\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{Reinforced conservative management of post-dural puncture headache in a
patient with a rare case of tethered cord syndrome using an abdominal
binder: a case report}



\author[1]{Seong-Ho Ok}%
\author[1]{Miyeong Park}%
\author[1]{Hokyung Yu}%
\author[1]{Jiyoung Park}%
\author[2]{Ju-Tae Sohn}%
\author[3]{Sunmin Kim}%
\author[3]{Kyeong Eon Park}%
\author[3]{Yeran Hwang}%
\affil[1]{Gyeongsang National University Changwon Hospital}%
\affil[2]{Gyeongsang National University College of Medicine}%
\affil[3]{Gyeongsang National University Hospital}%


\vspace{-1em}



  
  \date{June 14, 2024}


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





\selectlanguage{english}
\begin{abstract}
Careful physical examination of the site of procedure before the caudal
pain procedure should be performed because it could show the patient's
abnormal anatomical conditions. An abdominal binder could be used
effectively in a patient showing CSF leakage in the coccygeal area,
which is not controlled by conventional compressive dressing.%
\end{abstract}%



\sloppy


\textbf{Introduction}

In a study conducted in Korea, it was found that only 0.8\% of 2669
participants had an anatomical variation wherein the dural sac extended
below S3, while 1.6\% had pathological conditions like perineural cysts
and meningoceles.\textsuperscript{{[}1{]}} The incidence of post-dural
puncture headache (PDPH) after caudal epidural block due to these
anatomical abnormalities was low and its occurrence difficult to
predict.\textsuperscript{{[}2{]}} Epidural blood patch (EBP), considered
a gold standard treatment in PDPH, can lead to improvement of symptoms
in more than 90\% of cases.\textsuperscript{{[}3{]}} However, EBP cannot
be performed on PDPH patients who do not respond to conservative
management due to possible complications.

We report our experience in diagnosing rare tethered cord syndrome
(TCS), which could cause PDPH, secondary to CSF leakage through an MRI.
The MRI was performed to investigate the reason for subcutaneous
cerebrospinal fluid (CSF) collection suspected by physical examinations
and bedside sonography. PDPH accompanied by TCS was successfully
resolved after using an abdominal binder, as reinforced conservative
management, for a week.

\textbf{Case report}

A 59-year-old female visited our hospital due to posterior head and neck
pain that developed after a coccygeal procedure. She was referred to the
pain clinic for EBP.

She underwent the caudal approach procedure a week ago at a local clinic
due to pain in the left leg and foot. Reports about the procedure
performed from the clinic were inaccessible, but following the patient's
description, a caudal epidural block was presumed to have been used. One
day after the procedure, she found clear fluid leakage from the
coccygeal area and started experiencing posterior headache. She received
a disinfection and compressive dressing on the coccygeal area and was
prescribed bed rest and pain killers from the local clinic, but her
symptoms did not improve within a week.

The numerical rating scale (ranging from 0 to 10, with a score of 0
representing no pain and 10 corresponding to intractable pain) score for
pain severity was 5 and the pain was frequent, throbbing, and worsened
by the orthostatic position.

On physical examination, the compressive dressing looked clean except
for some swelling around the sacral area, and no leakage was observed.
Bedside ultrasound (US) showed subcutaneous edema and fluid collection
in the swollen peri-coccygeal area (Fig1). There were no suspicious
signs of meningeal irritation such as fever or neck stiffness.
Laboratory examination revealed no leukocytosis and normal C-reactive
protein levels.

We conducted the following treatments under suspicion of PDPH with
subarachnoid-cutaneous fistula after the caudal procedure.

First, she was admitted for absolute bed rest, hydration with
intravenous fluid, and was prescribed acetaminophen as a painkiller. A
neurologist referred her to a pain clinic for EBP because her headache
lasted for more than a week despite conventional conservative management
from a local clinic. We initially did not consider EBP through the
sacral hiatus as a treatment plan, because there was a possibility of
infection in the presence of a fistula or repeated CSF leakage. MRI was
scheduled for checking the presence of subcutaneous CSF leakage and its
cause. Additionally, we decided to disinfect the wound and apply
compressive dressing using an obliquely applied abdominal binder around
the patient's buttocks and the lower abdomen for better compact
compression than that with the previous conventional compressive
dressing, which comprised of adhesive tape and gauze.

After admission, a radiologist reported MRI, showed a low-lying conus
medullaris (which reached down to the inferior border of the S2
vertebra, Fig 2) with a thickened filum terminale, an 8 \selectlanguage{ngerman}× 7
cm\textsuperscript{2} sized fatty contiguous mass with a placode at the
sacrococcygeal area, and interspersed fluid around the mass. (Fig 3A and
B) This showed that she had a rare TCS that was now discovered late in
adulthood. A surgical procedure should have been considered for TCS, but
since she did not have any symptoms of the cauda equina syndrome or any
other severe neurologic symptoms, except the initial symptoms of left
leg and foot pain, we decided to use reinforced conservative management
with an abdominal binder first for a week.

On the 3rd day of hospitalization, the headache and neck pain decreased,
but reappeared after a short walk. On the 4th day, she could walk
without headache and neck pain, but she could still sometimes experience
mild posterior neck pain. On the 5th day, US still showed irregular and
mild hypoechoic subcutaneous edema around the coccyx area and
suspicious~fluid collection, but its size had decreased. On the 6th day,
she was discharged with an abdominal binder.

One week after discharge, she visited the pain clinic and had no
complaints even without the abdominal binder.

\textbf{Discussion}

TCS presents various symptoms depending on age and its cause. Neurologic
symptoms, sensory or motor dysfunction, dysuria, and deformation of the
feet or spine are usually developed by progressive stretching of and
increased tension in the spinal cord in
children.\textsuperscript{{[}4{]}} Cutaneous stigmata, hair tufts, skin
tags, dimple, nevi, hemangioma, lumps, lipomas, etc. could be surface
(cutaneous) findings that suspect TCS.\textsuperscript{{[}4{]}} Our
patient had subcutaneous fluid collection following the caudal approach
and it looked like it was concealing the existence of a subcutaneous
lipoma. If a physician at a local clinic had performed a physical
examination of the procedure site before the practice, he would have
found a lesion suspected as a lump or lipoma. We suspected that she had
a CSF leakage based on physical examination and bedside US and finally
confirmed a rare case of TCS and subarachnoid-cutaneous fistula by MRI.
Based on our experience, it is difficult to predict dural puncture
before caudal epidural block without expensive imaging studies such as
CT or MRI.

In children, an inelastic filum is attached to the caudal end of the
spinal cord which adds continuous tension to the spinal cord causing
severe neurological symptoms. Surgical treatment of TCS should maintain
neural functions and circulation of CSF, and reconstruct the dural sac;
additionally, there should be no adverse effects, such as infection or
adhesion.\textsuperscript{{[}5{]}} Major symptoms of TCS, such as pain,
bowel or urinary dysfunction, may not improve after surgery in adults,
and postoperative complications of old age may be three times higher
than that in young patients. Asymptomatic adult TCS patients could be
followed up only for symptoms, and conservative management could be
applied to non-aggressive and bearable
symptoms.\textsuperscript{{[}6-8{]}} There is a controversy over the
superiority of conservative management and surgical management. This
case showed that conservative management could improve symptoms in adult
TCS patients, therefore, we should be careful in our decision about
employing surgical treatment.\textsuperscript{{[}6-8{]}} In our patient,
surgical release of the filum terminale anchored to the spinal cord
could be considered, but we decided to maintain the conservative
management with an abdominal binder and observation of the symptoms
because her mild neurologic symptoms (pain of the left leg and foot)
improved and PDPH decreased.

In the structures of the epidural cavity, the incomplete lateral wall
could cause peritoneal pressure to be transmitted to the epidural cavity
through the intervertebral foramina. Therefore, increased abdominal
pressure could induce an increase in the epidural
pressure.\textsuperscript{{[}9{]}} In this respect, the compression of
the abdomen with an abdominal binder would be helpful to decrease the
PDPH after spinal tapping in postpartum patients by increasing the
peritoneal pressure. The preventive effects of an abdominal binder in
PDPH's occurrence were similar to that of the injection of normal saline
into the epidural space.\textsuperscript{{[}10{]}} We applied an
abdominal binder obliquely for two main purposes. First, we tried to
maintain high epidural pressure by increasing peritoneal pressure to
reduce pressure variations due to change in posture. Second, compressive
dressing with gauze and adhesive tape was insufficient, especially in
the coccyx area because the movable and soft buttocks would offset the
adhesive tape's compression. Considering these points, we applied an
abdominal binder to surround the lower abdomen, buttocks, and coccyx,
obliquely from the upper anterior pelvis to the lower posterior pelvis.
This placement, fastening the binder around the pelvis, prohibited
lateral movement of the buttocks and provided evenly distributed
compression of the lipoma and large subcutaneous CSF collection. The
abdominal binder was able to compress a surface area of 8 × 7
cm\textsuperscript{2}.

The EBP was a very effective treatment for spontaneous intracranial
hypotension; therefore, 90\% of the patients showed improvement
regardless of the presence or location of CSF
leakage.\textsuperscript{{[}11{]}} However, complications and/or side
effects---ranging from mild to severe---make EBP a challenging procedure
for anesthesiologists. Mild events include temporary nerve compression
syndrome, dizziness, vertigo, or tinnitus, while the rare, severe
adverse effects include epidural abscess or cord
compression.\textsuperscript{{[}12{]}} Our patient had a history of body
fluid leakage, which could be expected as the puncture site had swelling
and fluctuation on physical examination of the coccygeal area, which
appeared as a subcutaneous fluid collection. Because we confirmed that
this fluid collection communicated with the intrathecal space via MRI
(Fig 3A and B), we thought that the EBP through the sacral hiatus had a
high risk of meningitis. It was reported that the large volume of blood
infused into the epidural space ran in the cephalad direction during EBP
in MRI,\textsuperscript{{[}13{]}} and thus, the lumbar site for EBP in
our patients seemed ineffective and large volumes of blood would be
needed in contrast to that in the sacral approach if it was performed.

Based on the findings of this case, the following points should be kept
in mind during the caudal procedure.

\begin{enumerate}
\tightlist
\item
  Careful physical examination of the procedure site before the caudal
  pain procedure should be performed because it could show the patient's
  abnormal anatomical conditions.
\item
  When a subcutaneous fluid collection after a neuraxial procedure is
  suspected by peripheral swelling or fluctuation, fluid collection can
  be examined by easily accessible US. If it has been proven, CSF
  leakage must be ruled out by an MRI scan, and then EBP should be
  considered later.
\item
  An abdominal binder could be used effectively in a patient showing CSF
  leakage in the coccygeal area, which is not controlled by conventional
  compressive dressing.
\end{enumerate}

\textbf{Acknowledgments:} We would like to thank Editage
(www.editage.co.kr) for English language editing.

\textbf{Disclosures:} The authors have read the CARE Checklist (2013),
and the manuscript was prepared and revised according to the CARE
Checklist (2013).

\textbf{Ethical concerns:} The study was approved by the institutional
review board of Gyeongsang National University Changwon Hospital (GNUCH
2020-08-018)

\textbf{Conflict-of-interest:} The authors declare that they have no
conflict of interest.

\textbf{Author contributions} :

Miyeong Park : drafting the manuscript and primary treatment provider

Kyeong Eon Park, Yeran Hwang : drafting the manuscript

Hokyung Yu, Jiyoung Park, Sunmin Kim : co\selectlanguage{english}-authors responsible for the
literature review

Ju-Tae Sohn, Seong-Ho Ok : the senior authors managing the construction
and edits of the manuscript and guiding through the submission process.



\textbf{References:}

1. Joo J, Kim J, Lee J. The prevalence of anatomical variations that can
cause inadvertent dural puncture when performing caudal block in
Koreans: a study using magnetic resonance imaging.
\emph{Anaesthesia.}2010;65(1):23-26.

2. Kim SG, Yang JY, Kim DW, Lee YJ. Inadvertent Dural Puncture during
Caudal Approach by the Introducer Needle for Epidural Adhesiolysis
Caused by Anatomical Variation. \emph{The Korean journal of
pain.}2013;26(2):203-206.

3. So Y, Park JM, Lee PM, Kim CL, Lee C, Kim JH. Epidural Blood Patch
for the Treatment of Spontaneous and Iatrogenic Orthostatic
Headache.\emph{Pain Physician.} 2016;19(8):E1115-e1122.

4. Hertzler DA, DePowell JJ, Stevenson CB, Mangano FT. Tethered cord
syndrome: a review of the literature from embryology to adult
presentation. \emph{Neurosurgical focus.} 2010;29(1):E1.

5. Kang JK, Yoon KJ, Ha SS, Lee IW, Jeun SS, Kang SG. Surgical
management and outcome of tethered cord syndrome in school-aged
children, adolescents, and young adults. \emph{J Korean Neurosurg
Soc.}2009;46(5):468-471.

6. Duz B, Gocmen S, Secer HI, Basal S, Gonul E. Tethered cord syndrome
in adulthood. \emph{J Spinal Cord Med.} 2008;31(3):272-278.

7. Tu A, Steinbok P. Occult tethered cord syndrome: a
review.\emph{Child's Nervous System.} 2013;29(9):1635-1640.

8. Drake JM. Occult tethered cord syndrome: not an indication for
surgery. 2006;104(5):305-308.

9. Hogan QH. Epidural anatomy: new observations. \emph{Canadian Journal
of Anaesthesia.} 1998;45(S1):R40-R48.

10. Mosavy SH, Shafei M. Prevention of headache consequent upon dural
puncture in obstetric patient. \emph{Anaesthesia.} 1975;30(6):807-809.

11. Smith KA. Spontaneous intracranial hypotension: Targeted or blind
blood patch. \emph{J Clin Neurosci.} 2016;25:10-12.

12. White B, Lopez V, Chason D, Scott D, Stehel E, Moore W. The lumbar
epidural blood patch: A Primer. \emph{Applied
Radiology.}2019;48(2):25-30.

13. Beards S, Jackson A, Griffiths A, Horsman E. Magnetic resonance
imaging of extradural blood patches: appearances from 30 min to 18
h.\emph{BJA: British Journal of Anaesthesia.} 1993;71(2):182-188.

\textbf{Figure legends}

\textbf{Figure 1.} Sonographic longitudinal view of the coccygeal area,
showing the subcutaneous edema and fluid collection (orange arrow) in
the peri-coccygeal area.

\textbf{Figure 2.} The end of sunken conus medullaris (orange arrow)
extending to the S2 vertebra's inferior border with the thickened filum
terminale in the sagittal T2 weighted magnetic resonance imaging (MRI).

\textbf{Figure 3.} An approximately 8 x 7 cm\textsuperscript{2}-sized
fatty contiguous mass with neural placode at the sacrococcygeal area and
the interspersed fluid around the coccygeal mass as seen on horizontal
(A) and sagittal (B) sections on T1-weighted magnetic resonance imaging
(MRI).\selectlanguage{english}
\begin{figure}[H]
\begin{center}
\includegraphics[width=0.70\columnwidth]{figures/Figure-1/Figure-1}

\end{center}
\end{figure}\selectlanguage{english}
\begin{figure}[H]
\begin{center}
\includegraphics[width=0.70\columnwidth]{figures/Figure-2/Figure-2}

\end{center}
\end{figure}\selectlanguage{english}
\begin{figure}[H]
\begin{center}
\includegraphics[width=0.70\columnwidth]{figures/Figure-3A/Figure-3A}

\end{center}
\end{figure}\selectlanguage{english}
\begin{figure}[H]
\begin{center}
\includegraphics[width=0.70\columnwidth]{figures/Figure-3B/Figure-3B}

\end{center}
\end{figure}

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

