Oral cysticercosis—report of six cases

ORAL CYSTICERCOSIS—REPORT OF SIX CASES Sonu Nigam, MD, Tejinder Singh, MD, Archana Mishra, MBBS, K. Uma Chaturvedi, MD Department of Pathology, Maulana Azad Medical College, Bahadurshah Zafar Marg, C-367 Saraswati Vihar, Pitampura, Delhi, 34 India Accepted 10 October 2000

Abstract: Background. Cysticercosis cellulosae rarely involves the oral region in humans. Methods. Six cases of oral cysticercosis were diagnosed, three each in lips and buccal mucosa. Diagnosis was made by characteristic gross and histopathologic appearances. Results. Of 146 cases of cysticercosis, 6 (4.1%) were of the oral cavity. All 6 cases were initially seen with painless swellings. Clinical differential diagnosis ranged from retention cyst, lipoma, fibroma, neurofibroma, and angular stomatitis. Histopathologic examination revealed cysticercus larvae. Conclusions. The oral cavity is a rare site of involvement by cysticercosis, even in an endemic area. Histopathologic findings of the excised cyst are diagnostic of the lesion. © 2001 John Wiley & Sons, Inc. Head Neck 23: 497–499, 2001. Keywords: cysticercosis; oral; lips; buccal mucosa

Human cysticercosis (cysticercosis cellulosae) is due to the larval form of tapeworm (Taenia solium). It is often subcutaneous.1 Other tissues affected by cysticercosis in order of frequency are brain, muscle, heart, liver, lungs, and peritoneum.2 In humans, cysticercosis rarely involves the oral region, and often is seen as a diagnostic problem.3 Diagnosis is usually made postoperatively through histopathologic examination.4,5 Correspondence to: S. Nigam © 2001 John Wiley & Sons, Inc.

Oral Cysticercosis

Only 38 cases of oral cysticercosis have been reported in last half century.5 MATERIAL AND METHODS

Between 1986 and January 2000, cysticercosis was diagnosed on histologic examination in 146 cases from the Department of Pathology, Histopathology Division, Maulana Azad Medical College, New Delhi, India. Of these, 6 cases were of the oral cavity and 3 each in the lips and buccal mucosa. The most common presentation was as a subcutaneous nodule in 130 cases. Other sites involved were muscle (n ⳱ 7), eyelid (n ⳱ 2), and perinodal (n ⳱ 1) tissue. These nodules were excised and subjected to histopathologic examination. RESULTS

All 6 cases had painless swelling for a period of 2 months to 2 years. There was no history of seizures in any of these patients. The clinical profile of these patients is shown in Table 1. Nodules in all 6 patients were single, nontender, and of firm consistency in 4 and cystic in 2. All swellings were smooth and well defined, with intact overlying mucosa. No cervical lymph nodes were palpable. Clinical differential diagnosis included retention cyst, fibroma, neurofibroma, lipoma, and angular sto-

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Table 1. Clinical details of six cases of oral cysticercosis.

S. no.

Age (y)

Sex

Site

Reported duration

Size

Clinical diagnosis

1 2 3 4 5 6

23 25 20 18 24 43

F M M M F M

Buccal mucosa, left side Buccal mucosa, left side Buccal mucosa, left side Lips (right angle of mouth) Upper lip Upper lip

2y 3–4 mo 2y 2 mo 6 mo 18 mo

3 × 3 cm 3 cm 1 cm 1 cm 1.5 cm 0.5 cm

Lipoma Cysticercosis* Fibroma/neurofibroma Angular stomatitis Retention cyst/neurofibroma Retention cyst

*Cysticercosis was diagnosed on gross examination of excised cyst. F = female; M = male.

matitis. In 1 case (No. 2), the surgeon had suspected cysticercus after excision. There were no other similar nodules elsewhere in the body. Stool examination in all cases was normal. Pathology. Grossly, a gray white to yellow cyst (Fig.1, inset) ranging from 0.5 to 1 cm was seen. A cut section showed cysts to contain clear fluid with the characteristic pearly white structure of the larva. An H & E–stained section revealed the pale eosinophilic hyaline villus structure with scolex in the cystic space (Fig. 1). It was lined by fibrous capsule with inflammatory infiltrate composed of palisading histiocytes, eosinophils, and lymphocytes. Epithelioid cells and giant cells were seen in 2 cases. DISCUSSION

Pork tapeworm (Taenia solium) is endemic in many parts of the world, including Russia, China, India, Pakistan, Philippines, Indonesia, and Mexico.6,7 The larval or cysticercoid stage devel-

FIGURE 1. Section from cysticercus larva with scolex (S) with suckers. Inset shows naked eye appearance of larval cyst with pearly white structure of larva. (H & E, original magnification ×160; Inset-naked eye, ×4.5).

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ops in the muscles of the pig, but occasionally it may develop in man. Subcutaneous tissue and muscle are the most common sites of involvement.8 In this series of 146 cases, a subcutaneous nodule was also the most common clinical presentation (130 of 146 cases), followed by muscle (7 cases). Cysticercosis only rarely involves the oral cavity.4–6 A MEDLINE search revealed only 38 cases of oral cysticercosis published in the English literature.3–6,9–21 The age at discovery varies from 5 to 70 years, and there is no gender predilection.6 The duration of swelling usually ranges from 1 month to 4 years.5 The swellings are usually single and painless clinically,3–6,9–21 a feature also seen in all our 6 cases. In the oral region, the tongue is most commonly involved, followed by the upper and lower lips, the oral mucosa, and the submandibular and submental tissue.5,6,9 None of our cases had tongue involvement, with buccal mucosa and upper lip being affected equally. The clinical differential diagnosis of such an oral nodular lesion includes retention cysts, benign tumours such as lipoma, fibroma, hemangioma, myoma, or benign tumours of minor salivary gland.5,6 However, in this series angular stomatitis and neurofibroma were also considered as a differential diagnosis. Oral cysticercosis in most previous published data was diagnosed postoperatively by histopathologic examination of an excised specimen or was suspected after surgery by examination of the cyst. However, fine needle aspiration cytology (FNAC) is also a well-accepted procedure for reliable and quick preoperative diagnosis of cysticercosis.22–24 Larval parts seen on FNAC include bluish fibrillary material with interspersed small nuclei, fragments of wall or tegument, and the scolex of the parasite with sucker and hooklets.23 Calcareous spherules may also be seen within the fibrillary substance.23,24

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Oral cysticercosis is usually asymptomatic and solitary and the treatment of solitary oral cysticercosis is surgical excision.6 Medical treatment may be unnecessary in asymptomatic people.5 If no adult tapeworm or other cysticerci are found, patients can be followed up by monitoring antibody titers.6 Multicysts and neurocysticercosis are treated with praziquantel or albendazole.5

7. 8. 9. 10. 11.

CONCLUSION

Cysticercosis should be considered a differential diagnosis in solitary painless nodules of oral cavity, especially in patients from an endemic area. This series emphasizes the importance of histopathologic examination of excised tissue in confirming the diagnosis of cysticercosis, which would have been missed. Excision is curative, and further treatment is not required. The authors thank T. Mohanan for typing this manuscript and S. Ghosh for photographic assistance. Acknowledgments.

12. 13. 14. 15. 16. 17. 18. 19.

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