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September 2022 Volume 6 Issue 5

BRIEF ARTICLE

Eosinophilic Annular Erythema of Childhood: A Rare Case

Mariana Perez, MD1, Benjamin M. Witkoff, DO2, Moises Lutwak, MD3, Michael L. Cohn, MD5,
Eduardo Weiss, MD1-5

1Florida International University, Miami, FL
2Larkin Community Hospital Palm Springs Campus Dermatology Resident Program, Miami, FL

3Larkin Community Hospital South Miami Campus Dermatology Resident Program, Miami, FL
4Hollywood Dermatology & Cosmetic Specialists, Hollywood, FL
5Department of Pathology, Memorial Healthcare System, Miami FL

Eosinophilic annular erythema (EAE) of
childhood is a rare and recurrent skin
condition, with few cases described in the
literature. The trunk and proximal extremities
are mostly commonly affected.1–3 EAE of
childhood is characterized by the
appearance of persistent, non-pruritic,
urticarial annular lesions that enlarge in a
centrifugal pattern.1,2,4,5 Histopathology is
often required for diagnosis, with lesions
exhibiting a perivascular eosinophilic
infiltrate concentrated in the dermis.1–3 This
case report describes a rare case of EAE of
childhood in a newborn with associated
neonatal eosinophilic pustulosis and
laboratory abnormalities.

The patient is a three-week-old male who
was seen in the hospital for a generalized
eruption distributed on the trunk and
extremities, which developed four days
following birth. The lesions were initially
described as bright red targetoid plaques.
After a few days, the lesions improved for a
short period before recurring. Due to
worsening of the rash along with
concomitant diarrhea with streaks of blood
and concerns for dehydration, the patient
was admitted for further work-up and
supportive care. Upon dermatologic exam,
the patient had generalized annular
erythematous plaques with elevated borders
and without scaling (Figure 1-2). The mother
stated the lesions come and go and change

ABSTRACT

Eosinophilic annular erythema (EAE) of childhood is a rare and recurrent skin condition, with only a
few cases described in the literature. The etiology of EAE in children remains unclear. Clinical
presentation shows a persistent, non-pruritic, urticarial annular lesions that enlarge in a centrifugal
pattern. Biopsy is required for diagnosis which characteristically shows a perivascular eosinophilic
infiltrate concentrated in the dermis. Although there is considerable overlap between EAE and Annular
Erythema of Infancy (AEI), some proposed differences include age of onset, duration of lesions,
presence of mucin deposition of histopathology, and time to resolution. This case report describes a
rare case of EAE of childhood in a newborn with associated neonatal eosinophilic pustulosis and
laboratory abnormalities.

INTRODUCTION CASE REPORT

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in shape and distribution. In addition to the
lesions on the trunk and extremities, multiple
pustules over an eczematous base
appeared on the bilateral cheeks and
forehead. No lesions were noted on volar or
mucosal surfaces. Per the mother, the
patient did not appear irritable.

Figure 1. Annular erythematous plaques with
elevated borders on the abdomen.

The pregnancy was full term without history
of autoimmune disease. The patient’s vitals
remained stable throughout the hospital
course. Laboratory work-up showed
consistent hypereosinophilia, peaking at
7,560 µl (36%) and elevated total
immunoglobulin E (IgE). Subsequent
allergic, immunologic, hematologic,
gastrointestinal, and cardiac work-up was
unremarkable.

The clinical differential for the lesions on the
trunk and extremities included eosinophilic
dermatosis, EAE of childhood, annular

erythema of infancy (AEI), neonatal
eosinophilic pustulosis, erythema annularis
centrifugum (EAC), and neonatal lupus. A
3-0 punch biopsy was performed on the right
lower extremity. Histology showed mild
spongiosis without parakeratosis and
eosinophils interstitially and perivascularly in
the dermis (Figure 3a-c). PAS stain was
negative for fungi. There was no evidence of
vasculitis or other significant inflammatory
infiltrate. The histological differential
included hypersensitivity reaction and
allergic reaction.

Figure 2. Annular erythematous plaques with
elevated borders on the left lower extremity.

Based on clinical presentation and histologic
description, the patient was diagnosed with
EAE of childhood in addition to the clinical
diagnosis of neonatal eosinophilic
pustulosis. He was started on triamcinolone
0.1% cream twice daily for lesions on the
trunk and extremities and topical

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September 2022 Volume 6 Issue 5

clindamycin 1% twice daily to pustular
lesions on the face. At one-week outpatient
follow-up, lesions on both the face and body
had improved. Treatment was transitioned to
hydrocortisone 2.5% cream twice daily to
new lesions on the body.

Figure 3. Punch biopsy of the right lower
extremity (3a view at 100x, 3b view at 200x, 3c
view at 400x). Mild spongiosis without
parakeratosis is present. No interface dermatitis
is identified. 3b highlight dermal interstitial and
perivascular eosinophils (orange arrow) with
some lymphohistiocytic infiltrate as well (black
arrow).

EAE of childhood is an extremely rare
condition. There is considerable overlap
between EAE of childhood and AEI, with

some reports using the term interchangeably
in children younger than one year as well as
others describing EAE as a variant of
AEI.1,2,5 Some proposed differences include
age of onset, duration of lesions, presence
of mucin deposition on histopathology, and
time to resolution (Table 1).1,4–6 Although
there are features of AEI in our patient
clinically, histopathology favored EAE. The
biopsy demonstrated a predominant
eosinophilic perivascular infiltrate, typical of
EAE, vs. the perivascular interstitial
lymphohistiocytic infiltrate with some
eosinophils seen in AEI. Furthermore, while
early documented cases of AEI were not
typically associated with laboratory
abnormalities, some cases of EAE have
been found to be associated with peripheral
eosinophilia, as in our patient.3,7

EAE has also been described as an annular
variant of Well’s Syndrome (WS), or
eosinophilic cellulitis. WS, however, is
thought to be histopathologically
characterized by a diffuse inflammatory
infiltrate rather than perivascular
inflammatory infiltrate, eosinophilic
degranulation, the presence of flame figures
(deposits of major basic protein and
degenerated collagen) with surrounding
granulomatous reaction, and vasculitis.1,2,4,5
Additionally, resolution of WS lesions is
associated with hyperpigmentation and/or
residual atrophy, which is not typical for
EAE.1 Neonatal lupus may also be included
in the differential diagnosis, as it can present
with subacute cutaneous lupus
erythematosus-like annular erythematous
plaques.8 However, these lesions are
located in sun exposed areas (i.e. the face),
have fine scaling, and are often associated
with systemic findings such as congenital
heart block, hepatobiliary involvement, and
cytopenias. Additionally, histopathology
would show vacuolar interface changes,
which were not seen on biopsy in our

DISCUSSION

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patient. Finally, EAC is another migratory
annular erythema that can rarely occur in
newborns, but prominent eosinophils are not
noted on biopsy.4–6

The etiology of EAE is unclear. It has been
previously suggested that EAE may be due
to a hypersensitivity reaction, though no
antigen has been identified. In children, EAE
has been diagnosed in association with
asthma, though often no evidence for
underlying disease is found.1 In early
documented cases of AEI, associated
intestinal candida colonization and oral
candidiasis was also reported.7,9

Laboratory abnormalities can occur, though
they are not required for diagnosis.
Peripheral eosinophilia has been reported in
some cases;3 however, elevated IgE levels
have not been previously reported.
Interestingly, patients with neonatal
eosinophilic pustolosis frequently present
with peripheral eosinophilia.10 Further,
infants with hyperimmunoglobulin E (hyper-
IgE) syndrome can present with a crusting
pustular eruption on the face and scalp
similar to that of neonatal eosinophilic
pustulosis, which would be associated with
elevated IgE levels and persistent
eosinophilia.11 However, our patient lacked

Table 1. Comparison of eosinophilic annular erythema (EAE), annular erythema of infancy (AEI), Well’s
Syndrome (WS), and erythema annularis centrifugum (EAC)

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other findings of hyper-IgE syndrome, such
as coarse facial features, recurrent skin and
sinopulmonary infections, severe eczema,
and mucocutaneous candidiasis.

EAE may resolve spontaneously, be treated
in the case of persistent lesions, or be
recalcitrant to several therapies.2 Due to its
rarity, there are no established treatment
recommendations, and previous treatments
are based on the limited case reports with
varying levels of success. Previous reports
have attempted topical and/or systemic
steroids, hydroxychloroquine, chloroquine,
cyclosporine, dapsone, indomethacin,
tofacitinib, dupilumab, and UVB.1–4
Recurrence has been noted in several
reports. In our patient, topical mid-potency
steroids appeared to resolve the lesions.

To our knowledge, this is the earliest
reported case of EAE in a newborn and
among few cases of EAE reported in the
pediatric population. Our case is significant
for its association with neonatal eosinophilic
pustulosis, peripheral eosinophilia, and
elevated IgE. Overall, this report provides
new information to the limited knowledge
surrounding this rare condition.

Conflict of Interest Disclosures: None

Funding: None

Corresponding Author:
Edwardo Weiss, MD
Hollywood Dermatology & Cosmetic Specialists

3850 Hollywood Blvd Suite 403

Hollywood, FL 33021

Phone: (954) 961-1200

Fax: 954.963.0378

Email: eweiss1401@gmail.com

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