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CASE REPORT
Year : 2022  |  Volume : 15  |  Issue : 4  |  Page : 583-586  

An unusual cause of recurrence of cholesteatoma in a child


Department of ENT and Head and Neck surgery, Sri Ramachandra Institute of Higher Education and Research, Chennai, Tamil Nadu, India

Date of Submission14-Aug-2020
Date of Decision04-May-2021
Date of Acceptance04-May-2021
Date of Web Publication28-Jan-2022

Correspondence Address:
Prasanna Kumar Saravanam
65/2, East Colony, ICF, Chennai - 600 038, Tamil Nadu
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/mjdrdypu.mjdrdypu_452_20

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  Abstract 


Predicting the extent of cholesteatoma preoperatively in pediatric patients is a daunting task for the operating surgeons. Several times the operating surgeons come across unexpected findings that define the course of surgery inspite of appropriate preoperative workup. Bone wax is usually made of bee wax admixed and which is made pliable with paraffin or petroleum jelly. It is commonly used to achieve hemostasis during the course of ear surgeries in unexpected bleeding. In this case, we report a rare event in revision mastoid surgery where a large chunk of bone wax that was used in the previous surgery by another surgeon that had migrated into the external auditory canal and was a constant source of pain and discharge in the ear. Diagnostic difficulties and management of the same have been discussed.

Keywords: Bone wax, pediatric cholesteatoma, persistent otorrhea


How to cite this article:
Saravanam PK, Gajendran A. An unusual cause of recurrence of cholesteatoma in a child. Med J DY Patil Vidyapeeth 2022;15:583-6

How to cite this URL:
Saravanam PK, Gajendran A. An unusual cause of recurrence of cholesteatoma in a child. Med J DY Patil Vidyapeeth [serial online] 2022 [cited 2022 Jul 5];15:583-6. Available from: https://www.mjdrdypv.org/text.asp?2022/15/4/583/336737




  Introduction Top


Cholesteatoma is defined as accumulation of keratinizing squamous epithelium in the middle ear or mastoid with surrounding region of inflammation. Treatment and management of pediatric cholesteatoma have always been a challenge due to the aggressive nature of the disease along with its increased incidence of recurrence and the simultaneous need to preserve hearing in children.[1] Bone wax is highly hydrophobic and is often used during ear surgeries for achieving adequate hemostasis by arresting active bleeding and hematoma formation. Inspite of its various advantages, it has been found to interfere with new bone formation and wound healing, thereby acting as a potential source of infection and complication.[2] This case report emphasizes on complete removal of the underlying disease along with appropriate usage of hemostatic agents such as bone wax to avoid recurrence and on table complications in case of revision surgeries.


  Case Report Top


A 9-year-old male presented with complaints of persistent right ear pain associated with bloodstained, foul-smelling discharge for 1 and ½ years, he also gave a history of right ear surgery in another hospital 1 year back. On examination, patient had foul-smelling, scanty, blood-stained ear discharge. Aural toileting was done under otomicroscope which showed brown necrotic bony debris and cholesteatoma flakes occupying the external auditory canal in the posterosuperior region. Since the child had pain, he was agitated and further aural toileting could not be done.

High-resolution computed tomography temporal bone showed soft-tissue shadow along with bony necrosis in the right mastoid cavity extending up to the right sinodural angle posteriorly and filling the middle ear and external auditory canal anteriorly [Figure 1] and [Figure 2]. Revision surgery was done and intraoperatively it was found that the mastoid cavity and middle ear and external auditory canal was filled with bone wax which was removed in toto. Cholesteatoma was found deeper to bone wax and was completely removed [Figure 3], [Figure 4], [Figure 5], [Figure 6]. Breech in tegmen dura and partly drilled posterior canal wall secondary to the disease process or due to previous surgery was noted. The mastoid cavity was exenterated and exteriorized and type III tympanoplasty was done. Patient was followed up for 1 year postoperatively, he had dry and clean mastoid cavity with 30 dB hearing in the right ear.
Figure 1: Soft tissue density noted in right mastoid cavity

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Figure 2: Soft tissue density with bony erosions noted

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Figure 3: Bone wax occupying the middle ear

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Figure 4: Bone wax being removed from middle ear

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Figure 5: Cholesteatoma found deeper to bone wax

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Figure 6: Cholesteotoma removed in toto

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  Discussion Top


Term cholesteatoma was first described by Muller in 1838.[3] Cholesteatoma is broadly divided into congenital and acquired. Pediatric cholesteatomas are thicker when compared to the adult cholesteatoma.[4] The presence of increased angiogenesis in the prematrix of pediatric cholesteatoma showed that there is an increased inflammatory ongoing process associated with pediatric cholesteatoma when compared to adult cholesteatoma.[5] Cholesteatoma epithelium in children has a hyperproliferative nature which is associated with the aggressiveness of the disease in children.[6] Management of pediatric cholesteatoma requires prompt diagnosis and surgical intervention at the appropriate time. The surgical options include canal wall up mastoid surgery (posterior canal wall is preserved) and canal wall down mastoid surgery.

Bone wax was first introduced by Victor Horsley in 1892 as an antiseptic wax with a composition of seven parts of bee wax which was introduced into the surgical field by Parker in 1892.[7] It is soft, malleable, inexpensive, bioinert, nonbrittle material which has tamponade effect on bleeding vessels by having a mechanical effect on trabecular vascularization of the bone without any active hemostatic property such as activating the clotting factor. Bone wax is of two types, the absorbable and the nonabsorbable type. It comes in the form of putties and has minimal inflammatory reaction at the applied site. It is not soluble in bodily fluid and remains in the site of implantation for a long period of time. It is used in various fields of surgical medicine, including orthopedics, maxillofacial, cardiothoracic, and neurosurgery. Usually, in otology bone wax is used by surgeons in case of unexpected bleed during mastoid surgery.

Despite various advantages, bone wax has its own demerits. Although it remains at the site of implantation for long period, the retained or excess bone wax may migrate and cause local inflammatory reaction. Various other complications include ineffective bone healing (by inducing granulomatous inflammation, formation of fibrous tissue encapsulation, and by inhibiting osteogenesis), granuloma reaction (by causing increased inflammation and giant cell formation at the site of implantation), osteohypertrophy, pain, and thrombosis formation. Review of literature showed that bone wax can evoke a foreign body reaction and granuloma formation in the nasal cavity, orbit, and mastoid cavity surgery which can cause persistent ear discharge.[8],[9] It can serve as a nidus of infection at the site of implantation. This can also substantiate the cause of persistent otorrhea in our case.

Owning to the various above-mentioned complications associated with judicious usage of bone wax, it is recommended that it should be used only in necessary situations to achieve hemostasis and the excess bone wax should be removed meticulously and should not be left behind. The usage of bone wax has now been contraindicated in contaminated fields, in fusion sites (spinal surgery), and in patients with hyper immune sensitivity.

There are various other effective topical hemostatic alternatives which provide similar mechanical results with a property of being absorbed by the body without impairing wound healing. Gelatine foam (gel foam) is one of the most commonly used hemostatic agents in otologic procedures which arrests bleeding by acting as a physical barrier.[10] Nhan et al. reported Floseal, a hemostatic matrix which contains bovine-derived gelatin matrix along with human-derived thrombin isolated from pooled plasma which can be used as an effective alternative.[11] Orgill et al. reported the use of water-based resorbable agents such as polyethylene glycol which can be used as an effective hemostatic agent to avoid extensive foreign body reaction and granuloma formation.[12] Collagen, fibrin-collagen, and oxidized cellulose are other various hemostatic agents that are available.

We emphasize the importance of documentation of surgical findings and also the mention of what materials are used during surgery as that would definitely help in the management of revision cases. The use of bone wax needs to be justified and limited in mastoid surgery and cannot be used for obliterating the mastoid cavity as the bone wax by itself can evoke a foreign body reaction and persistent infection.

In this revision case, the mastoid cavity was found to be filled with bone wax with extensive cholesteatoma deeper to it. The bone wax used in the previous surgery could have probably migrated from the mastoid cavity into the external auditory canal leading to an inflammatory reaction and a discharging ear. It was also noted that tegmen tympani was breeched partially which might have been the reason for the use of bone wax in the previous surgery.


  Conclusion Top


This is a rare case report in which a chunk of bone wax was observed during revision mastoid surgery. A surgeon performing revision surgery should be aware of such possibility in the absence of documentation by the previous surgeon as it is difficult to diagnose bone wax clinically and radiologically. This case report highlights the importance of documentation and to expect the unexpected in a revision mastoid surgery and emphasizes the complete removal of cholesteatoma in pediatric age group before obliterating the mastoid cavity to prevent recurrence and unnecessary re-exploration in children.

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.
AlQudehy Z, Al-Abdulqader A, Alshaikh H. Pediatric Cholesteatoma Aggressiveness; a Comparative Study of Predictive Factors and Recurrence Rate. Otolaryngol Open Access J. 2016;1(2):25-32.  Back to cited text no. 1
    
2.
Tham T, Roberts K, Shanahan J, Burban J, Costantino P. Analysis of bone healing with a novel bone wax substitute compared with bone wax in a porcine bone defect model. Future Sci OA 2018;4:FSO326.  Back to cited text no. 2
    
3.
Olszewska E, Wagner M, Bernal-Sprekelsen M, Ebmeyer J, Dazert S, Hildmann H, et al. Etiopathogenesis of cholesteatoma. Eur Arch Otorhinolaryngol 2004;261:6-24.  Back to cited text no. 3
    
4.
Dornelles Cde C, da Costa SS, Meurer L, Rosito LP, da Silva AR, Alves SL. Comparison of acquired cholesteatoma between pediatric and adult patients. Eur Arch Otorhinolaryngol 2009;266:1553-61.  Back to cited text no. 4
    
5.
Morita Y, Yamamoto Y, Oshima S, Takahashi K, Takahashi S. Acquired cholesteatoma in children: Clinical features and surgical outcome. Auris Nasus Larynx 2014;41:417-21.  Back to cited text no. 5
    
6.
Bujía J, Holly A, Antolí-Candela F, Tapia MG, Kastenbauer E. Immunobiological peculiarities of cholesteatoma in children: Quantification of epithelial proliferation by MIB1. Laryngoscope 1996;106:865-8.  Back to cited text no. 6
    
7.
Horsley V. Antiseptic Wax. Brit MJ 1892;7:1165.  Back to cited text no. 7
    
8.
Low WK, Sim CS. Bone wax foreign body granuloma in the mastoid. ORL J Otorhinolaryngol Relat Spec 2002;64:38-40.  Back to cited text no. 8
    
9.
Patel RB, Kwartler JA, Hodosh RM. Bone wax as a cause of foreign body granuloma in the cerebellopontine angle. Case illustration. J Neurosurg 2000;92:362.  Back to cited text no. 9
    
10.
Wilkinson HA, Baker S, Rosenfeld S. Gelfoam paste in experimental laminectomy and cranial trephination: Hemostasis and bone healing. J Neurosurg 1981;54:664-7.  Back to cited text no. 10
    
11.
Nhan C, Bezdjian A, Alarfaj A, Daniel SJ. Can floseal™ be applied safely during otologic surgery? Assessment of ototoxicity in a chinchilla animal model. J Otolaryngol Head Neck Surg 2017;46:1-4.  Back to cited text no. 11
    
12.
Orgill DP, Ehret FW, Regan JF, Glowacki J, Mulliken JB. Polyethylene glycol/microfibrillar collagen composite as a new resorbable hemostatic bone wax. J Biomed Mater Res 1998;39:358-63.  Back to cited text no. 12
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6]



 

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