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Clinical Review & Education
JAMA Dermatology Clinicopathological Challenge
An Annular Eruption on the Trunk and Limbs
Clara Fernández-Sartorio, MD; Llúcia Alós, MD; José M. Mascaró Jr, MD
A Clinical image
B
Hematoxylin-eosin, original magnification ×200
C
Hematoxylin-eosin, original magnification ×200
Figure. A, Clinical image shows several erythematous annular patches on the flanks and periumbilical region. B, Histopathologic analysis of a punch biopsy sample
from the abdomen showing superficial lymphocytic infiltrates in the papillary and superficial reticular dermis. C, There are also prominent vacuolar changes at the
dermoepidermal junction, and in some areas mild lymphocytic exocytosis.
A man in his 20s presented with a 2-month history of itchy skin lesions on his trunk, arms,
and groin. He was otherwise healthy, with no other underlying conditions, and he was taking no medications. He had no history of atopic dermatitis or other eczematous dermatoses. There were no associated extracutaneous symptoms. His family medical history was
relevant only in that his mother had systemic lupus erythematosus.
On physical examination, there were several erythematous annular patches on the abdomen (flanks and periumbilical region), lower back, groin, wrists, and legs (Figure, A). The lesions
showed slightly raised red-brown borders and a clear center in some patches with no visible
scaling, hypopigmentation, induration, or atrophy. Results of laboratory studies including complete blood cell count, biochemical analysis, complement levels, antinuclear antibodies, antiRo, anti-La, anti-ribonucleoprotein, anti-Smith, and anti-dsDNA antibodies, as well as serologic
testing for syphilis and hepatitis B and C virus, were all normal or negative. A punch biopsy specimen was obtained for histopathologic evaluation (Figure, B and C).
WHAT IS YOUR DIAGNOSIS?
A. Pigmented purpuric dermatosis
B. Mycosis fungoides
C. Annular lichenoid dermatitis
of youth
D. Secondary syphilis
Diagnosis
Microscopic Findings and Clinical Course
C. Annular lichenoid dermatitis of youth
A biopsy specimen was taken from the margins of a lesion and
showed a bandlike lymphocytic infiltrate in the papillary dermis with
vacuolar changes at the dermoepidermal junction, and some areas
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(Reprinted) JAMA Dermatology Published online October 25, 2017
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E1
Clinical Review & Education JAMA Dermatology Clinicopathological Challenge
of the specimen showed mild exocytosis. Dermal lymphocytes expressed CD3, CD4, and CD8 with slight predominance of CD4positive over CD8-positive lymphocytes in the infiltrate with no immunophenotypic alterations. The clinicopathologic findings were
highly suggestive of annular lichenoid dermatitis of youth (ALDY).
The patient was treated with emollients, oral antihistamines for
pruritus, topical corticosteroids for a week, and then pimecrolimus
cream for maintenance. After 1 month of treatment, he had no pruritus, the lesions presented a marked clinical improvement, and most
lesions had almost disappeared. Eleven months later he was still in
complete remission.
Discussion
A rare clinicopathological entity, ALDY was first described in 2003
by Annessi et al.1 To date, 46 cases have been reported in the literature. It mainly affects children and young people but has also been
described in adults.2 The etiopathogenesis of ALDY is currently unknown, and no relationship has been detected with drug use, autoimmune diseases, infections, or neoplasms.1-6
Clinically, ALDY is characterized by erythematous annular
patches with centrifugal growth, slightly raised borders, and a clear
center.1,2,4 The lesions can be single or multiple and are primarily located on the trunk, abdomen, flanks, and groin. Lesions are usually
asymptomatic, but pruritus is occasionally present.1,6
Complementary tests such as blood tests (blood cell count, biochemistry, coagulation), as well as autoimmune studies, multiple infectious serologic analysis tests, and patch tests have had negative
results in all cases. Annular lichenoid dermatitis of youth usually presents a chronic course with frequent recurrences.1,3-6 The treatments used have shown variable responses. In some cases, treatment with topical corticosteroids or topical tacrolimus monohydrate,
0.03%, has achieved a complete resolution with no recurrences.4-6
Spontaneous resolution has also been described in some cases.
ARTICLE INFORMATION
Author Affiliations: Department of Dermatology,
Hospital Clínic, University of Barcelona, Barcelona,
Spain (Fernández-Sartorio, Mascaró); Department
of Pathology, Hospital Clínic, University of
Barcelona, Barcelona, Spain (Alós).
Corresponding Author: Clara Fernández-Sartorio,
MD, Department of Dermatology, Hospital Clínic
and Barcelona University Medical School, Calle
Villarroel 170, 08036 Barcelona, Spain
(clarafernandezsartorio@gmail.com).
Published Online: October 25, 2017.
doi:10.1001/jamadermatol.2017.4320
Conflict of Interest Disclosures: None reported.
Additional Contributions: We thank the patient for
granting permission to publish this information.
Self-assessment Credit: This article is eligible for
journal-based self-assessment (1 credit) for
Maintenance of Certification (MOC) from the
E2
Histopathological findings in ALDY show a lymphocytic
lichenoid infiltrate with basal vacuolization and the presence of
occasional necrotic keratinocytes.1,3,5,6 These changes are characteristically seen at the tip of the epidermal rete ridges.1,4,5 Immunohistochemical studies have shown lymphocyte infiltrates
with slight predominance of CD4-positive over CD8-positive
lymphocytes, and molecular analysis shows a polyclonal T-cell
rearrangement.1,5,6
Annular lichenoid dermatitis of youth is probably underdiagnosed given its clinical and histological similarity to other dermatoses, especially early mycosis fungoides (MF).1,2,4-6 Differential
diagnosis should be made with pigmented purpuric dermatoses,
secondary syphilis, and especially with early-stage MF, to which it
may present great clinical and histopathological similarity. Clinically, all cases present in a similar way, with asymptomatic erythematous macules on the trunk and extremities that can resolve
by themselves or with topical corticosteroid therapy. Histologically, they share a lichenoid inflammatory infiltrate of CD4-positive
T lymphocytes. Immunohistochemical analysis and molecular
studies of T-cell receptor gene rearrangement are not conclusive
because there is no monoclonality at the early stages of MF.
Regarding secondary syphilis, negative serological results and
negative immunohistochemical staining for Treponema pallidum,
in addition to the absence of plasma cell infiltrates, rule this out.
The most difficult differential diagnosis is with the early stages of
MF because the initial forms of MF are indistinguishable from
ALDY by clinical, histological, immunohistochemical, and molecular analysis. Clinical follow-up of these patients is essential to make
a correct and definitive diagnosis.
Annular lichenoid dermatitis of youth is a well-defined clinical and
pathological entity with an excellent prognosis. However, because of
its similarities with early MF, an accurate differential diagnosis and
follow-up are essential to determine adequate treatment.
American Board of Dermatology (ABD). After
completion of an activity, please log on to the ABD
website at www.abderm.org to register your
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REFERENCES
1. Annessi G, Paradisi M, Angelo C, Perez M, Puddu
P, Girolomoni G. Annular lichenoid dermatitis of
youth. J Am Acad Dermatol. 2003;49(6):1029-1036.
2. Cesinaro AM, Sighinolfi P, Greco A, Garagnani L,
Conti A, Fantini F. Annular lichenoid dermatitis of
youth ... and beyond: a series of 6 cases. Am J
Dermatopathol. 2009;31(3):263-267.
4. Di Mercurio M, Gisondi P, Colato C, Schena D,
Girolomoni G. Annular lichenoid dermatitis of
youth: report of six new cases with review of the
literature. Dermatology. 2015;231(3):195-200.
5. Kazlouskaya V, Trager JD, Junkins-Hopkins JM.
Annular lichenoid dermatitis of youth: a separate
entity or on the spectrum of mycosis fungoides?
case report and review of the literature. J Cutan
Pathol. 2015;42(6):420-426.
6. Vázquez-Osorio I, González-Sabín M,
Gonzalvo-Rodríguez P, Rodríguez-Díaz E. Annular
lichenoid dermatitis of youth: a report of 2 cases
and a review of the literature [in Spanish]. Actas
Dermosifiliogr. 2016;107(6):e39-e45.
3. Leger MC, Gonzalez ME, Meehan S, Schaffer JV.
Annular lichenoid dermatitis of youth in an
American boy. J Am Acad Dermatol. 2013;68(5):
e155-e156.
JAMA Dermatology Published online October 25, 2017 (Reprinted)
© 2017 American Medical Association. All rights reserved.
Downloaded From: by a United Arab Emirates University User on 10/25/2017
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