Annals of Medicine & Surgery Publish Ahead of Print

DOI:10.1097/MS9.0000000000000926
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Diagnostic Challenges in Paragonimus westermani Infestation: A Case Report from Nepal

Kamal Kandel1, Arun Gautam2, Saroj Poudel3, Prakash Banjade1, Prashant Mani Tripathi2
1

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Manipal College of Medical Sciences, Pokhara, Kaski, Nepal
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B.P. Koirala Institute of Health Sciences, Dharan, Nepal
3
Chitwan Medical College, Bharatpur, Chitwan, Nepal

Corresponding author
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Name: Kamal Kandel

Email address: Kamalkandel010@gmail.com

ORCID: 0000-0002-2413-9774

Phone Number: +9779857625613

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Permanent Address: Shreenagar Tole-2, Baglung, 33300, Nepal

Highlights
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 Paragonimiasis is a zoonotic infection that mainly affects Southeast Asia, with

Paragonimus westermani being common in Nepal.
 The most common presentation of paragonimiasis is pulmonary involvement, but sputum
examination is not always reliable. Serological tests are more accurate, and the multiple
dot ELISA is recommended for patients with eosinophilia and pulmonary symptoms.
 Paragonimiasis should be suspected in patients with eosinophilia and inconclusive
diagnostic findings, especially if they have a history of consuming crustaceans.

Copyright © 2023 The Author(s). Published by Wolters Kluwer Health, Inc. This is an open access article distributed
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 Introduction and Importance: Paragonimiasis is a parasitic infection that is endemic in many
parts of Asia, including Nepal, and is spread by consuming raw or undercooked crustaceans.
Pulmonary involvement is common, and extra-pulmonary involvement is rare but can occur,
with cerebral paragonimiasis being the most common. Diagnosis is challenging, as sputum
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examination has a high false negative rate, and serological tests are more reliable.
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Case presentation: We present the case of a 39-year-old female with a history of consuming
crab who presented with pulmonary symptoms, eosinophilia, and pleural effusion, and was
eventually diagnosed with Paragonimus westermani infection.

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Clinical discussion: Paragonimiasis, caused by Paragonimus westermani, is a zoonotic infection
common in Nepal transmitted through infected crustaceans. Clinical presentation varies, but
pulmonary symptoms are common and can mimic tuberculosis, making diagnosis challenging.

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Blood tests may show eosinophilia, but sputum examination has a high false negative rate.
Serological tests have high sensitivity and specificity, and multiple dot ELISA is recommended
in patients with eosinophilia and pulmonary findings. In resource-limited settings, a high index
of suspicion is crucial in diagnosing paragonimiasis, particularly in patients with a history of
crustacean intake and inconclusive diagnostic findings.
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Conclusion: We recommend that clinicians in endemic regions be aware of the possibility of
paragonimiasis in patients with eosinophilia and inconclusive diagnostic findings, particularly
those with a history of consuming crustaceans. Early diagnosis and treatment are critical in
preventing morbidity and mortality associated with this disease.

Keywords:
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Paragominus westermani, Steroids, Tropical disease, Eosinophilia

Abbreviations:
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ELISA: Enzyme linked Immune Sorbent Assay.

CDC: Centers for Disease Control and Prevention.

CT: Computed Tomography.

 Introduction:

Paragonimiasis is a parasitic infection caused by helminths of the genus Paragonimus, which is

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endemic in many East Asian countries, such as China, Japan, the Philippines, as well as some
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regions in Africa and America. 1 The majority of the burden of this disease is in the Asian region.
The parasite is acquired through ingestion of raw or undercooked crab or crayfish, which contain
the infective metacercariae. Upon ingestion, the metacercariae excyst in the duodenum and
penetrate the intestinal wall, eventually infiltrating the lung parenchyma through the peritoneum.

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The adult worm lays eggs that are either coughed up in sputum or swallowed and eventually
excreted in feces. While pulmonary disease is the most common manifestation, rare cases of
extrapulmonary involvement, such as cerebral paragonimiasis, have been reported.2,3 We present

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the case of a 39-year-old female, who presented to us with a persistent burning sensation over the
chest for 5 months, cough for 15 days, and cushingoid habitus. She had been previously treated
with steroids for 5 months for eosinophilia and eosinophilic pleural effusion. This case highlights
the challenges in diagnosing and managing paragonimiasis and emphasizes the importance of
considering parasitic infections in patients with eosinophilia and pleural effusion, particularly in
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areas where the disease is endemic. This case is written in accordance with the SCARE
guidelines.4

Case Report:

A 39-year-old female from Sunsari district, Nepal, presented to the outpatient department of our
hospital with a complaint of burning sensation over the chest and neck for 5 months which was
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gradual in onset, progressive with no aggravating and relieving factors identified and was
associated with shortness of breath on walking uphill. She also complained of cough for 15 days
which was insidious in onset, gradually progressive, each episode having prolong bouts of cough
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which was productive with blackish flakes and the cough was more in the morning. Upon review
of her medical history, we found that she had previously been treated with prednisolone and
albendazole for hypereosinophilic syndrome /eosinophilic pleural effusion for 4 months. The was
no significant past medical history like tuberculosis. Patient denied consumption of alcohol and
tobacco products. The patient frequently consumes crab since it is widely believed in their
culture to reduce joint pain.

The vitals were normal and on head-to-toe examination, the only notable finding was cushingoid
features. Systemic examination was normal except there were decreased breath sounds on the
right infra scapular and infra-axillary region.

On laboratory evaluation complete blood count revealed a total leukocyte count of 15200/cu.mm
with neutrophil 31%, lymphocyte 25%, eosinophil 40%, and monocyte 4%. Peripheral blood
smear shows normocytic normochromic red blood cells, marked eosinophilia with no abnormal
morphology. Liver and kidney function tests were normal. Routine urine and stool examination
 were normal. Antinuclear antibody (ANA) test was negative. Chest imaging on X-ray showed
bilateral pleural effusion (Fig 1) while CT revealed bilateral pleural effusion (right>left) and
reactive mediastinal lymph node. Sputum microscopy revealed a yellow brown ovoid structure
with operculum resembling the egg of Paragonimus westermani (Figure 2). Ultrasound guided
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pleural fluid aspiration was done and the analysis revealed exudative pattern with protein
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37gm/L, glucose 4mmol/L, and lactate dehydrogenase (LDH) 297.1 U/l. Pleural fluid cytology
showed, plenty of leucocytes with eosinophil predominance and red blood cells.

As the diagnosis of paragonimus westermani was confirmed the patient was admitted to our

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hospital and treated with praziquantel and diethylcarbamazine during the four-days. Upon
discharge, she was prescribed a prednisolone tapering dose for 3 weeks and diethylcarbamazine
for 3 days and was advised to follow-up after 2 weeks. Follow-up sputum examination showed

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no growth and eggs, and the chest x-ray showed clear lung fields bilaterally (Figure 3).

Discussion:

Paragonimiasis is a zoonotic infection caused by trematodes of the genus Paragonimus, with an

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estimated global burden of 22.8 million, primarily in Southeast Asia. Paragonimus westermani is
the most common type prevalent in this region, including Nepal.5 The disease is acquired by
ingesting freshwater crustaceans that are infected with metacercariae. Once ingested, the
metacercariae excyst in the duodenum and penetrate the abdominal wall, through the peritoneum,
to reach the lung parenchyma. The parasite lays eggs in the lungs, which are either coughed up in
sputum or swallowed and excreted in the stool.3
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Patients with paragonimiasis typically present with pulmonary involvement, including cough,
chest pain, and hemoptysis. In our case, the patient presented with black mucus flakes and
burning central pain. Blood investigations often show eosinophilia, and chest imaging typically
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shows pleural effusion. However, pericardial effusion and nodular opacities mimicking
tuberculosis and lung cancer have also been reported in the literature.6,7

Paragnonimiasis is a zoonotic infection with high burden in southeast Asia, while paragonimus
westermani being common in Nepal. Infected crustacean is the reservoir of the disease. Clinical
presentation is variable, however pulmonary symptoms are frequent. Blood picture shows
eosinophilia usual with any parasitic infections. Pulmonary paragonimiasis often mimics
tuberculosis which makes the diagnosis challenging. Sputum examination is not always reliable
as false negative rate is high. Serological test confers high sensitivity and specificity. If available
multiple dot ELISA should always be considered for patient with eosinophilia and pulmonary
findings like nodularity, opacities and effusion.8 However, in resource limited countries like
Nepal where sophisticated tests are not readily available there should be high index of suspicion
for paragonimiasis in patients with eosinophilia and inconclusive diagnostic findings,
particularly those with a history of crustacean intake.
 In our case, we were able to identify the golden-brown, ellipsoidal, or oval operculated
Paragonimus ova in the sputum, confirming the diagnosis. Although the presence of ova in
sputum is highly specific, the sensitivity is low (28-30%), and repeated testing can confer higher
sensitivity. Failure to obtain positive sputum sample is the reason for diagnostic delay in our
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case. In most cases, stool examination may not reveal ova, as was the case in our patient.
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Serological testing using ELISA has high sensitivity and specificity, but this is a constraint in
resource-limited countries like Nepal.9 If available, multiple dot ELISA should always be
considered for patient with eosinophilia with pleural effusion to rule out paragonimus
westermani.8

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Extra-pulmonary involvement is rare but has been reported, with cerebral paragonimiasis being
the most common. Short course steroids may be added to decrease the inflammatory response in

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cases of extra-pulmonary paragonimiasis.10 The diagnostic challenge encountered in our case
highlights the importance of considering paragonimiasis in the differential diagnosis of patients
with eosinophilia and respiratory symptoms, especially in endemic areas. The initial diagnosis of
hypereosinophilic syndrome and treatment with steroids led to a worsening of symptoms and the
development of cushingoid habitus, delaying the correct diagnosis. This is consistent with reports
of misdiagnosis and delayed diagnosis of paragonimiasis due to non-specific symptoms and low
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awareness among clinicians.11 Prompt diagnosis and treatment with praziquantel are essential for
successful clinical outcomes. The CDC recommends a 25mg/kg oral dose of praziquantel given
in three divided doses over two consecutive days, with triclabendazole as an alternative
medication.10 In our case, we administered praziquantel 600mg three times daily for three days,
which resulted in clinical recovery and negative sputum examination for the parasite/ova during
follow-up.10
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Overall, our case highlights the importance of considering paragonimiasis in the differential
diagnosis of patients with pulmonary symptoms, especially in areas where the disease is
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prevalent. Prompt diagnosis and treatment with praziquantel are essential for successful clinical
outcomes. Further research is needed to improve diagnostic methods and develop effective
interventions for paragonimiasis, particularly in resource-limited settings.

Conclusion:

Paragonimiasis should be considered as a differential diagnosis in patients from endemic areas

presenting with non-specific respiratory symptoms, peripheral eosinophilia, and non-specific
chest imaging findings like effusion and nodular opacities. Our case highlights the diagnostic
challenges in identifying this rare but treatable parasitic infection, which was initially
misdiagnosed as hypereosinophilic syndrome and treated with steroids. A high level of suspicion
and thorough evaluation are crucial in reaching a correct diagnosis, especially in resource-limited
settings where immunological tests are not readily available. Early diagnosis and prompt
 treatment with praziquantel can lead to complete recovery and prevent potential complications.
Therefore, healthcare providers should maintain a high index of suspicion for paragonimiasis in
patients with eosinophilia and inconclusive diagnostic findings, particularly those with a history
of crustacean intake in endemic regions.
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Patient Perspective: Patient was happy on diagnosis and effective management of her condition.

Ethical approval: N/A

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Source of funding: N/A

Conflict of interest: N/A.

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Research registration unique identifying number (UIN): N/A

Patient consent: Written informed consent was obtained from the patient for publication of this
case report and accompanying images. A copy of the written consent is available for review by
the Editor-in-Chief of this journal on request.
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Provenance and peer review: Not commissioned, externally peer- reviewed
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Figure 1: Chest X-ray with bilateral pleural effusion.

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Figure 2: yellow brown ovoid structure with operculum suggesting Paragominus westermani

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Egg.
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Figure 3: Chest x-ray on follow up-resolved bilateral pleural effusion.

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