Isolated Cysticercosis of Tongue: A Case Report Pratima Khare, M.D.,* Nidhi Chauhan, D.N.B., Rajeev Dogra, D.C.P., Pooja Kala, M.D., and Priyanka Chand, M.D.
Cysticercosis of the tongue is a rare disease caused by infestation with the larval stage of the pork tapeworm (Taenia solium) in which man acts as a secondary host rather than a primary host. Most of these lesions are asymptomatic. The patient usually reports to the physician with the complaint of swelling. The solitary swelling in the tongue of this young 12-year-old girl was not suspected clinically for cysticercosis. Fine needle aspiration cytology, used for pre-operative diagnosis, suggested the possibility of cysticercosis. Detailed medical evaluation was carried out which ruled out neural cysticercosis and other extraneural lesions. The complete removal of the lesion was achieved by simple excision. The histopathological examination confirmed the diagnosis of lingual cysticercosis. Diagn. Cytopathol. 2014;42:716–718. VC 2014 Wiley Periodicals, Inc.
larly common in less developed countries where pigs are raised as a source of food.3 The prevalence of the disease in India, which is a developing country with a large population below the poverty line, is high.4 Isolated extra neural cysticercosis presenting as a solitary mass in the tongue is an extremely rare occurrence. There are only 34 cases reported in world literature.5,6 For a clinician, the cysticercosis of tongue is a diagnostic dilemma because of its rarity. It is usually never a provisional diagnosis by the treating doctor at the first instance.7 The diagnosis of cysticercosis is made on fine needle aspiration cytology (FNAC)/magnetic resonance imaging (MRI)/serology/histopathological examination.
Key Words:
Case
cysticercosis; FNAC; histopathology; tongue
Tapeworm infection in human beings falls into two categories. In one category, human is a definitive host with adult tapeworm living in gastrointestinal tract, whereas in the other category, man is an intermediate host with larval stage parasite present in tissues. Taenia solium larva which causes cysticercosis can result from consumption of under cooked pork meat. More commonly it results from accidental ingestion of the eggs of T. solium through faeco-oral contamination. Thus, vegetarians and other people who do not eat pork can acquire cysticercosis.1 Cysticercosis is the most common parasitic infection of the central nervous system in the world.2 However, extra neural cysticercosis is also very common and may affect any site including muscles, heart, liver, lungs, peritoneum, eye, and subcutaneous tissue. The disease is particu-
A 12-year-old Hindu girl presented in ENT OPD with a painless progressively increasing swelling on the right lateral border of tongue at the junction of anterior two third and posterior one third (Fig. 1). The swelling was first noticed 8 months ago and had been gradually increasing in size. At the time of clinical presentation, the swelling measured about 2.5 3 2 cm. The overlying mucosa was intact without any obvious signs of inflammation. The swelling was firm, non-mobile, non-tender with diffuse margins. The movements of the tongue were normal. There was no cervical lymphadenopathy. ENT and systemic examination was normal. The patient was further investigated. All routine investigations including hemogram, peripheral smear, urine examination, and stool examination were within normal limits. The X-ray chest was normal. FNAC was advised by clinician.
Cytologic Findings Department of Pathology, Dr B.S. Ambedkar Hospital, New Delhi, 110085, India *Correspondence to: Pratima Khare, M.D., Department of Pathology, Dr B.S. A. Hospital, Rohini, New Delhi 110085, India. E-mail: [email protected] Conflict of Interest: None. Received 3 September 2013; Revised 17 November 2013; Accepted 9 January 2014 DOI: 10.1002/dc.23109 Published online 8 March 2014 in Wiley Online Library (wileyonlinelibrary.com).
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Diagnostic Cytopathology, Vol. 42, No 8
FNAC of swelling was done using a 22 gauge needle and 10 mL syringe. The material was smeared on the slides. The slides were air dried and fixed in methanol and stained with Geimsa stain. Thick blue spherules with pinkish blue fibrillary material and interspersed small blue nuclei giving honey comb appearance were seen. Cytological diagnosis of a parasitic lesion with possibility of cysticercus cellulosae, was made (Fig. 2). C 2014 WILEY PERIODICALS, INC. V
Diagnostic Cytopathology DOI 10.1002/dc
ISOLATED CYSTICERCOSIS OF TONGUE
Fig. 3. Histopathology section showing cysticercus larva within the bladder wall (H&E stain) (103). [Color figure can be viewed in the online issue, which is available at wileyonlinelibrary.com.]
The patient was put on high doses of Albendazole (400 mg twice daily) and surgical excision of the swelling was planned. The patient was operated upon after one week and complete excision of the cyst was carried out. The cyst was subjected to histopathological examination. Fig. 1. Clinical photograph showing swelling at lateral border of tongue. [Color figure can be viewed in the online issue, which is available at wileyonlinelibrary.com.]
Histopathological Examination A small 2 3 2 cm tissue was received and was processed for histopathological examination. The section showed cysticercus larva surrounded by a double layered membrane. Folding of the spiral canal and sucker of the scolex was seen (Fig. 3).
Follow up Immediate post-operative period was uneventful. The patient was additionally given a high dose of albendazole (400 mg twice daily) for 4 weeks more. At 1 year follow up, there was no evidence of any recurrence and the patient was free from any visceral or peripheral cysticercosis.
Discussion Fig. 2. FNAC smear showing fragments of parasite (Giemsa stain)(103). [Color figure can be viewed in the online issue, which is available at wileyonlinelibrary.com.]
Clinical Management In view of the findings on FNAC, the patient was subjected to further investigations to look out for any neural or visceral evidence of cysticercosis. Ultrasound abdomen was essentially normal with no evidence of involvement of liver and other viscera. Chest computed tomography (CT) was normal. To eliminate the possibility of neurocysticercosis, CT scan brain was also performed but no parasitic focus was detected.
Cysticercosis has been eradicated from most of the developed countries, but is still prevalent in Central and South America, South Asia, India, and China.8 According to the literature reports, the prevalence of oral cysticercosis is 4.1%. The most commonly involved intraoral sites are buccal mucosa, tongue, and lips.9 Cysticercosis of tongue is rare. In a recent study from India, among total 6,805 biopsies analyzed, there were 21 cases of cysticercosis in the soft tissues out of which there was only one case of cysticercosis of tongue.10 The disease is usually not suspected clinically and the diagnosis is usually made on histopathological examination. Risk factors for human cysticercosis include consumption of undercooked pork containing cysticerci, poor personal and house hygiene and history of Diagnostic Cytopathology, Vol. 42, No 8
717
Diagnostic Cytopathology DOI 10.1002/dc
KHARE ET AL.
passing tapeworm proglotids. Autoinfection may occur if an individual with an egg producing tapeworm ingests eggs derived from his or her own feces.11 The patient in our case report was vegetarian from a middle class background with good level of personal hygiene and no history of passing tape like proglotids. Most of the cases of cysticercosis present as painless, well circumscribed soft swelling that may mimic fluctuant lesions like mucocele.12 The swelling in our patient was more diffuse. The clinician considered the possibility of benign lesion like fibroma, leiomyoma, dermoid cyst, or a benign lesion of neural origin and referred the case for FNAC. The possibility of a parasitic lesion was not considered by the treating physician and it was only after FNAC which suggested the possibility of cysticercosis, further investigations and treatment was carried out. The FNAC, as a pre-operative diagnostic tool in oral cysticercosis, has been reported in literature.13 However, in most of the case reports of lingual cysticercosis the diagnosis was established only after histopathological examination.5,14 It has been proposed that every case of oral cysticercosis should be thoroughly investigated to determine the involvement of neural as well as extraneural sites, since there is high incidence of detecting additional lesions.7,13 Detailed investigations in our patient failed to detect any other foci of visceral or neural cysticercosis and the patient was diagnosed as being suffering from an isolated solitary cysticercosis of tongue.
Conclusion In spite of abundance of muscular tissue in the oral and facio-maxillary region, it is a rare site of occurrence for cysticercosis. Infestation in this region is relatively benign as compared to ocular or neurocysticercosis. Most of these patients present as an asymptomatic mass in the tongue. FNAC is well accepted modality for diagnosis of cysticercosis. Other diagnostic tools helpful in diagnosis are enzyme linked immunotransfer blot (ETIB) assay, CT scan, and MRI. However, absolute criteria for diagnosis of cysticercosis11 are the histopathological demonstration of parasite, visualization of parasite in eye by fundoscopy, and neuroradiologic demonstration of cystic lesion containing scolex. In view of the fact that ocular or cerebral
718
Diagnostic Cytopathology, Vol. 42, No 8
cysticercosis are serious diseases, all those cases diagnosed and treated for extra neural cysticercosis should be kept under regular follow up for occurrence of symptoms pertaining to these organs.
Acknowledgments The study is from the Department of Pathology, Dr. B. S. A. Hospital, Rohini, Delhi (India), which is run by Government of Delhi. All the services are provided free of cost to the patients.
References 1. Schantz PM, Moore AC, Munoz JL, et al. Neurocysticercosis in an Orthodox Jewish community in New York City. N Engl J Med 1992;327:692–695. 2. Schmidt DKT, Jordaan HF, Schneider JW, Cilliers J. Cerebral and subcutaneous cysticercosis treated with albendazole. Int J Dermatol 1995;34(8):574–579. 3. Garcia HH, Del Brutto OH. Taenia solium cysticercosis. Infect Dis Clin North Am 2000;14:97–119. 4. Vedantam R. Epidemiology of Taenia solium taeniasis/cysticercosis in India and Nepal Southeast Asian. J Trop Med Public Health 2004;35:247–251. 5. Pandey SC, Pandey SD. Lingual cysticercosis. Indian J Plast Surg 2005;38(2):160–161. 6. Wortman PD. Subcutaneous cysticercosis. J Am Acad Dermatol 1991;25:409–414. 7. Roth B, Gocht A, Metternich FU. Cysticercosis as a rare cause of a tumor of tongue. Laryngorhinootologie 2003;82:564–567. 8. Prasad KN, Prasad A, Gupta RK, Pandey CM, Uttam S. Prevalence and associated risk factors of T. solium taeniasis in a rural pig farming community of North India. Trans R Soc Trop Med Hyg 2007;101:1241–1247. 9. Jay A, Dhanda J, Chiodini PL, et al. Oral cysticercosis. Brit J Oral Max Surg 2007;45:331–334. 10. Agrawal R. Soft tissue cysticercosis: Study of 21 cases. J Clin Diagn Res 2012;10:1669–1671. 11. Kasper B, Fauci H, Longo J. Harrison’s principles of internal medicine. 16th ed. New Yok: McGraw Hill; 2008. Vol. 1. p 1260–1263. 12. Hansen LS, Allard RHB. Oral cysticercosis. JADA 1984;108:632– 636. 13. Gadbail AR, Korde S, Wadhwan V, Chaudhary M, Patil S. Oral cysticercosis: Report of two cases with review of literature. Oral Surgery 2010;3:51–56. 14. Elias FM, Martin MT, Folonda R, Jorge WA, Ara ujo NS. Oral cysticercosis: Case report and review of literature. Rev Inst Trop Sao Paulo 2005;47(2):95–98.
Cysticercosis of the tongue is a rare disease caused by infestation with the larval stage of the pork tapeworm (Taenia solium) in which man acts as a secondary host rather than a primary host. Most of these lesions are asymptomatic. The patient usually reports to the physician with the complaint of swelling. The solitary swelling in the tongue of this young 12-year-old girl was not suspected clinically for cysticercosis. Fine needle aspiration cytology, used for pre-operative diagnosis, suggested the possibility of cysticercosis. Detailed medical evaluation was carried out which ruled out neural cysticercosis and other extraneural lesions. The complete removal of the lesion was achieved by simple excision. The histopathological examination confirmed the diagnosis of lingual cysticercosis. Diagn. Cytopathol. 2014;42:716–718. VC 2014 Wiley Periodicals, Inc.
larly common in less developed countries where pigs are raised as a source of food.3 The prevalence of the disease in India, which is a developing country with a large population below the poverty line, is high.4 Isolated extra neural cysticercosis presenting as a solitary mass in the tongue is an extremely rare occurrence. There are only 34 cases reported in world literature.5,6 For a clinician, the cysticercosis of tongue is a diagnostic dilemma because of its rarity. It is usually never a provisional diagnosis by the treating doctor at the first instance.7 The diagnosis of cysticercosis is made on fine needle aspiration cytology (FNAC)/magnetic resonance imaging (MRI)/serology/histopathological examination.
Key Words:
Case
cysticercosis; FNAC; histopathology; tongue
Tapeworm infection in human beings falls into two categories. In one category, human is a definitive host with adult tapeworm living in gastrointestinal tract, whereas in the other category, man is an intermediate host with larval stage parasite present in tissues. Taenia solium larva which causes cysticercosis can result from consumption of under cooked pork meat. More commonly it results from accidental ingestion of the eggs of T. solium through faeco-oral contamination. Thus, vegetarians and other people who do not eat pork can acquire cysticercosis.1 Cysticercosis is the most common parasitic infection of the central nervous system in the world.2 However, extra neural cysticercosis is also very common and may affect any site including muscles, heart, liver, lungs, peritoneum, eye, and subcutaneous tissue. The disease is particu-
A 12-year-old Hindu girl presented in ENT OPD with a painless progressively increasing swelling on the right lateral border of tongue at the junction of anterior two third and posterior one third (Fig. 1). The swelling was first noticed 8 months ago and had been gradually increasing in size. At the time of clinical presentation, the swelling measured about 2.5 3 2 cm. The overlying mucosa was intact without any obvious signs of inflammation. The swelling was firm, non-mobile, non-tender with diffuse margins. The movements of the tongue were normal. There was no cervical lymphadenopathy. ENT and systemic examination was normal. The patient was further investigated. All routine investigations including hemogram, peripheral smear, urine examination, and stool examination were within normal limits. The X-ray chest was normal. FNAC was advised by clinician.
Cytologic Findings Department of Pathology, Dr B.S. Ambedkar Hospital, New Delhi, 110085, India *Correspondence to: Pratima Khare, M.D., Department of Pathology, Dr B.S. A. Hospital, Rohini, New Delhi 110085, India. E-mail: [email protected] Conflict of Interest: None. Received 3 September 2013; Revised 17 November 2013; Accepted 9 January 2014 DOI: 10.1002/dc.23109 Published online 8 March 2014 in Wiley Online Library (wileyonlinelibrary.com).
716
Diagnostic Cytopathology, Vol. 42, No 8
FNAC of swelling was done using a 22 gauge needle and 10 mL syringe. The material was smeared on the slides. The slides were air dried and fixed in methanol and stained with Geimsa stain. Thick blue spherules with pinkish blue fibrillary material and interspersed small blue nuclei giving honey comb appearance were seen. Cytological diagnosis of a parasitic lesion with possibility of cysticercus cellulosae, was made (Fig. 2). C 2014 WILEY PERIODICALS, INC. V
Diagnostic Cytopathology DOI 10.1002/dc
ISOLATED CYSTICERCOSIS OF TONGUE
Fig. 3. Histopathology section showing cysticercus larva within the bladder wall (H&E stain) (103). [Color figure can be viewed in the online issue, which is available at wileyonlinelibrary.com.]
The patient was put on high doses of Albendazole (400 mg twice daily) and surgical excision of the swelling was planned. The patient was operated upon after one week and complete excision of the cyst was carried out. The cyst was subjected to histopathological examination. Fig. 1. Clinical photograph showing swelling at lateral border of tongue. [Color figure can be viewed in the online issue, which is available at wileyonlinelibrary.com.]
Histopathological Examination A small 2 3 2 cm tissue was received and was processed for histopathological examination. The section showed cysticercus larva surrounded by a double layered membrane. Folding of the spiral canal and sucker of the scolex was seen (Fig. 3).
Follow up Immediate post-operative period was uneventful. The patient was additionally given a high dose of albendazole (400 mg twice daily) for 4 weeks more. At 1 year follow up, there was no evidence of any recurrence and the patient was free from any visceral or peripheral cysticercosis.
Discussion Fig. 2. FNAC smear showing fragments of parasite (Giemsa stain)(103). [Color figure can be viewed in the online issue, which is available at wileyonlinelibrary.com.]
Clinical Management In view of the findings on FNAC, the patient was subjected to further investigations to look out for any neural or visceral evidence of cysticercosis. Ultrasound abdomen was essentially normal with no evidence of involvement of liver and other viscera. Chest computed tomography (CT) was normal. To eliminate the possibility of neurocysticercosis, CT scan brain was also performed but no parasitic focus was detected.
Cysticercosis has been eradicated from most of the developed countries, but is still prevalent in Central and South America, South Asia, India, and China.8 According to the literature reports, the prevalence of oral cysticercosis is 4.1%. The most commonly involved intraoral sites are buccal mucosa, tongue, and lips.9 Cysticercosis of tongue is rare. In a recent study from India, among total 6,805 biopsies analyzed, there were 21 cases of cysticercosis in the soft tissues out of which there was only one case of cysticercosis of tongue.10 The disease is usually not suspected clinically and the diagnosis is usually made on histopathological examination. Risk factors for human cysticercosis include consumption of undercooked pork containing cysticerci, poor personal and house hygiene and history of Diagnostic Cytopathology, Vol. 42, No 8
717
Diagnostic Cytopathology DOI 10.1002/dc
KHARE ET AL.
passing tapeworm proglotids. Autoinfection may occur if an individual with an egg producing tapeworm ingests eggs derived from his or her own feces.11 The patient in our case report was vegetarian from a middle class background with good level of personal hygiene and no history of passing tape like proglotids. Most of the cases of cysticercosis present as painless, well circumscribed soft swelling that may mimic fluctuant lesions like mucocele.12 The swelling in our patient was more diffuse. The clinician considered the possibility of benign lesion like fibroma, leiomyoma, dermoid cyst, or a benign lesion of neural origin and referred the case for FNAC. The possibility of a parasitic lesion was not considered by the treating physician and it was only after FNAC which suggested the possibility of cysticercosis, further investigations and treatment was carried out. The FNAC, as a pre-operative diagnostic tool in oral cysticercosis, has been reported in literature.13 However, in most of the case reports of lingual cysticercosis the diagnosis was established only after histopathological examination.5,14 It has been proposed that every case of oral cysticercosis should be thoroughly investigated to determine the involvement of neural as well as extraneural sites, since there is high incidence of detecting additional lesions.7,13 Detailed investigations in our patient failed to detect any other foci of visceral or neural cysticercosis and the patient was diagnosed as being suffering from an isolated solitary cysticercosis of tongue.
Conclusion In spite of abundance of muscular tissue in the oral and facio-maxillary region, it is a rare site of occurrence for cysticercosis. Infestation in this region is relatively benign as compared to ocular or neurocysticercosis. Most of these patients present as an asymptomatic mass in the tongue. FNAC is well accepted modality for diagnosis of cysticercosis. Other diagnostic tools helpful in diagnosis are enzyme linked immunotransfer blot (ETIB) assay, CT scan, and MRI. However, absolute criteria for diagnosis of cysticercosis11 are the histopathological demonstration of parasite, visualization of parasite in eye by fundoscopy, and neuroradiologic demonstration of cystic lesion containing scolex. In view of the fact that ocular or cerebral
718
Diagnostic Cytopathology, Vol. 42, No 8
cysticercosis are serious diseases, all those cases diagnosed and treated for extra neural cysticercosis should be kept under regular follow up for occurrence of symptoms pertaining to these organs.
Acknowledgments The study is from the Department of Pathology, Dr. B. S. A. Hospital, Rohini, Delhi (India), which is run by Government of Delhi. All the services are provided free of cost to the patients.
References 1. Schantz PM, Moore AC, Munoz JL, et al. Neurocysticercosis in an Orthodox Jewish community in New York City. N Engl J Med 1992;327:692–695. 2. Schmidt DKT, Jordaan HF, Schneider JW, Cilliers J. Cerebral and subcutaneous cysticercosis treated with albendazole. Int J Dermatol 1995;34(8):574–579. 3. Garcia HH, Del Brutto OH. Taenia solium cysticercosis. Infect Dis Clin North Am 2000;14:97–119. 4. Vedantam R. Epidemiology of Taenia solium taeniasis/cysticercosis in India and Nepal Southeast Asian. J Trop Med Public Health 2004;35:247–251. 5. Pandey SC, Pandey SD. Lingual cysticercosis. Indian J Plast Surg 2005;38(2):160–161. 6. Wortman PD. Subcutaneous cysticercosis. J Am Acad Dermatol 1991;25:409–414. 7. Roth B, Gocht A, Metternich FU. Cysticercosis as a rare cause of a tumor of tongue. Laryngorhinootologie 2003;82:564–567. 8. Prasad KN, Prasad A, Gupta RK, Pandey CM, Uttam S. Prevalence and associated risk factors of T. solium taeniasis in a rural pig farming community of North India. Trans R Soc Trop Med Hyg 2007;101:1241–1247. 9. Jay A, Dhanda J, Chiodini PL, et al. Oral cysticercosis. Brit J Oral Max Surg 2007;45:331–334. 10. Agrawal R. Soft tissue cysticercosis: Study of 21 cases. J Clin Diagn Res 2012;10:1669–1671. 11. Kasper B, Fauci H, Longo J. Harrison’s principles of internal medicine. 16th ed. New Yok: McGraw Hill; 2008. Vol. 1. p 1260–1263. 12. Hansen LS, Allard RHB. Oral cysticercosis. JADA 1984;108:632– 636. 13. Gadbail AR, Korde S, Wadhwan V, Chaudhary M, Patil S. Oral cysticercosis: Report of two cases with review of literature. Oral Surgery 2010;3:51–56. 14. Elias FM, Martin MT, Folonda R, Jorge WA, Ara ujo NS. Oral cysticercosis: Case report and review of literature. Rev Inst Trop Sao Paulo 2005;47(2):95–98.
