently the " emergency " occupies a considerable part of a busy general hospital’s emergency resources. Moreover, the intensive care thus given probably reinforces attention- seeking behaviour and this prolongs the " coma ". 382 Clifton Drive North, St. Annes-on-Sea FY8 2PN. M. W. P. CARNEY. 1. Hopkins, A. Lancet, 1973, ii, 312. NEURAL-TUBE DEFECTS AND TWINNING SIR,-Dr Ian Leck’s comment (Feb. 2, p. 178) on Knox’s fetus/fetus interaction hypothesis is certainly supported by some clinical findings in spina-bifida babies. One swallow doesn’t make a summer, but one of the most striking cases that I have seen was a baby boy with a myelocele and an aborted twin attached at the site of the myelocele. The teratoid twin had well-formed digits which responded to stimulation of the legs of the host baby. A notable feature of most myeloceles is their gross excess of neural tissue and skin. Although this has been explained by some as merely representing a failure to check normal growth at the posterior neuropore, this finding supports the aborted-twin theory. Another feature that is not well recognised is that scalp-type hair grows around the myelocele and not normal body hair. This accords with the suggestion that the area represents the head of a second fetus. Clearly a detailed analysis of all twinning found in children with spina bifida and anencephaly is called for. 10 Kingswood Drive, Dulwich, London SE19 1UT. D. F. ELLISON NASH. DIGOXIN IN BENIGN INTRACRANIAL HYPERTENSION SIR,-Digoxin in normal doses substantially reduced the volume of cerebrospinal fluid (c.s.F.) obtained from the lateral ventricles of three patients with various central nervous system disorders.1 We have therefore attempted to use digoxin to control a persistent increase in C.S.F. pressure, refractory to more usual therapy, in a patient suffering from benign intracranial hypertension (B.l.H.). Moreover, as a result of repeated therapeutic lumbar puncture, we have been able to compare simultaneous plasma and c.s.F. digoxin levels, of interest to us especially since the report of moderate passage of digoxin from plasma to c.s.F. in four infants on maintenance digoxin therapy. 2 A 37-year-old man under the care of Prof. John Marshall had been evaluated 18 months earlier for symptoms of a few days’ intermittent, bilateral obscuration of vision, his previous health having been excellent. Examination revealed chronic bilateral papillredema (confirmed by fluorescein angiography) and an arcuate field defect in the region of the left blind spot, visual acuity remaining unimpaired. The presenting visual symptoms resolved shortly after his initial admission and have not subsequently recurred. Apart from a raised lumbar c.s.F. pressure of between 210 and 310 mm. c.s.F., determined on four occasions during the first admission, neurological investigation, including lumbar pneumoencephalography, demonstrated no abnormality, and a diagnosis was made of B.l.H. unassociated with any known precipitating factor. He was treated by repeated lumbar puncture and removal of sufficient c.s.F. to lower the pressure to 90-100 mm. c.s.F., and also with acetazol- amide (’Diamox’) and steroids, acetazolamide later being withdrawn when the patient had renal colic from a calculus. The steroids were gradually withdrawn after about three months. After follow-up for a year, during which time the c.s.F. pressure had ranged from 250 to 350 mm. c.s.F., another attempt to reduce this pressure was made by the reintroduction of steroids (oral dexamethasone 6 mg. daily). There had been little effect on the pressure, however, after a month’s treatment, and it was decided that oral digoxin should be introduced in addition. After digitilisation, the patient was maintained on digoxin (’ Lanoxin ’) 0-25 mg. 8-hourly, continued for sixweeks. Lumbar puncture was performed on seven occasions during this period, and digoxin was assayed on a sample of the c.s.F. obtained, a concurrent heparinised venous-blood sample also being assayed for digoxin. Plasma-digoxin was estimated by radioimmunoassay using the method of Smith et al.3 with minor modifications. The same method was used for the detection of digoxin in C.S.F,j

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358

crystalloid inclusions) which are known to develop in non-tumorous livers of women on contraceptive steroids.5 Ii

Although these fine structural changes are non-specificand may occur also in various other conditions, theywere thought to represent the fine-structural manifesta-tions of the protracted action of contraceptive steroids inour case. Our findings do not clarify whether or notcontraceptive steroids are involved in the causation ofbenign hepatomas. Further critical studies are needed.

Department of Pathology,St. Michael’s Hospital,University of Toronto,

Toronto, Ontario, Canada.

E. HORVATHK. KOVACSR. C. Ross.

1. Baum, J. K., Holtz, F., Bookstein, J. J., Klein, E. W. Lancet, 1973,ii, 926.

2. Contostavlos, D. L. ibid. p. 1200.3. ibid. p. 1481.4. Horvath, E., Kovacs, K., Ross, R. C. Digestion, 1972, 7, 74.5. Perez, V., Gorosdisch, S., De Martire, J., Nicholson, R., Di Paola,

G. Science, 1969, 165, 805.

PRETENDING TO BE UNCONSCIOUS

SIR,-Dr Hopkins’ article 1 prompts me to present thefollowing case-history.A young hospital worker was admitted overnight

after complaining of headache following involvement in aroad traffic accident in whjtch she was neither injured norrendered unconscious. She returned to duty next day butwas readmitted as an emergency after senior nursing staffhad seen her " staggering and bumping into things ". Afteradmission she had a " fit " and became inaccessible. The

duty house-physician, thinking her heart had stopped,resuscitated her with a blow on the chest and she wasthen monitored until next morning. Next morning theduty physician noted that there was a very emotionalatmosphere in her side-ward, where it was felt among thenursing staff that neurosurgical aid should have beensummoned long before. Others considered she might havetetanus, because of her posture which resembled

opisthotonos.The patient resisted attempts at examination, and her

attacks of hyperventilation alternated with breath-holdingto the point of cyanosis. The physician made a diagnosis ofhysteria but decided to cover himself by getting an anses-thetist to attend while she was sedated. A neurosurgeonfelt that there was no neurological damage, and the physi-cian then approached a psychiatrist, who agreed thatfurther exclusion of organic disease was called for. Electro-encephalography and lumbar puncture and some furtherblood-tests were done. No underlying disease was found.These tests occupied two days, during which time sheremained " unconscious ", her hydration being assuredby a drip. Then she suddenly became accessible andrational, but denied recollection of events since the accidentand seemed quite unconcerned. Subsequently it becameclear that she had escaped from an intolerable situationcaused by the stress of simultaneously coping with hercareer and supporting her family financially and emotionallywhile her parents planned divorce. She soon returned toduty.The case illustrates the reluctance to rely on a purely

clinical diagnosis, the efforts felt appropriate to excludeorganic disease, and the emotional involvement of otherstaff when a colleague is the patient. Consequently the" emergency " occupies a considerable part of a busygeneral hospital’s emergency resources. Moreover, theintensive care thus given probably reinforces attention-seeking behaviour and this prolongs the " coma ".

382 Clifton Drive North,St. Annes-on-Sea FY8 2PN. M. W. P. CARNEY.

1. Hopkins, A. Lancet, 1973, ii, 312.

NEURAL-TUBE DEFECTS ANDTWINNING

SIR,-Dr Ian Leck’s comment (Feb. 2, p. 178) on

Knox’s fetus/fetus interaction hypothesis is certainlysupported by some clinical findings in spina-bifida babies.One swallow doesn’t make a summer, but one of the most

striking cases that I have seen was a baby boy with amyelocele and an aborted twin attached at the site of themyelocele. The teratoid twin had well-formed digitswhich responded to stimulation of the legs of the hostbaby. A notable feature of most myeloceles is their grossexcess of neural tissue and skin. Although this has beenexplained by some as merely representing a failure tocheck normal growth at the posterior neuropore, thisfinding supports the aborted-twin theory. Another featurethat is not well recognised is that scalp-type hair growsaround the myelocele and not normal body hair. Thisaccords with the suggestion that the area represents thehead of a second fetus.

Clearly a detailed analysis of all twinning found inchildren with spina bifida and anencephaly is called for.

10 Kingswood Drive,Dulwich,

London SE19 1UT. D. F. ELLISON NASH.

DIGOXIN IN BENIGN INTRACRANIALHYPERTENSION

SIR,-Digoxin in normal doses substantially reduced thevolume of cerebrospinal fluid (c.s.F.) obtained from thelateral ventricles of three patients with various centralnervous system disorders.1 We have therefore attemptedto use digoxin to control a persistent increase in C.S.F.

pressure, refractory to more usual therapy, in a patientsuffering from benign intracranial hypertension (B.l.H.).Moreover, as a result of repeated therapeutic lumbar

puncture, we have been able to compare simultaneousplasma and c.s.F. digoxin levels, of interest to us especiallysince the report of moderate passage of digoxin fromplasma to c.s.F. in four infants on maintenance digoxintherapy. 2

A 37-year-old man under the care of Prof. John Marshallhad been evaluated 18 months earlier for symptoms of a fewdays’ intermittent, bilateral obscuration of vision, his previoushealth having been excellent. Examination revealed chronicbilateral papillredema (confirmed by fluorescein angiography)and an arcuate field defect in the region of the left blind spot,visual acuity remaining unimpaired. The presenting visualsymptoms resolved shortly after his initial admission and havenot subsequently recurred. Apart from a raised lumbar c.s.F.pressure of between 210 and 310 mm. c.s.F., determined on fouroccasions during the first admission, neurological investigation,including lumbar pneumoencephalography, demonstrated no

abnormality, and a diagnosis was made of B.l.H. unassociatedwith any known precipitating factor. He was treated byrepeated lumbar puncture and removal of sufficient c.s.F. to

lower the pressure to 90-100 mm. c.s.F., and also with acetazol-amide (’Diamox’) and steroids, acetazolamide later beingwithdrawn when the patient had renal colic from a calculus. Thesteroids were gradually withdrawn after about three months.After follow-up for a year, during which time the c.s.F. pressurehad ranged from 250 to 350 mm. c.s.F., another attempt to reducethis pressure was made by the reintroduction of steroids (oraldexamethasone 6 mg. daily). There had been little effect on thepressure, however, after a month’s treatment, and it was decidedthat oral digoxin should be introduced in addition.

After digitilisation, the patient was maintained on digoxin(’ Lanoxin ’) 0-25 mg. 8-hourly, continued for sixweeks. Lumbarpuncture was performed on seven occasions during this period,and digoxin was assayed on a sample of the c.s.F. obtained, aconcurrent heparinised venous-blood sample also being assayedfor digoxin. Plasma-digoxin was estimated by radioimmunoassayusing the method of Smith et al.3 with minor modifications.The same method was used for the detection of digoxin in C.S.F,j