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CORRESPONDENCE
Year : 2013  |  Volume : 9  |  Issue : 4  |  Page : 712-714

Sparganosis in a patient with diffuse large B cell lymphoma


1 Division of Medical Oncology, Seoul St. Mary's Hospital, Seoul, South Korea
2 Department of Plastic and Reconstructive Surgery, Uijeongbu St. Mary's Hospital, College of Medicine, The Catholic University of Korea, South Korea
3 Department of Oncology/Hematology, Daegu Fatima Hospital, Daegu, South Korea

Date of Web Publication11-Feb-2014

Correspondence Address:
Sang-Young Roh
Department of Plastic and Reconstructive Surgery, Uijeongbu St. Mary's Hospital, College of Medicine, The Catholic University of Korea, 271 Cheonbo-ro, Uijeongbu-si,Gyeonggi-do, 480-717
South Korea
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0973-1482.126462

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 > Abstract 

Sparganosis is the human infection by plerocercoid, the larvae of sparganum. Clinically, subjective symptoms do not occur in the incipient stage, but as the worm migrates, pruritus or tenderness may occur. On physical examination, soft, palpable, and sometimes migratory, subcutaneous nodules are found in sparganosis patients. As rare cases; sparganosis from the orbit, the abdominal viscera, and the breast have been reported. However, there have been no reports relating such disease to the patients' immunocompromised status.We experienced a case of sparganosis from a patient with lymphoma whose immune system was suppressed by anticancer therapy, suggesting that the immunosuppression might affect the onset and the exacerbation of the disease. We report our case with a review of the literature.

Keywords: Diffuse large B-cell lymphoma, lymphoma, sparganosis


How to cite this article:
Roh SY, Lee JY, Park KW, Jung SN. Sparganosis in a patient with diffuse large B cell lymphoma. J Can Res Ther 2013;9:712-4

How to cite this URL:
Roh SY, Lee JY, Park KW, Jung SN. Sparganosis in a patient with diffuse large B cell lymphoma. J Can Res Ther [serial online] 2013 [cited 2019 Dec 7];9:712-4. Available from: http://www.cancerjournal.net/text.asp?2013/9/4/712/126462


 > Introduction Top


Sparganosis is caused by the sparganum which infects the human body. It originates from the plerocercoid of the Spirometra mansoni, that is, the cestode of which the definitive host is the dog or the cat. [1],[2],[3] In most cases, movable or fixed subcutaneous nodules are clinically observed and it may be accompanied with pruritus and tenderness. [1],[2],[3],[4],[5] We observed a case of sparganosis from a patient with lymphoma whose immune system was suppressed by anticancer chemotherapy. Furthermore, the author supposed that the immunosuppression itself may have a great impact on the onset and the exacerbation of sparganosis, and we report the case with a literature review.


 > Case Report Top


A 47-year-old man admitted for epigastric pain and melena. He was diagnosed of diffuse large B cell lymphoma at the stomach by the endoscopic biopsy. The CT scan revealed a stomach wall thickening, multiple perigastric lymphadenopathies, and masses in the spleen.

After two cycles of CHOP (cyclophosphamide, doxorubicin, vincristine, prednisone), while the lymphoma of stomach, perigastric lymph nodes, and spleen were markedly regressed [Figure 1], multiple, 1-2.5 cm sized hard, non-movable subcutaneous masses were found on abdomen and chest wall as the new lesion. After four cycles of CHOP, the stomach wall thickening was markedly regressed, while the perigastric lymphadenopathies and the splenic masses completely disappeared. However, the subcutaneous masses of the chest and the abdomen tended to grow in size [Figure 2]. An excisional biopsy was performed on the abdominal subcutaneous mass. Histologic sections of the excised mass revealed the sparganum [Figure 3]. On hematologic examination, the WBC count was slightly low with 4,600/mm 3 and the eosinophil count slightly elevated by 33.1%. The antibody titer for sparganum measured 12-times greater than that of the control in an enzyme-linked immunosorbent assay (ELISA). On careful history taking, he had ingested flesh snakes several months before. The multiple masses, left over on the chest and the abdomen, were additionally excised. He was administered praziquantel 100 mg/kg for 2 days considering the possibility of undiscovered sparganum infestation in an immunocompromised patient on chemotherapy. On hematologic examination performed 2 months after administration of praziquantel, WBC count elevated to 5,600/mm 3 and eosinophils were normalized with 1.3%. No evidence of recurrence was found, and the patient is still under follow-up.
Figure 1: CT images. After two cycles of CHOP, three 1-2.5 cm sized subcutaneous non-movable hard masses were found at the abdomen and chest wall

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Figure 2: CT image. After four cycles of CHOP, the enlarged abdominal subcutaneous mass was noted

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Figure 3: Histologic findings (H and E, ×40). Sparganum (arrow)

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 > Discussion Top


Sparganums, the human parasites, have been found not only in the Southeast Asia but around the whole world since they were first discovered in the process of the autopsy of an Amoy native in 1882. [4] Human parasites invade the human bodies through the following three channels, [1],[2],[3],[4],[5] though they are rare cases, as in the following; first, drinking water containing cyclopses infected with procercoids. Second, eating secondary intermediate hosts on which the procercoids are parasitic, such as frogs, snakes, and rodents, without cooking. Lastly, applying the skin of the frog or snake, on which the procercoid is parasitic, to the wound as a folk remedy.

Clinically, they grow slowly and mostly develop fixed or migratory subcutaneous masses which are accompanied by tenderness. They can also invade the subconjunctival tissues and cause pruritus, pain, tenderness, edema, and subcutaneous hemorrhage. In some cases, they cause gastrointestinal perforation, peritonitis or brain abscess. In most cases, they are accompanied with leukocytosis and eosinophilia. [1],[2],[3],[4],[5]

Sparganosis may be diagnosed by clinical symptoms and habit of eating the raw, but the definite diagnosis is only made by detecting the larva in a wound or in an operative field. In case of worm necrosis or impossible extirpation, it is difficult to make a pathological diagnosis. In such cases, the serological diagnosis may be effective. The enzyme-linked immunosorbent assay (ELISA) can be effectively used in that it shows the elevation of the IgG antibody titer. [6] It has been reported that it takes several days to several years for patients to visit the hospitals from the onset of sparganosis; with the average of 3 years. [5] Interestingly, this case was discovered during the anticancer treatment. On the blood test performed previously to anticancer treatment, WBC count was normal (10,000/mm 3 ; eosiophil 4%), but 6 weeks after anticancer treatment, the lesions were found and WBC count dropped down to 4,600/mm 3 , while eosinophils increased to 33.1%.

Although, the size of the lymphomas of the stomach, the perigastic lymph nodes, and the spleen regressed due to the continuous anticancer treatments, the subcutaneous masses on the chest and the abdomen tended to grow.

It has been pointed out that immunosuppressed status is related to the onset of parasite infection and the fatal results, [7],[8] like cysticercosis that occurred concurrently with immunosuppressant in the deep chest and skin of patients with acute myelogenous leukemia, and strongyloidiasis that invaded the extensive skin of patients with non-Hodgkin lymphoma; and it was reported in an animal experiment that immunocompetence inhibits cysticercosis from fulminantly progressing and prevents reinfection through humoral and cellular immune mechanism. [9]

There is a possibility that sparganosis occur independently of anticancer therapy, but philological reviews suggest that anticancer therapy induced immunosuppression might actually cause the disease. Surgical excision is the most appropriate treatment. As alternative treatment options, oral administration of praziquantel or mebendazol, direct injection of aprocaine and 40% ethyl alcohol to the lesion, and the injection of novarsenobenzol into the vein have been reported. [10] In the case of our patient, surgical excision was performed followed by the oral administration of praziquantel.

As aforesaid, our experience on a case of sparganosis speculated that the immunosuppression caused by anticancer chemotherapy might influence the onset and exacerbation of the disease. We report our case with a review of the literature.

 
 > References Top

1.Chi JG, Chi HS, Lee SH: Histopathological study on human sparganosis. Kisaengchunghak Chapchi 1980;18:15-23.  Back to cited text no. 1
    
2.Chi JG, Sung RH, Cho SY. Tissue parasitic diseases in Korea. J Korean Med Sci 1988;3:51-62.  Back to cited text no. 2
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3.Lee SH, Chai JY, Hong ST. Clinical Parasite. 1 st ed. Seoul: Korea Medicine; 1996. p. 274.  Back to cited text no. 3
    
4.Paul CB, Rodney CJ, Eddie WC, editors. Clinical Parasitology. 9 th ed. Philadelphia: Lea and Febiger; 1984. p. 499-504.  Back to cited text no. 4
    
5.Lee BJ, Ahn SK, Kim SC, Lee SH. Clinical and histopathologic study of sparganosis. Korean J Dermatol 1992;30:168-74.  Back to cited text no. 5
    
6.Wang KC, Huh S, Hong ST, Chai JY, Choi KS, Lee SH. The fate of spargana inoculated into the cat brain and sequential changes of anti-sparganum IgG antibody levels in the cerebrospinal fluid. Kisaengchunghak Chapchi 1990;28:1-10.  Back to cited text no. 6
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7.Mauad T, Battlehner CN, Bedrikow CL, Capelozzi VL, Saldiva PH. Case report: Massive cardiopulmonary cysticercosis in a leukemic patient. Pathol Res Pract 1997;193:527-9.  Back to cited text no. 7
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8.von Kuster LC, Genta RM. Cutaneous manifestations of strongyloidiasis. Arch Dermatol 1988;124:1826-30.  Back to cited text no. 8
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9.Bojalil R, Terrazas LI, Govezensky T, Sciutto E, Larralde C. Thymus-related cellular immune mechanisms in sex-associated resistance to experimental murine cysticercosis (Taeia crassiceps). J Parasitol 1993;79:384-9.  Back to cited text no. 9
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10.Tsai MD, Chang CN, Ho YS, Wang AD. Cerebral sparganosis diagnosed and treated with sterotatic techniques. Report of two cases. J Neurosurg 1993;78:129-32.  Back to cited text no. 10
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