Download Cutaneous Larva Migrans, Sacroileitis, and Optic Neuritis Caused

Cutaneous Larva Migrans, Sacroileitis, and Optic Neuritis
Caused by an Unidentified Organism Acquired in Thailand
Israel Potasman, Michael Feiner, Eldad Arad, and Zvi Friedman
A relative afferent pupil defect (Marcus-Gunn pupil)
was also noted. A presumptive diagnosis of optic neuritis was made, but no treatment given.
Two weeks later she returned to the clinic with a
painful, photophobic right eye. Her visual acuity was 6/15’
meters with ecentric movements. The visual field in the
right eye showed peripheral scotomata. Slit lamp examination revealed keratic precipitates and a moving larva
attached at one end to the internal corneal membrane.
The larva, which measured less than 1 mm, was seen by
two senior ophthalmologists but quickly dlsappeared. Fundoscopy disclosed panuveitis and vitreitis. Infectious disease consultation was requested. A careful history revealed
that the patient had traveled to Thailand in October of
1995. Prior to travel she had been vaccinated against
Japanese B encephalitis, hepatitis A and B, and typhoid
fever (Vivotifa). During her 1 month trip she visited the
cities of Bangkok, Kanchanaburi, Chiang-Mai, and the
islands of Phuket and KO-Phi-phi. She strongly denied
consuming seafood, raw, or cooked, but recalled swimming in a stream. In December 1995 a pruritic, migrating, linear rash appeared on her right buttock. The rash
advanced at a rate of a few millimeters per day. Since it
continued for a few months a biopsy was carried out,
demonstrating eosinophils and flame figures compatible
with scabies or eosinophilic cellulitis, but no organisms
were seen. The lesion remained stable for 5 months and
the patient was not treated. In August 1996 she started
suffering from a lower backache. Radioisotopic bone scan
done 1 month later showed a slightly increased uptake
in the right sacroiliacjoint. Computerized tomography
of the lumbosacral region was completely normal, and
magnetic resonance imaging (MFU) of the hip and sacroiliac joints showed some fluid in the right pelvis. A
blood count revealed an eosinophilia of lo%, but rheumatologic serology was negative. Serum IgE was however
elevated to a level of 300 p/mL (n < 100).
Since a histologic preparation of the larva was
unavailable, and none of the involved physicians had
previously encountered such a case, we contacted several ID experts in Israel, the Centers for Disease Control (CDC) hotline in Atlanta, the National Institute of
Health (NIH), the Tropical School of Medicine in London, and the University of Mahidol in Thailand.
Three major questions were raised: (1) What is the
most likely diagnosis? (2) What would the optimal
Case Report
A 32-year-old immunologist presented to the ophthalmology outpatient clinic in August 1997 with a
headache of 3 weeks’ duration and blurred vision of the
right eye.
Her past medical history was remarkable for childhood asthma and penicillin and aspirin allergy. She was
in the 10th week of her first pregnancy, and afebrile. Fundoscopy revealed a swollen and edematous disk. The
visual field in the right eye demonstrated an enlargement
of the blind spot, and her visual acuity was 6/7 meters.
Israel Potasman, MD, Michael Feiner, MD, Eldad Arad, MD,
and Zvi Friedman, MD: Infectious Diseases Unit, and
Ophthalmology Department, Bnai Zion Medical Center, the
Rappaport School of Medicine,Technion, Haifa, Israel.
Reprint requests: Israel Potasman, MD: Head, Infectious
Diseases Unit, Bnai Zion Medical Center, PO Box 4940, Haifa
31048, Israel.
JTravel Med 1998; 5223-225.
223
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We report the case of a 32-year-old pregnant woman
with an unidentified intraocular parasite. The parasite,
which had been acquired in Thailand, caused cutaneous
larva migrans, sacroileitis, and 2 years later optic neuritis and panuveitis. The patient was successfully treated with
ivermectin and albendazole. The diagnostic possibilities
of this peculiar presentation are discussed.
Parasitic infections are a leading cause of medical
problems in travelers to tropical countries.’ While most
parasites cause gastrointestinal problems, some may
migrate throughout the body and lodge in critical organs.
Ocular parasitic infections may occur by direct inoculation onto the eye,’ or incidentally during systemic
migration. Subconjunctival parasites are easily diagnosed
by removal and careful microscopic e ~ a m i n a t i o nPar.~
asites, which lodge within the eye, are more difficult to
diagnose, especially if not removed. In this report we
describe a patient who presented with an intraocular parasite causing optic neuritis and panuveitis, 2 years after
travel to Thailand.
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Discussion
This patient had an unidentified ocular parasite that
caused optic neuritis and uveitis. Circumstantial evidence links the acquisition of the organism to Thailand
2 years prior to presentation.
An enigmatic case like the one presented can be
approached via several diagnostic paths. First, epidemiologically-where and when had she been traveling,
and what types of activities had she engaged in. Second,
parasitologically-what type of organism would survive
iil the body for 2 years and fit the description of the two
ophthalmologists. Third, clinically-what type of organism causing cutaneous larva migrans (CLM) and
sacroileitis,would end up in the eyeball and cause optic
neuritis.
The Gideon software4 lists 51 parasites indigenous
to Thailand, of which 12 have a life cycle that includes
migration through body organs. The most appealing
one is Gnathostoma spinigerum.5,6Gnathostoma is capable of surviving for long years in the human body and
causing an intraocular infe~tion,~.'
but has never been
reported to cause either optic neuritis or sacroileitis.
Gnathostoma is not uncommon among Thai stray dogs,
4.1% of which were found to harbor parasitic nodules
in their stomachs.' In fact, Gnathostoma is the most
common tissue parasite, and the second most common
ocular parasite in Thailand.'" The third stage larva of this
organism may find its way to the human gut after eating an infected second intermediate host-fish or frog5
but our patient denied consuming any marine animals
or drinlung tap water. One report, however, indicates that
Gnathostoma may be acquired after eating ducks or
chicken." This diagnostic possibility had essentially been
opposed in light of the size of the third stage larva which
usually measures 2.2-3.5 mm, and the negative serology
(the sensitivity of which is unknown). Another potential candidate was the larva of Angiostrongylns cuntonensis, the rat lung worm, whch is also endemic in Thailand.'"
A . cantonensis infection is also contracted by eating
infected marine products, which makes it an unhkely candidate. The fifth stage larva ofA. cantonensis measures 0.5
m m in length which fits our case, but ordinarily causes
an abrupt central nervous system disease within a mean
period of 2 weeks.'"
Other diagnostic possibilities such as anisakiasis,
heterophyiasis, hookworms, opisthorchis, paragonimus,
sparganosis," and trichinosis can be ruled out either on
epidemiologic grounds or on the morphological appearance of the larva seen in the anterior chamber. A different approach to this case is by looking at it as cutaneous
larva migrans. CLM, which was the first symptom of our
patient, can be caused by several organism^.'^ Ancylostomu
braziliensis, the most typical organism in this group, does
not generally penetrate to deeper tissues. However, a
closely related organism-A. caninum caused an epidemic of eosinophilic gastroenteritis in 93 cases in Australia,14attesting to the migratory capacity of Anylostoma.
Strongyloides stercoralis infection begins with exposure of
the skin to filariform larvae that reside in fecally contaminated moist soil. Our patient has indeed exposed herself by walking barefoot several times during the trip. A
syndrome of chronic, persisting infection with S. stercorulis causing cutaneous and enteric symptoms has been
described in World War I1 and Vietnam veterans. However, S. stevcovulis has not been reported to cause optic neuritis, and serology for this organism (90% sensitivity)
was negative. Bunastomum phlebotomum causes a papular
skin lesion and clears within 2 weeks. The possibility of
G. spinkerum as a cause for cutaneous larva migrans has
been described above.
Another organism commonly causing ocular larva
migrans is Toxocaru." Although serology for Toxocara was
tested by two different laboratories, and found negative,
it deserves consideration. Toxocara is a well-known agent
of visceral larva migrans, and may also cause chronic
urticaria," but not the typical form of cutaneous larva
migrans. In contrast, the eosinophilia exhibited by our
patient, and the fact that T canis is prevalent among stray
dogs in ThailandI7 makes it a diagnosis hard to exclude.
An entity closely related to toxocariasis is "diffuse
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therapy be? (3) How can the eye be saved? At this stage,
the outcome of the pregnancy was of secondary importance to the patient and the team.
The patient was afebrile and her physical examination, excluding the eye, was normal. Her complete
blood count (CBC) showed an eosinophilia of 5%;
chemistry was normal. A blood culture and three blood
films for Filaria taken at 14:00, 18:OO and 02:OO hours
were negative. Intravenous hydrocortisone was started
to reduce the inflammatory damage to the eye. After 48
hours, when some improvement was already evident she
received a single dose of 18 mg of Ivermectin. No
untoward effects were noted for the next 48 hours.
Since the diagnosis of gnathostomiasis had been entertained, it was decided to put her on oral albendazole 400
mg dady for 3 weeks. Although no proof of teratogenicity
has been claimed with this agent, the patient chose to
undergo an abortion.
In an attempt to solve the case using serologies, sera
were sent out for Filarial antibodies (EIA, at the NIH),
Gnathostoma (EIA+WB),Toxocara (EIA),Paragonimus
(Allin Thailand; Toxocara was also tested at the BenGurion University,Beer-Sheba) and Strongyloides (EIA,
at the CDC),but all returned negative. During the ensuing months her visual acuity has gradually improved
and by 3 months was 6/9 meters in the affected eye.
Journal o f Travel M e d i c i n e , Volume 5, N u m b e r 4
P o t a s m a n , C u t a n e o u s Larva Migrans, Sacroileitis, a n d Optic Neuritis
unilateral subacute neuroretinitis” (DUSN).18-2” Several
organisms have been described in association with this
entity: Toxocara,20 Alaria mesocercaria, Baylisascaris procyonis,” and others. Taken as a whole, the clinical picture
of our patient, excluding the ocular manifestations, does
not fit DUSN.
In summary, this extraordinary case has generated
two (perhaps conflicting) reflections. O n one hand,
there is a great temptation in unknown cases to favor
the possibility of a yet unreported organism. O n the other
hand, the possibility of a common organism (G.
spinigerum, for example) presenting uncommonly seems
equally appealing.
’’
Acknowledgments
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We are indebted to Drs. M. Ephros (Haifa),S. Berger
(Tel Aviv), A. Bryceson (London), P. Suntharasamai
(Bangkok), and P. M. Schantz, CDC, Atlanta for their
advice. We are also grateful to Drs. Thom B. Nutman,
Laboratory of parasitic diseases, NIH, M. Wilson, reference immunodiagnostic lab, C D C , Atlanta, and W.
Chaicumpa, Mahidol University, Bangkok, Thailand for
performing the serological tests.
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