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Atopic keratoconjunctivitis
A diagnostic dilemma—a case report
∗
Aipeng Li, MDa, Shang Li, MMb, Fang Ruan, MMc, Ying Jie, MDc,

Abstract
Rationale: Atopic keratoconjunctivitis (AKC) remains a difficult diagnosis despite advances in imaging technologies. This is a case
study of the diagnostic and treatment course for a patient with AKC.
Patient concerns: A 15-year-old male complained of progressively increasing pain, redness, watering and blurred vision in the
right eye. The medical history showed that the patient suffered from itching on the hands, knees, neck and the eye skin one year
before the onset of initial symptoms in the affected eye.
Diagnoses: A final diagnosis of stage III AKC with atopic dermatitis (AD) was reached.
Interventions: The patient was used 0.1% tacrolimus eye drops and 0.3% gatifloxacin eye gel after antimicrobial susceptibility test
was performed. In the presence of AD, 0.1% mometasone furoate cream and 0.03% tacrolimus ointment were applied twice daily.
Outcomes: One month after starting treatment, the conjunctivitis and corneal ulcer rapidly improved along with reduced lid
papillae. Macular grade corneal opacity was noticed with minimal thinning. The AD also rapidly improved. At the end of two months
patient was asymptomatic with a significant improvement in his quality of life.
Lessons: Proper diagnosis of AKC especially when associated with dermatological signs along with management of AD in
conjunction with dermatologist is necessary to prevent corneal involvement which can cause permanent visual disability is of utmost
importance. We also noticed that topical tacrolimus is a good option for the treatment of severe AKC with AD along with systemic
immunosupressants.
Abbreviations: AD = atopic dermatitis, AKC = atopic keratoconjunctivitis, VKC = vernal keratoconjunctivitis.
Keywords: atopic dermatitis, atopic keratoconjunctivitis, tacrolimus

1. Introduction from persistent inflammation can occur, possibly leading to

vision loss. Therefore, prompt and effective treatment is required.
In 1953, Hogan described a chronic allergic eye disease—atopic
We herein report a case of AKC (first misdiagnosed as viral
keratoconjunctivitis (AKC)—which although rare can have
stromal keratitis) in a 15-year-old male. Ethical approval was
serious consequences.[1] AKC has been described as a noninfec-
obtained from the Ethics Review Committee at the Beijing Tongren
tious chronic inflammatory ocular-surface condition associated
Hospital (Beijing, China). Written informed consent for publishing
with other atopic conditions. AKC can present along with the
the clinical details and images was obtained from the patient.
onset or course of associated atopy, although the severities of
both conditions may differ along with the involvement of the
cornea.[2] Corneal involvement can progress to frank epithelial 2. Case history
erosion, ulceration, formation of mucous plaques, and epithelial A 15-year-old male presented to our hospital complained of
filaments.[3] Moreover, corneal scarring and neovascularization progressively increasing pain, redness, and watering (epiphora) in
the right eye beginning 15 days previously. As he developed
blurred vision in the same eye during the last 5 days, he
Editor: Kaili Wu.
underwent an ocular examination elsewhere where he was
The authors have no conflicts of interest to disclose.
a
diagnosed with viral stromal keratitis and prescribed topical
Department of Ophthalmology, The First Hospital of Jilin University, Changchun,
b antivirals (ganciclovir eye drops 3 times a day in the right eye).
Department of Ophthalmology, Beijing You’an Hospital, c Beijing Tongren Eye
Centre, Beijing Tongren Hospital, Capital Medical University, Beijing However, the patient’s symptoms did not resolve, instead they
Ophthalmology and Visual Science Key Lab, Beijing, R.P. China. worsened. He was then referred to our hospital for further
∗
Correspondence: Ying Jie, Beijing Tongren Eye Centre, Beijing Tongren management.
Hospital, Capital Medical University, Beijing Ophthalmology and Visual Science The medical history showed that the patient suffered from
Key Lab, 1# Dongjiao Minxiang, Dongcheng, District, Beijing, R.P. China itching on the hands, knees, neck, and the eye skin 1 year before
(e-mail: jie_yingcn@126.com).
the onset of initial symptoms in the affected eye. The itchiness was
Copyright © 2018 the Author(s). Published by Wolters Kluwer Health, Inc. almost gone after 3 months of herbals treatment. One month
This is an open access article distributed under the terms of the Creative
Commons Attribution-Non Commercial-No Derivatives License 4.0 (CCBY-NC-
before the onset, he presented with skin itching again, and the
ND), where it is permissible to download and share the work provided it is symptoms did not response when treated with the same herbs.
properly cited. The work cannot be changed in any way or used commercially Slit lamp examination of the right eye showed a thickened and
without permission from the journal. inflamed eyelid margin with fissuring and hyperpigmentation,
Medicine (2018) 97:16(e0372) conjunctival giant papillary hypertrophy, obscured palpebral
Received: 17 December 2017 / Accepted: 20 March 2018 conjunctival vessels, and mucoid discharge (Fig. 1A). We also
http://dx.doi.org/10.1097/MD.0000000000010372 observed diffuse superficial conjunctival congestion, gelatinous

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Figure 1. (A) Conjunctival giant papillary hypertrophy, thickening, palpebral conjunctival vessels obscured and mucoid/ropy discharge. (B) Resolution of
conjunctival giant papillae following treatment.

Figure 2. (A) Diffuse superficial conjunctival congestion, gelatinous hyperplasia at the superior limbus (red arrow), shield ulcer in the inferior cornea (black arrow).
The superior of the shield ulcer showed dense multifocal anterior stromal infiltrate accompanied with necrotic tissue (yellow arrow). (B) Nebulomacular corneal scar
postresolution.

hyperplasia at the superior limbus (Fig. 2A, red arrow), and a lower eyelid was also found (Fig. 4A). The patient was referred to
shield ulcer in the inferior cornea measuring approximately 6 mm a dermatologist who diagnosed atopic dermatitis (AD).
 3 mm (Fig. 2A, black arrow). The superior of the shield ulcer Corneal scrapping was done and Grams stain showed a large
showed dense multifocal anterior stromal infiltrate accompanied number of cocci, occasional epithelial cells, and a few
with necrotic tissue (Fig. 2A, yellow arrow). No anterior chamber inflammatory cells. Staphylococcus aureus was identified
reaction was noticed. A fundus examination was deferred due to through bacterial culture, and drug sensitivity tests determined
photophobia. No obvious abnormality was found in the left eye that gatifloxacin was more effective than other antibiotics.
(Fig. 3), except the eyelid. Focal areas of dermatitis were found in Further corneal scraping for fungal culture was negative. A final
the right periorbital area, lips, and neck. Dermatitis on the left diagnosis of stage III AKC with AD was reached.

Figure 3. (A) Palpebral conjunctiva. (B) Bulbar conjunctiva and cornea.

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Figure 4. (A) Atopic dermatitis at the periorbital, around the lips and neck. (B) Resolution of atopic dermatitis following treatment.

The patient was started on 0.1% tacrolimus eye drops. AKC is a complex chronic inflammatory disease of the ocular
Initially, 0.5% levofloxacin eye drops was applied to the patient surface. Both conjunctival epithelial cells and inflammatory cells
for 7 days (3 times/day) and then changed to 0.3% gatifloxacin infiltrating conjunctival tissues (eosinophils, T lymphocytes, mast
eye gel after antimicrobial susceptibility test was performed. In cells, and basophils) are responsible for the secretion of both Th1
the presence of AD, 0.1% mometasone furoate cream and 0.03% and Th2 cytokines that induce progressive remodeling of the
tacrolimus ointment were applied twice daily. Systemic treatment conjunctival connective tissue—leading to mucus metaplasia,
(fexofenadine hydrochloride tablet twice a day and cetirizine conjunctival thickening, neovascularization, and scarring. These
hydrochloride tablet once at night) was prescribed as per the physiological mechanisms are responsible for the pathogenesis
dermatologist’s instruction. of corneal complications seen in vernal keratoconjunctivitis
One month after starting treatment, the conjunctivitis and (VKC).[6]
corneal ulcer rapidly improved along with reduced lid papillae, as AKC typically presents in the second or third decade and may
determined by slit-lamp examination. Macular grade corneal continue up to the fifth decade of life, although in rare cases may
opacity was noticed with minimal thinning (Figs. 1B and 2B). The present in childhood or in the adults in their late 50s.[3] Clinical
AD also rapidly improved (Fig. 4B). Treatment was gradually expression of AKC involves conjunctiva, eyelids, and cornea,
tapered over a period of 2 months after which time the patient with a wide spectrum of symptoms such as intense itching,
was asymptomatic with a significant improvement in his quality epiphora, redness, and loss of vision.[7] Different from the
of life. No adverse effects of treatment were noticed throughout infectious eye diseases which can affect only 1 eye, AKC is caused
his health care at our hospital. by “atopy,” a genetic condition, it is generally reported by
bilateral symptoms. However, we noticed only unilateral
involvement in our patient, which may have led to the
3. Discussion
misdiagnosis. This patient was initially misdiagnosed as viral
Both AD and AKC are manifestations of atopy. AKC is stromal keratitis due to the deceptive symptoms, including the
associated with AD in 95% of cases.[4] Conversely, only 20% unilateral onset, corneal ulcer with infiltration into corneal storm
to 43% of patients with AD have ocular involvement.[2] Patients as well as the secondary bacterial infection. However, this patient
with AD (similar to those with AKC due to their reduced innate did not respond to antiviral therapy. Also, giant papillary
immunity) are more susceptible to infections. Several reports of conjunctivitis, gelatinous limbal hyperplasia, shield ulcer,
staphylococcal and herpes simplex infections of the skin and eyes especially the atopic dermatitis occurred in this patient, which
have been published.[5] Similarly, we noticed that our patient were the typically indicators for the differential diagnosis.
developed a secondary staphylococcal bacterial corneal ulcer Generally, the differential diagnosis for AKC also includes:
following development of a shield ulcer, which resolved with use VKC, which has similar symptoms with AKC and often occurs in
of topical antibiotics. patients less than 20 years of age, but the patient developed in

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winter and was associated with atopic dermatitis; and seasonal noticed that topical tacrolimus plus systemic immunosupressants
allergic conjunctivitis, which was seasonal (caused by allergens is a good option for the treatment of severe AKC with AD.
like pollen) or perennial (caused by allergens like dust mites or
animal hair) ocular allergy, but does not affect the cornea. Author contributions
Topical antihistamines combined with mast cell stabilizers are
the cornerstone of ocular allergy treatment, but more aggressive Funding acquisition: ying jie.
treatments such as administrations with topical or systemic Supervision: ying jie.
immunosuppressive drugs (steroids, tacrolimus, and cyclosporin Data curation: Ai peng Li, Shang Li.
A) may be required in the most severe forms of disease.[8] Writing – original draft: Ai peng Li.
Tacrolimus, a competitive calcineurin inhibitor and macrolide Project administration: Shang Li.
antibiotic isolated from the soil fungus species Streptomyces Resources: Fang Ruan.
tsukubaensis, is commonly used in the management of ocular
allergy. Tacrolimus primarily acts by downregulating the activity References
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