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\begin{document}

\title{Omphalolith: An Underdiagnosed Entity?}



\author[1]{Wafa Jouini}%
\author[2]{Noureddine Litaiem}%
\author[1]{Yosra Zaimi}%
\author[3]{leila mouelhi}%
\author[1]{Radhouane Debbeche}%
\author[1]{Faten Zeglaoui}%
\affil[1]{Charles Nicolle Hospital}%
\affil[2]{University of Tunis El Manar Faculty of Medicine of Tunis}%
\affil[3]{Charles Nicolle Hospital, Gastroenterology department}%


\vspace{-1em}



  
  \date{June 17, 2024}


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\begin{abstract}
Omphalolith is a rare and underdiagnosed entity due to the accumulation
of sebum and keratin in the umbilicus. It usually occurs in the elderly
with deep and narrow umbilicus. Early recognition of omphalolith is
important to prevent complications, unnecessary investigations and
anxiety. We report herein two new cases of omphalolith.%
\end{abstract}%



\sloppy


\textbf{Omphalolith: An Underdiagnosed Entity}

\emph{Running header:} Omphalolith

\emph{Author names and affiliation:}

Wafa JOUINI\textsuperscript{1,2} (Corresponding Author), MD, Noureddine
LITAIEM\textsuperscript{1,2}, MD, Yosra ZAIMI\textsuperscript{2,3}, MD,
Leila MOUELHI\textsuperscript{2,3}, MD, Radhouane
DEBBECHE\textsuperscript{2,3}, MD, Faten ZEGLAOUI\textsuperscript{1,2},
MD

\begin{enumerate}
\tightlist
\item
  Department of dermatology, Charles Nicolle Hospital, Tunis, Tunisia
\item
  University of Tunis El Manar, Facult\selectlanguage{ngerman}é de Médecine de Tunis
\item
  Department of Gastroenterology, Charles Nicolle Hospital, Tunis,
  Tunisia.
\end{enumerate}

\emph{Corresponding author:}

Wafa JOUINI Department of dermatology, Charles Nicolle Hospital, Tunis,
Tunisia

\emph{wafajouini91@gmail.com}

\emph{Author emails:}

Wafa Jouini (\emph{wafajouini91@gmail.com}), Noureddine LITAIEM
(\emph{Noureddine.litaiem@gmail.com}), Yosra ZAIMI
(\emph{yosrazaaimi@hotmail.fr} ), Leila MOUELHI
(\emph{leilamouelhi@yahoo.fr} ), Radhouane DEBBECHE
(\emph{radhouane.debbeche@yahoo.fr}), Faten ZEGLAOUI
(\emph{Faten.zeglaoui@fmt.utm.tn})

\emph{Conflict of Interest Disclosure:} None Declared

\emph{Funding Sources:} None.

\emph{Patient's consent:} Written informed consent was obtained from the
patient to publish this report in accordance with the journal's patient
consent policy

\emph{Word count:} 590; \emph{Abstract} : 50. \emph{Figures:}
2,\emph{Tables:} 0, \emph{References:} 6

\emph{Keywords:} Omphalolith, umbilicus, metastasis, Mary Joseph's
nodule, endometriosis, Wilson's disease

\emph{}

\emph{Title:} \textbf{Omphalolith: An Underdiagnosed Entity}

\emph{Abstract}

Omphalolith is a rare and underdiagnosed entity due to the accumulation
of sebum and keratin in the umbilicus. It usually occurs in the elderly
with deep and narrow umbilicus. Early recognition of omphalolith is
important to prevent complications, unnecessary investigations and
anxiety. We report herein two new cases of omphalolith.

\emph{Introduction}

The umbilicus is a remaining scar in the center of the abdomen following
the removal of the umbilical cord (1). It has different shapes. Multiple
affections can raise in this area including metastasis (Mary Joseph's
nodule) and endometriosis. Less commonly reported yet benign conditions
are important to diagnose. Omphalolith is a rare and underdiagnosed
entity due to the accumulation of sebum and keratin in the umbilicus. It
usually occurs in the elderly with deep and narrow umbilicus. Neglect
and poor corporal hygiene represent the main risk factors. Early
recognition of omphalolith is important to prevent unnecessary
investigations and anxiety. Delayed diagnosis may lead to complications
including irritation, erosions, bleeding, pyogenic granuloma, abscess
formation, cellulitis or even peritonitis. We report herein two new
cases of omphalolith and provide a comprehensive summary of its
characteristics.

\emph{Case presentation}

\emph{Case 1:}

A 28-year-old female patient presented with a dark nodule on the
umbilicus. She didn't report pain, bleeding, discharge, or trauma and
denied recent fever or weight loss. Past medical history was consistent
with Wilson's disease with cirrhosis and hyperkinetic extrapyramidal
symptoms. She was entirely dependent on her mother for daily activities.
Clinical examination revealed a firm, keratotic, black nodule protruding
out of the umbilicus, measuring 2 x 1 cm (Figure 1A). The surrounding
skin was otherwise normal. Gentle traction of the nodule using dermal
forceps allowed its extraction (Figure 1B). The underlying skin didn't
show signs of inflammation. Histopathological examination revealed
laminated keratin (figure 1C). The diagnosis of omphalolith was made.
Proper hygiene was advised.

\emph{Case 2:}

A 54-year-old obese female patient with no medical history presented
with a firm, keratotic, brownish nodule protruding out of the umbilicus,
measuring 1,5 x 1 cm (Figure 2A). The surrounding skin was otherwise
normal. Dermoscopic examination revealed a dry crusted pigmented
lamellar keratotic material (Figure 2B). The diagnosis of omphalolith
was suspected. After 15mn application of petroleum jelly, gentle
traction using dermal forceps allowed its extraction (Figure 2C). It
revealed a narrow and deep umbilicus with no signs of inflammation or
ulceration (Figure 2D). The patient was reassured of the benign
character of the condition and proper body hygiene was advised.

\emph{Discussion:}

Omphalolith or umbilical concretion is a rare and benign affection due
to the accumulation of sebum and keratin in the umbilicus (2,3). It
usually presents as a firm black mass. The pigmented color of the
majority of omphaloliths is explained by melanin accumulation and lipid
oxidation (4).

Omphalolith mainly affects elderly women with deep and narrow umbilicus
and poor corporal hygiene (5). Obesity is another predisposing factor
(5,6). In the first case, a deep and retracted umbilicus associated with
insufficient hygiene led to the formation of the omphalolith in a young
and non-obese female patient. Having Wilson's disease with neurological
symptoms and being dependent on her mother may explain insufficient
hygiene in this case. In the second case, a narrow and deep umbilicus
associated with obesity predisposed to omphalolith.

Omphalolith usually remains asymptomatic for several years. I can be
noticed by chance or after secondary complications (3). Repetitive
trauma can cause irritation, erosions, bleeding, and pyogenic granuloma.
Secondary infection, abscess formation, cellulite, or even peritonitis
can also be the revealing symptoms of this condition (3,4,6) .

The diagnosis of omphalolith is based on clinical examination.
Dermoscopy shows aggregates of pigmented keratin. If a histopathological
examination is performed, it reveals laminated keratin associated with
amorphous sebaceous material.

Differential diagnoses include malignant tumors comprising Mary Joseph's
nodule and malignant melanoma. Benign affections can mimic umbilical
concretion such as keloids, umbilical endometriosis, dermatofibroma, and
persistent omphalomesenteric duct (1,2,6). Therefore, early recognition
of omphalolith is important to prevent unnecessary investigations.

The treatment consists of a gentle extraction of the calculus using
forceps (5,6). In our cases, it allowed the extraction of the entire
mass with no bleeding. Excision of an associated pyogenic granuloma can
be necessary in some cases. Patients' education on proper corporeal
hygiene is paramount to prevent recurrences (3,4,6). ~The excision of
the umbilicus may be recommended in some recalcitrant cases(1).

\emph{FIGURES:}

Figure 1: 1A: Black nodule of the ombilicus; 1B: Omphalolith after
removal. three black nodules with horny-like projections; 1C:
Histopathological aspect of the omphalolith showing laminated keratin
(HE x20).

Figure 2: 2A: Keratotic, brownish nodule protruding out of the
umbilicus; 2B: Omphalolith after removal: black keratotic nodule; 2C:
Narrow and deep umbilicus with no signs of inflammation or ulceration;
2D: Dermoscopic aspect: dry crusted pigmented lamellar keratotic
material.

\emph{REFERENCES:}

1. Gallouj S, Harmouch T, Amarti A, Fatima Z. Omphalolith: a rare entity
but important to recognize. Dermatol Online J. 2014 May 16;20(5):22641

2. Ichiki Y, Kitajima Y. Omphalith. Clin Exp Dermatol. 2009;34(3):420\selectlanguage{english}-1.

3. Bounouar M, Hatimi A, Meziane M, Mikou O, Mernissi F, Bennani A, et
al. Omphalolith: Une presentation deroutante. Pan Afr Med J. 2011;8:37.

4. Mahdi HR, El Hennawy HM. Omphalolith presented with peritonitis: a
case report. Cases J. 2009;2:8191.

5. Soriano LF, Pathmarajah P, Rajpopat S, Sahota A. Omphaloliths: a case
series and review of 29 cases in literature. Dermatol Online J.
2019;25(9):13030/qt1pj5r7s7.

6. Nakagawa H, Miyata Y. An Umbilical Mass in a 76-year-old Woman.
Intern Med. 2021;60(18):3059.\selectlanguage{english}
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