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Pediatrics and Neonatology (2013) 54, 56e59

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CASE REPORT

Eosinophilic Meningitis Caused by


Angiostrongylus cantonensis in an
Adolescent With Mental Retardation and Pica
Disorder
Chang-Wei Hsueh a, Huan-Sheng Chen b, Chen-Hua Li c,d, Yu-Wei Chen c,d,*

a
Department of Pediatrics, Landseed Hospital, Taoyuan, Taiwan
b
Division of Infection, Department of Internal Medicine, Landseed Hospital, Taoyuan, Taiwan
c
Department of Neurology, Landseed Hospital, Taoyuan, Taiwan
d
Department of Neurology, National Taiwan University Hospital, Taipei, Taiwan

Received Mar 9, 2011; received in revised form Aug 29, 2011; accepted Sep 14, 2011

Key Words Eosinophilic meningitis or encephalitis is a rare disorder and is most commonly caused by An-
acute urinary giostrongylus cantonensis. Humans are accidentally infected when they ingest raw snails or
retention; vegetables contaminated with the parasite larvae. Because of the improvement in sanitary
Angiostrongylus food handling practices, the occurrence of A. cantonensis eosinophilic meningitis has been
cantonensis; decreasing in Taiwan in recent decades. The common symptoms and signs of eosinophilic
eosinophilic meningitis are severe headache, neck stiffness, paresthesia, vomiting, nausea, and fever.
meningitis; Acute urinary retention is a rare presentation. We report a case of A. cantonensis eosinophilic
pica meningitis in an intellectually disabled patient who presented with acute urinary retention
without any other meningeal signs. The patient received supportive treatment with corticoste-
roid therapy and was discharged and received urinary rehabilitation at home.
Copyright 2012, Taiwan Pediatric Association. Published by Elsevier Taiwan LLC. All rights
reserved.

1. Introduction

Eosinophilic meningitis or myelitis can be found in various


* Corresponding author. Landseed Hospital, Number 77, Guangtai parasitic infestations of the central nervous system (CNS),
Road, Pingzhen City, Taoyuan County 324, Taiwan. in inflammatory diseases such as tuberculous meningitis,
E-mail address: chanyw@landseed.com.tw (Y.-W. Chen). cerebrospinal syphilis, viral and fungal meningitis, in drug

1875-9572/$36 Copyright 2012, Taiwan Pediatric Association. Published by Elsevier Taiwan LLC. All rights reserved.
http://dx.doi.org/10.1016/j.pedneo.2012.11.005
Eosinophilic meningitis with A. cantonensis 57

allergies, in multiple sclerosis, and in neoplasms such as


malignant lymphoma, Hodgkin disease, and leukemia.1
Among the parasitic agents, Angiostrongylus cantonensis
and Gnathostoma spinigerum are the most common path-
ogens in eosinophilic meningitis. G. spinigerum occurs in
Southeast Asian countries, mainly in rural communities
where the ingestion of raw snails or insufficiently cooked
poultry is the main source of this type of infection. Eosin-
ophilic meningitis caused by G. spinigerum has never been
reported in Taiwan.2e4
A. cantonensis is the most common parasitic cause of
eosinophilic meningitis in Taiwan.5 A. cantonensis is
a nematode that inhabits the pulmonary arteries and heart
of rodents. Humans may be accidentally infected by the
parasite. The common initial presentations of eosinophilic
meningitis are headache, neck stiffness, paresthesia,
vomiting, nausea, and fever. Acute urinary retention is
a symptom rarely associated with the parasitic infection.
We report here a case of a patient who showed few
meningeal signs but was finally confirmed to have A. can-
tonensis eosinophilic meningitis.

2. Case Report

A 15-year-old boy with moderate intellectual disability was


brought to the emergency department (ED) because of Figure 1 Radiograph of the kidneys, ureters, and bladder
progressive conscious disturbance for 2 weeks, reported by showing a pelvic mass with bowel displacement.
the attending teachers in the nursing home where he resided.
The associated symptoms included paucity of speech and On admission, the patient was afebrile but irritable,
decreased engagement in playing and other daily activity. He with E3V2M5 on the Glasgow Coma Scale. His general
had visited the pediatrics clinic and ED for irritability and physical examination was unremarkable, except for
abdominal pain six times within the preceding 2 weeks. tachycardia (111/min) and elevated blood pressure (140/
Physical examination at the first visit to the ED revealed 97 mmHg). His neck was supple. Neurologic examination
normal body temperature (36.2 C), elevated blood pressure showed isocoria with retained light reflex. Other examina-
(152/100 mmHg), and mildly distended abdomen. No neck tion with limited compression and cooperation showed
stiffness or increased chin-to-chest distance was present. A weakness of all four limbs (2e3/5 on the Medical Research
laboratory study showed leukocytosis (white blood cell Council Scale), which was greater in the lower extremities,
count: 16.75  109/L) with mild eosinophilia (5.6%). characterized by difficulty bending the knees. Increased
Nephrotic syndrome had been diagnosed 8 years previously, deep tendon reflex of knee jerks was recorded, without
and the patient was taking regular oral steroids (predniso- obvious Babinski sign bilaterally. There was no edema in his
lone, 45 mg/day) before admission. A radiograph of the extremities, face, or scrotum.
patients abdomen showed a normal bowel-gas pattern. He The patients leukocyte counts in peripheral blood kept
was then discharged from the ED. elevating, from 16.75  109/L at presentation to
In the following week, this patient came to the hospital 21.06  109/L at admission. The percentages of bandform
twice because of abdominal pain and shaking of the neutrophils increased simultaneously from 5.6% to 26%.
shoulders. A follow-up laboratory study 1 week later Serum albumin concentration was 3.7 g/dL. Routine
showed progression of leukocytosis (19.9  109/L) and urinalysis yielded negative results.
eosinophilia (16%). We suspected that the patients clinical symptoms may
Difficulty in urination had been noted on Day 8 of the have been caused by a central nervous system infection. A
illness and was associated with decreased speech. The lumbar puncture was performed with opening pressure of
patient came to our ED again, and abdominal plain film 210 mmH2O. The color of cerebrospinal fluid (CSF) was
showed a pelvic mass suggesting urinary bladder distension whitish turbid, and the analysis showed a white blood cell
with bowel displacement. Transurethral catheterization count of 0.837  109/L, with 86% eosinophils and 14%
revealed 1650 mL of residual urine (Figure 1). lymphocytes. The CSF chemistry showed a glucose level of
On Day 17, the patient became drowsy all day long and 2.5 mmol/L, and protein 106 mg/dL. Culture for CSF
was not able to stand up. He was admitted for further showed all negative results for virus, fungi, bacteria, and
management. Neither fever nor convulsion had been mycobacteria. Results of CSF polymerase chain reaction for
observed by his family and attending teacher since his first herpes simplex virus-1 and -2 were also negative. Magnetic
visit to our ED. However, the patients family and attending resonance imaging (MRI) study of the cervical spine showed
teacher reported that he had pica disorder, whereby he no compressive lesion (Figure 2), and MRI of the brain
consumed nonfood items outside the house. showed no enhancing or space-occupying lesions (Figure 3).
58 C.-W. Hsueh et al

hosts and rodents as definitive hosts. Human beings are


accidentally infected when they eat poorly cooked or raw
fish, slugs, or snails, or vegetables contaminated by infec-
ted rats. Most of the cases in Taiwan were reported before
the 1970s.12 Because of increasingly improved sanitary food
handling practices, only scattered cases have been re-
ported during the past several decades. In Taiwan, the
cases with eosinophilic meningitis caused by A. cantonensis
comprised children in the past three to four decades, and
adults in the past two decades. The decreased case
numbers in children may be related to the urbanization and
improvement of public health, leading to less frequent
exposure of children to snails and other intermediate host
animals.5
Institutionalized intellectually disabled children have
been reported to be prone to parasitic infection.14 Pica
disorder, defined as consumption of nonfood items, is one
of the most frequently observed eating disorders in people
with a developmental disability.15 The prevalence of pica in
institutionalized intellectually disabled people was re-
ported to be 25.8%.16 Patients with such an eating disorder
are prone to intake of the raw intermediate hosts of
A. cantonensis. Such patients require special supervision by
Figure 2 Sagittal T2-weighted MRI showing neither
staff, and several behavior interventions have been
compression nor intramedullary lesions.
reported.17
Diagnosis of A. cantonensis eosinophilic meningitis is
Based on the aforementioned clinical, laboratory, and confirmed by recovery of larvae from the central nervous
radiologic results, eosinophilic meningitis was demon- system, but the frequency of detecting these worms in
strated. The patients serum and CSF specimens were sent patients is very low.12 Presumptive diagnosis may be based
to the Department of Parasitology, College of Medicine, on laboratory findings in the blood and CSF, a history of
Kaohsiung Medical University. The antibody levels in the eating intermediate hosts, serologic tests, and clinical
serum and CSF of this patient were detected by enzyme- symptoms.6 The common symptoms and signs of A. canto-
linked immunosorbent assay by using an antigen with nensis eosinophilic meningitis are headache, neck stiffness,
a molecular weight of 204 kDa from fifth-stage worms paresthesia, vomiting, nausea, and fever. Abdominal pain
of A. cantonensis; that antigen had been purified using and weakness of the extremities are less common.18 Our
monoclonal antibodies.6 patient presented with acute urine retention, which is
After admission, we started empirical treatment with rarely reported.19,20
ceftriaxone, and 4 mg of dexamethasone was administered Acute urinary retention, which refers to the inability to
intravenously every 6 hours. A second lumbar puncture was void urine by oneself, is prevalent in older people and rare
performed 4 days after the first, and the follow-up CSF in children and adolescents.21 Here, we report a case of
analysis showed a white blood cell count of 0.171  109/L acute urinary retention that accompanied eosinophilic
with 25% eosinophils. After 10 days of dexamethasone meningitis in an adolescent with mental retardation who
administration, treatment was interrupted because of lived in a nursing home. The more common causes of acute
a urinary tract infection. The patients activities improved urinary retention in children include neurologic disorders,
and his sleep quality was better during hospitalization, with severe voiding dysfunction, urinary tract infection, con-
sequelae of urine retention. He was discharged with a Foley stipation, adverse drug effects, local inflammatory causes,
catheter inserted and received urinary rehabilitation at the locally invasive neoplasms, and obstructive lesions.22,23 The
nursing home for the disabled where he resided. neurologic disorders that accompany acute urinary reten-
tion include peripheral nervous diseases such as sacral
herpes and central nervous diseases such as aseptic
3. Discussion meningitis and eosinophilic meningitis.19,20,23 However, the
typical symptoms and neurologic signs of meningitis may be
Eosinophilic meningitis is a rare neurologic disorder that mild or even absent initially. As in our patient, there may
can be caused by infectious or noninfectious etiology. be no vomiting, fever, or neck stiffness initially, and the
Noninfectious causes include malignancy, intracranial CNS infection can be difficult to diagnose early. Most cases
foreign body, and medication.7 In eosinophilic meningitis of A. cantonensis eosinophilic meningitis are mild and self-
caused by infection, A. cantonensis and G. spinigerum are limiting. Lumbar puncture should be performed to relieve
the most common agents.8 Eosinophilic meningitis is most the headache caused by increasing intracranial pressure.
commonly caused by the rat lungworm A. cantonensis.9 It is Treatment is supportive and symptomatic and is based on
endemic in Taiwan, Southeast Asia, the Pacific islands, the use of analgesics with or without corticosteroids.12
Australia, and the Caribbean islands.10e13 The life cycle of The diagnosis could be difficult if the patient presents
A. cantonensis involves snails, slugs, or fish as intermediate few meningeal irritative symptoms, as in our case. Because
Eosinophilic meningitis with A. cantonensis 59

4. Schmutzhard E, Boongird P, Vejjajiva A. Eosinophilic meningitis


and radiculomyelitis in Thailand, caused by CNS invasion of
Gnathostoma spinigerum and Angiostrongylus cantonensis.
J Neurol Neurosurg Psychiatry 1988;51:80e7.
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