TRANSACTIONS OF THE ROYAL SOCIETYOF TROPICAL MEDICINEAND HYGIENE(2003) 97, 200-202 Imported cutaneous gnathostomiasis: report of five cases A. M~nard ~, G. Dos Santos 2, P. Dekumyoy 3, S. Ranque 4, J. Delmont 1, M. Danis 2, F. Bricaire e and E. Caumes 2 i Service des Maladies Infectieuses et Tropicales, Hdpital Nord, Marseille, France; 2 Ddpartement des Maladies Infectieuses et Tropicales, Hdpital de la Pitid-Salpdtri~re, Paris, France; 3Department of Helminthology, Faculty of Tropical Medicine, Mahidol University, Bangkok, Thailand; 4Laboratoire de Parasitologie, Facult~ de M~decine, La Timone, Marseille, France Abstract Gnathostomiasis has rarely been described outside endemic countries. We report on a series of 5 patients (4 females, 1 male, mean age 42.2 years) who returned to France from South-East Asia and presented with cutaneous gnathostomiasis. The cutaneous lesions appeared within a mean period of 62 d (range 10-150 d) after return. They consisted of creeping eruptions in 3 patients (in addition one also had papules, one had nodules and hepatitis, and one had hepatitis; all 3 had profound asthenia) and recurring migratory swellings in 2 patients. The mean eosinophil count was 1546/mm 3 (range 398-3245/mm3). Diagnosis was based on positive serological tests in 3 patients and seroconversion in 2 patients, and was confirmed by identification of Gnathostoma hispidum in a biopsy specimen from one of the seropositive patients. Albandazole (1-4 courses) was given as treatment. Recurrences may occur up to 24 months after apparent cure without reinfection. Gnathostomiasis should be considered when patients return from tropical countries and present with migratory swellings or creeping eruption that does not respond to the usual treatment for cutaneous larva migrans. Serological tests may be negative initially and thus need to be repeated to check for seroconversion. Treatment may require multiple courses of albendazole and a prolonged period of follow-up is necessary before cure can be confirmed. Keywords: cutaneous gnathostomiasis, Gnathostoma hispidum, cutaneous larva migrans, diagnosis, chemotherapy, albendazole, travel Introduction Gnathostomiasis is a well-known parasitosis in South-East Asia and Latin America where it is acquired by the ingestion of raw or insufficiently cooked meat of animal hosts (fish, amphibians, reptiles, birds and mammals) (Daengsvang, 1981). It is becoming more commonly observed in western countries due to in- creasing intercontinental travel and consumption of Asian food. Of the 8 cases of imported gnathostomiasis which have been previously reported in Western coun- tries 4 occurred in travellers returning from South-East Asia (Chabasse et al., 1988; Rusnack & Lucey, 1993; Jelinek et al., 1994; Crowley & K,im, 1995), 2 in travellers returning from Latin America (Chappuis et al., 2001; del Giudice et al., 2001), one was acquired in Spain through an unknown route (Montero et al., 2001), and one was acquired in Hungary after con- sumption of Asian food (Chandenier et al., 2001). These case reports reflected the clinical spectrum of gnathostomiasis, 7 were cutaneous gnathostomiasis and one was neurological gnathostomiasis (Chandenier et al., 2001). We report on a series of 5 travellers presenting with cutaneous gnathostomiasis after returning to France from South-East Asia. We discuss the diagnostic criter- ia used and the outcome for patients with no previous exposure to the disease. Patients and Methods We reviewed all cases of cutaneous gnathostomiasis observed in 2 referral centres for tropical diseases in France from April 1991 to April 2000. Patients were considered as possible cases if they (i) presented with cutaneous manifestations attributable to gnathostomia- sis such as creeping eruption, migratory swelling, nodule or plaque on the trunk; (ii) had returned from a trip to an endemic area; and (iii) were antibody-specific to Gnasthostoma spp. by immtmoblot assay. Diagnosis of certitude was made if a biopsy specimen of the cuta- neous lesion revealed a cross-section of the nematode. Immunoblot assays were performed at the Depart- Address for correspondence: Eric Caumes, D~partement des Maladies Infectieuses et Tropicales, H6pital de la Piti6- Salp&ri6re, 47-83 Boulevard de l'H6pital, 75013 Paris, France; phone +33 01 4216 0114, fax +33 01 4216 0165, e-mail eric.caumes@psl.ap-hop-paris, fr ment of Helminthology, Faculty of Tropical Medicine in Bangkok, Thailand using an antigen produced from infective stage larvae of G. spinigerum as previously described (Nopparatana et aI., 1988; Tapchaisri et al., 1991). The following information was recorded for each patient: age, gender, country of origin, recent travel history, country visited, mode of acquisition, time from return to clinical onset and presentation, clinical and biological signs, histological data, treatment and out- come. Cure was defined as the absence of recurrence (i.e. reappearance of cutaneous lesions related to gnathostomiasis) 2 years after the last effective treat- ment. Results All 5 patients (4 females, 1 male, mean age 42.2 years) had recently returned to France from South-East Asia (3 from Cambodia, one from Thailand and one from Viet Nam). The average length of stay was 34.2 d (range 15-90 d); the mean interval from the date of return to France to clinical onset was 62 d (range 10- 150 d); the mean interval from clinical onset to con- sultation was 180 d (range 20-730 d). Dietary histories were known for 4 patients who had eaten freshwater fish marinated in lemon juice while travelling. Four different clinical forms of cutaneous gnathosto- miasis were observed: creeping eruption (3 patients; one also with papules and one also with nodules) and migratory oedematous plaques (2 patients). Of the 3 patients who presented with a creeping eruption, 2 were initially diagnosed with cutaneous larva migrans and received 4 ineffective treatments (one received ivermectin and the other ivermectin, thiabendazole, flubendazole and short-course albendazole) before being diagnosed with gnathostomiasis. All 3 had hyper- eosinophilia and cutaneous signs were associated with alteration of their general condition: all had profound asthenia; one also had fever, one fever and hepatitis with cytolysis, and one hepatitis with cytolysis and a weight loss of 7 kg. Histological examination in this last patient showed an eosinophilic infiltrate in the dermis and a cross-section of a nematode's larva within the dermis. The nematode's larva was identified as third- stage G. hispidum because the intestinal canal consisted of about 35 intestinal cells and a large nucleus was also observed at the centre of each epithelium cell. This