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\begin{document}

\title{PARANEOPLASTIC JUVENILE IDIOPATHIC ARTHRITIS AS A MANIFESTATION OF A
BENIGN TERATOMA IN A FIVE-YEAR-OLD GIRL}



\author[1]{Shivani Kamal}%
\author[1]{Sukesh Sukumaran}%
\author[2]{Christopher LePhong}%
\author[1]{Karen Fernandez}%
\affil[1]{Valley Children's Healthcare}%
\affil[2]{Valley Children's Hospital}%


\vspace{-1em}



  
  \date{May 25, 2024}


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\textbf{PARANEOPLASTIC JUVENILE IDIOPATHIC ARTHRITIS AS A MANIFESTATION
OF A BENIGN TERATOMA IN A FIVE-YEAR-OLD GIRL}

\textbf{Authors:}

Shivani Kamal DO, Pediatric Residency Program, Valley Children's
Hospital affiliated with Stanford Medicine, Madera, California, USA

Sukesh Sukumaran MD, Pediatric Rheumatology, Valley Children's Hospital
affiliated with Stanford Medicine, Madera, California, USA

Christopher LePhong DO, Pediatric Pathology, Valley Children's Hospital
affiliated with Stanford Medicine, Madera, California, USA

Karen S. Fernandez MD, Pediatric Hematology/Oncology, Valley Children's
Hospital affiliated with Stanford Medicine, Madera, California, USA

\textbf{Correspondence}

Shivani Kamal DO, Pediatric Residency Program, Valley Children's
Hospital affiliated with Stanford Medicine, 9300 Valley Children's'
Place Madera, California, USA

Email: skamal1@valleychildrens.org

\textbf{Word Count:} 408 words

\textbf{Number of Figures:} 2 figures

\textbf{Short Running Title:} JUVENILE IDIOPATHIC ARTHRITIS AS A
MANIFESTATION OF A BENIGN TERATOMA IN A FIVE-YEAR-OLD GIRL

\textbf{Keywords:} teratoma; benign; arthritis; pediatric

\textbf{Abbreviations:}\selectlanguage{english}
\begin{longtable}[]{@{}ll@{}}
\toprule
Juvenile idiopathic arthritis & JIA\tabularnewline
\midrule
\endhead
Non-steroidal anti-inflammatory drug & NSAID\tabularnewline
Magnetic resonance imaging & MRI\tabularnewline
Computed tomography & CT\tabularnewline
\bottomrule
\end{longtable}

\textbf{Meeting Abstract:} Ovarian Teratoma masquerading as Idiopathic
Juvenile Arthritis~in a 5 year old girl.~Presented at the American
Society of Pediatric Hematology/Oncology (ASPHO) Annual Meeting.~ Fort
Worth, TX. May~ 10 - 13, 2023.~~ Poster\# 225. 2023 ASPHO Conference
Paper and Poster Index. Pediatric Blood Cancer, 70: e30390.
S101~https://doi.org/10.1002/pbc.30390

Juvenile idiopathic arthritis (JIA) is an auto-inflammatory disorder
that is the most common type of arthritis in children under the age of
16. Thirty percent of patients with JIA have seronegative polyarticular
disease. In adults, there are few reports of seronegative polyarthritis
associated with ovarian teratomas and sacrococcygeal teratomas; however,
to date, there are none reported in the pediatric
population.\textsuperscript{2,3,4}

A 4-year-old Caucasian female presented with 6 month-history of upper
and lower extremity joint pain and swelling associated with myalgias.
Family history of psoriatic arthritis, rheumatoid arthritis, and lupus
was reported. On examination, she had hypermobility in multiple joints
associated with swelling particularly in bilateral knees, right wrist,
and left elbow. Complete blood counts, comprehensive chemistry, lactate
dehydrogenase, and uric acid were normal. Initial erythrocyte
sedimentation rate was elevated at 15 mm/h. Antinuclear antibody and
human leukocyte antigen B27 markers were negative. The patient was
diagnosed with oligoarticular JIA and started on scheduled non-steroidal
anti-inflammatory drug (NSAID) therapy. Due to persistent bilateral knee
pain for 8 months despite NSAIDs, magnetic resonance imaging (MRI) of
bilateral knees was ordered to look for radiological inflammation or
erosive joint disease prior to starting disease modifying anti-rheumatic
drugs. As part of routine MRI screening, the mother reported that the
child may have swallowed a metallic object, prompting a chest X-ray,
which showed a lobular mass with unusual calcifications in the
epigastric area. Further investigation prompted computed tomography (CT)
and MRI of the abdomen, which showed an epigastric well-circumcised mass
(8.8 x 8.3 x 5.5 cm) with soft tissue, fat, and calcified components
including numerous tooth-like structures most consistent with teratoma.
The MRI of bilateral knees was negative for active inflammation. Tumor
markers alpha-fetoprotein and total beta-human chorionic gonadotropin
were negative. Tumor resection and histopathology confirmed benign
mature retroperitoneal teratoma without immature or malignant
components. After tumor removal, the patient's joint pain resolved.

The spontaneous resolution of an inflammatory seronegative arthritis
after the excision of the tumoral mass suggests a cause-and-effect
relationship between paraneoplastic manifestations and a benign
teratoma. It is unclear if the early manifestations of JIA resulted from
a genetic predisposition to autoimmune disorders as suggested by the
family history.

Teratomas are often found incidentally and most are asymptomatic. There
is no known association of JIA with benign teratoma; however, there are
abdominal tumors associated with other autoimmune conditions such as
juvenile dermatomyositis.\textsuperscript{1} This case highlights the
importance of thorough clinical evaluation in patients with seronegative
arthritis and consideration for teratoma.

\textbf{Figure 1} \textbf{Figure 2}\selectlanguage{english}
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\includegraphics[width=0.70\columnwidth]{figures/image1/image1}

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\textbf{Figure 1: Gross pathology of excised retroperitoneal teratoma,
consisting of soft tissue, bone, teeth, fat, hair, and other calcified
components}

\textbf{Figure 2: MRI abdomen showed teratomatous lesion extending for
hepatic vein confluence superiorly descending into the upper abdomen in
the porta hepatis region}

\textbf{References:}

\begin{enumerate}
\tightlist
\item
  Maria Ibarra, Pauline Chou, Lauren M. Pachman; Ovarian Teratoma
  Mimicking Features of Juvenile Dermatomyositis in a Child. Pediatrics
  November 2011; 128 (5): e1293--e1296. 10.1542/peds.2010-2115
\item
  Wiese W, Alansari H, Tranchida P, Madrid FF. Paraneoplastic
  polyarthritis in an ovarian teratoma. J Rheumatol. 2004
  Sep;31(9):1854-7. PMID: 15338513.
\item
  Smithers BM, Theile DE, Dickinson IC, Thomson DB, Axelsen RA.
  Malignant sacrococcygeal germ cell tumour in an adult. AustNZ J Surg
  1996;66:185-9.
\item
  Yu X, Qin D, Ma D, Yao Q. Adult dermatomyositis associated with benign
  ovarian teratoma: A case report. Oncol Lett. 2016 Apr;11(4):2611-2614.
  doi: 10.3892/ol.2016.4251. Epub 2016 Feb 19. PMID: 27073526; PMCID:
  PMC4812384.
\end{enumerate}

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