Clinical Characteristics of Idiopathic Orbital Inflammation Accompanied with Paranasal Sinusitis

Hyun Lee Ka; MD Jin Sook Yoon,

doi:10.3341/jkos.2017.58.7.776

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Ka and Jin Sook Yoon,: Clinical Characteristics of Idiopathic Orbital Inflammation Accompanied with Paranasal Sinusitis

Original Article

J Korean Ophthalmol Soc 2017;58(7):776-781.

Published online: 8 September 2017

DOI: https://doi.org/10.3341/jkos.2017.58.7.776

Clinical Characteristics of Idiopathic Orbital Inflammation Accompanied with Paranasal Sinusitis

Hyun Lee Ka, M.D.^1,², MD Jin Sook Yoon,, PhD^1,

¹The Institute of Vision Research, Department of Ophthalmology, Yonsei University College of Medicine, Seoul, Korea

²Department of Ophthalmology, Konyang University College of Medicine, Daejeon, Korea

Address reprint requests to Jin Sook Yoon, MD, PhD Department of Ophthalmology, Severance Hospital, # 50-1 Yonsei-ro, Seodaemun-gu, Seoul 03722, Korea Tel: 82-2-2228-3570, Fax: 82-2-312-0541 E-mail: yoonjs@yuhs.ac

* Conflicts of Interest: The authors have no conflicts to disclose.

Received 20 April 20173 June 2017 Accepted 25 June 2017

This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/by-nc/3.0/) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.

Abstract

Purpose

To investigate the clinical characteristics of idiopathic orbital inflammatory disease (IOI) with paranasal sinusitis.

Methods

This study is a retrospective, comparative case series of patients who were diagnosed with IOI between January 2009 and December 2016. This study included patients with available medical and radiologic data at diagnosis and who participated in follow-up for more than 12 months after treatment. The patients were divided into two groups according to accompaniment of paranasal sinusitis and were compared.

Results

Among 101 patients with IOI, 13 (12.9%) were identified to have paranasal sinusitis. The incidence of pain was higher in patients with sinusitis (69.2%) than in patients without sinusitis (25.0%, p = 0.003). More patients with paranasal sinusitis ex-perienced recurrence after systemic steroid therapy (69.2%) than in the other group (34.1%, p = 0.033). Additional im-munosuppressants and/or radiation therapy were needed only in 9.1% patients without sinusitis but in 38.5% patients with sinus-itis (p = 0.039).

Conclusions

IOI patients with sinusitis showed a significantly higher recurrence rate. More careful follow-up of patients during steroid tapering and treatment of sinusitis might be helpful to prevent recurrence of IOI.

Keywords: Idiopathic orbital inflammation, Sinusitis

References

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Figure 1.

Treatment and outcome for patients with orbital inflammatory disease. More patients with paranasal sinusitis experienced re-currence after systemic steroid therapy (69.2%) than in the other group (34.1%, p = 0.033). Additional immunosuppressants and/or radiation therapy were needed only in 9.1% patients without sinusitis but in 38.5% patients with sinusitis (p = 0.039). IOI = idiopathic orbital inflammation.

Figure 2

. Facial photograph and orbital computed tomography (CT) in a patient who visited our clinic with a 1-month history of pain and proptosis in the right eye, and diplopia (case 2). (A, B) Facial photograph at initial visit shows proptosis in the right eye. (C) Initial CT results revealed infiltration of the right inferior orbital area with ipsilateral maxillary sinusitis. She experienced repetitive inflammation despite of systemic steroid therapy and additional immunosuppressants. (D) The follow-up CT taken after functional endoscopic sinus surgery for maxillary sinusitis. She stopped systemic steroid and additional immunosuppressant, and maintained without recurrence.

Table 1.

Clinical manifestations in patients with idiopathic orbital inflammation

	Patients with sinusitis (13 patients)	Patients without sinusitis (88 patients)	p-value
Sex (male:female)	9:4	42:46	0.250
Age (years, mean)	48.3	45.5	0.587
Symptom duration (months, mean)	5.1	7.1	0.575
Total follow up (months, mean)	22.0	27.9	0.665
Unilateral: Bilateral	10:3	71:15	0.916
Initial symptom (n, %)
Eyelid swelling	10 (76.9)	51 (58.0)	0.317
Proptosis	6 (46.2)	25 (28.4)	0.331
Pain	9 (69.2)	22 (25.0)	0.003^†
Palpable mass	1 (7.7)	13 (14.8)	0.795
Diplopia	3 (23.1)	5 (5.7)	0.732
Decreased vision	1 (7.7)	18 (23.4)	0.701
Location (n, %)
Lacrimal gland	3 (23.1)	39 (44.3)	0.250
Intraocular muscle	4 (30.8)	33 (37.5)	0.872
Lacrimal gland + intraocular muscle	3 (23.1)	5 (12.5)	0.106
Diffuse infiltration of retrobulbar fat	3 (23.1)	11 (5.7)	0.548
Sclerosing disease (pathology)	1/7	8/22	0.271
IgG4-related disease (n, %)	4 (30.8)	12 (13.6)	0.114
Treatment (n, %)
Only systemic steroid	8 (61.5)	80 (90.9)	0.039^†
+ Additional immunosuppressants^*	1 (7.7)	2 (2.3)
+ Radiation	2 (15.4)	3 (3.4)
+ Additional immunosuppressants and radiation	2 (15.4)	3 (3.4)
Outcome (n, %)
Recurrence ≥1	9 (69.2)	30 (34.1)	0.033^†

Values are presented as n (%) unless otherwise indicated. IgG4 = immunoglobulin G4.

^* Per oral methotrexate, azathioprine, and cyclosporine in addition to systemic steroid;

^† Significant p-values. p-values < 0.05 are considered to be statistically significant.

Table 2.

Clinical manifestations at the initial visit in idiopathic orbital inflammation patients with sinusitis

Case	Sex/Age Ocular signs and symptoms	Symptom duration (months)	Radiologic finding		IgG4-RD	Follow up (months)
Case	Sex/Age Ocular signs and symptoms	Symptom duration (months)	Location of inflammation	Sinusitis	IgG4-RD	Follow up (months)
1	Male/54 Eyelid swelling and pain (OU)	4	Diffuse	M + E	O	40
2^*	Female/35 Eyelid swelling, pain and proptosis (OD)	1	Myositis	M + E	O	12
3	Male/48 Eyelid swelling, proptosis, pain and	3	Diffuse	M	X	35
	blurred vision (OS)
4	Male/71 Proptosis and pain (OD)	12	Dacryoadenitis + Myositis	M	X	96
5	Female/59 Eyelid swelling (OS), and diplopia	1	Dacryoadenitis + Myositis	M	X	60
6	Male/40 Pain (OD)	0.50	Myositis	M	X	40
7	Male/60 Eyelid swelling, proptosis (OS) and	36	Dacryoadenitis + Myositis	M	O	27
	diplopia
8	Female/41 Eyelid swelling, proptosis, pain (OS)	0.75	Diffuse	E	X	12
	and diplopia
9	Female/19 Eyelid swelling, pain and palpable mass (OD)	0.25	Dacryoadenitis	E	X	12
10	Female/41 Pain (OS)	0.50	Myositis	M	X	20
11	Male/50 Eyelid swelling, and pain (OU)	0.25	Myositis	M + E	X	72
12	Male/51 Eyelid swelling and proptosis (OU)	3	Dacryoadenitis	M + E	X	12
13	Male/45 Eyelid swelling (OD)	0.25	Dacryoadenitis	M + E	X	11

OU = oculus unitas; OD = oculus dexter; OS = oculus sinister; M = maxillary sinusitis; E = ethmoid sinusitis; IgG4-RD = immunoglobulin G4-related disorder.

^* Illustrations for radiological and histochemical analysis of Case 2 are provided in Figure 1.

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