|Year : 2011 | Volume
| Issue : 2 | Page : 183-186
Case of sparganosis: A diagnostic dilemma
S Duggal1, RK Mahajan2, N Duggal2, C Hans2
1 Department of Microbiology , Dr B. L. Kapur Memorial Hospital, New Delhi - 110 005, India
2 Department of Microbiology, PGIMER, Dr Ram Manohar Lohia Hospital, New Delhi - 110 001, India
|Date of Submission||29-Jul-2010|
|Date of Acceptance||23-Mar-2011|
|Date of Web Publication||2-Jun-2011|
Department of Microbiology , Dr B. L. Kapur Memorial Hospital, New Delhi - 110 005
Source of Support: None, Conflict of Interest: None
Sparganosis, also known as larval diphyllobothriasis, is a rare disease of humans as man is not a natural host in the life cycle of Spirometra spp. Diagnosis of the latter is difficult as it mimics other conditions that commonly cause subcutaneous or visceral fluid collection. Clinical diagnosis of this particular case was also erroneously labelled as tuberculosis but later labelled as a case of sparganosis. To the best of our knowledge, this is the first case from India where a sparganum-like parasite was isolated in drain fluid from the perinephric area.
Keywords: Host-parasite, perinephric, sparganum
|How to cite this article:|
Duggal S, Mahajan R K, Duggal N, Hans C. Case of sparganosis: A diagnostic dilemma. Indian J Med Microbiol 2011;29:183-6
| ~ Introduction|| |
Spirometra , a pseudophyllidean cestode, is rarely pathogenic in humans, but its plerocercoid larva, known as sparganum, is of public health significance. Man acts as an accidental dead-end host. Symptoms are caused by mass effect and are usually diagnosed as a space occupying lesion on imaging studies or after surgical removal. Such parasites that have a complex life cycle seem to have evolved in tandem, with their aquatic and human hosts producing a well-balanced association. However, the recent changing trends of human settlement, agricultural activities and expansion of population may have increased the transmission of the parasite, producing severe infections. Sparganosis is a rare disease as only about 450 cases are known, mostly from Japan, China, Korea and Southeast Asia,  but a careful understanding of the ecology and epidemiology coupled with careful examination of the patient and the relevant clinical samples can provide useful clues to its diagnosis. In this particular case, it was diagnosed only when the sparganum was recovered in the drain fluid from the perinephric area.
| ~ Case Report|| |
A 25-year-old male patient, resident of eastern Uttar Pradesh, presented to the Surgical Emergency with chief complaints of pain and a gradually increasing swelling in the left lumbar region for 3 months. On ultrasonography, the left kidney was echogenic and elongated with a large, loculated perinephric collection on the left side. Contrast-enhanced computed tomography (CT) for kidney, ureter and bladder showed a large hypodense collection in the perinephric region of the left kidney causing significant mass effect, displacing it anteriorly, medially and towards the right with deformed contour and flattening of the renal parenchyma. However, the renal parenchyma showed normal contrast excretion with no pelvicalyceal dilatation. The right kidney appeared normal with a mild perinephric collection. On the basis of a positive TB-polymerase chain reaction report from the fluid aspirate, he was diagnosed as a case of abdominal tuberculosis and was put on anti-tubercular therapy. Mantoux test and aspirate smear were negative for tuberculosis. A Double J (DJ) ureteric stent was placed for temporary drainage from the ureteropelvic junction to the bladder.
However, the patient presented again with similar features 2 months after the initial admission. The pain was dull aching, varying in severity, more with movement and radiating to the right. The patient was an accountant by profession, worked in the vicinity of a drain and often consumed the nearby tap water. Family history was unremarkable. There was no history of contact with pets. He was a non-vegetarian, mostly a fish eater, but had never consumed any other sea food.
On general physical examination, the patient was conscious and well oriented. His vital parameters were within normal range. The patient was however admitted for investigations and follow-up. His blood urea and creatinine levels were raised, being 65 mg/dL was 1.8 mg/dL, respectively. The erythrocyte sedimentation rate (ESR_ was also raised -58 mm in the 1 st hour. Urinary albumin secretion was 1+, with occasional hyaline casts and calcium oxalate crystals. Other biochemical and haematological parameters were within normal limits.
CT scan at this point of time revealed bilateral perinephric fluid collection, more on the left side, with a displaced DJ stent in the left perinephric collection. Urinary bladder appeared normal. An impression of perinephric uroma left lumbar region was made.
The stent was placed back and about 15 ml of drain fluid was sent to the Microbiology Department. On macroscopic examination, it was slightly cloudy, whitish in colour, containing a thin, white, elongated dead parasite-like structure that was white, wrinkled and ribbon-shaped, about 15 cm long and, on an average, 1.5-1.7 mm wide [Figure 1]. The posterior end was tapering, measuring approx 0.6 mm [Figure 2] and the anterior end was featureless [Figure 3], measuring approximately 1.7 mm.
|Figure 1: Sparganum as isolated from the perinephric fl uid collection, showing its thread-like structure and its length (about 15 cm)|
Click here to view
The parasite was sent to IVRI (Indian Veterinary Research Institute), Izatnagar, U.P., for identification. On the basis of the site of predilection and gross morphological appearance of the parasite, the parasites suspected were Dioctophyma renale or Dracunculus medinensis or a plerocercoid (sparganum). However, because the extremities were not well defined/damaged, on the basis of body tissue composition and orientation, the possibility of the parasite being a nematode was ruled out. Therefore, according to IVRI, the parasite resembled a plerocercoid (sparganum) of Spirometra spp.
Meanwhile, he was treated with Mebendazole at a dose of 100 mg BD for 3 days. No other parasites/spargana were found in the drain fluid on subsequent examinations. The patient was doing well on his follow-up after 1 year.
| ~ Discussion|| |
Sparganosis has been reported worldwide, especially in East Asia (China, Japan and Korea), South-east Asia (Malaysia, India and Philippines) and South-west US.  Experimental infection was done by Mueller and Coulston into themselves to observe the migration and penetration of spargana after inoculating themselves with the heads of these larvae subcutaneously. 
The incubation period ranges from 20 days to 14 months, although spargana can live up to 20 years in a human host.  Human sparganosis is an accidental extraintestinal infection. In nature, sparganosis is maintained primarily by contamination of natural or artificial bodies of water with faeces from felids and canids infected with Spirometra spp. The two species most commonly recovered are S. mansoni and S. proliferum. Common routes of infection are ingestion of contaminated water or of a second intermediate host such as a frog or snake or contact between a second intermediate host and an open wound or mucus membrane, seen especially in Vietnam and Thailand, where frogs are popularly believed to have an antiphlogistic effect and are applied over the wound as poultices. , However, under certain sociocultural conditions as in some regions of central Africa, it is suspected that man apparently acts as the only intermediate host and hyenas are the definitive hosts of Spirometra, the cycle being maintained as a result of the tribal custom of letting hyenas feed on human corpses.  In this particular case, the source of infection could be the drinking water from a tap in the vicinity of a sewage drain.
After consumption by man, the procercoids and plerocercoids fail to develop further and, therefore, they migrate through the intestinal wall and invade various tissues, releasing toxins and causing oedema. The main symptom is pruritus, sometimes accompanied by urticaria. Migration of the sparganum to internal organs can give rise to the visceral form of disease. Although it can localize in the brain, spinal cord, subcutaneous tissue, breast, scrotum, urinary bladder, abdominal cavity, eye and intestinal wall, the preferred locations include the intestinal wall, perirenal fat and the mesentery. After death of the larvae, severe local inflammation may develop around the larvae resulting in mass-like lesions. 
Specific diagnosis can be made only by removing the nodular lesion and confirming the presence of the plerocercoid larvae by studying its characteristic morphology. Sparganum looks like a bright white ribbon, with the undulating movement typical of a pseudosegmented cestode and with an invagination at the oral end. 
However, accurate species identification requires feeding the sparganum to a proper definitive host, e.g. a dog or cat, and recovering the adult parasite for further morphologic study. Diagnosis in definitive hosts infected with adult cestodes can be made by coprologic examination or autopsy.  In sparganosis, surgery is usually required for both diagnostic and therapeutic purposes. 
To prevent ingestion of Cyclops or Spirometra larvae, water in endemic areas should be boiled or treated with disinfectants. Currently, no effective treatment for sparganosis is known.  The only treatment is surgical excision of localized infection, although Praziquantel has been used with limited success. 
Sparganosis is rare in India, but the discomfort of the patient should not be aggravated by a wrong/delayed diagnosis and the lack of knowledge of its existence. Although sparganosis is most commonly and evidently diagnosed by recovery of the parasite from the patient, the importance of other surrogate findings like eosinophilia, antisparganum ELISA test, etc. along with relevant clinical history cannot be over-emphasized. Also, improvement in public health measures and provision of safe drinking water still remains an issue to be resolved.
| ~ Acknowledgements|| |
The authors sincerely acknowledge the help extended to them by Dr Jagvir Singh, Joint Director and Head, Dr D Bora, C.M.O. (NFSG), Department of Parasitic Diseases, National Institute of Communicable Diseases, Delhi and Dr JR Rao, Principal Scientist and Head, Division of Parasitology, Indian Veterinary Research Institute, Izatnagar, U.P., who helped in the identification of this parasite of great ecological importance.
| ~ References|| |
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|2.||Irizarry L, Phan P. Tapeworm Infestation. Brooklyn Hospital Center. Available from: http://emedicine.medscape.com/article/786292-overview. [updated on 2009 Nov 10, cited on 2010 Jun 21]. |
|3.||Mueller JF, Coulston F. Experimental Human Infection with the Sparganum Larva of Spirometra Mansonoides. Am J Trop Med 1941;21:399-425. |
|4.||Baily G. Other cestode infections: Intestinal cestodes, cysticercosis, other larval cestode infections. In: Cook GC, Manson P, Zumla AI, editors. Manson′s Tropical Diseases. China: Saunders Elsevier; 2009. p. 1569-84. |
|5.||Hughes AJ, Biggs BA. Parasitic worms of the central nervous system: An Australian perspective. Internal Med J 2001;32:541-3. |
|6.||Zoonoses and communicable diseases common to man and animals. In: Parasitoses. Vol. 3, 3rd ed. USA Pan American sanitary bureau, regional office of the World Health Organization; 2003. p. 210-3. |
|7.||Torres JR, Noya OO, Noya BA, Mouliniere R, Martinez E. Treatment of proliferative sparganosis with mebendazole and praziquantel. Trans R Soc trop Med Hyg 1981;75:846-7. |
[Figure 1], [Figure 2], [Figure 3]
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