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Journal of Indian Association of Pediatric Surgeons
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Table of Contents   
CASE REPORT
Year : 2019  |  Volume : 24  |  Issue : 4  |  Page : 300-302
 

Congenital dermal facial temporal sinus: Rare presentation of intraorbital dermoid in children


1 Department of Pediatric Surgery, Dr. D.Y. Patil Hospital, Navi Mumbai, Maharashtra, India
2 Department of Ophthalmology, Advanced Eye Hospital and Institute, Navi Mumbai, Maharashtra, India

Date of Web Publication29-Aug-2019

Correspondence Address:
Dr. Nandita A Saxena
Department of Pediatric Surgery, Dr. D.Y. Patil Hospital, Nerul, Navi Mumbai, Maharashtra
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jiaps.JIAPS_153_18

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   Abstract 


We present a rare case of an intraorbital dermoid which was associated with a small temporal region dermal sinus in a 3-year-old child. This got infected and the child presented with orbital cellulitis. Definitive surgery involved excision of all the dermal elements using a superficial and intraorbital approach. We stress the need to evaluate, apparently benign lateral facial dermal sinuses as they may be the pointers of the underlying pathological deep dermoid cysts to avoid complications.


Keywords: Dermal sinus, intraorbital dermoid, temporal region


How to cite this article:
Saxena NA, Nair AG, Kulkarni BK, Multani PM, Lahoti HN, Borwankar SS, Goel D. Congenital dermal facial temporal sinus: Rare presentation of intraorbital dermoid in children. J Indian Assoc Pediatr Surg 2019;24:300-2

How to cite this URL:
Saxena NA, Nair AG, Kulkarni BK, Multani PM, Lahoti HN, Borwankar SS, Goel D. Congenital dermal facial temporal sinus: Rare presentation of intraorbital dermoid in children. J Indian Assoc Pediatr Surg [serial online] 2019 [cited 2019 Nov 18];24:300-2. Available from: http://www.jiaps.com/text.asp?2019/24/4/300/265693





   Introduction Top


Congenital facial dermal sinuses are most commonly located in the midline and paranasal regions. Frontotemporal region sinuses are a rare occurrence. However, unlike their cystic counterpart, they notoriously have deeper extensions. Hence, they require detailed imaging and prompt treatment.

We report a case of this rare sinus in a 3-year-old boy which got infected and was found to have an intraorbital extension.

On review of literature, we found that only nine similar cases have been reported.


   Case Report Top


A 3-year-old boy presented with pus discharging from the right lower eyelid. He had a previous history of incision and drainage of a right lower-lid abscess done 2 months ago after which the wound had temporarily healed [Figure 1].
Figure 1: Clinical photograph showing the sinus along with lower-lid abscess

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A detailed clinical examination revealed that the child had a congenital right temporal region sinus which was communicating with this lower-lid abscess wound. Palpation also revealed a small defect in the temporal bone. There was no associated proptosis or squint.

To assess the anatomy in detail, a computed tomography (CT) scan of the face with three-dimensional (3D) reconstruction was done. This showed a bony defect in the orbital process of the right frontal bone. An intraorbital collection measuring 1.1 cm × 1.2 cm × 2.1 cm was noted causing erosion of the lateral orbital wall. It was abutting the anterior aspect of the lacrimal gland [Figure 2].
Figure 2: Computed tomography face showing intraorbital extension and three-dimensional-reconstructed computed tomography image showing bony defect in the right temporal bone

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A thorough debridement of the lower lid abscess was repeated following which the child was started on long-term antibiotic therapy for 6 weeks.

During definitive surgery, a lateral orbitotomy incision was used to expose the lateral intraorbital region. There was thick, pultaceous material within a cyst wall suggestive of a ruptured dermoid cyst.

The cyst was removed piecemeal and the bony defect was defined and curetted. The temporal sinus was cored out up to the bony defect and excised. The abscess cavity on the lower lid was also curetted via a separate incision on the lower lid.

The postoperative course was uneventful and the child remained asymptomatic at 3-month follow-up with no signs of recurrence.


   Discussion Top


The commonest location of congenital dermal sinuses in the face is the midline and paramedian region, with an incidence of 36%–78% reported in literature.[1]

The frontotemporal region is usually affected with a superficial cystic swelling, i.e., the external angular dermoid. The presence of a discharging sinus in this region is a rare occurrence with a rate of about 0.7% as reported by Bonavolontà et al.[1]

Our child had a small congenital sinus in the frontoorbital region with no associated swelling or any ophthalmic complaints and hence was ignored till it developed infectious complications. This stresses on the importance of early evaluation of these lesions even though they appear to be harmless.

The presence of a bony defect in the orbital process of the frontal bone led to the suspicion of an intracranial or intraorbital extension which prompted a CT evaluation. The importance of preoperative assessment for deeper extension in cases of temporofacial sinuses has also been stressed by Yan and Low.[2]

Although rare, there have been a few reports of sinus tracts associated with superficial frontotemporal dermoid cysts.[2],[3] Our case did not have any superficial swelling as the associated cyst was intraorbital. Nine similar cases have been reported in literature so far to the best of our knowledge [Table 1].
Table 1: Reported cases of frontotemporal sinuses with deep extensions

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Most of these lesions presented with complications of infection.[1],[2],[3],[4],[5],[6],[7],[8],[9],[10],[11]

In all the cases, involvement has been limited to just above the dura without any intradural extension.[1],[2],[3],[4],[5],[6],[7],[8],[9],[10],[11]

The approach to these sinuses has been a local exploration, a lateral orbitotomy, or a wider hemi coronal incision. For removal of the intracranial extension, a widening of the existing bony defect or a frontotemporal craniotomy has been sought.

In our case, we preferred the lateral orbitotomy approach with widening of the bony defect to remove the entire lesion albeit piecemeal. This gave a good cosmetic result postoperatively with minimal facial dissection.


   Conclusion Top


Congenital frontotemporal region sinuses are a rare occurrence. However, their presence is an indicator of deeper extension.

Further evaluation with radiological imaging helps to confirm deeper extension prior to surgery for complete removal.

Prophylactic surgical excision may be warranted for avoiding serious complications.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
Bonavolontà G, Tranfa F, de Conciliis C, Strianese D. Dermoid cysts: 16-year survey. Ophthalmic Plast Reconstr Surg 1995;11:187-92.  Back to cited text no. 1
    
2.
Yan C, Low DW. A rare presentation of a dermoid cyst with draining sinus in a child: Case report and literature review. Pediatr Dermatol 2016;33:e244-8.  Back to cited text no. 2
    
3.
Parag P, Prakash PJ, Zachariah N. Temporal dermoid – An unusual presentation. Pediatr Surg Int 2001;17:77-9.  Back to cited text no. 3
    
4.
Cullen JF. Orbital diploic dermoids. Br J Ophthalmol 1974;58:105-6.  Back to cited text no. 4
    
5.
Pollard ZF, Calhoun J. Deep orbital dermoid with draining sinus. Am J Ophthalmol 1975;79:310-3.  Back to cited text no. 5
    
6.
Hong SW. Deep frontotemporal dermoid cyst presenting as a discharging sinus: A case report and review of literature. Br J Plast Surg 1998;51:255-7.  Back to cited text no. 6
    
7.
Wells TS, Harris GJ. Orbital dermoid cyst and sinus tract presenting with acute infection. Ophthalmic Plast Reconstr Surg 2004;20:465-7.  Back to cited text no. 7
    
8.
Scolozzi P, Lombardi T, Jaques B. Congenital intracranial frontotemporal dermoid cyst presenting as a cutaneous fistula. Head Neck 2005;27:429-32.  Back to cited text no. 8
    
9.
Naderi S, Nejat F, Shahjouei S, El Khashab M. Cranial dermal sinus: Presentation, complications and management. Pediatr Neurosurg 2012;48:86-92.  Back to cited text no. 9
    
10.
Bliss M, Grant G, Tittler E, Loven T, Yeom KW, Sidell D, et al. Diagnosis and treatment of pediatric frontotemporal pits: Report of 2 cases. J Neurosurg Pediatr 2016;18:471-4.  Back to cited text no. 10
    
11.
Kwon JM, Lee JW, Ahn JH. Frontotemporal dermoid cyst with sinus tract in a child. J Korean Ophthalmol Soc 2017;58:208-12.  Back to cited text no. 11
    


    Figures

  [Figure 1], [Figure 2]
 
 
    Tables

  [Table 1]



 

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