2024 Volume 66 Issue 3 Pages 202-205
The present work reports a case of a female patient complaining of itching and painful lesions affecting the oral mucosa for 7 months. Buccal and lip mucosa showed swelling and erythema, with serpiginous tracks. The patient was diagnosed with oral larva migrans, and the lesions resolved after ivermectin administration. At 18-month follow-up, no sign of recurrence was observed. Larva migrans can represent a pitfall in oral diagnosis and a stressful condition for the patient. Oral health care providers should be aware of this and keep this disease in mind as a possible differential diagnosis in oral mucosa lesions.
Cutaneous larva migrans is a common dermatologic infection in tropical and subtropical countries worldwide, usually caused by the helminth Ancylostoma brasiliensis or Ancylostoma caninum [1,2]. Contamination happens after direct contact of the larvae with host skin, where the extremities are usually the most affected anatomical sites because of contact with contaminated soil [1]. These parasites cannot pass through the basal membrane of the epidermis, so they migrate irregularly in the epidermis, producing an elevated linear or serpiginous track called creeping eruption [2].
On rare occasions, the infection can occur in the oral mucosa [2,3], where patients report burning and itching sensation associated with the feeling that the lesion is crawling throughout the oral cavity [2,4]. The ingestion of contaminated food is one possibility to explain its occurrence at this anatomic site [5]. Considering the rarity and difficulty in diagnosing oral larva migrans, this study aimed to present a case report of this condition, focusing the discussion on its clinical features and differential diagnosis.
A 36-year-old female patient presented with a complaint of lesions in the oral cavity for the last 7 months, which had impaired eating and speaking. The patient reported a series of events that had occurred in her oral cavity. These started with an itching sensation combined with redness of the oral mucosa. Afterwards, the lesion migrated forming lines throughout the oral mucosa for about 5 days (Fig. 1). This happened in a cyclic manner, affecting different sites of the oral mucosa, which were associated with pain. The patient had been seen by several physicians and dentists, and subjected to blood tests, biopsy and treatment attempts. According to her, the lesions worsened during corticosteroid use and showed limited improvement with ketoconazole.
On clinical examination, discrete erythematous lesions were observed in the buccal, lip and oropharyngeal mucosa. The histological slides of the biopsy (hematoxylin and eosin, H&E were reviewed and new laboratory tests were requested (Table 1). At this time, the patient reported new lesions that had started directly after the last appointment, with substantial itching and the feeling that the lesion was migrating throughout the oral cavity. On oral examination, the buccal mucosa and upper and lower lip mucosa showed erythema and swelling, with serpiginous tracts (Fig. 2). The review of the H&E slides showed considerable vacuolization of the epithelium, exocytosis, spongiosis, hyperemia, dilated vessels/capillaries and substantial hemorrhage. In some areas of the microscopic field, it was possible to observe a hyaline structure compatible with a worm (Fig. 3). A diagnosis of larva migrans was established, and oral ivermectin, 6 mg/day, was prescribed for 12 days. At 2-month follow-up, no lesion was detected in the oral mucosa, and at 18-month follow-up, the patient showed no recurrence of the lesions.
| Test | Result | Test | Result |
|---|---|---|---|
| Anti-HIV | negative | RBC | 4.17 million/µL |
| Anti-HCV | negative | Hematocrit | 39.2% |
| FTA-ABS | negative | Hemoglobin | 13.5 g/dL |
| VDRL | negative | WBC | 5,800/µL |
| ANA | negative | Neutrophils | 3,770/µL |
| Anti-DNA | negative | Lymphocytes | 1,450/µL |
| B12 | 335 pg/mL | Monocytes | 406/µL |
| Folate | 11.10 ng/mL | Eosinophils | 116/µL |
| Iron | 139 µg/dL | Basophils | 0/µL |
| Fasting blood glucose | 78 mg/dL | Plasma cells | 0/µL |
| PT | 14.2 s | Platelets | 313,000/µL |
| aPTT | 24.4 s | ESR | 12 mm/h |
Helminthiasis is not a common condition of the oral cavity [2], and sometimes becomes a pitfall in oral diagnosis [3]. Oral helminth infections present a benign clinical picture and can resemble mucoceles or immune-mediated systemic conditions such as pemphigus vulgaris and pyostomatitis vegetans [2]. However, they are not usually included in the differential diagnosis due to the rarity of these conditions and scarce knowledge about their oral manifestations [2]. Besides, even though most patients recover spontaneously from larva migrans, usually within a month, some cases can persist for longer [3,4,5,6]. Therefore, it seems that the condition is underdiagnosed and that the true incidence, especially with oral involvement, is not known [4,5,7]. Only seven cases describing the involvement of the oral mucosa in larva migrans infection have been reported in the literature (Table 2) [2,3,4,5,6,8]. The present case was atypical not only because of the affected anatomical site but also because of the long period the patient remained infected. Similarly, a case of cutaneous larva migrans with persistence as long as 18 months has been described in the literature [9]. According to Thomé Capuano et al. [8], in cases where a wide area of the oral cavity is affected, probably more than one parasite creates the tracks, which, in the present case, is reinforced by the lesion persistence after biopsy. In addition, considering that the patient had no comorbidities, the possibility of multiple parasites would also explain the prolonged period of infection.
The oral mucosa is infected by larva migrans through direct contact with surfaces contaminated with dog and cat feces, such as fallen fruits, or contact with contaminated hands and objects [8]. The diagnosis of oral larva migrans is mainly based on clinical aspects and the patient’s history, which can be a difficult situation, considering the low incidence of the condition and consequent lack of experience of health care providers [2,7]. Biopsy may be helpful, especially when the parasite is detected. However, depending on the elected site, biopsy can only show nonspecific acute inflammation [4], since the migrating characteristic of the parasite hinders its detection [1,3]. Even in the absence of larvae, the epithelium can show tunnel-like areas associated with eosinophilic infiltration [1]. According to Faustino et al. [2], the nodule at the end of the serpiginous track should be elected for biopsy, because this is the probable lodging place of the larva. Biopsies performed along the trail of the larvae will probably show nonspecific ulcerations and inflammatory infiltrates determined by tissue destruction [2].
The blood tests played no role in the establishment of larva migrans diagnosis, since the patient did not show eosinophilia, an expected event in parasitic infections. Nevertheless, a case series study reported only 20% of cutaneous larva migrans patients showing eosinophilia [7], which means this is not an essential feature. An interesting fact reported by the patient was the worsening of the lesions during corticosteroid therapy and their partial remission after taking ketoconazole. The immunosuppressive effect of corticosteroid therapy can increase parasitic activity. Regarding the improvement with ketoconazole, a cross-response of helminth infections to antifungal therapy has been reported [10].
Albendazole, thiabendazole, mebendazole and ivermectin are treatment options for larva migrans [7,8]. Ivermectin was used considering its efficacy and good tolerance, leading to satisfactory results. Although oral larva migrans is not a severe disorder, it can be a stressful condition, due to the itching sensation and discomfort caused by the parasite migration. Also, until the diagnosis is confirmed, the patient suffers with the uncertainties of an undiagnosed disease.
| Authors | Age (years) | Gender | Time of evolution (months) | Chief complaints | Affected sites | Clinical signs | Differential diagnoses |
|---|---|---|---|---|---|---|---|
| Jaeger R, 1987, [6] | 50 | male | 6 | red wandering lines with itching and burning sensation | gingivae, buccal mucosa and lip | linear and circinate/serpiginous erythematous lesion | chemical burn |
| Andre, 1988, [5] | 40 | female | 3 | feeling of a mole burrowing underground Growing and decreasing edema |
lower lip and palate | edema and serpiginous tract | acute urticaria, pathomimia |
| Lopes, 1994, [3] | 39 | male | 3 | itching and burning tongue with exacerbation and remission of the symptoms | dorsum and ventral surface of the tongue, floor of the oral cavity | erythema; areas of depapillation on the dorsum of the tongue; tortuous whitish lines; erythematous irregular lines, serpiginous tracts; white track surrounded by erythema with a more elevated burrow at the end of the track | N/S |
| Thomé Capuano, 2006, [8] | 24 | female | N/S | itching and burning sensation of wandering lines; exacerbation and remission of the lesions | palate, dorsum of the tongue, lip and buccal mucosa | vesicular and ulcerative lesions; erythematous lines with serpiginous aspect | mucocele, chemical burn |
| Damante, 2011, [4] | 27 | female | N/S | oral canker sores that migrated in the oral cavity, that itch and give the impression of a bug crawling in the oral cavity; dyspnea | retromolar trigone, soft palate, buccal mucosa | linear erythematous plaque with hemorrhagic aspect | N/S |
| Damante, 2011, [4] | 35 | male | 3 | burning sensation Recurrent lesions |
palate, alveolar and buccal mucosa | linear fusiform erythematous plaque with an area more elevated and more erythematous area sensitive to palpation | N/S |
| Faustino, 2019, [2] | 38 | male | 1 | itching moving through the oral cavity Intermittent pain and burning sensation | buccal mucosa | linear ulcer with a small nodule perceptible by palpation | pyostomatitis vegetans, oral manifestations of gastrointestinal disorders |
ANA: antinuclear antibody; aPTT: activated partial thromboplastin time; DNA: deoxyribonucleic acid; ESR: erythrocyte sedimentation rate; FTA-ABS: fluorescent treponemal antibody absorption; H&E: hematoxylin and eosin; HCV: hepatitis C virus; HIV: human immunodeficiency virus; N/S: not specified; PT: prothrombin time; RBC: red blood cell count; VDRL: venereal disease research laboratory; WBC: white blood cell count
All procedures were in accordance with The Code of Ethics of the World Medical Association (Declaration of Helsinki) and carried out with the adequate understanding and written consent of the patient. The present study was approved by the Research Ethics Committee of Pontifical Catholic University of Rio Grande do Sul (CEP-PUCRS, protocol #49017221.4.0000.5336). The patient provided informed consent.
The authors declare that they have no competing interests.
This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.
VSK: conceptualization, methodology, data curation, investigation (clinical investigation, clinical follow up of the patient), writing – original draft, review and editing; FGS: methodology, data curation, investigation (clinical investigation, clinical follow up of the patient), writing – review and editing; ALHC: methodology, data curation, investigation (clinical investigation, histopathological analysis), writing – review and editing; FLDM: methodology, data curation, investigation (clinical investigation, histopathological analysis), writing – review and editing; MP: methodology, data curation, investigation (clinical investigation, histopathological analysis), writing – review and editing; KC: conceptualization, methodology, data curation, investigation (clinical investigation, histopathological analysis, clinical follow up of the patient), writing – original draft, review and editing
1)VSK: valesca.koth@edu.pucrs.br, https://orcid.org/0000-0002-5119-7347
1)FGS: fernanda.salum@pucrs.br, https://orcid.org/0000-0001-7842-619X
2)ALHC: ana.carvalho@pucrs.br, https://orcid.org/0000-0001-5012-5221
2)FLDM: fmaito@pucrs.br, https://orcid.org/0000-0001-8716-9578
2)MP: marcia.payeras@pucrs.br, https://orcid.org/0000-0002-9538-6131
1)KC*: karen.cherubini@pucrs.br, https://orcid.org/0000-0003-0913-477X
The authors are grateful to Dr. A. Leyva (USA) for English editing of the manuscript.
All data are available as part of the article.
