Indian Journal of Medical Microbiology Medknow Publications on behalf of Indian Association of Medical Microbiology ISSN: 0255-0857 EISSN: 1998-3646 Vol. 29, Num. 2, 2011, pp. 180-183

Indian Journal of Medical Microbiology, Vol. 29, No. 2, April-June, 2011, pp. 180-183

Case Report

A rare case of disseminated cysticercosis: Case report and literature review

A Banu¹, N Veena²

¹ Department of Microbiology, Bangalore Medical College and Research Institute, Bangalore, Karnataka, India
² Department of Medicine, Bangalore Medical College and Research Institute, Bangalore, Karnataka, India

Correspondence Address: A Banu Department of Microbiology, Bangalore Medical College and Research Institute, Bangalore, Karnataka India asima.banu@gmail.com

Date of Submission: 08-Dec-2010
Date of Acceptance: 23-Mar-2011

Code Number: mb11042

PMID: 21654117

DOI: 10.4103/0255-0857.81787

Abstract

Keywords: Cysticercosis, disseminated cysticercosis, Taenia solium

Introduction

Cysticercosis is caused by Cysticerus cellulosae, the larval form of the tape worm Taenia solium. Humans acquire cysticercosis through faeco-oral contamination with Taenia solium eggs from tape worm carriers.

Disseminated cysticercosis (DCC) is an uncommon manifestation of this common disease. ^[1] Widespread dissemination of the cysticerci can result in involvement of almost any organ of the body. The main features of DCC include intractable epilepsy, dementia, enlargement of muscles, subcutaneous and lingual nodules and a relative absence of focal neurological signs or obviously raised intracranial pressure, at least until late in the disease. Muscular pseudohypertrophy, a rare presentation, is caused by heavy infection of the skeletal muscles, which gives the patient a "Herculean appearance". Fewer than 50 cases have been reported worldwide, the majority being from India, ^[2] and all these cases had extensive symptoms.

We report a case of an immunocompetent patient with DCC who had diffuse involvement of the skeletal muscles, lungs, subcutaneous tissue and brain. This case is remarkable because, in spite of the diffuse involvement of multiple organs, the patient presented with only arthritis, and DCC was an incidental finding. To the best of our knowledge, this is the first such case reported.

Case Report

A 55-year-old Christian male from Bangalore presented with pain and swelling in the right knee since 2 months, which was gradual in onset, progressive, present throughout the day and aggravated on joint movement. He also had swelling of the ankle and shoulder joints associated with pain on movement. The patient had no history of recurrent fever, morning stiffness of joints or involvement of small joints. He did not have a history of seizures. He was on a mixed diet including pork and other meat. There was no history of chronic cough, chronic diarrhoea, weight loss, decreased appetite or any past history suggestive of diabetes, hypertension and tuberculosis.

On examination, the patient was alert, conscious and cooperative. He had non-pitting bilateral pedal oedema. All the joints were normal except for tenderness on movement. There was mild symmetrical hypertrophy of the limbs prominent in the calf muscles. His systemic and ophthalmic examinations were clinically normal.

Investigations revealed a Hb of 14.8 g/dl, total leucocyte count of 10,600 cells/cu.mm, with polymorphs 54%, lymphocytes 40%, eosinophils 5% and monocytes 2%. The erythrocyte sedimentation rate was 40 mm/h. Creatinine phosphokinase was 142 units/L (normal 200 units/L). Routine biochemical investigations revealed normal glucose levels and renal and liver function tests. RA factor and CRP was positive. ANA was negative. The patient did not have any other symptoms to suggest diagnosis of rheumatoid arthritis or lupus. Tests for human immunodeficiency virus 1 and 2, hepatitis B surface antigen and hepatitis C virus were negative. Electrocardiogram was normal. Plain radiographs of lower limbs [Figure - 1] showed multiple calcified lesions in the muscles and subcutaneous tissues. X-ray of the skull was normal. Chest X-ray [Figure - 2] showed a single calcified cyst in the right lung. Computerized tomography (CT) of the brain [Figure - 3] showed a single calcified cyst. CT of the limbs [Figure - 4], [Figure - 5] showed thousands of calcified cysts in the muscular planes. Magnetic resonance imaging (MRI) was not performed because of the lack of facility in our hospital. Muscle biopsy was taken from the gastrocnemius muscle under local anaesthesia. Histopathology showed calcified cysts of cysticercus. Blood ELISA (qualitative) was positive for IgG antibodies for cysticercosis. Stool examination was normal.

A final diagnosis of DCC was made and the patient was treated with a tapering dose of prednisolone started a week earlier to albendazole at a dose of 1 mg/kg body weight and albendazole 15 mg/kg body weight for 6 weeks. He was also started on anti-inflammatory drugs for his persistent pain. Surgery was not a feasible option due to the sheer number of cysts. The patient′s arthritis improved and he was advised to continue physiotherapy. He was also advised regarding hygienic dietary practices and educated regarding the disease. On follow-up after 6 months, the patient continues to do well. Follow-up plain radiographs of the limbs were similar to that at presentation.

Discussion

Human cysticercosis is caused by the dissemination of the embryos of Taenia solium from the intestine via the hepatoportal system to the tissues and organs of the body. The organs most commonly affected are subcutaneous tissues, skeletal muscles, the lungs, brain, eyes, liver and, occasionally, the heart.

Widespread dissemination of cysticerci throughout the body was reported as early as 1912 by the British Army medical officers stationed in India. ^[2] In 1961, a review of 450 cases of cysticercosis by Dixon and Lipscomb ^[3] reported only one case of dissemination. Kumar et al.^[1] and Wadia et al.^[4] reviewed 22 cases each. Involvement of lung and muscles is rare. ^[5]

The clinical features depend on the location of the cyst, the cyst burden and the host reaction. The syndrome of DCC is characterized by pseudomuscular hypertrophy (100%), palpable subcutaneous nodules (87%), seizures (78%) and abnormal mentation. There is diffuse symmetrical painful or painless enlargement of all groups of muscles associated with weakness and easy fatigability. ^[1] Although our patient had diffuse muscular involvement of the lower limbs, there was no remarkable hypertrophy and he was without associated symptoms and also did not have subcutaneous nodules. Cerebral cysts usually number seven to ten per patient, ^[6] but here, this patient had only one to two cysts, which is a rare entity. He only presented with arthritis and was positive for non-specific RA factor, which could indicate a probable hypersensitivity reaction.

CT scan and MRI are useful in anatomical localization of the cysts, CT being more sensitive than MRI in detecting small calcifications. However, MRI is more sensitive than CT as it identifies scolex and the cyst. ^[1] Serological tests for detecting antibodies against cysticercosis are used to confirm the diagnosis. Enzyme-linked immunoblot assay is more sensitive and specific than ELISA. ^[7] The antibody test for IgG was positive in our case. Sensitivity of serological tests tends to be high for patients with multiple cysts (94%), but substantially lower for patients with a single cyst or calcified cysts (28%). ^[8]

Management of DCC is symptomatic (antiepileptics and steroids), surgical (removal of cysts and ventriculoperitoneal shunt) and cysticidal. The role of treatment with albendazole (15 mg/kg/day for 30 days) or praziquantel (10-15 mg/kg/day for 6-21 days) is controversial. These drugs hasten the death of the cysts, which may occur even in the absence of such treatment. Neurocysticercosis is a serious disease with potentially life-threatening complications. Patients with active cysts remain at risk of serious complications. It is therefore recommended that all patients with multiple cysts should receive treatment with cysticidal drugs. ^[8] Following treatment, cysticidal syndrome, characterized by features of raised intracranial tension, may occur in 50% of the cases. Efficacy of treatment should be monitored by repeat CT after 3 months.

There is no role for cysticidal drugs in inactive neurocysticercosis, i.e. calcified cysts, because the parasites are dead. But, still, we treated this patient with the standard treatment owing to the extensive involvement of other tissues and the presence of active inflammation as evidenced by the biomarkers.

Cysticercosis, thus, should always be part of the differential diagnosis of subcutaneous and intra-muscular swellings in India and, especially, Karnataka state, which happens to be a moderately endemic area. The disseminated form, although rare, should particularly be kept in mind. The usefulness of a detailed physical examination cannot be overemphasized, as illustrated in this case, wherein a subtle finding like mild calf hypertrophy led to the detection of a treatable condition like cysticercosis. A case of human cysticercosis with such extensive dissemination and with arthritis as the only presenting symptom is indeed very unusual.

Acknowledgement

The authors are thankful to the Superintendent of B & LCH and HOD of Radiology, Dr Satish Chandra for the radiography images.

References

1.	Kumar A, Bhagwani DK, Sharma RK, Kavita, Sharma S, Datar S, et al. Disseminated cysticercosis. Indian Pediatr 1996;33:337-9.   Back to cited text no. 1  [PUBMED]
2.	Bhalla A, Sood A, Sachdeve A, Varma V. Disseminated Cysticercosis: A case report and review of the literature. J Med Case Rep 2008;2:137.  Back to cited text no. 2
3.	Dixon HBF, Lipscomb FM. Cysticercosis: An analysis and follow-up of 450 cases. Med Res Counc Rep Ser 1961;299:1-58.   Back to cited text no. 3
4.	Wadia N, Desai S, Bhatt M. Disseminated cysticercosis: New observations, including CT scan findings and experience with treatment by praziquantel. Brain 1988;111:597-614.  Back to cited text no. 4  [PUBMED]  [FULLTEXT]
5.	Jain BK, Sankhe SS, Agrawal MD, Naphade PS. Disseminated cysticercosis with pulmonary and cardiac involvement. Indian J Radiol Imaging 2010;20:310-3.  Back to cited text no. 5  [PUBMED]
6.	King CH. Cestodes (Tapeworms). In: Mandell GL, Bennett JE, Dolin R, editors. Mandell, Douglas and Bennett's Principles and Practice of Infectious Diseases, 5th ed, Vol 2. Churchill Livingstone; 2000. p. 2956-64.   Back to cited text no. 6
7.	Stacker A, SampairoSilva M. Active neurocysticercosis, parenchymal and extra-parenchymal: A study of 38 patients, J Neurol 1993;241:15-21.  Back to cited text no. 7
8.	Wilson M, Bryan RT, Fried JA. Clinical evaluation of the cysticerciosis enzyme, linked immunoelectro transfer blot in patients with neurocysticercosis. J Infect Dis 1991;164:1007-9.  Back to cited text no. 8

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