EXPERIENCE AND REASON-BRIEFLY RECORDED
areflexic paralysis. Laboratory data, including
CSF, are normal. EMG reveals decreased
mo-tor nerve conduction velocity and muscle-ac-tion potential amplitude which return to nor-mal as the patient recovers in a few days.8’#{176}A mortality rate in a large series of patients with
this disease from British Columbia was 10%
and was due to respiratory paralysis.5 Although a few patients have died after tick removal,1’2’10 rapid recovery within hours or days is usual.
The precise nature of the toxin is not known but it is present in greatest quantity in the tick’s eggs and ovaries.4 It impairs conduction most markedly in small motor nerve fibers and motor nerve end-plates.8’9
Care must be taken to remove the mouth
parts of the tick which contain toxic saliva.4 A
good method for removal of the tick is as
follows: Raise the skin adjacent to the tick with sterile forceps, making a “tent;” then under-mine area with a sterile needle or scalpel blade; as the tick is removed with forceps, the superficial layer of skin will be detached with the mouth parts intact. The finding of one tick should not bring to a halt a thorough search of
the patient’s entire surface, since more than
one tick may be attached and engorging.
FRANKLIN L. DEBUSK, M.D.
SusAN O’CoNNoR, M.D.
Department of Pediatrics University of Florida
College of Medicine
Gainesville, Fkrida 32601
This study was supported in part by a Develop-mental Physiology Training Grant NIH Ti-HD0054.
\Ve are indebted to Dr. George A. Dell for refer-ring this patient.
REFERENCES
1. Abbott, K. H. : Tick paralysis: A review. I. Proc. Staff Mayo Clinic, 18:39, 1943. 2. Stanbury, J. B., and Huyck, J. H. : Tick
paraly-sis: A critical review. Medicine, 24:219, 1945.
3. McCue, C. M., Stone, J. B., and Sutton, L. E., Jr. : Tick paralysis; Three cases of tick ( Der-macentor variabiis Say) paralysis in Vir-ginia. With a summary of all the cases reported in the Eastern United States.
Prni-ATRICS, 1:174, 1948.
4. Costa, J. A. :Tick paralysis on the Aflantic Sea-board. Amer. J. Dis. Child., 83:336, 1952. 5. Schmitt, N., Bowmer, E. J., and Gregson,
1. D. : Tick paralysis in British Columbia. Canad. Med. Ass. J., 100:417, 1969.
6. Mulherin, P. A. : Ataxia due to bite of Amen-can dog tick ( Dermacentor variabilis Say).
1.
Pediat., 16:86, 1940.7. Jaffe, E., and Perlmutter, I. : Tick paralysis
(
successfully treated in the stage of ataxia): Report of a case. J. Pediat., 45:98, 1954.8. Cherington, M., and Snyder, R. D. : Tick paral-ysis: Neurophysiologic studies. New Eng. J. Med., 278:95, 1968.
9. Murnaghan, M. F. : Site and mechanism of tick paralysis. Science, 131 :418, 1960.
10. Alexander, R. M.: Tick paralysis: Report of a case in Florida. J.A.M.A., 149:931, 1952.
Survival
in Anthrax
Meningitis
For the past two decades, infection with the
anthrax bacillus has become increasingly rare;
the incidence of meningitis as a complication
of this disease is even rarer.1
In spite of the rarity of this disease, its sa-lient features are worth mentioning since some cases may now be cured.’
In most cases, meningo-anthrax, an uncom-mon finding in human anthrax, is caused by
an-thrax septicemia resulting from primary
cuta-neous or visceral involvement.2 This
complica-tion of anthrax often results in sudden death.
Pathologists and forensic experts at the end of
the 19th century were the first to describe
men-ingo-encephalitic involvement in human
an-hr2
The reports of slightly more than 100 cases
of anthrax meningitis in the medical literature
with only six survivals prompted the present
report as a brief review of the literature and a
description of the clinical details in our patient who survived together with an additional note
on new therapy in this usually fatal disease.
CASE REPORT
H.N., an 83k-year-old Iranian girl from Shiraz, was
admitted to the Pediatric Service of the Nemazee Hospital for the first time in a semicomatose state
of 2 days duration on October 1, 1967.
The present illness dates back 5 days prior to
admission when the patient developed headache
330
and was brought to the emergency room of the hospital.
Because of convincing evidence of meningeal
signs, a lumbar puncture was performed; the cere-brospinal fluid was found to be cloudy. Admission to the hospital was advised but the parents refused until 2 days later when she was transferred here.
Pa8t History: The most significant information obtained was that the child had been in close con-tact with many sheep and goats.
Physical Examination: The patient was a well
developed, but undernourished, semicomatose, and restless child who was acutely ill. The temperature was 39#{176}C,pulse rate 130/mm, respiratory rate 40/mm, weight 18 kg, height ill cm, head circum-ference 52 cm, chest circumference 58 cm; blood pressure was 90 mm Hg systolic but diastolic pres-sure could not be obtained.
The principal findings on the initial physical ex-amnination were limited to a black eschar of 2 to 3 cm in diameter located behind the right ear lobe
(Fig. 1
),
surrounded by edema extending to the right half of the face anteriorly and to the right side of the neck inferiorly and posteriorly. The pu-pils were dilated and reacted poorly to light. Fun-duscopy showed 1+ papilledema of one diopter inthe night eye. The neck was stiff; Kernig and
Brudzinski’s signs were positive, and the deep ten-don reflexes were sluggish bilaterally.
Laboratory Findings: The smear and culture of the cerebrospinal fluid obtained on admission to hospital revealed bacillus anthracis (Fig. 2
),
CSF pressure was elevated, sugar was 39 mg/100 ml; the blood sugar obtained at the same time was 210 mg/i00 ml ( patient was receiving intravenous glucose) CSF protein was 672.6 mg/i#{174} ml; CSF red blood cells was 1,100/mm’, white blood cells was 2,654/mm’, with 70% segment. The hemoglo-bin concentration was 12.5 gm/100 ml; hematocnit (Hct) was 38%; the leucocyte count was 5,400/ mm’, with 60% neutrophiles, 4% band forms and3% metamyelocytes, 30% lymphocytes and 3%
monocytes. Wound swab and blood cultures on ad-mission also revealed B. anthracis. Total blood pro-tein was 6.4 gm/100 ml; protein electrophoresis revealed serum albumin to be 3.8 gm/100 ml, al-phai globulin of 0.39 gm/100 ml, alpha2 globulin
of 0.48 gm/100 ml, beta globulin of 0.54 gm/100 ml, and gamma globulin of 1.08 gm/100 ml. Se-rum electrolytes showed CO of 31.4 mEq/i, Na
140 mEq/1, Cl 100 mEq/i, K 2.5 mEq/1. Chest x-ray and urinalysis were within normal limits.
Hospital Course: Patient was first treated with intravenous triple therapy ( sulfonamide, chloram-phenicol, and penicillin ). Upon identification of
the microorganism in the cerebrospinal fluid, the above regimen was changed to 2 million units aqueous penicillin immediately, followed by 1
mil-lion units every 2 hours and 100 mg hydrocortisone semisuccinate (solucortef) in 1,000 cc fluid intra-venously. This regimen was continued for the first 3 days, up to October 4, when the patient’s general condition improved and she could respond to yen-bal commands. At this time, lumbar puncture re-vealed protein of 396.4 mg/100 ml, RBC 1,100
mm’, WBC 380 mm’, segment 9%, lymphocyte
91%, sugar 50 mg/100 ml; simultaneous blood sugar was 119 mg/100 ml; CSF culture revealed no growth. Aqueous penicillin, 1 million units ev ery 4 hours, was then given intramuscularly ac-companied with 10 mg prednisone by mouth every 8 hours.
The patient’s general condition improved mark-edly. Temperature dropped to normal on the 6th day of hospitalization and there was only slight ev-idence of meningeal signs at this time. Facial edema was much less and the patient could now feed herself and answer questions intelligently. At this time penicillin was reduced to 1 million units every 6 hours I.M. and prednisone was tapered off to 5 mg every 6 hours for the next 6 consecutive days. Prednisone was discontinued on the 12th day after admission. Aqueous penicillin was changed to procaine penicillin 800,000 units every 12 hours I.M. and was continued up to 23rd hospital day. Repeated lumbar puncture 1 day prior to discharge yielded clear cenebrospinal fluid; RBC 4 mm’, WBC 75 mm3, all lymphocytes. Protein (CSF) was 29.9 mg/100 ml, and sugar 46 mg/100 ml.
The patient was discharged in good general con-dition without any neurological sequelae, to be
followed up in the Outpatient Department. The
last CSF examination, 3 weeks later, was within normal limits.
DISCUSSION
The first reported cases of anthrax meningitis were those of Wagner who, in 1874, described its pathological changes. Following his first
mention of three cases of hemorrhagic meningi-tis, there have been isolated case reports, most
of them in the Cerman literature.
In 1881, Greenfield reported three cases of
anthrax meningitis following woolsorter’s
dis-ease in England and, subsequently, single case
summaries have appeared.
Bruce and Sherman,6 in 1910, were able to
find in the literature only 26 verified instances
of meningitis due to bacillus anthracis to which they added one of their own. In 1920, HouseT
collected 45 cases and reported three of his
own with detailed descriptions of the gross and
EXPERIENCE AND REASON-BRIEFLY RECORDED
Haight1 described some 95 cases of anthrax meningitis up to 1952; but, of these only 70 were reported in sufficient detail to allow corn-prehensive analysis. We can add one case of anthrax meningitis that Bocquiens reported in
1949 and Haight had not mentioned.
Charn-l)OIi reported two cases in 1955, Miller10 four
cases in 1962, and finally one case was
re-ported b Boudin3 et a!. in 1964. One newborn infant died of hemorrhagic anthrax meningitis which was presumed to have been acquired
from the l)lood stream of the mother who was infected with the anthrax bacillus. In 1953 one 15-year-old boy was reported to have
men-ID nph1 2
Several points are worth mentioning in our case. It was remarkable to occur following cu-taneous anthrax and presumably after septice-mia. The cutaneous lesion was still present when menmgeal symptoms and signs appeared and medical help was sought.
A review of the literature reveals that among 70 cases of anthrax meningitis, the primary fo-cus was skin in 52.8% of cases.’ Very seldom rneningo-anthrax may be a primary manifesta-tion of the infection.2 All patients, like our
case, had positive CSF culture for Bacillus
an-thracis and 70 had positive blood culture.I
Though peripheral \VBC among their cases
ranged from 10,000 to 80,000 mm3,’ it was
Oh the low side (5,400/mm3) in our patient
with almost a normal differential count.
\Vhether this finding is due to the previous par-tial antibiotic therapy or is a part of the picture of the disease in this case has to be determined.
Hemorrhagic meningitis was present in all of
these cases. Cortical hemorrhage was present
ill 50% of cases.’ Apart from the lactescent hemorrhagic aspect of the cerebrospinal fluid which is considered to be the classical type, in the past two decades reports have been pub-lished on cases of meningo-anthrax with a pu-rulent spinal fluid.2 It was interesting to note that the CSF in our case was purulent rather than being hemorrhagic.
The other point of interest was our patient’s age and her survival without sul)sequent
neuro-logical sequelae. Six cases have So far survived.3
Fic. 2. A pure culture of Bacillus anthracis in chain fashion grown from patient’s cerebrospinal
fluid.
Shanahan3”3 reported one survival with anti-biotic and serotherapy. Czyhlarz3”4 reported one survival with serotherapy alone. Kindler2#{176}
reported one and Miller3’10 three survivals
re-spectively. Our case who was treated with mas-sive penicillin and steroid therapy presents the seventh survival.
Like other cases,15 our patient showed evi-dence of trismus which is usually a character-istic sign of anthrax meningitis. The pathologic alteration observed in other cases supports the
following conclusions regarding pathogenesis
based in turn on previously reported
experi-mental work.1,2,b6
Septicemia and dissemination of either spore
containing macrophages or vegetative forms of
bacillus anthracis from cutaneous lesions result
in such secondary manifestations as edema,
hemorrhage, or meningitis. It is presumed that this phenomena was operating in the pathogen-esis of meningitis in our case. In almost every
reported case with good clinical
documenta-tion, the symptoms of meningitis or cerebral ir-ritation preceded death by a matter of hours.17
In our case the meningeal irritation and signs
were most probably present 3 to 4 days prior
to admission and treatment.
The incidence of meningitis is quite rare in
this disease. Shanahan1 and his associate, in
their report of 66 cases of cutaneous anthrax at
a rug manufacturing company, had four
pa-tients with meningitis, or about 5%. Smyth,17
in a United States Public Health Service sur-vey, found that there were 60 to 80 cases of anthrax annually and that 95% of these were of the cutaneous variety. He did not mention a single case of meningitis in this group. Wolff and Heimann18 did not mention a single case
of meningitis in a group of over 2,400 cases
studied, while 98% of these had the cutaneous
variety.
Although specific serum therapy has lowered
the mortality rate in anthrax from 28 to 6.7%,
it has not influenced the prognosis in meningo-anthrax cases
(
100% mortality;2 a fact which may be accounted for by the low permeabilityof the hematomeningeal barrier for antibody
globulin. In view of the high susceptibility of
the anthrax bacillus to many antibiotics and of
the possibility of obtaining effective concentra-tions of some antibiotics in the cerebrospinal
fluid, the theoretical premises have been
cre-ated for the application of combined serum
and antibiotic therapy in anthrax of C.N.S. The
association of anti-inflammatory corticoid ther-apy likewise appears to be theoretically well
founded as it was demonstrated clearly in our
previous report2”9 of patients with malignant
cutaneous anthrax who all survived with this
latter regimen.
There is no clear explanation as to why
men-ingitis and other manifestations of dissemina-tion in this case should develop. It is thought that death results from the effects of secondary
shock attributable to blood and fluid loss
caused by a toxin elaborated by the B.
anthra-dr.1619 It is presumed that combined I.V. fluid
and corticosteroid therapy prevents this
phe-nomena and in turn increases the survival rate
as it has been demonstrated in our case.’#{176}
SUMMARY
An 8-year-old child with anthrax
septice-mia and meningitis presumably secondary to
the cutaneous lesion was presented. The salient
features in our case such as purulent spinal fluid, relative peripheral blood leukopenia.
and her survival, in contrast to the majority of
cases in world literature, were stressed. The
patient was treated successfully with the IV.
fluids-high doses of penicillin in association with new method of corticosteroid therapy.
A. C’srnus TAHERNIA, M.D. CH. HASHEMI, M.D. Department of Pediatrics
Pahiavi University School of Medicine
The Nemazee Hospital
Shiraz, Iran
The authors wish to thank Mr. M. A. Shahbazi for his assistance with the laboratory studies.
REFERENCES
1. Haight, T. H. : Anthrax meningitis, review of literature and report of two cases with au-topsies. Amer. J. Med. Sci., 224 :57, 1952. 2. Vita, A., Secu, A., Cuciureanu, G., Leibovici,
M., Bejenariu, C., and Cutu, G. : Consider-ation on three cases of meningoencephalitis due to anthrax bacilli. Rum. Med. Rev., 5: 36, 1961.
3. Boudin, C., Lauras, A., and Vaillant, C. : Etat de mal epileptique revelateur d’une men-ingite charbonneuse etude anatomo-clinique. Societe Medicale des Hopitaux de Paris, 115: 183, 1964.
4. Wagner, E. : Die intestinalmvkose und ihre Beziehung zum Milzbrand. Arch. d. Heilk. Leipz, 15:1, 1874.
fur-EXPERIENCE AND REASON-BRIEFLY RECORDED
then investigations on anthrax and allied dis-eases in man and animals. Bnit. Med. J., 2:
1007, 1880 and 1 :3, 81, 1881; also Lancet,
2:965, 1880 and 1:391 and 163, 1881.
6. Bruce, A., and Sherman, T. : A case of acute haemorrhagic meningitis due to anthrax. Rev. Neurol. Psychiat, Edinb., 8:521, 1910. 7. House, S. J.:Hemorrhagic meningoencephalitis
in anthrax: A report of three cases. J. Infect. Dis., 27:513, 1920.
8. Bocquien, Y. : Sur une epidemic de charbon animal et humain observee dans le Morbi-han. Bull. Acad. de Med., p. 585, 1950. 9. Chambon, L. and Dutrenit, J.: Note sun une
epidemic de charbon humain avec deux cas de meningite charbonneuse. Bull. Soc. Path. Exot., 48:544, 1955.
10. Miller, J.: Human anthrax in New York State. New York J. Med., 61:2046, 1961.
11. Marchand, F.: Ueber einen merkwurdigen Fall von Milzbrand, bei einer schwangeren mit todtlicher Infection des kindes. Virchow. Arch. Path. Anat., Berl, 9:86, 1887.
12. Rapor, A. B.: Anthrax meningo-encephalitis, E. Afr. Med. J., 30:339, 1953.
13. Shanahan, R. H., Griffin, J. R., and Von Auers-perg, A. P. : Anthrax meningitis, report of case of internal anthrax with recovery. Amer. J. Clin. Path., 17:719, 1947.
14. Czyhlanz, E.: Beitrag zur Lehr von der Milz-brandmeningitis. Wien. Klin. Wchnschr.. 29:768, 1916.
15. Plotkin, S. A., Brachman, P. S., Utell, M., Bum-ford, F. H., and Atchison, M. M. : An epi-demic of inhalation anthrax the first in the twentieth century I-clinical feature. Amer. J.
Med., 29:992, 1960.
16. Aibrink, \V. S., Brooks, S. M., Biron, R. E.,
and Kopel, M. : Human inhalation
an-thrax, a report of three fatal cases. Amer. J. Path., 36:457, 1960.
17. Smyth, H. F.: U.S. Public Health Service Sur-vey. Cited in Cecil’s Textbook of Medicine. Philadelphia: W. B. Saunders Co., 1947, pp. 268-274 (also by Wolff and Heimann). 18. Wolff, A. H., and Heimann, H.: Industrial
an-thrax in Tjnited States; epidemiologic study. Amer. J. Hg., 53:80, 1951.
19. Tahernia, A. C. : Treatment of anthrax in chil-dren. Arch. Dis. Child., 42:181, 1967. 20. Kindler, D. : Recovery from anthrax meningitis.
md. Med., 21 :487, 1952.
Mobitz
Type
IIAtrioventricular
Block in a Newborn
Patients with congenital complete heart
block and no other cardiac lesion usually are asvmptomatic and have a normal axis and QRS
pattern on the electrocardiogram.1 The site of the block is usually in the region of the AV
node.2 Another less common type of congenital
AV block has an abnormal QRS complex on the
electrocardiogram.3 Death from Stokes-Adams attack has been recorded in infancy in this group.4 Mobitz Type II block is very rare in
infancy but may precede complete heart block
which requires ventricular pacing. The purpose of this report is to illustrate Mobitz Type II
heart block in a newborn which progressed to
complete block. Because of Stokes-Adams
at-tacks, ventricular pacing was required.
CASE REPORT
K.M., a female, was born September 22, 1969 bs’ caesarian section because of slow fetal heart rate.
The mother’s pregnancy was uneventful but
throughout she took fluphenazine (Prolixin) for schizophrenia. The birth weight was 2.6 kg and the Apgar score was 8, increasing to 9 five minutes later. The slow heart rate persisted and ranged
be-tveen 35 and 60. She was an active and acyanotic
infant with a good volume, regular pulse of 40 per minute. There were no signs of congestive failure. The left ventricular apex was prominent and there was a grade Il/VI mid-systolic murmur and occa-sional 4th heart sounds. The chest x-ray showed mild cardiomegaly. The initial electrocardiogram at 6 hours showed varying atrio-ventricular block at 2:3: 1 or 4: 1 ( Fig. 1 ) with left axis deviation, right branch bundle block. There is also fixed coupling
(ventricular Bigeminy) and 2:1 heart block with right bundle block. At 13 hours a stable pattern was recorded ( Fig. 2 ). Isoprenaline infusion at a
rate of 0.09 mg/minute increased the ventricular rate to 90 and the atrial to 200. Atropine also in-creased atrial and ventricular rate. Continuous electrocardiographic monitoring over the next 48 hours did not show any episodes of ventricular tachvcardia or asvstole. Cardiac catheterization was performed at 2 weeks of age. The pulmonary artery pressure was 52/3 ( mean 10 ) and the right ventricle 55/7 mm Hg. These high pressures were interpreted as resolving pulmonary hypertension in the newborn; and the large stroke volume from the slow heart rate. The right atrium had a mean of 2 and the left mean of 6 mm Hg. A right atrial cineangiogram was normal as were all other cham-bers subsequently visualized. Attempts were made to record the bundle of His by electrography but this was unsuccessful. The infant, on no therapy, was followed at regular intervals and at about 3 months developed complete heart block ( Fig. 3) with a rate of 40. At the age of 18 months she was again admitted following a syncopal episode. Her
resting heart rate was 35 beats/minute. Some 18
