Case Report

Brucellar Meningitis in an Infant—Evidence for Human Breast

Milk Transmission
by Nitin V. Tikare,a Basappa G. Mantur,b and Laxman H. Bidaria
a
Dr. Bidari’s Ashwini Institute of Child Health and Research Centre, Bijapur – 586103, Karnataka, India

Downloaded from https://academic.oup.com/tropej/article-abstract/54/4/272/1650418 by guest on 08 November 2019

b
Department of Microbiology, Belgaum Institute of Medical Sciences, Belgaum – 590001, Karnataka, India

Summary
Brucellar meningitis is relatively uncommon, especially in children younger than 1 year. We present
another case of meningitis due to Brucella melitensis biotype 1 in a 11-month-old infant transmitted by
breast milk. This is the first report of successful isolation of B. melitensis from the breast milk in the
literature. Babies of infected nursing mother should be monitored closely for evidence of infection since the
breast milk is the source of transmission of brucellosis. It would be prudent to abstain from breast feeding
until infection of nursing mother has been eradicated. One should be aware of this in endemic areas.

Key words: Brucellosis, Meningitis, Infant, Breast milk transmission, Breast milk culture, Brucella
melitensis.

Introduction abscess, chorea, facial palsy and ischemic attacks have

Brucellosis is one of the world’s major zoonoses that
still is of veterinary public health and economic
concern in many parts of the world. Human infection
can occur through consumption of infected raw milk,
raw milk products and raw meat. Brucellosis is almost
invariably transmitted to man from infected domestic
animals. Transmission from human to human, mainly
mother to child, has been reported but is very rare.
Human brucellosis is a multi-system disease that may
present with a broad spectrum of clinical manifesta-
tions, and its complications can affect almost all
organs and systems with varying incidence [1].
Neurological complications of brucellosis are uncom-
mon and occur in <5% of adults [2] and in <1% of
pediatric cases [3]. The nervous system can be involved
at the onset of illness, during convalescence, or
months after recovery from acute infection.
Meningitis, encephalitis, meningoencephalitis, brain
Acknowledgements
We thank Dr Satish K Amarnath, Medical Director
from Manipal Cure and Care, Bangalore, India, for
critical reviewing of the manuscript.
Correspondence: Dr Nitin V Tikare, Dr. Bidari’s Ashwini
Institute of Child Health and Research Centre, B.L.D.E
Road, Bijapur - 586103, Karnataka, India.
Tel: (91) 8352 - 263428, 261328; Fax: (91) 8352-262324;
E-mail <nitintikare@rediffmail.com>.
all been reported [4–7]. Brucellar meningitis is rela-
tively uncommon, especially in children younger than
1 year, with only three cases reported in the literature
[8–10]. We describe herein a case of meningitis due to
brucellosis that has occurred in a child of younger
than 1 year transmitted by breast milk.
Case Report
A 11-month-old, previously healthy exclusively
breast-fed female baby was admitted to Dr Bidari’s
Ashwini Institute of Child Health and Research
Centre, Bijapur with 7-day history of fever. She had
one episode of generalized tonic-clonic convulsions
and altered sensorium of 1 day duration. Physical
examination on admission revealed a temperature of
100 F. She was conscious but irritable with bulging
fontanellae. There was no rash or lymphadenopathy.
Liver was 4 cm and spleen was 2 cm below the costal
margins. There were no focal neurodeficits. There
was no history of contact with tuberculosis. Baby’s
father was a shepherd.
On investigation, haemoglobin was 8 gm%, leuco-
cyte count was 6700 mm 3 and platelet count was
73,000/mm3. Peripheral blood smear for malarial
parasites and Widal test were negative. Blood sugar
was 140 mg dl 1 and serum electrolytes were normal.
A cranial computed tomographic (CT) scan and chest
X-ray were normal. Examination of CSF sample
obtained by lumbar puncture disclosed a white blood
cell count of 210 cells mm 3 with 65% polymorphs
and 35% lymphocytes, a glucose level of 50 mg dl 1

ß The Author [2008]. Published by Oxford University Press. All rights reserved. For Permissions, please email: journals.permissions@oxfordjournals.org 272
doi:10.1093/tropej/fmn017 Advance Access Published on 21 February 2008
CASE REPORT
and a protein concentration of 320 mg dl 1.
Stains and cultures for bacteria, mycobacteria and
fungi were all negative. The patient was started on
Ceftriaxone (100 mg kg 1 per day in two divided
doses) intravenously. In view of the occupation of
parents, her serum was sent for Brucella antibodies
which was found positive at a dilution of 1:320 by
standard tube agglutination test (SAT). Blood and
bone marrow specimens were sent for Brucella culture.
Two days later, examination of CSF showed a WBC
count of 200 cells mm 3, glucose level of 60 mg dl 1
and a protein concentration of 300 mg dl 1. A rose
bengal plate agglutination (RBPT) and SAT in CSF
were negative on initial evaluation. Culture of CSF for
Brucella was advised. As the baby was exclusively on
breast feed and was not exposed to raw animal milk or
animals in house and mother also had fever on and
off, mother’s serum for Brucella antibody and blood
and breast milk cultures for Brucella were sent.
With a diagnosis of brucellar meningitis, the patient
was started on gentamycin intravenously (7.5 mg kg 1
per day in two divided doses), oral trimethoprim
(10 mg kg 1 per day), sulphamethoxazole (50 mg kg 1
per day) and rifampicin (20 mg kg 1 per day) to which
she became afebrile on 7th day and her activities
improved. Bone marrow specimens of baby inocu-
lated onto Castaneda’s biphasic medium consisting of
trypticase soy agar and broth grew Brucella melitensis
biotype1; however, blood and CSF samples yielded no
growth. The SAT performed on mother’s blood
was positive for Brucella antibodies (titer,1:1280).
Four milk samples (5 ml each) and two blood samples
(5 ml each) cultured onto trypticase soy agar and
broth were found positive for B. melitensis biotype 1 in
two samples of milk and both samples of blood.
She was started on injectable streptomycin 1 gm
for initial 21 days together with oral tetracycline
2 gm in four divided doses for 45 days and was advised
to stop breast feeding. A repeat lumbar puncture
performed on 10th day of treatment showed 12
cells mm 3 (all lymphocytes), protein 140 mg dl 1,
and sugar was 70 mg dl 1, and now the CSF was
positive for Brucella antibodies by RBPT and
demonstrated agglutinins to a dilution of 1:160 by
SAT. Brucella antibodies by SAT in serum were
1:5120 on 10th day of treatment. As parents wanted to
go home early, the patient was discharged on request
with injectable streptomycin (20 mg kg 1 per day 2
months), oral trimethoprim–sulphamethoxazole and
rifampicin (4 months). It was planned to followup the
infant for antibodies and monitor for renal, hepatic
and auditory functions, but the infant was lost for
follow-up.
Discussion
Brucellosis is primarily a disease of domestic animals:
dogs, pigs, sheep, goats and camels. Humans may be
infected by exposure to raw meat, to fresh or
Journal of Tropical Pediatrics Vol. 54, No. 4
inadequately pasteurized milk or milk products or
by direct exposure to infected animals. The species of
Brucella that may infect man are: B. melitensis,
B. suis, B. abortus and B. canis. B. melitensis
colonizes ovine stock and is the most frequent
cause of brucellosis, globally in humans; it is the
only agent of brucellosis in this part of the country
[1, 5]. Brucellosis is said to be a rare disease in
childhood—a puzzling phenomenon as children are
large consumers of milk and milk products. The
Downloaded from https://academic.oup.com/tropej/article-abstract/54/4/272/1650418 by guest on 08 November 2019
explanation for this is unclear. However, in areas,
where B.melitensis is endemic, pediatric cases are
seen [5, 11] occurring mainly in school age group.
Important routes of transmission for brucellosis in
school age group are the ingestion of contaminated
dairy products, close contact with infected animals or
inhalation of infected aerosols. Although person to
person transmission of brucellosis is rare in children,
there have been few cases reported in the literature
including breast feeding [12–15], but invariably one
should think of person to person transmission like
congenital, neonatal blood transfusion/bone marrow
transplantation and breast feeding [16], when bru-
cellosis occurs in children especially younger than
1 year of age. Although possible breast milk
transmission has been documented for Brucellae,
the Brucellae could not be isolated from the breast
milk in any of the reported cases. To our knowledge,
we describe the first case of brucellosis in a baby
transmitted by breast feeding with successful isola-
tion of B. melitensis from the breast milk. The
reasons for efficiently picking up Brucella from milk
in the case reported here could be attributed to
collection of milk for culture before administration of
antibiotics, the use of biphasic medium with the
prolonged incubation period of 30 days, and multiple
samples cultured. Given that the mother had active
brucellosis, the baby had no known other risk factors
predisposing her to B. melitensis infection, and breast
milk had grown B. melitensis, and isolates from the
samples of both mother and baby were indistinguish-
able biochemically and serologically, breast feeding is
the route of transmission in the case presented here.
Our observations, in accordance with previous
published reports concerning breast feeding trans-
mission, support the possibility of person to person
transmission by breast feeding. The baby in our case
responded well to IV gentamycin given for initial 7
days along with oral co-trimoxazole and rifampicin.
Brucellosis is known for complications. The
incidence of neurologic involvement in brucellosis is
not well known. The range reported in the literature
varies from 0 to 25% and depends on the criteria
required to make the diagnosis [2]. Most of the
authors, nowadays, require evidence of direct central
nervous system involvement by infecting Brucellae as
the diagnostic criterion [2]. On this basis, the
incidence of neurobrucellosis is at present <5% of
the adult cases of brucellosis and <1% of pediatric
273

patients of brucellosis. Neurobrucellosis, especially,
meningitis due to Brucellae is very uncommon in
children younger than 1 year of age. A MEDLINE
search (1966–2007) revealed only three cases con-
cerning the meningitis of brucellar origin in children
younger than 1 year.
It is well recognized that, in the majority of cases,
the diagnosis can be made after history taking alone
[17]. The history of travel to endemic countries along
with exposure to animals and exotic foods are usually
critical to making the clinical diagnosis of brucellosis
[18, 19]. The baby’s parents’ occupational exposure
to the animals, in this case, alerted us to the strong
possibility of exposure of mother to Brucella. After
this history became known, serologic and culture
studies for Brucella of mother as well as baby were
obtained leading to the correct diagnosis, otherwise
the diagnosis would have been missed or delayed.
The suspicion for the diagnosis of neurobrucellosis
should always be entertained in patients, living in or
coming from endemic areas, who present with
obscure, confusing and mixed constitutional or
neurologic symptoms for which no obvious explana-
tion is readily apparent. Fever was the only out-
standing constitutional feature of our patient’s
illness. If these non-specific complaints are associated
with specific focal neurologic manifestations, the
establishment of the diagnosis may not be difficult.
The neurologic manifestations like convulsions,
altered sensorium noted in our patient along with
the presence of biochemical parameters and Brucella
antibodies and absence of other pathogens have
suggested brucellar meningitis. The CSF picture of
polymorphonuclear pleocytosis seen in our case,
which is in contrast to lymphocytic pleocytosis
reported by others [2], could be due to acute history
and early detection of meningitis. The delayed
detection of brucellar antibodies in the CSF can be
attributed to less sensitive nature of agglutination
tests for neurobrucellosis. It is worthwhile noting
that the systemic physical signs of brucellosis were
inconspicuous: hepatosplenomegaly was observed,
other abnormal findings such as arthritis and lymph
adenopathy, thought to be present in patients with
generalized brucellosis, were not detected. There are
no data in the medical literature that permit the clear
delineation of an ideal regimen and duration of
therapy for brucellar meningitis in children. After
initiation of therapy, the clinical and biochemical
parameters of the patient returned slowly to normal
and improved steadily.
In summary, babies of infected nursing mother
should be monitored closely for evidence of infection
since the breast milk is the source of transmission of
brucellosis, and it would be prudent to abstain from
breast feeding until infection of nursing mother has
been eradicated and one should be aware of this in
endemic areas. Brucellosis may be a deceptive
infectious disorder, particularly if it invades the
274
CASE REPORT
nervous system. Neurobrucellosis is not a commonly
encountered infection, fortunately, especially in
children younger than 1 year, but should be
considered carefully in the differential diagnosis,
especially in endemic areas like India where brucel-
losis has not been eradicated. Effective antibiotic
therapy is available so that early diagnosis and
treatment are matters of great importance.
Downloaded from https://academic.oup.com/tropej/article-abstract/54/4/272/1650418 by guest on 08 November 2019
References
1. Mantur BG, Biradar MS, Bidri RC, et al. Protean
clinical manifestations and diagnostic challenges of
human brucellosis in adults: 16 years’ experience in an
endemic area. J Med Microbiol 2006;55:897–903.
2. Bouza E, Garcia de la, Torre M, et al. Brucellar
meningitis. Rev Infect Dis 1987;9:810–22.
3. Lubani MM, Dudin KI, Araj GF, et al. Neurobrucel-
losis in children. Pediatr Infect Dis J 1989;8:79–82.
4. Shakir RA, Al-Din AS, Araj GF, et al. Clinical
categories of neurobrucellosis: a report on 19 cases.
Brain 1987;110:213–23.
5. Mantur BG, Akki AS, Mangalgi SS, et al. Childhood
brucellosis – a microbiological, epidemiological and
clinical study. J Trop Pediatr 2004;50:153–7.
6. Tsolia M, Drakonaki S, Messaritaki A, et al. Clinical
features, complications and treatment outcome of
childhood brucellosis in central Greece. J Infect
2002;44:257–62.
7. Bingol A, Togay–Isikay C. Neurobrucellosis as an
exceptional cause of transient ischemic attacks. Eur J
Neurol 2006;13:544–8.
8. Rangel Guerra R, Martinez HR, de Leon Flores L.
Neurobrucellosis. Report of five cases and literature
review. Rev Invest Clin 1982;34:62–8(In Spanish).
9. Swick HM. Brucella meningoencephalitis in childhood.
Neuro- pediatrics 1981;12:330–6.
10. Bethenod M, Nivelon JL, Rouchon A, et al. Malta
fever in a pure meningeal form in an infant. Pediatrie
1966;21:606–8(In French).
11. Sharda DC, Lubani M. A study of brucellosis in
childhood. Clin Pediatr 1986;25:492–5.
12. Lubani M, Sharda D, Helin I. Probable transmission of
brucellosis from breast milk to a newborn. Trop Geogr
Med 1988;40:151–2.
13. Palanduz A, Palanduz S, Guler K, et al. Brucellosis in a
mother and her young infant: probable transmission by
breast milk. Int J Infect Dis 2000;4:55–6.
14. Arroyo Carrera I, Lopez Rodriguez MJ, Sapina AM,
et al. Probable transmission of brucellosis by breast
milk. J Trop Pediatr 2006;52:380–1. Epub 2006 May 30.
15. Singer R, Amitai Y, Geist M, et al. Neonatal brucellosis
possibly transmitted during delivery. Lancet
1991;338:127–8.
16. Lubani MM, Dudin KI, Sharda DC, et al. Neonatal
brucellosis. Eur J Pediatr 1988;147:520–2.
17. Platt R. Two essays on the practice of medicine. Manch
Univ Med Sch Gaz 1947;27:139–45.
18. Young EJ. An overview of human brucellosis. Clin
Infect Dis 1995;21:283–90.
19. Dames S, Tonnerre C, Saint S, Jones SR. Clinical
problem-solving. Don’t know much about history.
N Engl J Med 2005;352:2338–42.
Journal of Tropical Pediatrics Vol. 54, No. 4