LGCmain
Indian Journal of Pathology and Microbiology
Home About us Instructions Submission Subscribe Advertise Contact e-Alerts Ahead Of Print Login 
Users Online: 894
Print this page  Email this page Bookmark this page Small font sizeDefault font sizeIncrease font size
IJPM is coming out with a Special issue on "Genitourinary & Gynecological pathology including Breast". Please submit your articles for these issues


 
  Table of Contents    
BRIEF COMMUNICATION  
Year : 2016  |  Volume : 59  |  Issue : 1  |  Page : 56-58
Pleuropulmonary paragonimiasis with migrated lesions cured by multiple therapies


1 Department of Infectious Disease, Union Hospital, Tongji Medical College, Huazhong University of Science and Technology, Wuhan 430022, China
2 Department of Radiology, Union Hospital, Tongji Medical College, Huazhong University of Science and Technology, Wuhan 430022, China

Click here for correspondence address and email

Date of Web Publication9-Mar-2016
 

   Abstract 

Paragonimiasis is an infectious disease caused by Trematodes of the genus Paragonimus that is endemic in Asia, Africa, and South America. Most patients with paragonimiasis are cured by standard praziquantel treatment. However, several cases have been reported to have unsatisfactory responses to the standard praziquantel treatment. To probe the clinical characteristics, possible cause, and management of the paragonimiasis individuals improved by multiple therapies, we present a 12-year-old Chinese boy, who was infected with Paragonimus accompanied by arachnoid cyst involvement, as not having typical clinical symptoms, but repeatedly presenting with migrated lesions between the lung and pleura. He responded to treatment with 3 cycles of praziquantel and 1 cycle of albendazole.

Keywords: Albendazole, migrated lesions, multiple therapies, paragonimiasis, praziquantel

How to cite this article:
Hu Y, Qian J, Yang D, Zheng X. Pleuropulmonary paragonimiasis with migrated lesions cured by multiple therapies. Indian J Pathol Microbiol 2016;59:56-8

How to cite this URL:
Hu Y, Qian J, Yang D, Zheng X. Pleuropulmonary paragonimiasis with migrated lesions cured by multiple therapies. Indian J Pathol Microbiol [serial online] 2016 [cited 2019 Nov 22];59:56-8. Available from: http://www.ijpmonline.org/text.asp?2016/59/1/56/174820



   Introduction Top


Paragonimiasis is an infectious disease caused by lung flukes of the genus Paragonimus and is endemic in Asia, Africa, and South America. [1] The nationwide prevalence in China is estimated at 1.71%. [2] In Hubei Province, an epidemiological survey revealed that paragonimiasis is widespread with a mean prevalence of 10-20%. [1] Transmission to humans occurs by the ingestion of inadequately cooked crabs or crayfish that are infected with Paragonimus. The life cycle of Paragonimus involves crabs or crayfish as intermediate hosts and humans, cats, dogs, or foxes as definitive hosts. The clinical manifestations of paragonimiasis characteristically present in the chest, abdomen, or brain. The pulmonary manifestations are a predominately cough, hemoptysis, chest pain, dyspnea, and eosinophilia.

Praziquantel is the most commonly used the drug for the treatment of human paragonimiasis in China. [3] About 80-90% of [4],[5] paragonimiasis cases can be successfully treated with praziquantel. Here, we report a 12-year-old Chinese boy, who was infected with Paragonimus accompanied by arachnoid cyst involvement, as not having typical clinical symptoms, but repeatedly presenting with migrated lesions between the lung and pleura, which was resistant to standard treatment, but improved with 3 cycles of praziquantel treatment and 1 cycle of albendazole treatment.


   Case report Top


The patient is a 12-year-old boy who resides in Enshi City, China. He was first admitted to a local hospital in September 2012 with a 1-week history of marasmus, fatigue, anorexia, and occasional headache. Blood tests showed that his total white blood cell count (WBC) was 18.38 × 10 9 cells/L with 8.2% eosinophilia. The enzyme-linked immunoabsorbent assay (ELISA) for Paragonimus species was positive. The report of computed tomography (CT) scan of the chest revealed right lung infiltrate with a pleural effusion on the right side (the image was not available). The abdominal sonography indicated a small amount of ascites. Upon further questioning, he had visited his relatives in the mountain area of Enshi, Hubei, China, in July 2012, and consumed two incompletely cooked freshwater crabs. His cousins, who ate more crabs, were asymptomatic. The patient was diagnosed with paragonimiasis and treated with praziquantel (150 mg/kg in 3 divided doses for 3 days). Five days after treatment, the chest CT scan showed significant improvement except for the small residual pleural effusion on the right side. The ascites had disappeared. At discharge, he did not complain of any discomfort. At the follow-up visit in December 2012, his WBC was elevated (17.12 × 10 9 cells/L) and the eosinophil percentage was 54.3%. The chest CT showed a right lung nodule and a pleural effusion on the right side [Figure 1]a. He was readmitted to the local hospital and received an additional cycle of praziquantel treatment (150 mg/kg in 3 divided doses for 3 days). His WBC decreased to 11.77 × 10 9 cells/L and the eosinophil count came down to 13.9%. The chest CT showed the resolution of the right lung nodule, improvement of the right pleural effusion and clearing of the lung fields. At follow-up examination in January 2013, the patient's WBC was elevated to 13.23 × 10 9 cells/L and the eosinophil count was back to 20.9%. The chest CT scan at that time indicated that the lesions had migrated to the left lower lobe.
Figure 1: The changes of the chest computed tomography scans over time in a 12-year-old boy who had migrated lesions between the lung and pleura. (a) Before the second praziquantel treatment, computed tomography scan of the chest showing a right lung nodule and a pleural effusion on the right side. (b) Before the third praziquantel treatment, computed tomography scan of the chest with intravenous contrast showing multiple lung nodules and a mass of the upper left lung

Click here to view


The patient was referred to our hospital for further treatment in February 2013. His initial examination showed normal temperature, blood pressure of 106/66 mm Hg, the pulse of 85 beats/min, and a respiratory rate of 20 breaths/min. No other outward significant signs were found. However, his total WBC count was 13.84 × 10 9 cells/L, with 27.7% eosinophilia, and the erythrocyte sedimentation rate was 29 mm/h. The CT scan of his chest with intravenous contrast showed multiple lung nodules and a mass of the upper left lung [Figure 1]b. The magnetic resonance imaging (MRI) of his brain showed an arachnoid cyst (size 6.2 cm × 5.5 cm × 6.2 cm) of the left temporal lobe. Serum was sent to several different laboratories for a Paragonimus antibody test and the results were positive. Other parasite infections such as Schistosomiasis Japonica, Trichinosis, and Cysticercosis were excluded. The serological tests were negative for Rickettsia, Ehrlichia, and Strongyloides species, Epstein-Barr virus, herpes simplex virus, Cytomegalovirus, and human immunodeficiency virus. Tuberculin skin testing and immunological tests for tuberculosis were negative. The patient's parent refused bronchoscopy. The diagnosis of paragonimiasis was confirmed based on the patient's epidemiology history, eosinophilia, serological results, and chest findings. The brain MRI results were reviewed by a radiologist, a neurosurgeon, and a neurologist, and the arachnoid cyst was diagnosed as a benign cyst and not associated with the paragonimiasis. The patient was treated with a third cycle of praziquantel therapy (150 mg/kg in 3 divided doses for 3 days). The next day after treatment, the chest CT indicated that the nodules in the left lung had disappeared and only a very small pleura effusion was present on the left side. The WBC and eosinophil remained elevated.

In March 2013, the patient's WBC remained elevated (WBC, 15.61 × 10 9 cells/L; eosinophil, 33.2%). The chest CT indicated a large left pleural effusion. Therefore, he was readmitted to our hospital. The pleural fluid was aspirated by thoracentesis and laboratory tests on the pleural fluid showed the following findings: WBC, 11,075 cells/μL (20% neutrophils, 8% lymphocytes, 7% monocytes, and 65% eosinophils); pH, 7.135; proteins, 8.8 g/dl; albumin, 2.5 g/dl; and glucose, 6 mg/dl. The eggs of Paragonimus were not found in the pleural fluid. Triclabendazole is the second line drug that can be used for paragonimiasis treatment; however, it is not available in China. The patient was thus given a cycle of albendazole treatment (8 mg/kg in 2 doses each day for 7 days). Following the albendazole treatment, in over 1-month, the WBC was back to normal and eosinophil gradually went back to normal levels. The chest sonography showed no pleural effusion.


   Discussion Top


The incidence of paragonimiasis has decreased in many endemic countries. However, paragonimiasis is increasing in some regions. [1],[2] In our case, the patient had always resided in an urban area that is not considered endemic. He only visited the mountain area in Enshi once and was infected with Paragonimus after only eating two undercooked freshwater crabs. Other members of his family, who also consumed freshwater crabs at the same time, had no symptoms. This may indicate that persons who live in the endemic area may have a protective immunity to Paragonimus. Persons who live in nonendemic areas may be more sensitive to Paragonimus infection.

The typical symptoms of pleuropulmonary paragonimiasis include fever, cough, blood-tinged sputum, dyspnea, abdominal pain, and chest pain. [6] Our patient did not present with these typical symptoms. He only complained of anorexia, marasmus, fatigue, and occasional headache. It was doubtful that he had a parasite infection due to the marked peripheral blood eosinophilia and dietary history. Further tests showed that only the antibody to Paragonimus was positive and the results were confirmed by several different laboratories, including two reference laboratories. Other parasite infections such as Schistosomiasis japonica, Trichinosis, and Cysticercosis were excluded. The CT image indicated that the patient might have a parasite infection. The patient's mild symptoms did not match with his severe lesions in the lung. The predominant Paragonimus species are Paragonimus westermani and Paragonimus skrjabini in China. [1] The ELISA kit used for detecting the antibody to Paragonimus cannot differentiate these two species. We tried to get the eggs of Paragonimus from the pleural fluid aspirate and feces, but they could not be detected.

When humans eat parasitized crayfish or crabs, the metacercaria excyst in the duodenum can migrate to the lung and ectopic locations such as brain or subcutaneous tissues. [7],[8] Most paragonimiasis case reports only present a 1-time CT image, but the dynamic change of these lesions is not known. This patient had dynamic CT images and they clearly showed that the lesions migrated from the upper right lung to the lower left lung and then moved to the upper left lung. When the lesions in the lung disappeared, the pleural effusion appeared. The brain MRI showed an arachnoid cyst, but it was concluded that it was congenital and not associated with paragonimiasis based on the patient's clinical symptoms and the MRI characteristics.

The reasons that patients are resistant to standard praziquantel are not clear. The chronicity of paragonimiasis or the severity of paragonimiasis could be the reason for the requirement of additional doses of treatment. [9] Although this patient did not present typical clinical symptoms, the lesions were severe and this may be the reason that he did not respond well to standard treatment. Triclabendazole is another WHO-recommended drug for treatment of paragonimiasis; however, this drug cannot be obtained in China. A search of the published literature indicated that albendazole is also useful in the treatment of paragonimiasis. [10]

Since this patient did not show typical symptoms, it was difficult to conclude whether he was completely cured or not based on the clinical symptoms. The experience obtained from this patient should remind physicians that it is very important to have follow-up examinations after every treatment.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
   References Top

1.
Liu Q, Wei F, Liu W, Yang S, Zhang X. Paragonimiasis: An important food-borne zoonosis in China. Trends Parasitol 2008;24:318-23.  Back to cited text no. 1
    
2.
Coordinating Office of the National Survey on the Important Human Parasitic Diseases. A national survey on current status of the important parasitic diseases in human population. Zhongguo Ji Sheng Chong Xue Yu Ji Sheng Chong Bing Za Zhi 2005;23 5 Suppl: 332-40.  Back to cited text no. 2
    
3.
Chen MG. New developments in the clinical study of praziquantel. Ji Sheng Chong Xue Yu Ji Sheng Chong Bing Za Zhi 1984;2:193-5.  Back to cited text no. 3
[PUBMED]    
4.
Ibáñez N, Jara C. Experimental paragonimiasias: Therapeutical tests with praziquantel - First report. Mem Inst Oswaldo Cruz 1992;87 Suppl 1:107.  Back to cited text no. 4
    
5.
Udonsi JK. Clinical field trials of praziquantel in pulmonary paragonimiasis due to Paragonimus uterobilateralis in endemic populations of the Igwun Basin, Nigeria. Trop Med Parasitol 1989;40:65-8.  Back to cited text no. 5
    
6.
Jeon K, Koh WJ, Kim H, Kwon OJ, Kim TS, Lee KS, et al. Clinical features of recently diagnosed pulmonary paragonimiasis in Korea. Chest 2005;128:1423-30.  Back to cited text no. 6
    
7.
Chen J, Chen Z, Lin J, Zhu G, Meng H, Cui G, et al. Cerebral paragonimiasis: A retrospective analysis of 89 cases. Clin Neurol Neurosurg 2013;115:546-51.  Back to cited text no. 7
    
8.
Dainichi T, Nakahara T, Moroi Y, Urabe K, Koga T, Tanaka M, et al. A case of cutaneous paragonimiasis with pleural effusion. Int J Dermatol 2003;42:699-702.  Back to cited text no. 8
    
9.
Oh IJ, Kim YI, Chi SY, Ban HJ, Kwon YS, Kim KS, et al. Can pleuropulmonary paragonimiasis be cured by only the 1 st set of chemotherapy? Treatment outcome and clinical features of recently developed pleuropulmonary paragonimiasis. Intern Med 2011;50:1365-70.  Back to cited text no. 9
    
10.
Liu WJ. Effect of albendazole on the paragonimiasis. Zhongguo Ji Sheng Chong Xue Yu Ji Sheng Chong Bing Za Zhi 1987;5:309-10.  Back to cited text no. 10
    

Top
Correspondence Address:
Xin Zheng
Department of Infectious Disease, Union Hospital, Tongji Medical College, Huazhong University of Science and Technology, Jiefang Avenue 1277, Wuhan 430022
China
Login to access the Email id

Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0377-4929.174820

Rights and Permissions


    Figures

  [Figure 1]

This article has been cited by
1 Pulmonary larval paragonimiasis mimicking lung cancer
V. V. Ermilov,A. V. Smirnov,G. L. Snigur,R. S. Dudin,S. S. Popov
Arkhiv patologii. 2018; 80(2): 60
[Pubmed] | [DOI]



 

Top
 
 
  Search
 
    Similar in PUBMED
   Search Pubmed for
   Search in Google Scholar for
 Related articles
    Email Alert *
    Add to My List *
* Registration required (free)  


    Abstract
   Introduction
   Case report
   Discussion
    References
    Article Figures

 Article Access Statistics
    Viewed2009    
    Printed33    
    Emailed1    
    PDF Downloaded58    
    Comments [Add]    
    Cited by others 1    

Recommend this journal