Indian Journal of Dermatology
  Publication of IADVL, WB
  Official organ of AADV
Indexed with Science Citation Index (E) , Web of Science and PubMed
 
Users online: 4384  
Home About  Editorial Board  Current Issue Archives Online Early Coming Soon Guidelines Subscriptions  e-Alerts    Login  
    Small font sizeDefault font sizeIncrease font size Print this page Email this page


 
Table of Contents 
CASE REPORT
Year : 2018  |  Volume : 63  |  Issue : 5  |  Page : 415-417
Bullous pemphigoid induced by metamizole in a pediatric patient


1 Department of Dermatology, Dicle University Medical Faculty, Diyarbakir, Turkey
2 Department of Pathology, Dicle University Medical Faculty, Diyarbakir, Turkey

Date of Web Publication31-Aug-2018

Correspondence Address:
Dr. Isa An
Department of Dermatology, Dicle University, Sur/Diyarbakir
Turkey
Login to access the Email id

Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijd.IJD_209_17

Rights and Permissions

   Abstract 


Bullous pemphigoid (BP) is an autoimmune bullous disease commonly seen in adult population but rarely encountered in the pediatric population. Although the exact etiology of BP remains unclear, various vaccines, infectious agents, and drug use have been blamed in the etiology of BP. Here, we present a 14-year-old patient who developed BP after using metamizole and whose diagnosis was clinically and histopathologically confirmed.


Keywords: Bullous pemphigoid, dipyrone, metamizole


How to cite this article:
An I, Ucmak D, Ibiloglu I. Bullous pemphigoid induced by metamizole in a pediatric patient. Indian J Dermatol 2018;63:415-7

How to cite this URL:
An I, Ucmak D, Ibiloglu I. Bullous pemphigoid induced by metamizole in a pediatric patient. Indian J Dermatol [serial online] 2018 [cited 2020 Jul 12];63:415-7. Available from: http://www.e-ijd.org/text.asp?2018/63/5/415/240095





   Introduction Top


Bullous pemphigoid (BP) is a subepidermal autoimmune bullous dermatosis characterized by linear deposition of immunoglobulin G (IgG) and C3 along the dermoepidermal junction. BP180 and BP230 are major antigens in patients with BP.[1]


   Case Report Top


A previously healthy 14-year-old boy presented to our clinic with the complaints of bullous lesions in the face, scalp, neck, upper chest, and back. The patient noticed the lesions 24 h after receiving intramuscular metamizole due to the complaint of headache. The patient had no systemic symptom except for itching and had no history of infection or other drug use within the last 1 month. Moreover, he had no family history of bullous disease. Skin examination revealed multiple tense bullae in the face, scalp, neck, upper chest, back, and genital region. In addition, numerous targetoid lesions were also present on the back [Figure 1]am[Figure 1]b,[Figure 1]c. No lesion was detected in the oral mucosa. Routine laboratory workup including complete blood count and liver function tests was normal. Histopathologic evaluation of the lesion site indicated subepidermal disintegration and lymphocyte infiltration accompanied by eosinophils in the upper dermis [Figure 2]a. Direct immunofluorescence (DIF) test of the perilesional area revealed a linear deposition of IgG and C3 in the basal membrane [Figure 2]b and [Figure 2]c. The patient was diagnosed as having pediatric BP depending on the clinical, histopathologic, and immunofluorescence findings. The patient was initiated on oral methylprednisolone (1 mg/kg/day) and all the lesions resolved within the first 3 days. At week 1, the methylprednisolone treatment was gradually terminated. No recurrence occurred within the 10-month follow-up.
Figure 1: (a) Multiple tense bullae in the face, scalp, neck, and upper chest; (b) multiple tense bullae and numerous targetoid lesions on the back and (c) multiple tense bullae in the genital region

Click here to view
Figure 2: (a) Subepidermal disintegration (large arrow) and lymphocyte infiltration accompanied by the eosinophils in the upper dermis (small arrow) (H and E; ×200) (b) direct immunofluorescence showing linear deposition of immunoglobulin G in the epidermal basement membrane (×100) (c) direct immunofluorescence showing linear deposition of C3 in the epidermal basement membrane (×100)

Click here to view



   Discussion Top


BP is an autoimmune subepidermal bullous disease frequently seen in elderly patients and rarely encountered in pediatric patients.[2] The incidence of pediatric BP remains unclear. Pediatric BP has two peak incidences, one in the 1st year of life and the other at around 8 years of age.[3] BP is seen equally in women and men in adult patients, whereas it is more common in girls in pediatric patients.[4]

BP has different clinical manifestations; the face, palms, and soles are commonly involved in children aged less than a year, whereas these regions are rarely affected in older children. Although the involvement of the genital region is rarely seen in newborn children, it is presented by almost 50% of older children, mostly in girls. In addition, BP may be limited to the genital region alone in some patients.[3]

The exact pathogenesis of BP remains unknown. However, in the literature, BP has been reported after the first set of vaccinations (tetanus, diphtheria, pertussis, and polio) and hepatitis B vaccine.[1],[2] In addition, a BP case that developed after homeopathy treatment including sulfur, mercury, and Rhus (Toxicodendron) had also been reported.[5] The literature also shows that BP may be triggered by infectious factors such as scabies in adult patients.[6] In addition, BP has also been shown to be associated with Coombs'-positive hemolytic anemia, membranous glomerulonephritis, ulcerative colitis, and hyperimmunoglobulin E syndrome and to develop after liver transplantation.[7],[8]

The diagnosis of BP is usually established by a combination of clinical history, histopathologic features of the disease, and immunofluorescence examination. BP mostly presents as multiple tense bullae. Histopathologic examination of BP mostly reveals eosinophilic infiltration and subepidermal bullae. DIF test of perilesional skin samples commonly indicates linear deposition of IgG and C3 in the basal membrane.[1],[8]

Systemic corticosteroids such as prednisone (1–2 mg/kg/day) constitute the mainstay treatment of pediatric BP.[3] BP commonly yields good prognosis. Patients respond well to early treatment, and most of them achieve complete remission within a few months from the initiation of therapy. Recurrence rarely occurs after the termination of corticosteroid therapy.[1] In resistant cases, some other drugs including dapsone, azathioprine, cyclosporine, mycophenolate mofetil, rituximab, and intravenous immunoglobulin can also be used.[1],[2],[3]

Metamizole, also known as dipyrone, is a nonsteroidal anti-inflammatory drug commonly used as an analgesic or antipyretic agent.[9] Commonly reported side effects of metamizole include agranulocytosis, anemia, acute kidney failure, and severe thrombocytopenia. The cutaneous side effects of metamizole include diaphoresis, urticaria, angioedema, morbilliform, erythematous and purpuric rash, fixed drug eruption, DRESS syndrome, acute generalized exanthematous pustulosis, and toxic epidermal necrolysis.[9] Brenner et al. reported three cases of pemphigus vulgaris induced by metamizole.[10]

Clinicians should be aware of this side effect of metamizole which is a common drug used in clinical practice.

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.
Yang B, Wu M, Yan X, Bao F, Yue Z, Pei Z, et al. Childhood bullous pemphigoid: A report of three cases in China. Int J Dermatol 2016;55:691-4.  Back to cited text no. 1
    
2.
Ister M, Pouessel G, Ythier H, Catteau B, Carpentier O. Postvaccinal, corticosteroid-resistant bullous pemphigoid in infancy: Treatment with intravenous immunoglobulin. Pediatr Dermatol 2014;31:e94-5.  Back to cited text no. 2
    
3.
Ramos L, Tellechea O, Moreno A, Martins L, Neves N, Januário G, et al. Childhood bullous pemphigoid: Report of 2 cases. J Eur Acad Dermatol Venereol 2016;30:330-2.  Back to cited text no. 3
    
4.
Reis-Filho EG, Silva Tde A, Aguirre LH, Reis CM. Bullous pemphigoid in a 3-month-old infant: Case report and literature review of this dermatosis in childhood. An Bras Dermatol 2013;88:961-5.  Back to cited text no. 4
    
5.
Kuenzli S, Grimaître M, Krischer J, Saurat JH, Calza AM, Borradori L, et al. Childhood bullous pemphigoid: Report of a case with life-threatening course during homeopathy treatment. Pediatr Dermatol 2004;21:160-3.  Back to cited text no. 5
    
6.
Chung SD, Lin HC, Wang KH. Increased risk of pemphigoid following scabies: A population-based matched-cohort study. J Eur Acad Dermatol Venereol 2014;28:558-64.  Back to cited text no. 6
    
7.
Kerkar N, Cohen S, Dugan C, Morotti RA, Phelps RG, Herold B, et al. Bullous pemphigoid after liver transplantation for liver failure. Liver Transpl 2006;12:1705-10.  Back to cited text no. 7
    
8.
Erbagci Z. Childhood bullous pemphigoid in association with hyperimmunoglobulin E syndrome. Pediatr Dermatol 2008;25:28-33.  Back to cited text no. 8
    
9.
Nikolova I, Petkova V, Tencheva J, Benbasat N, Voinikov J, Danchev N. Metamizole: A review profile of a well-known “forgotten” drug. Part II: Clinical profile. Biotechnol Biotechnol Equip 2013;27:3605-19.  Back to cited text no. 9
    
10.
Brenner S, Bialy-Golan A, Crost N. Dipyrone in the induction of pemphigus. J Am Acad Dermatol 1997;36:488-90.  Back to cited text no. 10
    


    Figures

  [Figure 1], [Figure 2]



 

Top
Print this article  Email this article
 
 
  Search
 
  
    Similar in PUBMED
   Search Pubmed for
   Search in Google Scholar for
 Related articles
    Article in PDF (1,027 KB)
    Citation Manager
    Access Statistics
    Reader Comments
    Email Alert *
    Add to My List *
* Registration required (free)  


    Abstract
   Introduction
   Case Report
   Discussion
    References
    Article Figures

 Article Access Statistics
    Viewed1546    
    Printed23    
    Emailed0    
    PDF Downloaded55    
    Comments [Add]    

Recommend this journal