open access

Vol 8 (2023): Continuous Publishing
Case report
Published online: 2023-03-31
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Orbital angiolymphoid hyperplasia with eosinophilia in a pediatric patient: a case report

Alex Weaver1, Aaron Ross Harris2, Nathan Albrecht3, Daniel Lattin4, Guangiu Luo5
·
Ophthalmol J 2023;8:22-24.
Affiliations
  1. University of Florida College of Medicine, Gainesville, FL, United States
  2. University of Miami Miller School of Medicine, Miami, FL, United States
  3. Department of Pediatrics, University of Florida College of Medicine, Jacksonville, FL, United States
  4. Nemours Children’s Health, Jacksonville, FL, United States
  5. Children’s Healthcare of Atlanta, Atlanta, GA, United States

open access

Vol 8 (2023): Continuous Publishing
CASE REPORTS
Published online: 2023-03-31

Abstract

A 7-year-old female presented to an outpatient eye clinic with a six-week history of left upper eyelid edema and erythema in the absence of trauma without compromise of visual acuity or extraocular muscle movements and was initially treated as a case of pre-septal cellulitis with antibiotic therapy. Failure of resolution with new onset nasal bleeding after two weeks prompted computed tomography (CT) and magnetic resonance imaging (MRI), showing an orbital mass with radiologic concern for lymphoma or rhabdomyosarcoma. Upon biopsy, the histopathological analysis diagnosed angiolymphoid hyperplasia with eosinophilia (ALHE), a rare condition that infrequently presents with ocular manifestations and more rarely presents in the pediatric population. Most therapeutic management for cutaneous and non-cutaneous ALHE have high rates of recurrence. This case was refractory to oral prednisone treatment but responded to a combination of methotrexate and adalimumab therapy with an 18.5% reduction in volume after three months.

Abstract

A 7-year-old female presented to an outpatient eye clinic with a six-week history of left upper eyelid edema and erythema in the absence of trauma without compromise of visual acuity or extraocular muscle movements and was initially treated as a case of pre-septal cellulitis with antibiotic therapy. Failure of resolution with new onset nasal bleeding after two weeks prompted computed tomography (CT) and magnetic resonance imaging (MRI), showing an orbital mass with radiologic concern for lymphoma or rhabdomyosarcoma. Upon biopsy, the histopathological analysis diagnosed angiolymphoid hyperplasia with eosinophilia (ALHE), a rare condition that infrequently presents with ocular manifestations and more rarely presents in the pediatric population. Most therapeutic management for cutaneous and non-cutaneous ALHE have high rates of recurrence. This case was refractory to oral prednisone treatment but responded to a combination of methotrexate and adalimumab therapy with an 18.5% reduction in volume after three months.

Get Citation

Keywords

ALHE; epithelial hemangioma; images

About this article
Title

Orbital angiolymphoid hyperplasia with eosinophilia in a pediatric patient: a case report

Journal

Ophthalmology Journal

Issue

Vol 8 (2023): Continuous Publishing

Article type

Case report

Pages

22-24

Published online

2023-03-31

Page views

2153

Article views/downloads

283

DOI

10.5603/OJ.2023.0007

Bibliographic record

Ophthalmol J 2023;8:22-24.

Keywords

ALHE
epithelial hemangioma
images

Authors

Alex Weaver
Aaron Ross Harris
Nathan Albrecht
Daniel Lattin
Guangiu Luo

References (8)
  1. Wells GC, Whimster IW. Subcutaneous angiolymphoid hyperplasia with eosinophilia. Br J Dermatol. 1969; 81(1): 1–14.
  2. Azari AA, Kanavi MR, Lucarelli M, et al. Angiolymphoid hyperplasia with eosinophilia of the orbit and ocular adnexa: report of 5 cases. JAMA Ophthalmol. 2014; 132(5): 633–636.
  3. Urabe A, Tsuneyoshi M, Enjoji M. Epithelioid hemangioma versus Kimura's disease. A comparative clinicopathologic study. Am J Surg Pathol. 1987; 11(10): 758–766.
  4. Adler BL, Krausz AE, Minuti A, et al. Epidemiology and treatment of angiolymphoid hyperplasia with eosinophilia (ALHE): A systematic review. J Am Acad Dermatol. 2016; 74(3): 506–12.e11.
  5. Bahloul E, Amouri M, Charfi S, et al. Angiolymphoid hyperplasia with eosinophilia: report of nine cases. Int J Dermatol. 2017; 56(12): 1373–1378.
  6. McEachren TM, Brownstein S, Jordan DR, et al. Epithelioid hemangioma of the orbit. Ophthalmology. 2000; 107(4): 806–810.
  7. Buder K, Ruppert S, Trautmann A, et al. Angiolymphoid hyperplasia with eosinophilia and Kimura's disease - a clinical and histopathological comparison. J Dtsch Dermatol Ges. 2014; 12(3): 224–228.
  8. Baker MS, Avery RB, Johnson CR, et al. Methotrexate as an alternative treatment for orbital angiolymphoid hyperplasia with eosinophilia. Orbit. 2012; 31(5): 324–326.

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