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|||�|||hhhhhh����Title: Concurrent thyroglossal duct cyst and dermoid cyst in a pediatric patient. 
Keywords: thyroglossal duct cyst; dermoid cyst; Sistrunk procedure; midline neck mass 
Authors: 
Leenoy Hendizadeh BS1, Soroush Zaghi MD1, Dana Haydel MD 2, Soroush Farnoosh BS3, Jeffrey Koempel MD, MBA3 
Department of Head and Neck Surgery, David Geffen School of Medicine at the University of California, Los Angeles; Los Angeles, CA 
Department of Pathology, Children�s Hospital Los Angeles at the Keck School of Medicine at the University of Southern California, Los Angeles, CA
Division of Otolaryngology � Head and Neck Surgery, Children�s Hospital Los Angeles at the Keck School of Medicine of the University of Southern California, Los Angeles, California
Corresponding Author: 
Leenoy Hendizadeh, B.S. 10833 LeConte Avenue, Rm. 62-132, CHS Los Angeles, CA 90095-1624
Email: leenoyhendi@gmail.com 
Phone: 001-310-775-1451
Fax:  HYPERLINK "tel:(310)%20206-5106" \t "_blank" 001-310-206-5106
This work has not been previously presented. There were no conflicts of interests and there was no funding for this work. 






Abstract: 
Among the most frequently encountered congenital midline cervical masses in the pediatric population are thyroglossal duct cysts and dermoid cysts.  While both of these entities should be considered in the differential diagnosis of an anterior midline neck mass, the two may occur simultaneously in rare instances.  We present the case of a 4 year-old girl who presented with what appeared to be a single, anterior neck mass that extended into the floor of mouth on pre-operative imaging.  During surgical excision, two completely separate cystic masses were identified; one in the floor of the mouth and another immediately superior to the hyoid bone.  We present this case to increase awareness to the rare circumstance of a dual diagnosis in the evaluation of a pediatric neck mass and to illustrate the importance of preparing the patient and the parents for a Sistrunk procedure pre-operatively.  We recommend that surgeons prepare for a Sistrunk operation when considering an excision of any mass in the mid-line, near mid-line, or floor of mouth in a pediatric patient. 











Introduction:

Among the most frequently encountered congenital midline cervical masses in the pediatric population are thyroglossal duct cysts (TGDCs) and dermoid cysts (DCs) [1].  TGDCs are asymptomatic congenital masses that are derived from the persistence of a thyroglossal duct remnant.  They typically present in the midline and can be located as far superior as the tongue base and as far inferior as the manubrium of the sternum [2,3].  DCs are masses that are filled with sebum-like material and evidence of skin derivatives [4].  DCs may also present as midline neck masses in the submental region [5,6].  While both TGDCs and DCs should be included in the differential diagnosis of an anterior midline neck mass in a pediatric patient, in some rare cases, these two entities may occur simultaneously [2]. 
We present the case of a 4 year-old girl who presented with what appeared to be a single, anterior neck mass on pre-operative imaging that extended into the floor of mouth. Upon surgical excision, two completely separate cystic masses were identified; one in the floor of mouth as well as one located immediately superior to the hyoid bone. 
We present this case to demonstrate the possibility of a dual diagnosis in the evaluation of a pediatric neck mass; specifically to bring awareness to the possibility of a concurrent TGDC and DC.  We also illustrate the importance of preparing for a Sistrunk procedure in the surgical treatment of any mass in the mid-line, near mid-line, or floor of mouth in a pediatric patient.  


Case: 
A 4 year-old girl presented to our office with a three-month history of a mass in the floor of mouth with swelling that resulted in a perceived change in her breathing.  The mass was first noticed by her parents when she was three years old and increased in size over a three-month period. 
A CT of the neck with contrast was obtained and read by the radiologist as a single 5.7 cm multi-loculated, fluid filled mass in the midline under the tongue and anterior to the hyoid bone (Figure 1). 
The patient was taken to the operating room for surgical excision.  An incision was made in the mucosa of the tongue just posterior to the sublingual gland duct openings.  A monopolar electrosurgical unit with a Colorado tip was used to raise the tongue musculature superiorly.  A cystic mass was immediately obvious under the mucosa.  Because the mass could not be visualized in entirety through the oral cavity, a combined external approach was utilized.  A curvilinear incision was made in the skin within a relaxed skin tension line overlying the hyoid bone.  The median raphe of the strap muscles was divided.  Dissection began at the level of the inferior portion of the thyroid cartilage and proceeded superiorly.  Several millimeters of the medial portion of both sides of the strap muscles were taken with the specimen as the dissection proceeded superiorly towards the hyoid bone.  A ruler was used to measure a 1 cm midportion of the hyoid bone using the laryngeal prominence as a mark for the midline.  A cut was made on the hyoid bone at each end of this marking and both visual inspection and palpation were used to confirm that the midportion of the hyoid bone was completely resected.  A 0.5 cm diameter portion of tongue musculature superior to the hyoid was resected as well to complete the Sistrunk procedure.  Finally, the mass was retracted superiorly and resected as one single complete specimen along with the midportion of the hyoid bone and the soft tissue extension to the base of tongue.  The cystic mass itself measured 3 cm by 3 cm. 
A portion of the mylohyoid muscle was divided revealing a second, distinctly separate cystic mass. This second mass was larger, measuring 5 cm by 3 cm, and less translucent in nature as compared to the previously resected cystic mass. 
Upon pathologic analysis, the 5 cm by 3 cm mass which was in the floor of the mouth was found to be a dermoid cyst; composed of squamous epithelium, adnexal structures, sebaceous glands, and keratin debris at surface tissue (Figure 2).  The 3 cm by 3 cm mass that was immediately superior and dorsal to the hyoid bone was found to be a thyroglossal duct cyst lined by ciliated respiratory epithelium (Figure 3 and Figure 4). 

Discussion:

The differential diagnosis for an anterior midline neck mass in a pediatric patient includes the following:  branchial cleft cyst, laryngocele, ranula, thyroid mass, paraganglioma, teratoma, vascular malformation, inflammatory mass, lymphadenopathy, neoplastic lesion, thymic cyst, lipoma, benign skin cyst, dermoid cyst, and thyroglossal duct cyst [7].  DCs and TGDCs are among the most commonly encountered lesions in the pediatric population and should be at the top of the list of differential diagnoses for a congenital midline neck mass [1]. 
TGDCs typically present as asymptomatic masses that are painless, non-tender, and not fluctuant.  They are most often located adjacent to the hyoid bone; they are either on the hyoid bone or immediately above or below the hyoid bone [1,3].  Movement of the cyst upon swallowing is often cited as an important diagnostic sign.  However, misdiagnosis may occur due to similar behavior of deep DCs.  TGDCs are derived from the aberrant persistence of an embryonic remnant anywhere along the migration pathway of the thyroid diverticulum as it descends from the tongue base to the thyroid gland�s normal location anterior to the first several tracheal cartilages [2].  This pathway normally obliterates by the 5th to 8th week of gestation to leave a proximal remnant, the foramen cecum, at the base of the tongue and a distal remnant, the pyramidal lobe of the thyroid gland [2].  Incomplete obliteration of this embryonic thyroglossal pathway would result in a thyroglossal duct remnant or TGDC [2].
DCs are often initially noticed during the first few years of life but may also go unnoticed for a long period of time [5].  They present as benign, painless, non-tender, superficial, and sometimes mobile swellings.  DCs occur in the head and neck region with an incidence of about 7% [8].  DCs form along the lines of embryologic fusion since they originate from entrapment of epithelium in deep tissue during embryogenesis or as a result of traumatic implantation [9].  They consist of stratified squamous epithelium lining and skin adnexae like hair follicles or sebaceous glands [1,10,11].  DCs are filled with keratinous material while the more solid dermoid tumor forms are usually pedunculated and filled with a matrix of connective tissue and thick fibro-fatty material [5]. 
Preoperatively, it is often difficult to distinguish between TGDCs and DCs [1,6,12].  This difficulty in distinguishing between DCs and TGDCs was demonstrated in a review of 75 cases which found that 17 midline cervical cysts were misdiagnosed intra-operatively as TGDCs when they were actually found to be DCs or mixed cysts upon pathological diagnosis (deMello 10).  On physical exam, TGDCs move with deglutition and tongue protrusion because they are often attached to the hyoid bone. However, like TGDCs, DCs may also be adherent to the hyoid bone and move with deglutition and tongue protrusion.  On ultrasound, DCs are usually more homogeneous as compared to TGDCs which are more heterogeneous with complex mixed appearances [2].  Both DCs and TGDCs can increase in size over time.  However, these two structures can be easily distinguished intra-operatively by creating a small opening into the cystic mass.  TGDCs usually contain mucus or a mucopurulent material while DCs usually contain a keratinaceous material [3].  Although some surgeons might choose to open the cysts intra-operatively, others have warned against such a maneuver due to the potential of local infection from the spillage of cyst contents [10].  It is our practice to open these masses if there is any question about the diagnosis. 
The recommended treatment for both DCs and TGDCs is excision in order to prevent an infection or abscess formation and in the case of TGDCs, to remove a potentially malignant lesion such as a papillary thyroid carcinoma which has been reported in rare cases world-wide.  Determining the correct diagnosis does affect surgical management.  Masses identified as TGDCs are treated with a Sistrunk procedure; complete excision of the mass including the one centimeter mid-portion of the hyoid bone as well as removal of some tongue musculature superior to the hyoid.  Surgical management of DCs, on the other hand, involves excision of the mass alone.  A TGDC removed by a Sistrunk procedure reduces the recurrence rate to as low as 4% while cyst excision alone can result in a recurrence rate as high as 50% [1,13].  A simple resection is curative for a typical DC [1,10].  Erroneously diagnosing a TGDC as a DC, however, would result in a simple cyst excision and likely lead to recurrence and another surgery [13]. 
Atypical presentations of midline neck masses, such as the sublingual mass in our patient, are especially susceptible to a misdiagnosis as demonstrated in a 300 patient retrospective review in which 20 of the 28 misdiagnosed lesions were TGDCs that presented in atypical sites [13, 14].  Therefore, it is important for the surgeon to consider a Sistrunk procedure in order to best avoid a recurrence.  The possibility of a second mass should also be considered despite any pre-operative imaging that might lead one to believe that only one lesion is present.  Care should be taken during excision to identify the presence of a concurrent lesion. 
Other case reports have pointed out the association between TGDCs and DCs through their simultaneous, yet contiguous, presentations.  Such associations were demonstrated in the following publications: seven reports of �mixed� TGDC and DCs which presented as midline cervical cysts since they contained characteristics of classic TGDCs (i.e. an epithelial tract or thyroid follicles) and classic DCs (i.e. skin appendages) [10,11], one report of a TGDC attached to a DC in the anterior neck line with no communication between the two cysts [15], a contiguous DC and TGDC in the anterior floor of the mouth [16], an anterior neck lesion comprised of two DCs and a TGDC [12].
Two cases of concurrent, non-contiguous DC and TGDC similar to our patient were recently described by Lin et al [2].  An 11-year old boy had with two separate cysts; a DC in the submandibular space and a TGDC in continuity with the hyoid bone [2].  These separate cystic structures were revealed preoperatively by CT and MRI imaging studies. And similar to our patient, a 17-day-old baby boy who had a DC on the floor of the mouth and a TGDC just left of the anterior neck and above the hyoid bone [2]. The baby�s cervical mass went undetected preoperatively and intra-operatively since the clinically noted mass was intraoral and the CT scan did not include the neck region.  The baby returned 18 months after the intraoral mass� excision for the excision of a newly noticed submental mass which was found to be TGDC.  
Although the floor of mouth is a rare location for a DC, a Sistrunk procedure in our case resulted in the identification of two distinct masses without violation of the capsule of either cystic structure.  Resection of the mid-portion of the hyoid bone should be considered by the surgeon and included in the informed consent discussion for a patient with any mid-line, near mid-line, or floor of mouth mass.  Furthermore, the presence of two distinct masses concurrently is a rare and potentially underappreciated occurrence despite pre-operative imaging that may only identify one single lesion. 


References: 
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13.  HYPERLINK "http://www.ncbi.nlm.nih.gov/pubmed?term=T%C3%BCrkyilmaz%20Z%5BAuthor%5D&cauthor=true&cauthor_uid=15043670" T�rkyilmaz Z,  HYPERLINK "http://www.ncbi.nlm.nih.gov/pubmed?term=S%C3%B6nmez%20K%5BAuthor%5D&cauthor=true&cauthor_uid=15043670" S�nmez K,  HYPERLINK "http://www.ncbi.nlm.nih.gov/pubmed?term=Karabulut%20R%5BAuthor%5D&cauthor=true&cauthor_uid=15043670" Karabulut R, Demirgoullari B, Sezer C, et al. Management of thyroglossal duct cysts in children.  HYPERLINK "http://www.ncbi.nlm.nih.gov/pubmed/15043670" \l "#" \o "Pediatrics international : official journal of the Japan Pediatric Society." Pediatr Int. 2004 Feb;46(1):77-80.14. Solomon JR, Rangecroft L. Thyroglossal-duct lesions in childhood. J. Pediatr. Surg.1984; 19: 555"61. HYPERLINK "info:pmid/6502427" \o "Link to external resource: " 

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Figure 1: Axial and sagittal C.T. scans showing a multi-loculated mass at the floor of mouth. 

Figure 2: Photomicrograph of intraoral specimen, at 4 x magnification demonstrating DC traits; showing the cyst cavity (C.C.) with keratin in the lumen lined by stratified squamous epithelium (S.S.E.) that connects with�multiple underlying pilosebaceous units (P.S.). Hematoxylin and eosin stain.

Figure 3: Photomicrograph of specimen found superior to the hyoid bone, at 4 x magnification showing the cyst cavity (C.C.) lined by ciliated respiratory epithelium (C.R.E.), mucous glands (M.G.) and hyoid bone fragment (H.B.) characteristic of a TGDC. Hematoxylin and eosin stain.

Figure 4: Photomicrograph specimen found S]�������				6
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