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Table of Contents   
CASE REPORT  
Year : 2021  |  Volume : 32  |  Issue : 4  |  Page : 537-540
Management of oral mucosal lesions in salicylate sensitive stevens–Johnson syndrome – A case report


Department of Pediatric and Preventive Dentistry, Sri Ramachandra Institute of Higher Education and Research, Chennai, Tamil Nadu, India

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Date of Submission07-Apr-2020
Date of Decision25-Feb-2022
Date of Acceptance10-Mar-2022
Date of Web Publication18-May-2022
 

   Abstract 


In the present era, the use of drugs is an important paradigm of health care. Reactions to drugs range from minor cutaneous reactions to potentially lethal conditions like Stevens–Johnson syndrome. A 13-year-old, male patient, known case of Stevens–Johnson Syndrome was referred from the pediatric ward for the management of oral mucosal lesions, post consumption of the antibiotic combination of sulfamethoxazole and trimethoprim. Failure of lesion regression led to the change in the treatment plan, speculating an allergic reaction to one of the components of the initial treatment medication (choline salicylate) as well. Identification and withdrawal of the offending medication and rendering supportive care along with treatment of the lesions with topical corticosteroids form the outline of management. This case report demonstrates the approach undertaken by the pediatric dentist to cure the oral mucosal lesions in symbiosis with pediatricians, ophthalmologists and nutritionists to cure this life-threatening condition.

Keywords: Drug allergy, Salicylates, Sensitive Stevens–Johnson syndrome, Sulfonamides, Trimethoprim

How to cite this article:
Panchanadikar N, Balasubramanian S, Nirmal L, Haridoss S, Muthu MS. Management of oral mucosal lesions in salicylate sensitive stevens–Johnson syndrome – A case report. Indian J Dent Res 2021;32:537-40

How to cite this URL:
Panchanadikar N, Balasubramanian S, Nirmal L, Haridoss S, Muthu MS. Management of oral mucosal lesions in salicylate sensitive stevens–Johnson syndrome – A case report. Indian J Dent Res [serial online] 2021 [cited 2022 Dec 7];32:537-40. Available from: https://www.ijdr.in/text.asp?2021/32/4/537/345419



   Introduction Top


Allergies to various drugs, especially antibiotics are not very common. They range from mild cutaneous wheals to major life-threatening conditions like Stevens–Johnson Syndrome (SJS). Hebra in 1866 described SJS as a 'self-limiting, acute inflammatory reaction of the skin and mucous membranes'.[1] It forms a part of the spectrum of diseases of erythema multiforme (EM) family. The mortality rate of SJS is around 5%.[2] SJS lies in the spectrum between EM Minor – involving less than 10% of body surface area and toxic epidermal necrolysis (TEN), which is characterized by mucocutaneous involvement affecting 30-100% of the body surface area.[3]

Painful oral lesions in the form of erosions and crusting over the mucous membranes prove to have a significant impact upon the patient. Palliative care often follows identification and withdrawal of the offending drug which forms the first line of treatment. The following case report presents an illustration of step-by-step management of oral mucosal lesions in a pediatric patient with SJS.


   Case Report Top


A 13-year-old male diagnosed with drug-induced SJS was referred to the Department of Pediatric and Preventive Dentistry for the management of oral mucosal lesions. After suffering from fever for the past 1 day, the patient had self-prescribed syrup Septran (Sulfamethoxazole + Trimethoprim), obtaining it as an over-the-counter drug. He complained of development of painful oral mucosal lesions on the labial (both maxillary and mandibular) and lingual mucosae, immediately after consumption of one dose of Septran. Lesions persisted for around 21 days in total. Oral lesions were preceded by the skin and ophthalmic lesions. Prodromal symptoms included fever, malaise and photophobia. Management was initiated by the pediatricians with Injection hydrocortisone 50 mg IV stat., Tablet cetrizine 10 mg for 15 days, Syrup azithromycin 200 mg per 5 mL for 5 days and tablet paracetamol 250 mg for 15 days. His medical, personal and family histories were non-contributory. Although investigations of complete blood count, albumin levels, liver function tests, blood microbial culture did not detect any abnormalities, an increase in erythrocyte sedimentation rate (ESR) value (21 mm/h) was noted. Nikolsky sign or lesion biopsy was not performed for the patient. He was referred to the Department of Pediatric Dentistry 4 days after the initial management.

Extraoral examination revealed peri-orbital encrustations and flat, atypical lesions on the limbs and side of the neck [Figure 1]. Intraoral examination revealed severe erosions and hemorrhagic crusts with whitish slough on the maxillary and mandibular labial mucosae, flat atypical lesions on the dorsum of the tongue [Figure 2], thick sputum and whitish slough on the right and left buccal mucosae. The patient was symptomatic with severe pain, reduced mouth opening (less than 10 mm), reduced oral intake and poor oral hygiene.
Figure 1: (a) Periorbital encrustation. (b) Flat atypical lesions on the side of the neck

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Figure 2: (a) Erosions and hemorrhagic crusts with whitish slough on the labial mucosa. (b) Flat atypical lesions on the dorsal surface of the tongue

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On referral to our department, the patient was prescribed perioral and intraoral application of topical anesthetic-analgesic agent (Mucopain gel) and Vaseline – to prevent dryness of the mucosa.[4] Compressions with saline and 0.12% chlorhexidine mouth rinse alternately after every 2 hours were advised. The patient was instructed to consume a soft diet with enough liquid intake to avoid dehydration. The parents were instructed not to forcefully remove the encrustations so as to act as a biological membrane.

Severe edema on the labial mucosa and intraoral lesions persisted even after 10 days of therapy [Figure 3]. It was then noticed that the patient had been prescribed ointment Zytee (benzalkonium chloride and choline salicylate) for the labial mucosal lesions by the pediatricians on day 4 of admission. The overall clinical picture showed that the possible reason for lesion exacerbation was the application of ointment Zytee on the labial mucosae. The patient was advised to discontinue Zytee and was prescribed topical corticosteroid – triamcinolone acetonide (Ointment Kenacort) thrice a day, for 15 days in a tapering dose. Improvement was evident, and the lesions regressed within 10 days [Figure 3].
Figure 3: Lesion exacerbation and lesion regression

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   Discussion Top


Erythema multiforme (EM) includes a spectrum of diseases characterized by target or iris lesions on the skin or mucous membrane.[5] SJS is characterized by 'sudden onset of inflammatory bullous lesions on mucous membranes, mainly on the oral mucosa, lips and conjunctiva'.[6] Several other mucocutaneous lesions with the similar clinical course are mentioned in [Table 1]. The clinical classification of EM lesions is described in [Table 2].[7]
Table 1: Differential Diagnosis of acute ulcerative lesions

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Table 2: Clinical Classification of EM lesions

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The causative agents for SJS are infectious agents and drugs. Drugs associated with SJS, mainly include antibacterial (sulfonamides), anticonvulsants, (valproic acid), nonsteroidal anti-inflammatory drugs, antimalarials and allopurinol.[8]

Diagnosis of SJS is based on clinical manifestations. Lab investigations like elevated blood sedimentation rate, mild to moderate elevation of liver enzymes, blood urea nitrogen, fluid and electrolyte imbalances, leukocytosis, eosinophilia and microalbuminuria can be nonspecific.[8]

The management of SJS involves first and foremost withdrawal of the offending drug. Various management protocols have been suggested by various authors including palliative and supportive therapy.

The use of systemic corticosteroids in managing such patients is controversial.[5] For the management of oral mucosal lesions, it has been suggested that debridement should not be performed routinely in children.[9] Blisters though fragile should be left in place or only be punctured. Treatment of erosions can be done with chlorhexidine, octinesept or polyhexanide solutions and impregnated non-adhesive mesh gauze.[10] Disinfectant mouthwashes should be used to treat oral lesions, and mild ointment like dexpanthenol can be applied on hemorrhagic crusting on the labial mucosa.[10] Viscous lidocaine is applied on the lips for symptomatic relief.[9] Saline solution soaks should be applied on the lips, three-four times daily, followed by petroleum jelly.[9] Application of petroleum jelly and sterile saline compressions promotes re-epithelization of the tissues.[3]

Nutritional support should be given parenterally to patients with poor oral intake.[9] Adequate fluid intake is of utmost importance. Anti-inflammatory, anesthetic and topical corticosteroid suspensions provide symptomatic relief for painful oral lesions.[5]

It has been said that salicylates have been implicated in SJS and their use should be avoided.[1] As in our case, the patient was applying ointment Zytee, which is a combination of choline salicylate and benzalkonium chloride, the patient was asked to immediately discontinue the application. Topical application of triamcinolone acetonide was advised, and positive results were noted immediately.

The uniqueness of this case report lies in its management protocol. As oral mucosal lesions aggravated after the initial use of Zytee, it was speculated that the patient was sensitive to salicylates. Once the use of the offending drug was discontinued, rapid improvement was seen in the health of the oral mucosa. Identification of the offending drug and change in treatment plan played a key role in the regression of the lesions.

Management of ocular and skin lesions was performed by the ophthalmologists and pediatricians utilizing topical applications of antimicrobials.


   Conclusion Top


The occurrence of mucocutaneous diseases with oral manifestations is rare in children. The diagnosis is entirely dependent on history and clinical presentation. Identification and withdrawal of the offending drug along with careful and judicious use of treatment medications form the basis of management. Palliative and supportive treatment should be the first line of treatment of such lesions. These lesions must be monitored on a daily basis to understand the clinical course and thus formulate a treatment plan. Treatment with a multi-disciplinary approach has to be advocated taking into consideration the age and the psychological status of the young patient to obtain a better prognosis of this life-threatening condition.

Key message

Allergies to drugs are not uncommon. A severe manifestation of these allergies should be treated with a multidisciplinary approach after identification and elimination of the offending drugs.

Acknowledgements

Authors wish to thank the Department of Paediatrics, Sri Ramachandra Institute of Higher Education and Research for their support in the management of skin as well as ophthalmic lesions for the patient.

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.
Bryant BG, Mathews BL. Stevens-Johnson syndrome. Drug Intell Clin Pharm 1986;20:489–93.  Back to cited text no. 1
    
2.
Forman R, Koren G, Shear NH. Erythema multiforme, Stevens-Johnson syndrome and toxic epidermal necrolysis in children: A review of 10 years experience. Drug Saf 2002;25:965–72.  Back to cited text no. 2
    
3.
Hazin R, Ibrahimi OA, Hazin MI, Kimyai-Asadi A. Stevens-Johnson syndrome: Pathogenesis, diagnosis, and management. Ann Med 2008;40:129–38.  Back to cited text no. 3
    
4.
Creamer D, Walsh SA, Dziewulski P, Exton LS, Lee HY, Dart JKG, et al. UK Guidelines for the management of Stevens-Johnson syndrome/toxic epidermal necrolysis 2016. J Plast Reconstr Aesthet Surg 2016;69:736-41.  Back to cited text no. 4
    
5.
Williams PM, Conklin RJ. Erythema multiforme: A review and contrast from Stevens-Johnson syndrome/toxic epidermal necrolysis. Dent Clin North Am 2005;49:67–76.  Back to cited text no. 5
    
6.
Vanfleteren I, Van Gysel D, De Brandt C. Stevens-Johnson syndrome: A diagnostic challenge in the absence of skin lesions. Pediatr Dermatol 2003;20:52–6.  Back to cited text no. 6
    
7.
Bastuji-Garin S. Clinical classification of cases of toxic epidermal necrolysis, Stevens-Johnson syndrome, and erythema multiforme. Arch Dermatol 1993;129:92-6.  Back to cited text no. 7
    
8.
Fritsch PO, Sidoroff A. Drug-induced Stevens-Johnson syndrome/toxic epidermal necrolysis. Am J Clin Dermatol 2000;1:349–60.  Back to cited text no. 8
    
9.
Prendiville JS, Hebert AA, Greenwald MJ, Esterly NB. Management of Stevens-Johnson syndrome and toxic epidermal necrolysis in children. J Pediatr 1989;115:881–7.  Back to cited text no. 9
    
10.
Mockenhaupt M. The current understanding of Stevens–Johnson syndrome and toxic epidermal necrolysis. Expert Rev Clin Immunol 2011;7:803–15.  Back to cited text no. 10
    

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Correspondence Address:
Dr. Noopur Panchanadikar
Department of Pediatric and Preventive Dentistry, Sri Ramachandra Institute of Higher Education and Research, Chennai, Tamil Nadu – 600 116
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijdr.IJDR_322_20

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    Figures

  [Figure 1], [Figure 2], [Figure 3]
 
 
    Tables

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   Case Report
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