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\begin{document}

\title{A tale about an unusual cause of pygalgia in ankylosing
spondyloarthritis}



\author[1]{Rim Dhahri}%
\author[1]{Hiba Bettaieb}%
\author[1]{Maroua SLOUMA}%
\author[1]{Yasmine Khrifech}%
\author[1]{youssef Mallat}%
\author[1]{Khalil Amri}%
\author[1]{Ahmed Harbaoui}%
\author[1]{Leila Metoui}%
\author[1]{Imen Gharsallah}%
\author[1]{Bassem Louzir}%
\affil[1]{University of Tunis El Manar Faculty of Medicine of Tunis}%


\vspace{-1em}



  
  \date{May 24, 2024}


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\selectlanguage{english}
\begin{abstract}
Neurinomas are tumors of Schwann cells of the peripheral nerve sheath.
Sacral location is rarely reported especially in spondyloarthritis
patients. Herein, we report a case of uncommon pygalgia in a 25-year-old
man with history of a non-radiographic axial spondyloarthritis and in
whom the diagnosis of sacral neurinoma was established.%
\end{abstract}%



\sloppy


\textbf{TITLE: A tale about an unusual cause of pygalgia in Ankylosing
Spondyloarthritis}

\textbf{Authors:}

Rim Dhahri 1, Hiba Bettaieb 1, Maroua Slouma1, Yasmine Khrifech 2,
Youssef Mallat 3, Khalil Amri 3, Ahmed Harbaoui 4, Leila Metoui1, Imen
Gharsallah 1, Bassem Louzir2

\textbf{Authors affiliation:}

\begin{enumerate}
\tightlist
\item
  Rheumatology department, Military Hospital of Tunis, Tunisia
\item
  Internal Medicine department, Military Hospital of Tunis, Tunisia
\item
  Orthopedics' department, Military Hospital of Tunis, Tuni
\item
  Neurosurgery Department, Military Hospital of Tunis, Tunisia
\end{enumerate}

\emph{Corresponding author}\textbf{~: Hiba Bettaieb}

\begin{itemize}
\tightlist
\item
  \textbf{Dr. Rim Dhahri:} Associate Professor , Rheumatology
  department, Military Hospital of Tunis,
  Tunisia\textbf{Email:}rimdhahri@ymail.com
\item
  \textbf{Dr Hiba Bettaieb} : Rheumatologist, , Rheumatology department,
  Military Hospital of Tunis, Tunisia\selectlanguage{ngerman}\textbf{The corresponding
  author+216 56~179~617Adress: 08 Street de la sirène. 2070. Tunis.
  TunisiaEmail} : hibahera@gmail.com
\item
  \textbf{Dr Maroua Slouma :} University Hospital Assistant,
  Rheumatology department, Military Hospital of Tunis,
  Tunisia\textbf{Email:}maroua.slouma@gmail.com
\item
  \textbf{Dr Yasmine Khrifech} ~: Intern, internal medicine department,
  Military Hospital of Tunis, Tunisia\textbf{Email} :
  yessminkhrifech@gmail.com
\item
  \textbf{Dr Youssef Mallat} : University Hospital Assistant,
  Orthopedics' department, Military Hospital of Tunis,
  Tunisia\textbf{Email} : dr.youssef.mallat@gmail.com
\item
  \textbf{Dr Khalil Amri} ~: University Hospital Assistant, Orthopedics'
  department, Military Hospital of Tunis, Tunisia\textbf{Email} :
  akhalil.kh@gmail.com
\item
  \textbf{Dr Ahmed Harbaoui} ~: Associate Professor, Neurosurgery
  department, Military Hospital of Tunis,
  Tunisia\textbf{Email:}ahmed.harbaoui@gmail.com
\item
  \textbf{Prof Leila Metoui~} : Associate Professor, Rheumatology
  department, Military Hospital of Tunis,
  Tunisia\textbf{Email:}leila.metoui@gnet.tn
\item
  \textbf{Prof Imene Gharsallah} ~: Professor, head of Rheumatology
  department , Military Hospital of Tunis, Tunisia\textbf{Email} :
  imengharsallah@hotmail.fr
\item
  \textbf{Prof Bassem Louzir~:} Professor, head of internal medicine
  department , Military Hospital of Tunis,
  Tunisia\textbf{Email:}louzir.bassem@yahoo.fr\textbf{Declarations
  section:}
\end{itemize}

\begin{itemize}
\tightlist
\item
  \textbf{Consent for publication:} Patient was informed and fully
  consented to publication.
\item
  \textbf{Availability of data and material:} The datasets used and/or
  analyzed during the current study are available from the corresponding
  author on reasonable request.
\item
  \textbf{Competing interests~: None to declare}
\item
  \textbf{Funding~: None}
\item
  \textbf{Pharmaceutical / industry support: NoneABSTRACT:} Neurinomas
  are tumors of Schwann cells of the peripheral nerve sheath. Sacral
  location is rarely reported especially in spondyloarthritis patients.
  Herein, we report a case of uncommon pygalgia in a 25-year-old man
  with history of a non-radiographic axial spondyloarthritis and in whom
  the diagnosis of sacral neurinoma was established.\textbf{KEY WORDS:}
  Neurinoma; Schwannoma; spondyloarthritis; pygalgia\textbf{KEY CLINICAL
  MESSAGE} :
\end{itemize}

\begin{itemize}
\tightlist
\item
  Neurinoma of peripheral nerve roots is a rare entity.
\item
  Neurinoma and spondyloarthritis is an unusual association
\item
  Differential diagnosis of sacroiliac joint pain is made with S1 nerve
  roots tumors such as Neurinoma.\textbf{PRESENTATION:}A 25-year-old man
  with history of a non-radiographic axial spondyloarthritis admitted
  for persistent right buttock pain. The sacroiliac radiographs showed a
  slight enlargement of the first right sacral foramen (Figure A). An
  MRI of the lumbosacral spine and the sacroiliacs was done in the
  background to objectify a sacroiliitis in flare. However, the MRI had
  shown a soft tissue mass lateral to the left S1/S2 foramina. This was
  consistent with a neurinoma measuring 18mm * 22 mm. The patient was
  operated on with total resection of the neuroma. Histological findings
  have confirmed the diagnosis (Figure F). The evolution was good with
  disappearance of pygalgias.\textbf{DISCUSSION:}The incidence of spinal
  schwannoma, according to recent studies, varies between 3 and 4 cases
  / 1,000 000 people per year {[}1{]}. MRI is the gold standard for the
  diagnosis of spinal schwannoma, it allows structural and spatial
  analysis of the lesion {[}2{]}. The average age of the appearance of
  neurinomas is between 30 and 50 years old. In our observation, the age
  was younger than usual. The most clinical signs are the pain of both
  the spine and root system reflecting the lesion syndrome. Surgery is
  the gold standard of treatment for spinal schwannoma
  {[}3{]}.\textbf{REFERENCES:}{[}1{]} Emel E, Abdallah A, Sofuoglu OE,
  Ofluoglu AE, Gunes M, Guler B, Bilgic B. Long-term Surgical Outcomes
  of Spinal Schwannomas: Retrospective Analysis of 49 Consecutive Cases.
  Turk Neurosurg. 2017;27(2):217-225. doi:
  10.5137/1019-5149.JTN.15678-15.1. {[}2{]} Amezyane T, Pouit B, Bassou
  D, Lecoules S, Desramé J, Blade JS, Béchade D, Algayres JP. Une cause
  rare de lombosciatique {[}A rare cause of sciatica{]}. Rev Med
  Interne. 2006 ;27(6):494-6. doi: 10.1016/j.revmed.2005.10.001. {[}3{]}
  Chang, Ung-Kyu, et al. Radiosurgery Using the Cyberknife for Benign
  Spinal Tumors: Korea Cancer Center Hospital Experience. Journal of
  Neuro-oncology, vol. 101, no. 1, 2011, pp. 91-9.\textbf{FIGURE'S
  LEGEND:}Figure A: Enlargement of the first right sacral foramen on a
  pelvic x ray. Figure B, C: Frontal T1 and T2 sacroiliac sequences
  showing targeted appearance of the mass consistent with neurinoma'
  diagnosis. Figure D, E: Sagittal lumbar spine T1, T2 and STIR weighted
  sequences showing high signal soft tissue mass on the right S1 nerve
  root on T2 and STIR weighted sequences and low signal on T1 weighted
  sequences. Figure F: Spindle cell tumor proliferation showing
  characteristic nuclear palisades.
\end{itemize}\selectlanguage{english}
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