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 Table of Contents  
CASE REPORT
Year : 2021  |  Volume : 22  |  Issue : 3  |  Page : 271-273

Oral mucosal Myiasis: A rare association with pediatric SJS-TEN overlap


1 Department of Dermatology, Venereology and Leprosy, IMS and SUM Hospital, Siksha O Anusandhan University, Bhubaneswar, Odisha, India
2 Department of Paediatrics, IMS and SUM Hospital, Siksha O Anusandhan University, Bhubaneswar, Odisha, India

Date of Submission15-Jun-2020
Date of Decision16-Mar-2021
Date of Acceptance04-Apr-2021
Date of Web Publication30-Jun-2021

Correspondence Address:
Annie Raizada
Department of Dermatology, Venereology and Leprosy, IMS and SUM Hospital, Kalinga Nagar, Bhubaneswar - 751 003, Odisha
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijpd.IJPD_28_20

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  Abstract 


Stevens-Johnson syndrome (SJS) and toxic epidermal necrolysis (TEN) overlap is a severe cutaneous adverse reaction with predominantly drug-related etiology which is an immune complex mediated hypersensitivity disorder. Oral myiasis is defined as an intra-oral infestation of the living tissues of animals or humans by dipteral larvae. Herein, we report a rare case of an eight-year-old female child who presented to us with SJS-TEN overlap and was accidentally diagnosed with oral myiasis during her course of hospitalisation. This case report gives an overview of oral mucosal myiasis in childhood as a rare association with SJS-TEN overlap.

Keywords: Childhood oral myiasis, ivermectin, oral mucosal maggots, SJS-TEN overlap


How to cite this article:
Raizada A, Ghosh T, Panda M, Mahapatra A. Oral mucosal Myiasis: A rare association with pediatric SJS-TEN overlap. Indian J Paediatr Dermatol 2021;22:271-3

How to cite this URL:
Raizada A, Ghosh T, Panda M, Mahapatra A. Oral mucosal Myiasis: A rare association with pediatric SJS-TEN overlap. Indian J Paediatr Dermatol [serial online] 2021 [cited 2021 Jul 24];22:271-3. Available from: https://www.ijpd.in/text.asp?2021/22/3/271/319944




  Introduction Top


 Stevens-Johnson syndrome More Details and toxic epidermal necrolysis (SJS-TEN) is an immune complex-mediated hypersensitivity disorder, predominantly of drug-induced etiology. The term myiasis was first coined by Frederick William Hope and is derived from the Latin word “muia” and “iasis” meaning fly and disease, respectively.[1] Here, we present a rare case report of an 8-year-old female child who presented to us with SJS-TEN overlap and during the course of hospitalization was accidentally discovered with oral myiasis. To the best of our knowledge, this association has not been described in the literature before in the pediatric population.


  Case Report Top


An 8-year-old female child presented to us with chief complaints of erythematous to purpuric rash with overlying bullous lesions covering predominantly the chest and back, painful hemorrhagic crusts over the entire lips, and peeling of skin around the periorbital lesion. The patient had a history of intake of oral paracetamol tablets 2 weeks back for fever; 4 days after which, there was the appearance of well-defined erythematous to purpuric macules and few vesicles containing clear fluid over face and trunk. Gradually, the lesions increased in extent and severity and predominantly involved the face, chest, and back and sparsely over the extremities with body surface area (BSA) involvement of around 25%. Pseudo Nikolsky's sign was positive.

A sheet of well-defined dusky erythema covering almost the entire face [Figure 1] and erosions with adherent hemorrhagic crusts and oozing on the lips and adjacent skin [Figure 2] were present along with tender erosions and ulcerations on the inner lower lip, bilateral buccal mucosa, and base of the tongue with restricted mouth opening and inability to take liquid diet. Desquamation was present around the periorbital region with crusting along the eyelids [Figure 2] due to which the patient was unable to open the eyes completely. This was also associated with watery discharge from eyes.
Figure 1: Well-defined dusky erythema covering the face

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Figure 2: Hemorrhagic crusts and oozing on the lips and the adjacent skin and desquamation around periorbital region and crusting along the eyelids

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The patient was alert, febrile, and her vitals were stable. No history of any previous infection, intake of any other drug, and any similar episode in the past was noted.

Patient was able to open her mouth partially after three days of admission. Accidently, intraoral examination revealed multiple maggots crawling within the necrotic lesions over the mucocutaneous junction of the lips and the gingiva, for which an oro maxillary facial consultation was done. Maintaining aseptic conditions, the affected site was explored thoroughly and the wound was flushed with normal saline and irrigated with turpentine oil. The larvae were then eliminated with the help of the forceps which were 5–7 mm long, cylindrical with the tapering end toward the head, and creamy whitish in color. More than 50 maggots were manually removed [Figure 3].
Figure 3: Maggots after the manual removal

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The patient was managed with linezolid antibiotic to prevent secondary bacterial infection, short-course intravenous dexamethasone, Vitamin A capsules, and antihistaminic. For myiasis, tablet ivermectin 6 mg for 3 days orally was given. Over the next 7 days, there was a drastic resolution of both cutaneous [Figure 4] and oral lesions along with the disappearance of the maggots.
Figure 4: Drastic resolution of cutaneous lesions after the treatment

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  Discussion Top


SJS-TEN overlap is a rare variant of severe cutaneous adverse drug reactions characterized by painful erythematous macules evolving to mucous membrane erosions and epidermal detachment due to epithelial cell apoptosis.[2] According to the classification of severity, SJS is defined as epidermal detachment of <10% BSA, SJS-TEN overlap 10%–30% BSA, and TEN >30% BSA. The pathogenesis is considered to be immune mediated.[2] The frequent cause of SJS-TEN syndrome is exposure to drugs, as seen in our patient where paracetamol was found to be the culprit drug. In literature, Paracetamol has been mentioned to be one of the offending drugs to cause SJS-TEN syndrome.[3] Diagnosis mainly relies on clinical signs and histopathology is usually confirmatory.

Bhola et al. defined myiasis as an infestation by dipterous larva on live human and vertebrate animals, which feeds on host's dead or living tissue, liquid body secretions, and fluids or ingested food for a certain period of its lifecycle,[4] and it is usually a disease of tropical countries. Predisposing factors for oral myiasis are poor oral hygiene, mouth breathing during sleep, ulcerative lesion, open bite in the anterior part, and periodontal pockets, and most of the patients are the ones living in poor conditions, mentally retarded, senile, alcoholics or immunocompromised,[5] cerebral palsy, neglected mandibular fracture, patients on mechanical ventilation, and those using narcotics.[6] The most common anatomic sites for myiasis are the nose, eye, lung, ear, anus, and vagina,[1] as the flies responsible for myiasis prefer warm and humid environment.[5] The oral mucosa rarely involved. Oral myiasis is most frequently seen in the anterior maxillary region due to the easy accessibility of the site to the flies.[6] Myiasis is generally self-limiting and in many cases not dangerous to the host; however, complications such as secondary bacterial infection, regional lymphadenopathy, and/or systemic symptoms may arise.

Diagnosis of myiasis is based on the identification of maggots, and in our case, it was a common Indian housefly Musca domestica. Their removal is the mainstay of treatment, published previously in two case reports.[4],[5] Local application of turpentine oil, creosote, mercuric chloride, iodoform, ethyl chloride, and systemic butazolidine and thiabendazole has been used for the treatment.[6] In our case, the local application of turpentine oil and oral ivermectin was done. The former acts as an asphyxiating agent for the larvae which can be subsequently removed as they come out due to asphyxia,[6] and the later acts by blocking the nerve impulses in the nerve ending through the release of gamma-aminobutyric acid, linking to the receptors and causing palsy and death of the parasites.[6]

Oral myiasis has been previously reported in a 12-year-old[4] and 13-year-old[5] children patients associated with neurological deficit and mental retardation, respectively, and both the aforesaid patients were treated with combination therapy of oral ivermectin, turpentine oil application locally, and oral antibiotics to prevent possible secondary infections.


  Conclusion Top


We report this case of mucosal myiasis in a pediatric patient in association with SJS-TEN overlap because of its unusual presentation and rare association. This case report also reviews the incidence, presentation, and therapeutic management of myiasis in childhood cases.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient's parents have given their consent for her images and other clinical information to be reported in the journal. The patient's parents understand that her 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.
Sharma D, Kumar S, Parashar P, Naphade VV. Oral gingival myiasis: A rare case report and literature review. Contemp Clin Dent 2015;6:548-51.  Back to cited text no. 1
[PUBMED]  [Full text]  
2.
Moitra S, Sen S, Banerjee I, Das P, Tripathi SK. Diclofenac-serratiopeptidase combination induced Stevens–Johnson syndrome - A rare case report with review of literature. J Clin Diagn Res 2014;8:YD08-11.  Back to cited text no. 2
    
3.
Das S, Ramamoorthy R. Stevens-Johnson syndrome and toxic epidermal necrolysis in children. Indian J Paediatr Dermatol 2018;19:9-14.  Back to cited text no. 3
  [Full text]  
4.
Bhola N, Jadhav A, Borle R, Adwani N, Khemka G, Jadhav P. Primary oral myiasis: A case report. Case Rep Dent 2012;2012:734234.  Back to cited text no. 4
    
5.
Ali FM, Patil K, Kar S, Patil AA, Ahamed S. Oral myiasis affecting gingiva in a child patient: An uncommon case report. Case Rep Dent 2016;2016:2197450.  Back to cited text no. 5
    
6.
Shah SA, Kadam SG, Padawe DS, Takate VS. Oral myiasis of maxilla and mandible: A case report. J Indian Acad Oral Med Radiol 2015;27:143-6.  Back to cited text no. 6
  [Full text]  


    Figures

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



 

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