\documentclass[10pt]{article}

\usepackage{fullpage}
\usepackage{setspace}
\usepackage{parskip}
\usepackage{titlesec}
\usepackage[section]{placeins}
\usepackage{xcolor}
\usepackage{breakcites}
\usepackage{lineno}
\usepackage{hyphenat}





\PassOptionsToPackage{hyphens}{url}
\usepackage[colorlinks = true,
            linkcolor = blue,
            urlcolor  = blue,
            citecolor = blue,
            anchorcolor = blue]{hyperref}
\usepackage{etoolbox}
\makeatletter
\patchcmd\@combinedblfloats{\box\@outputbox}{\unvbox\@outputbox}{}{%
  \errmessage{\noexpand\@combinedblfloats could not be patched}%
}%
\makeatother


\usepackage{natbib}




\renewenvironment{abstract}
  {{\bfseries\noindent{\abstractname}\par\nobreak}\footnotesize}
  {\bigskip}

\titlespacing{\section}{0pt}{*3}{*1}
\titlespacing{\subsection}{0pt}{*2}{*0.5}
\titlespacing{\subsubsection}{0pt}{*1.5}{0pt}


\usepackage{authblk}


\usepackage{graphicx}
\usepackage[space]{grffile}
\usepackage{latexsym}
\usepackage{textcomp}
\usepackage{longtable}
\usepackage{tabulary}
\usepackage{booktabs,array,multirow}
\usepackage{amsfonts,amsmath,amssymb}
\providecommand\citet{\cite}
\providecommand\citep{\cite}
\providecommand\citealt{\cite}
% You can conditionalize code for latexml or normal latex using this.
\newif\iflatexml\latexmlfalse
\providecommand{\tightlist}{\setlength{\itemsep}{0pt}\setlength{\parskip}{0pt}}%

\AtBeginDocument{\DeclareGraphicsExtensions{.pdf,.PDF,.eps,.EPS,.png,.PNG,.tif,.TIF,.jpg,.JPG,.jpeg,.JPEG}}

\usepackage[utf8]{inputenc}
\usepackage[ngerman,english]{babel}



\usepackage{float}








\begin{document}

\title{Pulmonary Mucormycosis and Hydatid Cyst; a Case Report}



\author[1]{Samira Akbarieh}%
\author[1]{Behrooz  Naghili}%
\author[1]{Hamed  Valizade}%
\author[1]{Samad Beheshtirouy}%
\author[1]{Behnam Sajedi}%
\author[2]{Morteza Haramshahi}%
\affil[1]{Tabriz University of Medical Sciences Faculty of Medicine}%
\affil[2]{Tabriz University of Medical Sciences}%


\vspace{-1em}



  
  \date{June 03, 2024}


\begingroup
\let\center\flushleft
\let\endcenter\endflushleft
\maketitle
\endgroup





\selectlanguage{english}
\begin{abstract}
The patient was a 52-year-old woman with approximately 6 years' history
of uncontrolled diabetes mellites and hypothyroidism, who presented with
cough, sputum and dyspnea 2 months ago.%
\end{abstract}%



\sloppy


\textbf{Pulmonary Mucormycosis and Hydatid Cyst; a Case Report}

1. \textbf{Samira Akbarieh} : Tabriz University of Medical Sciences,
Tabriz, Iran

(Email: samiraakbarieh1365@gmail.com)

2. \textbf{Behrooz Naghili} : Tabriz University of Medical Sciences,
Tabriz, Iran

(Email: naghili\_b@yahoo.com)

3. \textbf{Hamed Valizade} : Tabriz University of Medical Sciences,
Tabriz, Iran

(Email: H\_VALLEY78@YAHOO.COM)

4. \textbf{Samad Beheshtirouy} : Tabriz University of Medical Sciences,
Tabriz, Iran

(Email: s\_beheshty@yahoo.com)

5. \textbf{Behnam Sajedi} : Tabriz University of Medical Sciences,
Tabriz, Iran

(Email: behnamsajedi1365@gmail.com)

6. \textbf{Morteza Haramshahi} : Tabriz University of Medical Sciences,
Tabriz, Iran

(Email: mortezaharamshahi53@gmail.com)

\textbf{Corresponding Author}

\textbf{Samira Akbarieh} : Subspecialty Fellowship in Nosocomial
Infections

\textbf{Tabriz University of Medical Sciences, Tabriz, Iran}

Address: Central Building, Tabriz University of Medical Sciences,
Tabriz, Iran.

Phone: +984133357310

Email: samiraakbarieh1365@gmail.com

\section*{Abstract}

{\label{abstract}}

\textbf{Background} : Mucormycosis is a group of life-threatening
diseases caused by a fungus of Mucoraceae family and has a higher
mortality rate compared to other known fungal infections. Hydatid cyst,
caused by Echinococcus, is crucial a health concern in endemic areas and
the disease is characterized by slow-growing cysts in liver, lungs, or
other organs. In this report, a woman with coexistence of hydatid cyst
and Mucormycosis is introduced.

\textbf{Case Report} : The patient was a 52-year-old woman with
approximately 6 years' history of uncontrolled diabetes mellites and
hypothyroidism, who presented with cough, sputum and dyspnea 2 months
ago. On the initial auscultation of the lungs, there was a decreased
sound at the base of the left lung and she had fever. In blood tests,
she had a high titer of erythrocyte sedimentation rate (ESR) and 3+
C-Reactive Protein (CRP). The symptoms in favor of hydatid cyst were
observed in lung computed tomography and in pleural needle biopsy,
hydatid cyst was confirmed. With this indication, she undergone wedge
resection, resection of the left lower lung cyst. Two samples are taken
from the cyst side and the pathology report was consistent with
Mucormycosis (wide filaments with a 90-degree angle). The patient was
immediately treated with liposomal amphotericin for a 4-week period.
Lung CT scan was performed before and after treatment. Albendazole was
treated in order to treat hydatid cyst. After discharge, the treatment
of the patient continued with oral Posaconazole, and after the treatment
finalization, the general condition of the patient was good and she did
not have any complaints.

\textbf{Conclusion:} In pulmonary diseases that do not respond
significantly to surgical treatment (such as hydatid cyst), fungal
disease (mucor) must be considered simultaneously. Mucormycosis has more
prevalence in patients with uncontrolled diabetes and it is necessary to
be considered if these patients were infected to pneumonia and their
symptoms did not improve with usual treatments.

\textbf{key words:} Mucormycosis, Cyst Hydatid, Lung Infection, Case
Report.

\textbf{Introduction}

Mucormycosis is an uncommon and rapidly progressive invasive fungal
infection that can in case of lack of timely diagnosis and treatment,
lead to mortality. It is usually caused by members of the Mucoraceae
family of fungi, which its subgroups include Rhizopus, Mucor, and
Absidis. Mucormycosis is transmitted through the air (1). This fungus is
not normally pathogenic and only occurs when the patient has underlying
diseases such as immunodeficiency and diabetic ketoacidosis (2). Other
conditions of immunodeficiency and the use of drugs that weaken the
immune system are the other predisposing factors.

Pulmonary Mucormycosis is the second most common manifestation of
mucositis, after the Rhinocerebral form. The symptoms of this infection
include dyspnea, cough, chest pain, and hemoptysis. Vascular invasion
causes necrosis, cavitation, or hemoptysis. Lobar opacity, solitary
mass, nodular disease, cavity, or wedge-shaped infarcts may be seen on
chest X-rays (3, 4). The diagnosis of Mucormycosis is a medical
challenge and autopsy studies have shown that up to half of cases were
diagnosed only after death (5).

Hydatid cyst, caused by Echinococcus, is a cestode parasite of the
tapeworm variant. Humans are mostly affected by two species: granulosus
and multilocularis which cause two major types of the disease; cystic
echinococcosis and alveolar echinococcosis (6). Hydatid cyst is a
crucial health problem in endemic areas and the disease is characterized
by slow-growing cysts.

The lungs are the second most prevalent site of this infection after the
liver. Manifestations of pulmonary hydatid cyst vary depending on
whether the cyst is intact or ripped and their clinical manifestations
mostly depend on size and location. Unripped cysts have no specific
symptoms. Large cysts may cause signs of dry cough, pressure on adjacent
organs, and upper superior vena cava syndrome. Also, atelectasis may be
seen due to the compressive effect on the bronchi. Diagnosis of a cystic
mass in a person who has a history of contact with dogs and lives in
endemic areas strongly suggests a hydatid cyst (7, 8). The treatment
method for hydatid cyst of the lung is surgery and drug treatments are
used in cases where surgery is not possible for any reason (9).

Coexistence of pulmonary Mucormycosis and hydatid cyst is a rare
phenomenon and best of our knowledge this is the first-ever reported
case of coexistence of these two pathogens. Here we report the symptoms,
diagnosis, treatment and follow-up details of our presented case of
coexistence of pulmonary Mucormycosis and hydatid cyst in Imam Reza
Medical Research and Training hospital of Tabriz University of Medical
Science.

\textbf{Case presentation}

A 52-years-old woman with a history of uncontrolled diabetes, HbA1C
level of 12\%, and hypothyroidism for about 6 years, was referred to the
hospital due to productive cough, hemoptysis, fever, and chills started
two months before, and it was accompanied by shortness of breath. The
drug history was metformin and oral Glibenclamide. At the time of
admission, she had a fever of 38\selectlanguage{ngerman}° centigrade, a 95\% oxygen saturation
level, a respiratory rate of 23, and a heart rate of 90. In the physical
examination, she was pale; the cardiac auscultation was normal and, in
the skin examination the scars of the abdomen were evident. On the
initial auscultation of the lungs, there was a decreased sound at the
base of the left lung and she had fever. In blood tests, she had a high
titer of erythrocyte sedimentation rate (ESR) and 3+ C-Reactive Protein
(CRP).

Due to pulmonary symptoms, she was repeatedly treated with antibiotics
during this period which didn't have a clinical response. In our
hospital, after observing the symptoms in favor of hydatid cyst in lung
computed tomography (CT-scan) (Figure 1. A), the patient underwent a
biopsy, and after reporting a positive serology for hydatid cyst,
medical treatment with two albendazole tablets, once daily, was
initiated. The patient's symptoms continued until she underwent a
thoracotomy and resection of the left lower lung cyst. During the
surgery a sample was taken from another cavity located next to the
resected cyst. (Figure 1. B)

In pathology report in left chest wall lesion resection ''\emph{consist
with hydatid cyst accompanied by foreign body reaction} '' and in the
left lung, lower lesion resection, ''\emph{lung tissue with hemorrhage,
congestion, infiltration cells with neutrophil predominance and groups
of large, nonseptic hyphae with variable width, 90-degree angle
branching and non-parallel walls consistent with Mucormycosis} '' were
noted (Figure 2).

After surgery and confirmation of coexistence of lung hydatid cyst and
Mucormycosis, the patient was treated with liposomal amphotericin B
5mg/kg=300 mg daily for 4 weeks and hydatid cyst medical therapy also
continued with albendazole 80 mg daily. After 4 weeks of treatment with
amphotericin B, lung CT-scan reported an improvement in the condition
(Figure 1. C). During the four weeks treatment period, the patient's
symptoms were stable and she did not have fever and chills and did not
report any complaints about the previous symptoms.

The oral treatment continued and the patients was discharged for 2 weeks
with good general condition. For maintenance therapy, the patient
received itraconazole 200mg tablets, BID to complete 6 weeks of
treatment. At the end of 6 months of follow-up, she had no pulmonary
complaints and clear radiological evidence was noted in lung CT-scan.
(Figure 1. D) and after two weeks, she was revisited without any
particular complaints.

\textbf{Discussion}

Here we reported a poorly controlled patient with diabetes mellitus and
coexistence of lung Mucormycosis and hydatid cyst, which she was treated
successfully with medical and surgical treatments. Based on imaging
evidence and clinical findings, finally, the appropriate therapeutic
response to treatment was observed. Best of our knowledge this is the
first reported case of coexistence of pulmonary Mucormycosis and hydatid
cyst.

In our reported case, the patient was taken to the operating room with
diagnosis of hydatid cyst, which is an indication for thoracotomy, and
the pathology finding was consistent with Mucormycosis. The most
accurate diagnostic method for fungi infections is to perform a biopsy
and observe vascular invasion and subsequent tissue thrombosis with
evidence of acute and chronic inflammation in the sample. In our case
the surgical biopsy was sent for pathologic assessments and 90-degree
angle branching and non-parallel walls consistent with Mucormycosis was
reported.

In patients with symptoms of pneumonia who do not respond to the
treatment, Mucormycosis should be considered from a diagnostic
perspective. The presence of this infection and the lack of a standard
protocol for treatment is experimental for now (10). In general, dealing
with patients with Mucormycosis includes natural treatment and
elimination of the underlying factor. Because of underlying conditions,
in the present case, amphotericin b consumption was started.

The prognosis of Mucormycosis depends on several factors, including
rapid diagnosis recovery, and the underlying factor. Studies in this
regard have also yielded favorable results from the administration of
hyperbaric oxygen and liposomal nystatin. Liposomal amphotericin is the
most effective drug known for the medical treatment of Mucormycosis. The
duration and dose of the drug depend on various factors, including the
side effects, patient tolerance, and the progress of the disease (11).
The Survival rate is higher using the combination of medical and
surgical treatment (12). Our patient was treated with liposomal
amphotericin B and itraconazole 200mg as a maintenance therapy.
According to the patient's improvement in the treatment process and also
other studies in this field, it turns out that it was an appropriate
treatment (13).

\textbf{Conclusion}

Mucormycosis and hydatid cyst can be presented together. In diabetic
patients or other immunosuppressive conditions with symptoms of
pneumonia who do not respond appropriately to the usual treatments,
Mucormycosis should be considered. In Mucormycosis patients with
underlying conditions such as diabetes mellitus, quick diagnosis and
initiation of the treatment improve the survival rate of the patients.



\textbf{Ethical Approval and Consent to participate}

Patient was informed from this case report and written consent form was
obtained.

\textbf{Consent for publication}

Written informed consent was obtained from the patient for publication
of this case report and any accompanying images. A copy of the written
consent is available for review by the Editor-in-Chief of this journal.

\textbf{Availability of supporting data}

Not applicable.

\textbf{Competing interests}

None

\textbf{Funding}

None

\textbf{Acknowledgements}

None

\textbf{Authors' contributions}

SA, BN, HV, SB, BS and MH were at the treatment team of the patients. BS
and SA drafted the manuscript and BN, HV, SB and MH critically revised
it. SA, HV and BS prepared the figures. All authors read and approved
the final manuscript.



\textbf{References}

1. Lee FY, Mossad SB, Adal KA. Pulmonary mucormycosis: the last 30
years. Archives of Internal Medicine. 1999;159(12):1301-9.

2. Yamin HS, Alastal AY, Bakri I. Pulmonary mucormycosis over 130 years:
a case report and literature review. Turkish thoracic journal.
2017;18(1):1.

3. Dave SP, Vivero RJ, Roy S. Facial cutaneous mucormycosis in a
full-term infant. Archives of Otolaryngology--Head \& Neck Surgery.
2008;134(2):206-9.

4. Javadi M, Sh M, Daneshi A. Rhinorbital cerebral mucormycosis: case
Report. RJMS. 2001;26(8):397-404.

5. Spellberg B, Edwards Jr J, Ibrahim A. Novel perspectives on
mucormycosis: pathophysiology, presentation, and management. Clinical
microbiology reviews. 2005;18(3):556-69.

6. Wen H, Yang W-G. Public health importance of cystic echinococcosis in
China. Acta Tropica. 1997;67(1-2):133-45.

7. Usluer O, Kaya SO, Samancilar O, Ceylan KC, Gursoy S. The effect of
preoperative albendazole treatment on the cuticular membranes of
pulmonary hydatid cysts: should it be administered preoperatively?
Kardiochirurgia i torakochirurgia polska= Polish journal of
cardio-thoracic surgery. 2014;11(1):26.

8. Katan Y. Intrabiliary rupture of hydatid cysts of liver. Ann R Coll
Surg Engl. 1977;59:108-14.

9. Morar R, Feldman C. Pulmonary echinococcosis. European Respiratory
Journal. 2003;21(6):1069-77.

10. Hadzri M, Azarisman S, Fauzi A, Kahairi A. Invasive rhinocerebral
mucormycosis with orbital extension in poorly-controlled diabetes
mellitus. Singapore medical journal. 2009;50(3):107-9.

11. Mileshkin L, Slavdst M, Seymour J, McKenzie A. Successful treatment
of rhinocerebral zygomycosis using liposomal nystatin. Leukemia \&
lymphoma. 2001;42(5):1119-23.

12. Kwan LP, Choy CB, Chan TM, Suen WS, Yap DY. Successful treatment of
pulmonary rhizopus infection with surgical resection and posaconazole in
a renal transplant recipient. Nephrology. 2013;18(1):74-5.

13. Patterson TF. Advances and challenges in management of invasive
mycoses. The Lancet. 2005;366(9490):1013-25.\selectlanguage{english}
\begin{figure}[H]
\begin{center}
\includegraphics[width=0.70\columnwidth]{figures/image1/image1}

\end{center}
\end{figure}

Figure 1 -- CT scans (A: before surgery; B: After surgery; C: After
surgery and amphotericin B therapy; D: 6 months after the treatment and
follow up).\selectlanguage{english}
\begin{figure}[H]
\begin{center}
\includegraphics[width=0.70\columnwidth]{figures/image2/image2}

\end{center}
\end{figure}

Figure 2 -- pathology slides of the patient.

\selectlanguage{english}
\FloatBarrier
\end{document}

