Annals of Tropical Medicine and Public Health
Home About us Ahead Of Print Instructions Submission Subscribe Advertise Contact e-Alerts Editorial Board Login 
Users Online:2338
  Print this page  Email this page Small font sizeDefault font sizeIncrease font size
 


 
Table of Contents   
CASE REPORT  
Year : 2013  |  Volume : 6  |  Issue : 3  |  Page : 317-320
Disseminated cysticercosis: Uncommon presentation of a common disease


1 Department of Medicine, Himalayan Institute of Medical Sciences, Dehradun, Uttarakhand, India
2 Department of Medicine, School of Medical Sciences and Research, Sharda University, Greater Noida, Uttar Pradesh, India
3 Department of Pathology, Himalayan Institute of Medical Sciences, Dehradun, Uttarakhand, India

Click here for correspondence address and email

Date of Web Publication7-Nov-2013
 

   Abstract 

Cysticercosis is an important disease of the developing world and is a common cause of seizures and neurological morbidity. Neurocysticercosis (NCC) is the most common manifestation of the disease involving the central nervous system (CNS). Disseminated cysticercosis (DCC) is an uncommon manifestation of this disease and less than 50 cases have been reported; the majority being from India. Here we report a case with extensive dissemination of C cellulosae, involving all possible sites, namely, the brain, eyes (extra- and intraocular), muscles, tongue, and subcutaneous tissues. She had innumerable cerebral cysts giving a starry sky appearance on imaging, and subcutaneous nodules, as yet unreported in the existing literature, to the best of our knowledge. The patient developed an anaphylactic reaction on receiving albendazole, despite being properly preconditioned with steroids. Presently she is in follow-up and is asymptomatic.

Keywords: Disseminated cysticercosis, neurocysticercosis, seizure, starry sky

How to cite this article:
Dhar M, Ahmad S, Srivastava S, Shirazi N. Disseminated cysticercosis: Uncommon presentation of a common disease. Ann Trop Med Public Health 2013;6:317-20

How to cite this URL:
Dhar M, Ahmad S, Srivastava S, Shirazi N. Disseminated cysticercosis: Uncommon presentation of a common disease. Ann Trop Med Public Health [serial online] 2013 [cited 2020 Jan 23];6:317-20. Available from: http://www.atmph.org/text.asp?2013/6/3/317/120994

   Introduction Top


Human cysticercosis is an important cause of seizures and neurological morbidity in developing countries. Cysticercosis results in humans, after ingestion of the eggs of Taenia solium, usually after a close contact with a tapeworm carrier. The larval stage of the pig tapeworm can invade many tissues in the body, but it is most commonly detected in the brain, skeletal muscle, subcutaneous tissue, or eye. Neurocysticercosis (NCC) is considered to be the most common parasitic infestation of the central nervous system (CNS), although most of the NCC cases are symptomatic, a few may be asymptomatic and detected incidentally on imaging. Symptoms and signs may be related both to the parasite and to the inflammatory-immunological response of the host. Simultaneous and extensive involvement of the brain, spinal cord, eyes, muscles, and subcutaneous tissues is extremely rare. Disseminated cysticercosis (DCC) is an uncommon manifestation of the disease, and less than 50 cases, mostly from India, have been reported worldwide. Dixon et al. reported only one case of disseminated disease in a study of 450 patients. [1]

Here we report a case of disseminated cysticercosis in a host from the State of Uttarakhand. The patient had novel clinical manifestations in the form of a nodule over the dorsum of the tongue and soft tissue calcification, besides the extensive dissemination. There are only a few studies from Uttarakhand highlighting mainly the clinical pattern of neurocysticercosis. [2],[3] Ours is the first case report of disseminated cysticercosis from this region.


   Case Report Top


A 40-year-old vegetarian lady, farmer by occupation, presented with complaints of moderate-to-high grade fever of a 15-day duration, followed by an onset of multiple seizures and altered sensorium, two days prior to hospitalization. She was a known case of seizure disorder since six years, and was on treatment for the same; the last episode of seizures was three months before presentation. On examination, she was a female of average build, with nodular enlargement of both upper limbs. She also had multiple nodules all over her body, subcutaneously on the neck, chest, abdominal wall, all four limbs [Figure 1], and a large one on the dorsum of her tongue [Figure 2]. The pulse was regular at 46 beats per minute and blood pressure was 100/60 mm Hg. The Glasgow Coma Scale score [E4M4V2] was 10, with left-sided sixth and seventh cranial nerve palsies. Focal neurological deficit and other signs of raised intracranial pressure were absent. The systemic examination was otherwise unremarkable.
Figure 1: Subcutaneous nodules on the forearm

Click here to view
Figure 2: Subcutaneous nodule on the dorsum of the tongue

Click here to view


Her hemoglobin was 10.2 gm%, total leucocyte count (TLC) 10900/cu.mm, absolute eosinophil count (AEC) 2180, and erythrocyte sedimentation rate (ESR) 80 mm/first hour. The renal and liver functions were within normal limits. Computed tomographic scan of the brain showed multiple punctuate calcifications with a starry sky appearance [Figure 3]. Magnetic resonance imaging (MRI) showed multiple cysts in different stages of the life cycle in the brain [Figure 4]. Soft tissue calcifications were evident in plain radiographs of the limbs [Figure 5]. Cerebrospinal fluid (CSF) examination revealed 10 leucocytes per cu.mm, sugar 38 mg/dl, and protein 100 mg/dl. An electrocardiogram (ECG) showed sinus bradycardia, however, echocardiography ruled out any structural cardiac lesion. A larval cyst was seen on histological examination of a nodule excised from the limb [Figure 6]. The immunological enzyme-linked immunosorbent assay (ELISA) test for cysticercal antibodies was positive.
Figure 3: Multiple punctuate calcifications on the CT scan

Click here to view
Figure 4: MRI showing cysts in different stages of the life cycle

Click here to view
Figure 5: Soft tissue calcification on a plain radiograph

Click here to view
Figure 6: Invaginated scolex of Taenia with birefringent hooklets

Click here to view


A diagnosis of disseminated cysticercosis was made and the patient was managed with intravenous fluids, steroids, and anti-epileptics. The patient improved clinically within 48 hours of admission and the fever subsided in the next three days. On day seven, albendazole 15 mg/kg, in three divided doses, was initiated. On receiving two doses of albendazole, she developed anaphylactic shock, from which she recovered in two days. Finally, the patient was discharged on anti-epileptics and steroids. During the last two months of Outpatient Department (OPD) follow-up, she has been doing well and has been asymptomatic.


   Discussion Top


Disseminated cysticercosis is described by diffuse symmetrical painful or painless enlargement of all group of muscles associated with weakness and easy fatigability, although, very little has been published about this rare entity. Priest et al., in 1926, probably described the first case of extensive somatic dissemination of C. cellulosae in a British soldier, who had swelling of his muscles, epileptic seizures, mental dullness, and widespread subcutaneous nodules. [4] The diagnosis of disseminated cysticercosis was made by the presence of C. cellulosae in the brain and subcutaneous and muscular tissues, simultaneously. A computed tomographic (CT) scan and Magnetic Resonance Imaging (MRI) were useful in the anatomical localization of cysts and in the documenting of natural history. MRI was more sensitive than CT, as it identified scolex and live cysts.

There are only few reports of cysticerci from northern India and only two-to-three studies from the state of Uttarakhand. A pilot study from the state of Uttarakhand had reported a high prevalence of NCC in the state. [2] Another study done from the state had shown that NCC accounts for approximately 35% of the cases of epilepsy in this region. [3]

A series of 22 cases was reported by Wadia et al., who described the syndrome of DCC as pseudomuscular hypertrophy (100%), palpable subcutaneous nodules (87%), seizures (78%), and abnormal mentation (65%). [5] Cysticerci can lodge themselves in any part of the ocular tissues, extraocular tissues, and associated brain parenchyma. A case of orbital cysticercosis, associated with multiple cysts in the brain and subcutaneous tissue, was also reported by Pushker et al. [6] Chadha et al. described disseminated cysticercosis as having simultaneous intraocular and extraocular cysticerci, and reported the first case in a 21-year-old male. [7] Dysautonomia is a well-known phenomenon caused by mesial temporal lobe and insular NCC. [8]

In our case we had extensive dissemination of C. cellulosae, virtually involving all sites, namely, brain, eyes (extra- and intraocular), muscles, tongue, and subcutaneous tissues. Moreover, the innumerable cerebral cysts and subcutaneous nodules seen in our patient, is unreported in the existing literature, to the best of our knowledge. Bradycardia at presentation can be attributed to what is known as the neurogenic heart. [9]

Management of DCC includes symptomatic treatment of the central nervous system lesions using steroids and anti-epileptics. The cysticidal drugs and praziquantel and albendazole, reduce the parasite burden that may occur even in the absence of such treatment. Severe reactions resulting from the treatment may result from the enlargement of cysts, massive release of antigens, and generalized anaphylactic reaction, as in our case. Priming with corticosteroids, before starting the cysticidal drug decreases the incidence of such complications.

Our patient developed anaphylaxis to albendazole, despite receiving parenteral steroids for a week before starting the cysticidal drug. Hence, the therapy needs to be individualized and close monitoring is required when cysticidal drugs are initiated. [10]

Limitation of the Study

In view of the widespread dissemination, assessment of tapeworm infection should have been ruled out, which was not done in the present study.

 
   References Top

1.Dixon HBF, Lipscomb FM. Cysticercosis: an analysis and follow-up of 450 cases. Medical Research Council Special Report Series 1961; 299:1-58.  Back to cited text no. 1
    
2.Varma A, Gaur KJ. A clinical spectrum of neurocysticercosis in Uttaranchal region. J Assoc Physicians India 2002;50:1398-400.  Back to cited text no. 2
    
3.Goel D, Dhnai JS, Agarwal A, Melhotra V, Saxena V. Neurocysticercosis and its impact on crude prevalence of epilepsy in Indian community. Neurol India 2011;59:37-40.  Back to cited text no. 3
[PUBMED]  Medknow Journal  
4.Priest R. A Case of Extensive Somatic Dissemination of Cysticercus Cellulosae in Man. Br Med J 1926;2:471-2.  Back to cited text no. 4
    
5.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. 5
    
6.Pushker N, Bajaj MS, Balasubramanya R. Disseminated cysticercosis involving orbit, brain and subcutaneous tissue. J Infect 2005;51:245-8.  Back to cited text no. 6
    
7.Chadha V, Pandey PK, Chauhan D, Das S. Simultaneous intraocular and bilateral extraocular muscle involvement in a case of disseminated cysticercosis. Int Ophthalmol 2005;26:35-7.  Back to cited text no. 7
    
8.Foyaca S.H, Ibañez-Valdés LdeF. Vascular dementia type Binswanger's disease in patients with active neurocysticercosis. Electron J Biomed 2002; 1(1):1-12  Back to cited text no. 8
    
9.Foyaca SH, Ibañez-Valdés LdeF. Generalized cysticercosis with cardiac involvement. The Internet Journal of Neurology 2007; 7 (2).   Back to cited text no. 9
    
10.García HH, Evans CA, Nash TE, Takayanagui OM, White AC Jr, Botero D, et al. Current Consensus Guidelines for Treatment of Neurocysticercosis. Clin Microbiol Rev 2002;15:747-56.  Back to cited text no. 10
    

Top
Correspondence Address:
Minakshi Dhar
Department of Medicine, Himalayan Institute of Medical Sciences, Swami Ram Nagar, Jolly Grant, P. O. Doiwala, Dehradun, Uttarakhand
India
Login to access the Email id

Source of Support: None, Conflict of Interest: None


DOI: 10.4103/1755-6783.120994

Rights and Permissions


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6]

This article has been cited by
1 Albendazole
Reactions Weekly. 2014; 1484(1): 5
[Pubmed] | [DOI]



 

Top
 
  Search
 
    Similar in PUBMED
   Search Pubmed for
   Search in Google Scholar for
 Related articles
    Email Alert *
    Add to My List *


    Abstract
   Introduction
   Case Report
   Discussion
    References
    Article Figures

 Article Access Statistics
    Viewed4099    
    Printed92    
    Emailed0    
    PDF Downloaded20    
    Comments [Add]    
    Cited by others 1    

Recommend this journal