J Am Board Fam Pract: first published as 10.3122/jabfm.5.5.533 on 1 September 1992. Downloaded from Urinary Gnathostomiasis In A Laotian Refugee

William A. Norcross, M.D., Bryan N.jobnson, M.D., Theodore G. Ganiats, M.D., and Suzanne M. Siedler, CFNP

Gnathostoma spinigerum is an uncommonly en­ the swnmer he also noticed occasional lower ab­ countered parasite, even in a practice such as ours, dominal pain, which resolved spontaneously. His which hosts a Traveler's Clinic and provides care medical history was pertinent only for a "left in­ for a relatively large Southeast Asian population. guinal groin abscess" treated by incision and Gnathostomiasis is most commonly reported in drainage in Laos in 1974. He was afebrile and the Asia, particularly Thailand and Japan. 1 N onethe­ bowel sounds were active. The abdomen was soft less, gnathostomiasis is a potentially serious, even and nontender without mass or organomegaly. life-threatening, parasitosis that can be seen in The stool was positive for occult blood. No skin any primary care setting that provides health care lesions or subcutaneous masses were noted. Both for Southeast Asian refugee populations. As of 30 urine and stool were examined for ova and para­ September 1990, the Southeast Asian refugee sites. The California State Medical Laboratory population of the United States was estimated to identified Gnathostoma spinigerum in the urine total 957,100, with refugees in every state and specimen. Further studies failed to provide evi­ large populations in California (378,500), Texas dence of other parasitic infections. The patient (71,800), Washington (44,900), Minnesota declined blood studies and was lost to follow-up (34,900), and New York (34,300).2 Consequently, after the second clinic visit despite nwnerous ef­ although this parasite is reportedly rare in the forts to contact him. United States, family physicians could well en­ counter Southeast Asian refugee patients and Comment should be familiar with the clinical presentations Gnathostomiasis is most commonly reported from of this parasite. Southeast Asia and japan, but sporadic cases have We present a case of urinary gnathostomiasis in occurred in many other areas of the world, includ­ a Laotian refugee. To our knowledge, this case is ing Europe, Africa, Mexico, and South America. only the second reported in the United States. Hwnans are infected through the ingestion of raw The first case of urinary gnathostomiasis in the fish, which contains encysted laIVae in its flesh. United States was reported in 1984.3 Feral and domestic cats and dogs are the natural http://www.jabfm.org/ hosts of G. spinigerum, harboring the adult worms Case Report in the stomach wall and excreting eggs in the feces. A 36-year-old male native of Laos immigrated to The eggs then hatch in fresh water, and the mo­ the United States via a refugee camp in Thailand tile first-stage larvae penetrate cyclops, a cope­ during summer 1990. On 7 November 1990 he pod, which is eaten by such intermediate hosts as

came to the Family Medical Center of the Uni­ freshwater fish, frogs, snakes, and rats. When these on 26 September 2021 by guest. Protected copyright. versity of California, San Diego, Medical Center animals are ingested by feral or domestic dogs and with the chief complaint of passing bright red cats, the larvae migrate to the stomach wall and blood and worms in his urine approximately 20 mature to the adult stage within approximately times during the past swnmer. He had been 6 months. The cycle is then completed (Figure 1). asymptor:natic for the previous 2 months. During Hwnans are incidental hosts, infected by the inges­ tion of raw or undercooked fish. Man is infected by the third-stage larvae of Submitted, revised, 24 January 1992. From the Department of Community & Family Medicine, G. spinigerum, which are then incapable of further University of California, San Diego, School of Medicine, and the maturation. The third-stage larvae, which are University of California, San Diego, Medical Center, La Jolla. very active, are capable of migrating widely Address reprint requests to William A. Norcross, M.D., Depart­ ment of Community & Family Medicine, Family Medicine Resi­ throughout the body, producing myriad clinical dency Program H-8809, UCSD School of Medicine, 225 Dick­ manifestations. Therefore, there is no single or inson Street, San Diego, CA 92103-9981. typical presentation of gnathostomiasis. In fact,

Urinary Gnathostomiasis S33 J Am Board Fam Pract: first published as 10.3122/jabfm.5.5.533 on 1 September 1992. Downloaded from

Man

Domestic CatsJDogs

IIqsr (3rd Stage Larva)

Cyclops (freshwater Copepod) (2nd stage larva)

Figure 1. Life c:ytle of the fiIuIlbosloma splnlgerum. some infections are likely to be subclinical or shows a mild-to-moderate pleocytosis with high minimally symptomatic and remain undiag­ percentages of eosinophils, elevated protein, nor­ nosed.I Urinary tract involvement, as docu­ mal glucose, and a mildly elevated opening pres­ mented in our patient, is apparendy an unusual sure.s Diagnosis rests upon identification of the http://www.jabfm.org/ manifestation of this disease.} The most dreaded parasite in the appropriate tissues or body fluids. manifestation of G. spinigerum infection is in­ Third-stage larvae are believed to survive for volvement of the central nervous system (CNS) as up to 10 years in the human host. 12 Infection with manifested by an eosinophilic myeloencephalitis, G. spinigerum is extremely unlikely in North cerebral hemorrhage, spinal cord involvement America; however, it is important for primary care with radiculopathy, cranial nerve palsies, and sub­ physicians, particularly those caring for Southeast on 26 September 2021 by guest. Protected copyright. arachnoid hemorrhage.4•s There are, however, a Asian patients, to recognize this entity. Unfortu­ wide variety of other manifestations, including nately, no consistendy effective medical therapy is creeping eruption {pruritic migratory swellings available for this parasitosis. 'When appropriate caused by larval migration),6 uveitis,7 skin and and feasible, surgical excision of the parasite is the subcutaneous nodules,8 gastrointestinal granulo­ treatment of choice. The parasite has been suc­ matous masses,9 cervical infection,IO penile infec­ cessfully excised from the skin,6 uveal tract,7 tion,l1 and pneumonitis. I2 Eosinophilia is often colon,9 uterine cervix,1O and penis. I I Infection can present in the peripheral blood smear, sometimes be disseminated, however, and surgical excision as high as 90 percent of leukocytes, I but serologic impossible, particularly for gnathostomal infec­ studies and other diagnostic efforts are frequendy tions of the central nervous system. Steroids can inconclusive. In clinical syndromes of CNS help cerebral edema, but no anthelmintic agent gnathostomiasis, the cerebrospinal fluid typically has proved effective for eNS gnathostomiasis.s In

534 JABFP Sept.-Oct. 1992 Vol. 5 No.5 one series of patients with eNS gnathostomiasis cially symptoms and signs of central nervous sys­ J Am Board Fam Pract: first published as 10.3122/jabfm.5.5.533 on 1 September 1992. Downloaded from reported from Thailand, none of whom were tem disease, including eosinophilic meningitis, treated with anthelmintics, 54 percent recovered radiculitis, encephalitis, and cerebral hemor­ fully, 38 percent recovered with neurologic re­ rhage. The third-stage larvae of G. spinigerum are sidua, and 8 percent died.s "strangers in a strange land" in the human host It is unknown what percentage of patients with and could migrate to any tissue in the body. undiagnosed infection caused by G. spinigerum remain asymptomatic or minimally symptomatic. As a result and because of the relative ineffective­ The authors thank Heidi Lenz, Debbie Johnston, and David ness of available pharmacologic therapies, pa­ Dixon for assistance in preparing this paper. tients in whom the parasite can be excised, but who are otherwise asymptomatic, should not References undergo investigation for foci of asymptomatic 1. BunnagT. Gnathostomiasis. In: Strickland Gf, edi­ infection. ' tor. Hunter's tropical medicine. Philadelphia: WB Saunders, 1991:764-7. The lack of convincing data in support of 2. Frelick B, Hamilton V, editors. Southeast Asian efficacy notwithstanding, the current recom­ refugees. Refugee Reports 1991; 12:16. mended pharmacotherapy for gnathostomiasis is 3. Horohoe JJ, Ritterson AL, Chessin LN. Urinary mebendawle 200 mg every 3 hours orally for gnathostomiasis.JAMA 1984; 251:255-6. 6 days.l3 Using mebendawle as therapy for 4. Punyagupta S, Bunnag T, Juttijudata P. Eosinophilic meningitis in Thailand. Clinical and epidemiological is considered investigational. gnathostomiasis characteristics of 162 patients with myeloencepha­ Other agents that have been used with varying litis probably caused by Gnathostoma spinigerum. success include diethylcarbamazine citrate, thia­ J Neurol Sci 1990; 96(2-3):241-56. bendazole, and albendazole.14 Bunnag, I who has 5. Schmutzhard E, Boongird P, Vejjajiva A. Eosino­ had extensive experience with gnathostomiasis, philic meningitis and radiculomyelitis in Thailand is not convinced that any anthelmintic agent caused by CNS invasion of Gnathostoma spinigerum and AngiostrungyJus CIlntunensis. J Neural Neurosurg has been proved efficacious. Nevertheless, Psychiatry 1988; 51:80-7. mebendawle is relatively well tolerated and has 6. Bhaibulaya M, Charoenlarp P. Creeping eruption an excellent safety profile. Side effects are limited caused by Gnathostoma spinigerum. Southeast Asian to abdominal pain and diarrhea, which have gen­ J Trop Med Public Health 1983; 14:266-8. erally occurred when treating patients infected 7. Kittiponghansa S, Prabriputaloong A, Pariyanonda S, Ritch R Intracameral gnathostomiasis: a cause of with large burdens of (nongnathostomal) intes­ anterior uveitis and secondary glaucoma. Br J Oph­ tinal parasites. is Because mebendawle was tera­ thalmo11987; 71:618-22. http://www.jabfm.org/ togenic in rats, it is not recommended for preg­ 8. Raturi U, Burkhalter W. Gnathostomiasis extema: a nant women. IS case report.J Hand Surg 1986; 11(5):751-3. The disease can be prevented by thoroughly 9. Sirikulchayanonta V, Chongchimant N. Gnathosto­ cooking seafood, fowl, and reptiles. Family physi­ miasis, a possible etiologic agent of eosinophilic granuloma of the gastrointestinal tract. Am J Trap cians should counsel travelers that gnathostomiasis Med Hyg 1979; 28:42-4.

can be prevented by avoiding uncooked fish and 10. Hadidjaja P, Margono SS, Moeloek FA. Gnathostoma on 26 September 2021 by guest. Protected copyright. fowl. Although travelers might be especially vigilant rpinigerum from the cervix of a woman inJakarta. Am in the third world nations of Southeast Asia, it is J TropMed Hyg 1979; 28:161-2. important to recall that gnathostomiasis is also en­ 11. Nitidandhaprabhas P, Sirimachan S, Chamvises K. A case of penile gnathostomiasis in Thailand. Am demic in Japan, where sushi is very popular. J Trop Med Hyg 1978; 27:1282-3. The ingestion of uncooked seafood carries the 12. Manson-Bahr PEC, Bell DR Manson's tropical dis­ risk of infection with cholera, Anisakis marina, fish eases. 19th ed. London: Bailliere Tindall, 1987:561. tapeworm, Paragonimus flukes and liver flukes, in 13. AbramowitzM, editor. Drugs for parasitic infections. addition to G. spinigerum. Additionally, physicians Med Lett Drugs Ther 1990; 32:26. caring for Southeast Asian refugees or other pa­ 14. Ramalingam S, Sinniah B, Krishnan U. Albendawle, an effective single dose, broad spectrum anthelmin­ tients with a history of travel in endemic areas tic drug. AmJ Trop Med Hyg 1983; 32:984-9. should include gnathostomiasis in the differential 15. Vermox, Janssen Pharmaceuticals. In: Physicians' diagnosis of patients with fever, eosinophilia, rash desk reference. Montvale, NJ: Medical Economics suggestive of cutanea larva migrans, and espe- Data, 1992:1149-50.

Urinary Gnathostomiasis 535