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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 Downloaded from http://jtm.oxfordjournals.org/ by guest on November 18, 2016 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. 224 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 Downloaded from http://jtm.oxfordjournals.org/ by guest on November 18, 2016 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 References 1. Wittner M, Tanowitz H. Intestinal parasites in returned travelers. Med Clin North Am 1992; 76(6):1433-1448. 2. Newman P, Beaver P, Kozarsky P, Waring G. Fly larva adherent to corneal endothelium.Am J Ophthalmol 1986; 102(2): 2 11-2 16. 3. Ottesen E. Filariases and tropical eosinophilia. In: Warren K, Mahmoud A, eds. Tropical and geographical medicine. New York: McGraw-Hill, 1984:390-412. 4. GIDEON-Global Infectious Diseases and Epidemiology Network [PC program]. 3.5 version. Raniat Hasharon, Israel: CY Informatics, 1997. 5. Bunnag T. Gnathostomiasis. In: Strickland G, ed. Tropical medicine. 7th Ed. Phladelphia: WB Saunders,1991:764-767. 6. Rusnak J, Lucey D. Clinical gnathostomiasis:case report and review of the Enghsh-language literature. Clin Infect Dis 1993; 16:33-50. 7. BiswasJ, Gopal L, Sharma T,Badrinath S. Intraocular Gnathostoma spinigerum. Retina 1994; 14:438-444. 8. Funata M, Custis P, Cruz Zdl, Juan Ed, Green W. Intraocular gnathostomiasis.Retina 1993; 13:240-244. 9. Maleewong W, Pariyanonda S, Sitthithaworn P, et al. Seasonal variation in the prevalence and intensity of canine Gnathostoma spinigerum infection in northeastern Thailand. J Helminthol 1992; 66(1):72-74. 10. Teekhasaenee C, Ritch R , Kanchanaranya C. Ocular parasitic infections in Thdand. Rev Infect Dis 1986;8(3):35G356. 11. Daengsvang S, Thienprasitthi P, Chomcherngpat €? Further investigations of natural and experimental hosts of larvae of Gnuthostoma spin&erirm in Thailand. Am J Trop Med Hyg 1966; 15:727-729. 12. Kittiponghansa S, Tesana S, Ritch R. Ocular sparganosis: a cause of subconjunctival tumor and deafness. Trop Med Parasitol 1988;39(3):247-248. 13. Neafie R, Meyers W. Cutaiieous larva migrans. In: Strickland G, ed. Tropical niedcine. 7th Ed. Philadelphia:WE3 Saunders, 1991:773-775. 14. Porciv Ci-oeseJ. Human eosinophilic enteritis caused by dog hookworm Ancylostoma caninum. Lancet 1990; 335 (8701): 1299- 1302. 15. Overgaauw €? Aspects of Toxocara epidemiology: human toxocariasis. Crit Rev Microbiol 1997; 23(3):215-231. 16. Humbert P, Buchet S, Barde T. Toxocariasis. A cosniopolitan parasitic zoonosis.AUerg Immunol 1995;27(8):284-291. 17. Hinz E. Intestinal helminths in Bangkok stray dogs and their role in public health. Zentralbl Bakteriol Mikrobiol Hyg 1980; 171(1):79-85. 18. McDonald H, Kazacos K, Schatz H, Johnson R . Two cases of intraocularinfection with Alaria mesocercaria (Trematoda). Am J Ophthalmol 1994; 117(4):447-455. 19. Goldberg M, Kazacos K,Boyce W, Ai E, Katz B. Diffuse unilateral subacute neuroentinitis. Morphometric, serologic, and epidemiologic support for Baylisascaris as a causative agent. Ophthalmology 1993; 1OO(11):1695-1701. 20. Oppenheim S, Rogell G, Peyser R. Diffuse unilateral subacute neuroretinitis. Ann Ophthalmol 1985; 17(6):336-338. Downloaded from http://jtm.oxfordjournals.org/ by guest on November 18, 2016 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. 225