\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[english]{babel}



\usepackage{float}








\begin{document}

\title{Chyloma Formation After Anterior Cervical Disc Fusion}



\author[1]{Theodore Klug}%
\author[1]{Courtney Shires}%
\author[2]{Merry Sebelik}%
\affil[1]{West Cancer Center}%
\affil[2]{Affiliation not available}%


\vspace{-1em}



  
  \date{May 27, 2024}


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





\selectlanguage{english}
\begin{abstract}
Chyle leak is serious sequela of head and neck surgery and has been
reported after esophageal, parathyroid, and thyroid surgery. However, it
has only been reported postoperatively once before after anterior
cervical disc fusion (ACDF). This case discusses the formation of a
chyloma following an anterior cervical spinal procedure.%
\end{abstract}%



\sloppy


Case Report: Chyloma Formation After Anterior Cervical Disc Fusion

Theodore Klug, MD, MPH\textsuperscript{1}; Merry Sebelik, MD,
FACS\textsuperscript{1}; Courtney B. Shires, MD, FACS\textsuperscript{1}

\textsuperscript{1} West Cancer Center, 7945 Wolf River Blvd,
Germantown, TN 38104

Corresponding Author:

Theodore Klug, MD, MPH

West Cancer Center

7945 Wolf River Blvd, Germantown, TN 38104

901-491-1721

theodklu@gmail.com

Author Contributions

Theodore Klug, MD, MPH: Collected data, wrote and edited article

Merry Sebelik, MD, FACS:~Collected data, wrote and edited article

Courtney B. Shires, MD, FACS: Collected data, wrote and edited article

\textbf{Financial Support:} This particular research received no
internal or external grant funding.

\textbf{Conflicts of Interest:} The authors report no relevant financial
disclosures related to this current work.

\textbf{Ethical Considerations:} All issues related to ethics were taken
into consideration throughout the study design and proposal and
implemented during the research study itself. Informed consent was
obtained, beneficence was made a top priority, and respect for
confidentiality and privacy were upheld during the study and its various
analysis and information assertation components.

\textbf{Abstract}

\textbf{Background}

Chyle leak formation is an uncommon but serious sequela of head and neck
(H\&N) surgery, including esophageal, parathyroid, and thyroid surgery.
It has only been reported postoperatively twice after anterior cervical
disc fusion (ACDF) to our knowledge. One prior case was repaired
immediately during the initial spine surgery, and one prior case
required postoperative needle aspiration.

\textbf{Case Presentation}

A 62-year-old obese male patient underwent anterior cervical disc fusion
(ACDF) via a left sided neck approach. There were no issues
intraoperatively and no chyle leak was noted. Four months later he
presented to the clinic with lower left neck pain and swelling inferior
to the incision. Exam and CT imaging showed a 4x4 cm left lower cervical
abscess. He underwent transcervical incision and drainage. He required a
second return trip to the operating room with ligation of the thoracic
duct in the chest. He returned to a regular diet and had no further
issues more than 5 years following the index operation.

\textbf{Conclusions}

We encountered a patient with a chyloma 4 months after ACDF. With neck
pain and swelling following ACDF, chyloma should be considered as a part
of the differential diagnosis. This is the only reported case that
required open repair with ligation of the thoracic duct in the chest.{}

\textbf{Key Words}

Chyloma; thoracic duct; chyle; chyle leak; anterior cervical disc
fusion; head and neck

\textbf{Key Clinical Message}

Chyle leak from iatrogenic thoracic duct injury is a rare but serious
complication of head and neck surgery. The chyloma in this case took
months to recognize and required open thoracic ligation. He fully
recovered.~

\textbf{Case Presentation}

The patient is a 62-year-old male with a BMI 24.2 who presented with
cervical spinal disc disease. He had a herniated nucleus pulposus with
associated radiculopathy at the level of C5-C6. He had hypertension, but
no other comorbidities at the time of the initial surgery. He underwent
anterior cervical disc fusion (ACDF) via a left sided neck approach.
There were no complications intraoperatively and no chyle leak was
noted. The patient was discharged home following the operation in stable
condition. Four months later he presented to clinic with lower left neck
pain and swelling inferior to the incision. Exam showed indurated and
erythematous left neck and upper chest skin (Figure 1). He had no
accompanying symptoms of fever or dysphagia. CT imaging showed a 4x4 cm
left lower cervical abscess (Figures 2 and 3). He underwent
transcervical incision and drainage. A JP drain was placed at that time.
This drain had \textgreater{}1000 cc of cloudy output per day. ENT was
consulted. The drainage was felt to be chyle. The patient was taken back
to the OR with ENT and thoracic surgery. A fluid cavity with a capsule
was noted. The chyloma was felt to have walled itself off. As this
capsule was opened, copious chyle was released. The thoracic duct was
noted inferior and lateral to the subclavian vein. This was ligated in
the chest. Another JP drain was placed. The drain was observed
postoperatively, and no further chyle leak was noted. The drain was
removed after 5 days. He returned to a regular diet and had no further
issues greater than 5 years following the index operation.



\textbf{Discussion}

The vast majority of lymphatic injury during spine surgery occurs in the
thoracic and lumbar regions. Injury due to the close proximity of the
thoracic duct and the cisterna chyli to the vertebral column results in
chylothorax or chyloretroperitoneum. ~The incidence of thoracic duct
injury following cervical spine surgery is substantially lower in than
in thoracolumbar surgery. This may be due to the longer anatomic course
of the thoracic duct in the thoracolumbar area as compared to the
cervical spine. ~The height of the arch of the thoracic duct in the root
of the neck also varies, and can be inferior to, at, or superior to the
clavicle.\textsuperscript{1} Langford et al reported that a thoracic
duct can extend as much as 5 cm above the clavicle\textsuperscript{2},
and Hart et al described a case in which the arch was situated 7 to 8 cm
above the clavicle.\textsuperscript{3} Also, there is significant
deviation in the termination pattern of the thoracic duct as it joins
the systemic circulation. It may end as single or multiple outlets into
the left internal jugular vein, the left subclavian vein, the left
external jugular vein, the left brachiocephalic vein, the left
transverse cervical vein, or the right internal jugular
vein.\textsuperscript{1,3-5}{}

Intraoperative identification of lymphatic injury can be difficult due
to the usual fasting state of patients prior to surgery, which
diminishes lymphatic production and transport. Thus cloudy lymphatic
material may not be visible in the wound even if an injury exists.
Valsalva administration or placing the patient in the Trendelenburg
position may assist in identifying an intraoperative leak. {}

During head and neck surgery, chyle leak from iatrogenic thoracic duct
injury is a rare but serious complication that occurs in 0.5--1.4\% of
thyroidectomies\textsuperscript{6-10} and 2--8\% of neck
dissections\textsuperscript{2, 6, 11-13}. However, it has only been
reported postoperatively twice before following an anterior cervical
disc fusion (ACDF). \textsuperscript{14} Our case is the third case of
chyle leak after cervical spine surgery.

Of the 3 reported cases of iatrogenic thoracic duct injury from ACDF
(Table 1), 2 were male and 1 was female. All three of the patients were
undergoing surgery focused on level C5-6 and were approached from the
left side.

The patients were relatively healthy, with patient 1 being in an MVA,
patient 2 having no comorbidities and patient 3 having hypertension.
None of the patients were obese. The size of the postoperative chyle
collection on imaging was 2.3 cm for Patient 2 and 4 cm for Patient 3.
One of the 3 leaks was noted intraoperatively and was repaired
immediately with clips. One of the leaks was noted after 2 months and
was treated with needle aspiration. The third leak was noted 4 months
after the initial surgery and required intrathoracic ligation of the
thoracic duct. In the 2 patients with delayed leaks, there was no noted
direct sizeable injury to the thoracic duct intraoperatively during
initial surgery. Given the delayed presentation and return after 2 and 4
months following ACDF, the differential diagnosis before imaging and
exploration was the formation of a post-op hematoma, seroma, or chyloma.
Following imaging, drainage, and surgery, the suspicion of a chyloma was
confirmed. We believe the chyloma likely occurred secondary to a
retraction injury on a portion of her thoracic duct. There was a small
leak that over time created a cystic capsule which contained the leak.
Patient 3 had immediate release of significant chyle (after a drain had
already collected 1000 mL) once this capsule was opened during
subsequent repair, but Patient 2 was able to heal after a simple needle
aspiration of 3.5 mL. These 3 patients show the range of leak severity
after injury during ACDF. {}

Two patients were discharged the day after their initial ACDF, and one
was discharged the same day as ACDF. As the ACDF surgery is trending
towards an outpatient procedure, postoperative chyle leaks will often
more likely be discovered in a delayed manner during an office visit,
like Patients 2 and 3.

Presenting symptoms of the delayed leaks were nontender, mobile neck
mass in Patient 2 and neck pain and swelling in Patient 3. No patient
had postoperative fever, dysphagia, hoarseness, shortness of breath, or
electrolyte abnormalities.

The 3 patients are doing well with follow up periods of 80 days, 3.5
years, and 5 years. Patient 3 who underwent thoracic duct ligation had
no subsequent nutrition deficiency, as has been reported
previously.\textsuperscript{15} After being addressed, patients may
recover completely from this complication. None of these 3 patient has
long-term side effects.

\textbf{Conclusions}

Chyle leaks can occur intraoperatively or become evident months after
ACDF. Three cases have reported, with an incidence of
\textless{}0.001\%. These all occurred after surgery at the C5-6 level
and a left sided approach. They present as lower neck swelling and pain.
These can be treated postoperatively with needle drainage if small or
open ligation of the thoracic duct if severe. Patients can experience a
full recovery.

\textbf{List of Abbreviations}

Head and neck (H\&N); anterior cervical disc fusion (ACDF); anterior
cervical corpectomy and fusion (ACCF); postoperative day (POD)

\textbf{References}

1. Van Pernis PA. Variations of the thoracic duct. Surgery. 1949;26:
806-809.

2. Langford R. J., Daudia A. T., Malins T. J. A morphological study of
the thoracic duct at the jugulo-subclavian junction.~Journal of
Cranio-Maxillo-Facial Surgery.~1999;27(2):100--104. doi:
10.1016/S1010-5182(99)80021-3.~

3. Hart AK, Greinwald JH, Shaffrey CI, Postma GN. Thoracic duct injury
during anterior cervical discectomy: a rare complication. Case report. J
Neurosurg. 1998;88:151-154.

4. Gottlieb MI, Greenfield J. Variations in the terminal portion of the
human thoracic duct. AMA Arch Surg. 1956;73:955-959.~

5.~ Kinnaert P. Anatomical variations of the cervical portion of the
thoracic duct in man. J Anat. 1973;115:45-52.

6.~ Delaney SW, Shi H, Shokrani A, Sinha UK. Management of Chyle Leak
after Head and Neck Surgery: Review of Current Treatment
Strategies.~\emph{Int J Otolaryngol}. 2017;2017:8362874.
doi:10.1155/2017/8362874

7. Lee Y. S., Kim B.-W., Chang H.-S., Park C. S. Factors predisposing to
chyle leakage following thyroid cancer surgery without lateral neck
dissection.~Head \& Neck.~2013;35(8):1149--1152. doi:
10.1002/hed.23104.~

8. Lorenz K., Abuazab M., Sekulla C., Nguyen-Thanh P., Brauckhoff M.,
Dralle H. Management of lymph fistulas in thyroid surgery.~Langenbeck's
Archives of Surgery.~2010;395(7):911--917. doi:
10.1007/s00423-010-0686-2.~

9. Roh J.-L., Yoon Y.-H., Park C. I. Chyle leakage in patients
undergoing thyroidectomy plus central neck dissection for differentiated
papillary thyroid carcinoma.~Annals of Surgical
Oncology.~2008;15(9):2576--2580. doi: 10.1245/s10434-008-0017-9.~

10.~Rammal A., Zawawi F., Varshney R., Hier M. P., Payne R. J., Mlynarek
A. M. Chyle leak: a rare complication post-hemithyroidectomy. Case
report and review of literature.~Otolaryngologia
Polska.~2014;68(4):204--207. doi: 10.1016/j.otpol.2014.03.003.~

11. Dhiwakar M., Nambi G. I., Ramanikanth T. V. Drain removal and
aspiration to treat low output chylous fistula.~European Archives of
Oto-Rhino-Laryngology.~2014;271(3):561--565. doi:
10.1007/s00405-013-2534-9.~

12. Nussenbaum B., Liu J. H., Sinard R. J. Systematic management of
chyle fistula: the Southwestern experience and review of the
literature.~Otolaryngology---Head and Neck Surgery.~2000;122(1):31--38.
doi: 10.1016/s0194-5998(00)70140-9.~

13. Crumley R. L., Smith J. D. Postoperative chylous fistula prevention
and management.~The Laryngoscope.~1976;86(6):804--813. doi:
10.1288/00005537-197606000-00008.~

14. Derakhshan A, Lubelski D, Steinmetz MP, et
al.~\href{https://pubmed.ncbi.nlm.nih.gov/28451482/}{Thoracic~Duct~Injury~Following
Cervical Spine Surgery: A Multicenter Retrospective Review.} Global
Spine J. 2017 Apr;7(1 Suppl):115S-119S. doi: 10.1177/2192568216688194.
Epub 2017 Apr.

15. Bonavina L, Saino G, Bona D, Abraham M, Peracchia A. Thoracoscopic
management of chylothorax complicating esophagectomy. J Laparoendosc Adv
Surg Tech A. 2001;11:367-369.\selectlanguage{english}
\begin{figure}[H]
\begin{center}
\includegraphics[width=0.70\columnwidth]{figures/Figure-1/Figure-1}

\end{center}
\end{figure}

Figure 1. Indurated and erythematous left neck and upper chest skin 4
months after initial surgery.

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

\end{center}
\end{figure}

\begin{quote}
\begin{quote}
\begin{quote}
\begin{quote}
\begin{quote}
Figure 2. Sagittal CT imaging showing a 4x4 cm left lower cervical
abscess.

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

\end{center}
\end{figure}

\begin{quote}
\begin{quote}
\begin{quote}
\begin{quote}
\begin{quote}
Figure 3. Axial CT imaging showing a 4x4 cm left lower cervical abscess.

\par\null
\end{quote}
\end{quote}
\end{quote}
\end{quote}
\end{quote}

Table 1.\selectlanguage{english}
\begin{longtable}[]{@{}llllllllllll@{}}
\toprule
Patient & age of pateint & sex & BMI & level of spine surgery~ &
approach side & time leak noted & method of repair & size of fluid
collection on imaging & date of initial discharge & symptoms & length of
follow up\tabularnewline
1 & NA & M & NA & C5-6 & left & intraoperative & clips & NA &
postoperative day 1 & NA & 80 days\tabularnewline
2 & NA & F & 21.9 & C5-6 & left & 2 months postoperatively & needle
aspiration & 2.3 cm & same day & nontender, mobile neck mass & 3.5
years\tabularnewline
3 & 62 years old & M & 24.2 & C5-6 & left & 4 months postoperatively &
ligation of thoracic duct in the chest & 4 cm & postperative day 1 &
neck pain and swelling & 5 years\tabularnewline
\bottomrule
\end{longtable}

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

