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 Table of Contents  
CASE REPORT
Year : 2021  |  Volume : 22  |  Issue : 3  |  Page : 250-253

Eosinophilic annular erythema in childhood: A distinct entity


1 Department of Dermatology, Venereology and Leprology, SBH Govt Medical College, Dhule, Maharashtra, India
2 Department of Dermatology, Venereology and Leprology, Krishna Institute of Medical Sciences, Karad, Maharashtra, India
3 Department of Paediatrics, SBH Govt Medical College, Dhule, Maharashtra, India

Date of Submission31-May-2020
Date of Decision16-Jun-2020
Date of Acceptance30-Mar-2021
Date of Web Publication30-Jun-2021

Correspondence Address:
Chetan D Rajput
Department of Dermatology, Venereology and Leprology, SBH Govt Medical College, Dhule, Maharashtra
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijpd.IJPD_98_20

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  Abstract 


Introduction: Many conditions included under figurate erythemas have similar clinical appearance which makes diagnosis difficult. Among these, eosinophilic annular erythema (EAE) is a rare, uncommon, underreported entity in the pediatric population. Case Report: Herein, we report an 11-year-old boy who presented with asymptomatic recurrent, erythematous-edematous annular plaques with central hyperpigmentation. Discussion: Review of the literature shows scarce reporting of such cases globally and none from India. Clinicopathological findings of our case, suggest that eosinophilic annular erythema is a distinct entity rather than a type of Wells' syndrome.

Keywords: Child; Annular erythema; Eosinophils; Dermatology; Hydroxychloroquine, eosinophilic annular erythema, hydroxychloroquine, Wells' syndrome


How to cite this article:
Rajput CD, Nikam BP, Hatkar NK. Eosinophilic annular erythema in childhood: A distinct entity. Indian J Paediatr Dermatol 2021;22:250-3

How to cite this URL:
Rajput CD, Nikam BP, Hatkar NK. Eosinophilic annular erythema in childhood: A distinct entity. Indian J Paediatr Dermatol [serial online] 2021 [cited 2021 Jul 24];22:250-3. Available from: https://www.ijpd.in/text.asp?2021/22/3/250/319958




  Introduction Top


Many annular dermatological conditions have a diagnostic clinical appearance; however, figurate erythema is a broad group of dermatoses that spread centrifugally to form annular, polycyclic, arcuate, or serpiginous lesions or remain fixed. This group encompasses many conditions that vary from benign annular erythema of infancy to neonatal lupus erythematosus with systemic involvement.[1] Eosinophilic annular erythema (EAE) is an uncommon benign eosinophilic dermatosis of unknown etiology having recurrent erythematous-edematous annular plaques with tissue eosinophilia without flame figures or granulomatous inflammation.[2],[3] We herein describe this rare entity which shared clinical and histopathological features that enabled diagnosis. We also provide insight into the relationship between different eosinophilic dermatosis in childhood.


  Case Report Top


An 11-year-old boy was referred to the dermatology outpatient department for the third episode of asymptomatic rash. On dermatological examination, erythematous-edematous annular, polycyclic, arcuate nonscaly plaques with central hyperpigmentation were noted over the trunk, abdomen, lower extremities, and scrotum. There was also a plaque over the upper back which had minimal scaling [Figure 1]a, [Figure 1]b, [Figure 1]c, [Figure 1]d. He had similar episodes twice in the last three months which were treated with systemic steroids. The child was otherwise healthy . There was no relevant history for any allergies or drug intake. There was no recent travel history or any history of insect bites. There were no systemic complaints. Laboratory investigations including complete blood count, erythrocyte sedimentation rate, and C-reactive protein were normal there was no evidence of peripheral eosinophilia. Other hematological and biochemical tests, thyroid function, urine, and stool analysis were normal. Potassium hydroxide mount from the minimally scaly lesion over the upper back did not show any hyphae. Other tests such as antinuclear antibodies, double-stranded DNA, anti-Ro/anti-La, antithyroid antibodies, rheumatoid factor, complement level, and serology for Borrelia, hepatitis, herpes simplex, venereal disease research laboratory, and human immunodeficiency virus (HIV) were negative. Radiological investigations (chest X-ray and ultrasonography) were also normal. Skin biopsy from the rim of the annular lesion was performed for histopathological examination and direct immunofluorescence (DIF). The hematoxylin and eosin-stained skin biopsy showed mild focal epidermal spongiosis and diffuse interstitial and perivascular lymphohistiocytic infiltrate, dermal mucin deposits, and abundant eosinophils involving entire upper and mid dermis without eosinophilic degranulation, flame figures, vasculitis, or granulomatous inflammation [Figure 2]a and [Figure 2]b. No immunoreactant deposits were seen on DIF. Temporary clearance was obtained with oral prednisolone but relapse occurred after discontinuation. Analysis of history, clinical, and histopathological findings resulted in a diagnosis of EAE. To reduce anxiety and needless fear of parents, the child was started on hydroxychloroquine 200 mg/day for 6 weeks. Lesions subsided in 6 weeks with mild residual pigmentation, and the child was child was followed up for one year without any recurrence.
Figure 1: (a-d) Erythematous-edematous annular, polycyclic, arcuate plaques with central hyperpigmentation over trunk, abdomen, lower extremities and scrotum

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Figure 2: (a) Diffuse interstitial and perivascular infiltrate of lymphocytes and eosinophils (H and E, ×10). (b) Numerous interstitial eosinophils without flame figures (H and E, ×40)

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  Discussion Top


EAE is a rare, benign, asymptomatic, and recurrent condition. Although described in children, adult cases have been reported and the first case was described by Kahofer et al. in 2000.[4],[5] Clinically, two types of asymptomatic lesions are featured: figurate or centrifugum type pattern; and the annular or urticarial type, and they fade away with erythema or pigmentation but no atrophy or scarring.[6],[7] Histologically, it has tissue eosinophilia with basal melanosis reflecting central hyperpigmentation.[8]

EAE needs to be differentiated morphologically from various causes of figurate erythema and urticarial lesions. Figurate erythema is a group of dermatoses which encompasses erythema annulare centrifugum, erythema chronic migrans, erythema marginatum, annular erythema of infancy, familial annular erythemas, neonatal lupus, and its variant erythema gyratum atrophicans transiens neonatal while erythema gyratum repens is not yet reported in pediatric age.[9] The urticarial annular type needs to be distinguished from urticarial vasculitis, Sweet's syndrome, acute hemorrhagic edema of infancy, and erythema multiforme. Differentiation of these conditions is of immense importance for management and depends upon the age of onset, duration, course of disease, morphology, distribution, associated features, and family history. Differential diagnosis of figurate and urticarial lesion conditions in contrast to EAE is summarized in [Table 1].[1],[9],[10],[11]
Table 1: Differential diagnosis of figurate and urticarial lesions in childhood[1],[9],[10],[11]

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Similarly, histopathologically, eosinophilic infiltrate in EAE requires differentiation from other eosinophilic dermatoses such as arthropod bite, drug reaction, annular erythema of infancy, Wells' syndrome (eosinophilic cellulitis), and early urticarial stage of bullous pemphigoid. Our patient was an adolescent boy with late-onset of lesions, which helps distinguish the lesions from annular erythema of infancy. Eosinophilic infiltrate without epidermal changes differentiate it from arthropod bite and drug reaction. Asymptomatic lesions and negative DIF rule out the early urticarial stage of bullous pemphigoid. The absence of vasculitis differs from recurrent cutaneous eosinophilic vasculitis.

Wells' syndrome is uncommon in childhood, and classically presents with prodromal burning, painful edema progressing to infiltrated plaques resembling cellulitis. There is peripheral eosinophilia. However, annular variant has also been reported.[12] Histologically it is characterized by superficial and deep perivascular infiltrate of lymphocytes and eosinophils extending up to hypodermis, eosinophilic degranulation, flame figures, vasculitis, and granulomatous inflammation around flame figures.[12] Our case lacked classical clinical presentation of Wells' syndrome, and infiltrate was confined to the only the upper and mid dermis with basal vacuolar change and dermal mucin deposition as additional findings. These features distinguish it as a distinct entity, rather than a subtype of Wells' syndrome. Weedon also stated that EAE may not be a variant of Wells' syndrome.[13]

Localized variants of eosinophilic annular erythema over the neck and palms and soles have also been reported.[14],[15] Although condition responds to steroids temporarily, various drugs have been used for treatment such as chloroquine, hydroxychloroquine, dapsone, indomethacin, cyclosporine, dupilumab, and UVB phototherapy.[3],[4],[7] We treated our case with hydroxychloroquine due to its action of inhibition of chemotaxis of eosinophils, and complete response was achieved in 6 weeks without any recurrence in one year of follow up.

Declaration of patient consent

The authors certify that they have obtained all appropriate consent forms, duly signed by the parent(s) of the patient. In the form the parent(s) has/have given his/her/their consent for the images and other clinical information of their child to be reported in the journal. The parents understand that the names and initials of their child 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.
Palit A, Inamadar AC. Annular, erythematous skin lesions in a neonate. Indian Dermatol Online J 2012;3:45-7.  Back to cited text no. 1
[PUBMED]  [Full text]  
2.
Peterson AO Jr, Jarratt M. Annular erythema of infancy. Arch Dermatol 1981;117:145-8.  Back to cited text no. 2
    
3.
Karataş Toğral A, Seçkin D. Eosinophilic annular erythema: A late but complete response to hydroxychloroquine. Australas J Dermatol 2017;58:228-30.  Back to cited text no. 3
    
4.
Kahofer P, Grabmaier E, Aberer E. Treatment of eosinophilic annular erythema with chloroquine. Acta Derm Venereol 2000;80:70-1.  Back to cited text no. 4
    
5.
Heras MO, Muñoz NP, Sancho MI, Millet PU. Eosinophilic annular erythema in adults: Report of two cases and review of the literature. An Bras Dermatol 2017;92:65-8.  Back to cited text no. 5
    
6.
Rongioletti F, Fausti V, Kempf W, Rebora A, Parodi A. Eosinophilic annular erythema: An expression of the clinical and pathological polymorphism of Wells syndrome. J Am Acad Dermatol 2011;65:e135-e137.  Back to cited text no. 6
    
7.
Thomas L, Fatah S, Nagarajan S, Natarajan S. Eosinophilic annular erythema: Successful response to ultraviolet B therapy. Clin Exp Dermatol 2015;40:883-6.  Back to cited text no. 7
    
8.
Nakazato S, Fujita Y, Shinkuma S, Nomura T, Shimizu H. Eosinophilic annular erythema is clinically characterized by central pigmentation reflecting basal melanosis: A clinicopathological study of 10 cases. J Eur Acad Dermatol Venereol 2017;31:1916-23.  Back to cited text no. 8
    
9.
Saha A, Seth J, Pradhan S. Annular erythema in pediatric population. Indian J Paediatr Dermatol 2016;17:167-72.  Back to cited text no. 9
  [Full text]  
10.
Toledo-Alberola F, Betlloch-Mas I. Annular erythema of infancy. Actas Dermosifiliogr. 2010;101:473-84.  Back to cited text no. 10
    
11.
Hamidi S, Prose NS, Selim MA. Neutrophilic figurate erythema of infancy: A diagnostic challenge. J Cutan Pathol 2019;46:216-20.  Back to cited text no. 11
    
12.
Howes R, Girgis L, Kossard S. Eosinophilic annular erythema: A subset of Wells' syndrome or a distinct entity? Australas J Dermatol 2008;49:159-63.  Back to cited text no. 12
    
13.
Weedon D. Weedon's Skin Pathology. 3rd ed. New York: Elsevier (Churchill Livingstone); 2010. p. 940-1.  Back to cited text no. 13
    
14.
Lee HS, Yang JY, Kim YC. Eosinophilic Annular Erythema Localized to the Palms and the Soles. Ann Dermatol 2016;28:769-71.  Back to cited text no. 14
    
15.
Hioki T, Takama H, Makita S, Shibata A, Akiyama M, Watanabe D. Eosinophilic annular erythema localized on the neck. Eur J Dermatol 2017;27:673-4.  Back to cited text no. 15
    


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