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\title{Hydropneumothorax as a Rare Complication of Secondary Hypereosinophilic
Syndrome: A Case Report}



\author[1]{Prakash Sapkota}%
\author[2]{Bhuwan  Thapa}%
\author[2]{Aseem Bhattarai}%
\author[2]{Sujata  Chalise}%
\author[2]{Sajana KC}%
\author[2]{Ashish Tamang}%
\affil[1]{Dhulikhel Hospital}%
\affil[2]{Kathmandu University School of Medical Sciences}%


\vspace{-1em}



  
  \date{April 25, 2026}


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\textbf{Title: `Hydropneumothorax as a Rare Complication of Secondary
Hypereosinophilic Syndrome: A Case Report'}

Prakash Sapkota, Bhuwan Thapa, Aseem Bhattarai, Sujata Chalise, Sajana K
C, Ashish Tamang

\textbf{Abstract} :

We present a case of a young man who presented with cough and shortness
of breath for two weeks with peripheral eosinophilia of unknown cause
leading to left-sided pleural effusion. He was initially administered
antiparasitic drugs (Ivermectin and Praziquantel). After one week the
patient presented with an acute increase in SOB and was diagnosed with
bilateral hydropneumothorax. A bilateral pigtail was inserted. Pleural
fluid analysis was done which was purulent with lymphocyte predominance
and high adenosine deaminase. The patient was treated with
anti-tubercular therapy and steroids. After six months of therapy, the
patient was asymptomatic. The hypereosinophilic syndrome can be
presented in several ways and this study presented in a distinct way via
hydropneumothorax.

\textbf{Introduction}

Hypereosinophilic syndrome (HES) is a rare group of disorders marked by
sustained overproduction of eosinophils and eosinophil-mediated organ
damage or dysfunction.\textsuperscript{1} Based on the underlying
etiologies, HES is classified as primary (clonal), reactive, and
idiopathic HES.\textsuperscript{2} The World Health Organization (WHO)
has defined HES as a sustained increase in the absolute eosinophil count
(AEC) above 1500/\selectlanguage{greek}µ\selectlanguage{english}L for longer than 6 months and tissue
damage.\textsuperscript{3} Though the underlying pathophysiology remains
unclear, clonal eosinophilic expansion and overproduction of
eosinophilopoietic cytokines and their enhanced activity-mediated rise
in eosinophils are the most accepted theories.\textsuperscript{2} Since
the onset is usually insidious, the disease might remain obscured for a
long period.\textsuperscript{3} Thus, it is extremely important to
identify the subtle clinical features along with performing routine
blood investigations for early diagnosis and
management.\textsuperscript{3} Limited studies have presented the case
details of the rare condition with eosinophil-mediated organ damage or
dysfunction. The clinical and other features of the case presented in
this study will contribute to expanding our understanding of this kind
of rare health condition focusing on the resources-limited settings
where tertiary care accessibility has several challenges. This case
study aims to present an adult man, a case of secondary hyper
eosinophilic syndrome in a tertiary care center in Nepal.

\textbf{Keywords:} Case Report, Hydropneumothorax, Peripheral
eosinophilia, Secondary Hypereosinophilic Syndrome

\textbf{Case History}

A 38-year-old male, migrant worker, a returnee from Malaysia presented
to the outpatient Department of Internal Medicine at Dhulikhel Hospital,
Nepal with a history of dry cough and exertional shortness of breath for
two weeks. In investigations, the laboratory findings suggested
peripheral eosinophilia (AEC: 8000/\selectlanguage{greek}µ\selectlanguage{english}L) and his chest x-ray showed
left-sided pleural effusion (Figure 1).

Suspecting a potential parasitic infestation, he was initially managed
with oral antiparasitic medications that included tablet praziquantel
75mg/kg/day in three divided doses for 3 days and tablet Ivermectin 12
mg once daily for 3 days. As he was progressing gradually during
hospital admission, he got discharged after three days of admission with
the prescription of oral steroids (prednisolone 1mg/kg/day) for 14 days.

A week after, the patient revisited the emergency department with
complaints of sudden onset shortness of breath for a few hours. A Chest
X-ray was repeated, which revealed bilateral hydro-pneumothorax with
collapsed lungs (Figure 2). A diagnostic and therapeutic tube
thoracotomy was performed by inserting chest tubes on both sides.
Purulent pleural fluid was present, whose analysis showed increased
eosinophil and lymphocyte with adenosine deaminase (ADA) value of
\textgreater{}100IU/L. (Table 2). Then, the case started receiving
anti-tubercular therapy (ATT) and continued the prednisolone. This was
based on the pleural fluid analysis reports keeping pleural tuberculosis
as the differential diagnosis through his sputum gene xpert was negative
for \emph{Mycobacterium tuberculosis} . After this medication, his
shortness of breath and other clinical status were improving. Despite
the completion of the initiation phase of the ATT,~his cough could not
subside, and his AEC remained more than 25000/\selectlanguage{greek}µ\selectlanguage{english}L. These findings raised
the suspicion of eosinophilic leukemia prompting a bone marrow
aspiration and biopsy along with Fluorescent In-situ Hybridization
(FISH) analysis--the results of which have been mentioned below in table
1.

\textbf{Discussion}

Hypereosinophilic Syndrome (HES) is defined by an abnormal
overproduction and deposition of eosinophils in the blood or peripheral
organs leading eosinophil - mediated organ damage or
dysfunction.\textsuperscript{4} HES has been further divided based on
etiology into primary (stem cell, myeloid or eosinophilic malignancy),
secondary (parasitic infections, solid tumors, T cell lymphoma), or
idiopathic.\textsuperscript{3} According to various sources idiopathic
HES is an extremely rare disease and its incidence ranges from 0.04 to
0.17 per 100,000 persons and the prevalence ranges from 0.15 to 0.89
cases per 100,000 persons.\textsuperscript{5} Due to advances in
diagnostic techniques, causes of eosinophilia can be identified in a
proportion of cases that in the past would have been classified as
idiopathic. However, due to the unavailability of services in Nepal,
some of the investigations of the indexed case were sent to India for
further evaluation. The hypereosinophilic syndrome reports male
predominance, with a male-to-female ratio of 9:1.\textsuperscript{5} HES
is commonly diagnosed in patients aged 20-50 years, with a peak
incidence in the fourth decade. This study also exemplified that 38
years-old male case presented with rare clinical manifestations of
hypereosinophilic syndrome.

The most common cause of eosinophilia in the United States is an
allergic reaction or allergic disease.\textsuperscript{6} Worldwide, the
most common cause of eosinophilia is
parasitosis.\textsuperscript{7}Several mechanisms have been proposed for
the pathogenesis of hypereosinophilic syndrome, including the
overproduction of eosinophilopoietic cytokines, their enhanced activity,
and defects in the normal suppressive regulation of
eosinophilopoiesis.\textsuperscript{8} Eosinophils amplify the
inflammatory cascade by secreting chemoattractants that recruit more
eosinophils.\textsuperscript{9} Organ damage-induced HES is due to the
eosinophilic infiltration of the tissues accompanied by the mediator
(major basic protein, eosinophil peroxidase, eosinophil-derived
neurotoxin, and eosinophil cationic protein) release from the eosinophil
granules.\textsuperscript{9} The case presented in this study was
suspected of the parasitic infestation and later further investigations
reported that the cause could be eosinophilic leukemia. A similar case
of hypereosinophilic syndrome case was presented with features of
dilated cardiomyopathy as a dilemma to approach and treatment, as our
case with the presentation of bilateral hydro pneumothorax as
presentation.\textsuperscript{\textbf{10}}

HES is a heterogeneous disease process with multiple manifestations that
may occur simultaneously or individually.\textsuperscript{4} The
presentation can be sudden and dramatic, with cardiac, neurologic, or
thrombotic complications, but, more often, the onset is insidious. HES
can involve any organ system but commonly involves the respiratory
tract, skin, central nervous system, and cardiac.\textsuperscript{4}Many
patients experience fever and night sweats.~Some sources identify
anorexia and weight loss as common presenting manifestations. Cardiac
involvement is the leading cause of mortality and common symptoms of
cardiac involvement include chest pain, dyspnea, or orthopnea. A
chronic, persistent cough, usually nonproductive, is the most common
respiratory symptom reported in hypereosinophilic syndrome.~Rhinitis or
angioedema is sometimes a presenting feature. Dyspnea may occur in
cardiac or respiratory involvement. Embolic or thrombotic strokes or
transient ischemic episodes may occur and are often the initial
manifestations. Some patients with HES experience encephalopathy caused
by CNS dysfunction. Peripheral neuropathies account for about 50\% of
all neurologic symptoms in HES.

Arthralgia, myalgia, pruritus, diarrhea, and fatigue are relatively
common manifestations of HES but are non-specific. However, the case in
this study demonstrated some of the abovementioned clinical such as
shortness of breath, dry cough, and chest pain.

The decision to use a drug for the treatment of HES depends partly on
the presence or absence of organ involvement.\textsuperscript{4} So, the
initial evaluation of the patient with eosinophilia should include tests
that facilitate the assessment of target organ damage. In asymptomatic
patients' treatment is postponed till a diagnostic workup is completed
and a specific diagnosis is made. Early initiation of therapy is
reasonable in clonal eosinophilia associated with imatinib
mesylate---sensitive molecular markers (eg,~\emph{FIP1L1-PDGFRA, PDGFRB}
~rearrangement,~\emph{BCR-ABL} ). In idiopathic HES no evidence to
support early drug treatment. Periodic follow-up with monitoring of
complications is reasonable (no evidence to support
this).\textsuperscript{2} Symptomatic patients with clonal HES imatinib
mesylate are the first drug to be considered in the presence
of\emph{FIP1L1- PDGFRA} ~or~\emph{PDGFRA/PDGFRB} ~translocations. For
induction of remission and maintenance 100mg/d of Imatinib mesylate is
recommended. In the case of rare mutant \emph{FIP1L1-PDGFRA,
imatinib-resistant, other tyrosine kinase inhibitors or interferon-alpha
is sensitive for induction of remission.} In refractory cases,
allogeneic hematopoietic cell transplant needs to be
considered.\textsuperscript{8} The case in this study firstly was
treated with oral antiparasitic medications and steroids, later he
underwent anti-tubercular therapy.

In 2018 RN Das et al reported the first case in Nepal of idiopathic HES
presented with fatal eosinophilic cardiomyopathy and
dysphagia.\textsuperscript{11} They reported the case of a farmer
presenting with symptoms of persistent fever, cough, dyspnea, and
refractory heart failure for 7 months with peripheral eosinophilia. They
found no cause for eosinophilia despite a thorough
evaluation.\textsuperscript{12} In 1971, similarly Rickles et al
reviewed 16 cases of hypereosinophilic syndrome, most of which were
doubtful eosinophilic leukemia.\textsuperscript{13} In 2009, a
multicenter study conducted by Princess U Ogbogu in 161 patients with
eosinophilia found FIP1L1-PDGFRA test positive in 18 patients.
\textsuperscript{11}

The strength of this case study is that this has reported a rare case,
and this has been reported in a tertiary care hospital in Nepal. This
case report has been presented with several limitations. This case
report reported only one case; thus, several such case studies would
help to provide us with comprehensive information. Due to the
limitations in the equipment and other resources, timely diagnosis has
been one of the major challenges. Many investigations were sent to India
due to unavailability in Nepal. So, the studies highlighting the
challenges might contribute to disseminating the information and filling
up the gap.

\textbf{Conclusion}

The hypereosinophilic syndrome can be presented in several ways and this
study presented in a distinct way via hydropneumothorax. The
unavailability of sufficient equipment and the materials to test and
diagnose the hypereosinophilic led to a delay in the diagnosis. The case
underwent several investigations and therapy to get it diagnosed. Thus,
further studies and future researchers should emphasize the requirement
of early and prompt diagnostic studies to contribute to minimizing this
challenge.

\textbf{Conflict of Interest} All authors declare that they have no
conflicts of interest in the context of this work.

\textbf{Consent Statement:}

Written informed consent was obtained from the patient to publish this
report in accordance with the journal's patient consent policy.

\textbf{References:}

\begin{enumerate}
\tightlist
\item
  Shehwaro N, Langlois AL, Gueutin V, Izzedine H. Renal involvement in
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\item
  Sreedharanunni S, Varma N, Sachdeva MUS, Naseem S, Malhotra P, Bansal
  D, et al. The spectrum of hypereosinophilia and associated clonal
  disorders -- a real-world data based on combined retrospective and
  prospective analysis from a tropical setting. Mediterr J Hematol
  Infect Dis. 2018;10(1):e2018052. doi: 10.4084/MJHID.2018.052
\item
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  management. Am J Hematol. 2022;97(1):129-148. doi: 10.1002/ajh.26352
\end{enumerate}

\par\null

\begin{enumerate}
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\item
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\item
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\item
  Weller PF, Lim K, Wan HC, Dvorak AM, Wong DT, Cruikshank WW, et al.
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\item
  Valent P, Gleich GJ, Reiter A, Roufosse F, Weller PF, Hellmann A, et
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\item
  Rothenberg, M. E. Eosinophilia.~N Engl J Med. 1998;338(22):1592-1600.
\item
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\item
  Ogbogu PU, Bochner BS, Butterfield JH, Gleich GJ, Huss-Marp J, Kahn
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\end{enumerate}

Table 1: Findings of ANCA, FISH, and bone marrow aspiration\selectlanguage{english}
\begin{longtable}[]{@{}llll@{}}
\toprule
\textbf{Parameters} & \textbf{Findings} & \textbf{Findings} &
\textbf{Remarks}\tabularnewline
\textbf{Anti Neutrophil Cytoplasmic Antibodies (IFA)} & \textbf{Anti
Neutrophil Cytoplasmic Antibodies (IFA)} & \textbf{Anti Neutrophil
Cytoplasmic Antibodies (IFA)} & \textbf{Anti Neutrophil Cytoplasmic
Antibodies (IFA)}\tabularnewline
c-ANCA & c-ANCA & 1.37 U/ml & Negative\tabularnewline
p-ANCA & p-ANCA & - & Negative\tabularnewline
\textbf{Fluorescent In situ hybridization (FISH)- Eosinophilic Leukemia
Panel} & \textbf{Fluorescent In situ hybridization (FISH)- Eosinophilic
Leukemia Panel} & \textbf{Fluorescent In situ hybridization (FISH)-
Eosinophilic Leukemia Panel} & \textbf{Fluorescent In situ hybridization
(FISH)- Eosinophilic Leukemia Panel}\tabularnewline
PDGFRA rearrangement (4q12) & PDGFRA rearrangement (4q12) & 0 &
Negative\tabularnewline
PDGFRB rearrangement (5q33) & PDGFRB rearrangement (5q33) & 0 &
Negative\tabularnewline
FGFR1 rearrangement (8p11.22-11.23) & FGFR1 rearrangement
(8p11.22-11.23) & 0 & Negative\tabularnewline
CBFB rearrangement (Inv 16) & CBFB rearrangement (Inv 16) & 0 &
Negative\tabularnewline
\textbf{Bone marrow examination} & \textbf{Bone marrow examination} &
\textbf{Bone marrow examination} & \textbf{Bone marrow
examination}\tabularnewline
Cellularity & Cellularity & Hypercellular &\tabularnewline
Erythroid series & Erythroid series & Present with normoblastic
maturation &\tabularnewline
Myeloid series & Myeloid series & Increased with progressive maturation
up to neutrophils. Markedly increased eosinophils and precursors were
noted, which show dysplasia in the form of abnormal segmentation and
irregular distribution of granules. & Myeloid hyperplasia\tabularnewline
\bottomrule
\end{longtable}

We referred the patient to another hospital for consultation. However,
the hospital restarted the treatment with Praziquantel and Ivermectin.
This made us doubtful of the judgment to continue ATT.

Table 2: Report on laboratory findings of the indexed case\selectlanguage{english}
\begin{longtable}[]{@{}ll@{}}
\toprule
\textbf{Parameters} & \textbf{Value}\tabularnewline
Glucose & 0.8 mg/dL (Low)\tabularnewline
Albumin & 2.9 g/dL\tabularnewline
Protein & 6.2 g/dL\tabularnewline
Adenosine deaminase (ADA) & 34 U/L\tabularnewline
Total Leukocyte count (TLC) & 21.9x10\^{}3/uL\tabularnewline
Differential Leukocyte count (DLC) & (2 months later)\tabularnewline
Neutrophil & 22\%\tabularnewline
Lymphocyte & 18\tabularnewline
Monocyte & 3\tabularnewline
Eosinophils & 57\tabularnewline
Basophils & 0\tabularnewline
Hemoglobin & 13.5 g/dl\tabularnewline
Platelet count & 333x10\^{}3/uL\tabularnewline
\bottomrule
\end{longtable}\selectlanguage{english}
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\includegraphics[width=0.70\columnwidth]{figures/image1/image1}

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