On-line version ISSN 1806-4841
An. Bras. Dermatol. vol.84 no.4 Rio de Janeiro July/Aug. 2009
Christiane Maria de Castro DaniI; Karina Frias MotaII; Paola Vizcaichipi SanchoteneIII; Juan Piñeiro MaceiraIV; Cláudia Pires Amaral MaiaV
specialist, Brazilian Dermatology Society. Policlínica Geral do Rio de
Janeiro, Rio de Janeiro (RJ), Brazil
IIDermatology specialist, Brazilian Dermatology Society. Policlínica Geral do Rio de Janeiro, Rio de Janeiro (RJ), Brasil
IIIDermatology specialist pela Brazilian Dermatology Society. Policlínica Geral do Rio de Janeiro, Rio de Janeiro (RJ), Brazil
IVAssociate Professor. PhD. Pathology Dept, Universidade Federal do Rio de Janeiro (UFRJ), Rio de Janeiro (RJ), Brazil
VAssistant Professor of Carlos Chagas Medical Graduate Program - Policlínica Geral do Rio de Janeiro. Master of Dermatology, Universidade Federal do Rio de Janeiro (UFRJ), Rio de Janeiro (RJ), Brazil
Gnathostomiasis is an endemic disease in Asian countries caused by the ingestion of larval stage of Gnathostoma sp roundworms that may be acquired by eating raw fresh-water fish. It can reach many organs and the skin is one of them, characterized by creeping eruptions and/or migrating erythema. Since 1970 the number of patients affected seems to be increasing in Central and South America countries like Mexico and Peru. This is the first notification and publication of gnathostomiasis in Brazil, and it serves as an alert to the possibility of the emergence of this disease in non-endemic areas. We report a case of a man who traveled to Peru and developed cutaneous features accordant with gnathostomiasis.
Keywords: Gnathostoma; Helminth; Panniculitis
Gnathostomiasis is a parasite disease caused by the roundworm Gnathostoma sp., characterized by creeping eruptions and/or migrating erythemas associated to local skin edema. It is acquired by eating raw or undercooked contaminated foods.1-6 It is endemic in Asian countries like Thailand and Japan where it is common to eat raw fresh water fish. Central America countries have already reported a significant number of cases of this disease due to the habit of eating a typical dish called ceviche, consisting of marinated raw fish.2,4,5 The larval stage of Gnathostoma sp. may be an exclusively cutaneous disease or it may disseminate to other organs and systems like the eyes, lungs, and central nervous system.4,6,7 This reports is presenting the case of a patient who traveled to Peru where he ate ceviche, and two weeks later presented a clinical condition compatible with Gnathostomiasis. In Brazil, so far, there has not been any record of this disease, and this case was reported to ANVISA (National Healthcare Agency) in March 2008.
Twenty-nine-year-old man with an erythematous-edematous skin lesion with pruritus, with two weeks of evolution, in the infraumbilical region and which later evolved to the right side of the abdomen (Figures 1 and 2). He reported having traveled to Peru where he had eaten ceviche three weeks before the emergence of the symptoms. Lab tests showed 17% eosinophilia (1530/mm3). The histopathology of the lesion showed eosinophilic cellulites (Figure 3 and 4) associated to disperse foci of cellular degeneration looking like "burning figures", compatible with the diagnostic hypothesis. No larvae were found in the biopsied tissue. Based on the clinical status and epidemiological information, in addition to compatible lab tests, we diagnosed gnathostomiasis. The patient was treated with two cycles of 200 mcg/Kg ivermectin, with a 7 day interval. He evolved with the emergence of a new more superficial and well-delimited creeping lesion (Figure 5) and then was prescribed albendazole 800 mg for 21 days, with complete resolution of the lesions and normalization of the peripheral eosinophilia (value 320/ mm3- 5%). Serology was performed with specimen drawn one month after treatment, and samples were tested with different antigens. Only one of the bands (34-35 Kda) of the four immunogenic peptides tested was positive for the tertiary larva of Gnathostoma binucleatum by Western Blot (Figure 6). The ELISA for the somatic extracts with antigens to stage A Gnathostoma binucleatum larvae, adult Gnathostoma doloresi, and adult Gnathostoma turgidum were negative with optical density below 0.500.
Gnathostomiasis is acquired by eating undercooked foods like fish, lobster, frog, crab, or chicken, contaminated with the tertiary larva of the roundworm Gnathostoma sp. Approximately 12 species of gnathostomas have been identified so far.8 G. spinigerum was considered for a long time the main agent of the disease in humans; however, new species like G. hispidum, G doloresi, G nipponicum and G binucleatum have been often detected. 1,9
The definitive hosts of the parasite are dogs, cats and lions, in which its adult form lives forming tumor-like structures on the gastric wall. Their eggs hatch in the GI tract and eliminated through feces. After one week, the eggs release larvae that infect small crustaceans of the genus Cyclops, which are then eaten by fishes, frogs, toads, or birds, subsequently contaminating humans. In these, the larva penetrates through the gastrointestinal tract into the blood stream after three to four weeks, and afterwards may migrate to many other organs. 1,8
The skin condition is characterized by a migrating edema with a snake-like shape and pruritus, which shows the migration of the larvae. 4 Its usual location is the skin but the may migrate to other organs and systems, like the eyes, lungs, and central nervous system.5,6,7 In Thailand, gnathostomiasis is one of the leading causes of subarachnoid hemorrhage, accounting for 6% of the cases in adults.10,11 Gnathostoma sp infection is diagnosed through its typical skin lesion, associated to peripheral eosinophilia, and to histological findings of dense eosinophil infiltrate involving the dermis and hypodermis, and the parasite may or may not be found.5,12 In the literature, the larva is identified in 23 to 34% of histopathological specimens. 1,12
In the case reported here, diagnostic was based on the compatible clinical-epidemiological status, excellent therapeutic response, significant peripheral eosinophilia, and on the histopathological examination, which showed a significant inflammatory reaction both on the dermis and hypodermis, with absolute predominance of eosinophil infiltrate (90%). The predominance of the inflammatory infiltrate on the dermis only shows aspects related to the migration of larva, and is an evidence of the uniqueness of this case in comparison to others commonly described, usually located more deeply on the hypodermis. There were no lesions on the epidermis, thus ruling out the possibility of diagnosing larva migrans caused by Ancylostoma braziliensis. The detection of antibodies through serologic methods (ELISA and Western Blot) has been used for diagnostics and epidemiological surveys in Thailand and Japan. 9 In Brazil these assays are not available yet. Therefore, the serological analysis was done in Mexico, with serum drawn one month after the treatment. ELISA and Western Blott were used to detect antibodies against the parasite, the first being negative and the second positive for only one of the four 4 antigens tested. Likewise, there are reports of cases in which serological tests were negative although the larva was found in the histopathological test.3,9 According to the aforementioned authors, probably the insufficient quantity of larvas and time of evolution of the disease could produce an indictable immune response.3,9 On our case, we interpreted negative serological results due to the modified immune response that may occur after treatment. It has to be mentioned that the patient's serum was drawn 30 days after the end of the treatment, but serological tests were done more than 8 months later, because of the difficulties to send the specimen abroad.
The drug of choice for the treatment is albendazole at the dose of 400-800 mg/day for 21 days with cure rates of 93.9% and 94.1%.8,10 Studies have shown that a single dose of ivermectin is also effective; however, it has a higher relapse rate.11 The treatment of the disease is relatively simple, although its cure usually requires more than one course of medication. There are reports that after the beginning of the treatment with antiparasites, sometimes the larva may migrate to a more superficial level, which explains the occurrence of new lesions after the cycle of ivermectin, in the case herein presented. 5,11,13,14
The incorporation of the habit of eating raw or undercooked foods in the culture of many countries such as Brazil, and the intense flow of travel around the globe have been causing the emergence of little known diseases in our environment, such as gnathostomiasis, which occur in regions distant from their endemic areas, thus leading to the need of having a broader knowledge of these diseases.
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Cláudia Pires Amaral Maia
Rua das Laranjeiras, 91, aptº 203 - Laranjeiras.
CEP: 22240-002 - Rio de janeiro - RJ
Conflict of interest:
Financial funding: None
How to cite this article: Dani CMC, Mota KF, Sanchotene PV, Piñeiro-Maceira J, Maia CPA. Gnatostomíase no Brasil Relato de caso. An Bras Dermatol. 2009;84(4):400-4.