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Case Reports

Ophthalmic Plast Reconstr Surg, Vol. 35, No. 3, 2019

FIG. 2.  Hematoxylin and eosin stains at various magnifications: (A) 200×, (B) 40×, (C) 40×, (D) 100×. The mass shows mature
adipose tissue (*), with branching capillaries (◁) and thick-walled vessels (★). Fibrosis (◆) and thrombi (■) were evident (B and D), but
no necrosis.

REFERENCES old woman with a history of systemic sarcoidosis previously


1. Carruth BP, Meyer DR. Angiolipoma of the orbit: a rare tumor in an treated with numerous immunosuppressive medications
unusual location. Ophthalmic Plast Reconstr Surg 2015;31:e142–5. who was examined for bilateral infraorbital nodules and
2. Feinfield RE, Hesse RJ, Scharfenberg JC. Orbital angiolipoma. swelling that were unresponsive to treatment. She underwent
Arch Ophthalmol 1988;106:1093–5. a bilateral anterior orbitotomy through a transconjunctival
3. Arenaz Búa J, Luáces R, Lorenzo Franco F, et al. Angiolipoma in approach with mass excision. The histologic analysis was
head and neck: report of two cases and review of the literature. Int consistent with foreign-body granulomata juxtaposed
J Oral Maxillofac Surg 2010;39:610–5.
to implantable material, specifically ArteFill, which
4. Tripathy D, Mittal R. Spindle cell lipoma of the orbit. Ophthalmic
Plast Reconstr Surg 2015;31:e53–5. was injected many years prior. There were no separate
5. Sah K, Kadam A, Sunita J, et al. Non-infiltrating angiolipoma of the noncaseating granulomas to suggest sarcoidosis as the
upper lip: a rare entity. J Oral Maxillofac Pathol 2012;16:103–6. underlying etiology. It is important to consider prior filler
6. Borrelli M, Buhlbuck D, Strehl A, et al. Leiomyolipoma of the or- injections in patients with sarcoidosis who present with
bit. Ophthalmic Plast Reconstr Surg 2012;28:e21–3. subcutaneous nodules as this changes management and may
prevent the need for more aggressive immunosuppressive
Foreign-Body Granulomata treatment.

Caused by Injected Permanent


Filler Masquerading as Cutaneous T he use of injectable soft-tissue fillers has risen dramatically
over the last few years with 2.7 million injections performed
in 2017 based on data from the American Society of Plastic
Sarcoidosis Surgeons.1 Several types of dermal fillers are commercially
Stacy M. Scofield-Kaplan, M.D.*, Sagar Y. Patel, M.D.*, available ranging from nonpermanent hyaluronic acid gel to
Andrew Mueller, M.D.*, W. Cameron Ford, B.S.*, permanent synthetic microspheres. Although injectable fillers
Bret M. Evers, M.D., Ph.D.†, R. Nick Hogan, M.D., Ph.D.*†, have an overall good safety profile, they can be associated with
and Ronald Mancini, M.D.* a variety of adverse events and complications including swell-
ing, infection, pain, nodule formation, granuloma formation,
skin necrosis, and blindness.2,3 Foreign-body granuloma forma-
Abstract: Foreign-body granuloma formation following filler
tion has been most commonly seen with permanent fillers; these
injections is most commonly seen with permanent fillers;
reactions can occur years after filler injection and may require
these reactions can occur years following the injections and
intralesional steroid injection or surgical excision.3–5 There have
often require either an intralesional steroid injection or
been a few reports of patients with systemic sarcoidosis devel-
surgical excision. The authors present a case of a 75-year-
oping subcutaneous nodules/granulomas.3,5,6 The authors report
a case of a patient with systemic sarcoidosis and significant
*Department of Ophthalmology, UT Southwestern Medical Center; and facial nodules that did not respond to numerous immunosup-
†Department of Pathology, UT Southwestern Medical Center, Dallas, Texas,
U.S.A. pressive medications aimed at treating cutaneous sarcoidosis
Accepted for publication March 13, 2019. nodules. She ultimately underwent a bilateral orbitotomy with
The authors have no financial or conflicts of interest to disclose. excisional biopsy, which revealed foreign-body granulomas
Address correspondence and reprint requests to Ronald Mancini, M.D., reactive to foreign-body material consistent with prior Artefill
Department of Ophthalmology, University of Texas Southwestern Medical
Center, 5323 Harry Hines Blvd, Dallas, TX 75390-9057. E-mail: Ronald. (Suneva Medical, San Diego, CA, U.S.A.) filler injections.
Mancini@utsouthwestern.edu This case was Health Insurance Portability and Accountability
DOI: 10.1097/IOP.0000000000001385 Act compliant with protection of any identifiable information.

e82 © 2019 The American Society of Ophthalmic Plastic and Reconstructive Surgery, Inc.

Copyright © 2019 The American Society of Ophthalmic Plastic and Reconstructive Surgery, Inc. Unauthorized reproduction of this article is prohibited.
Ophthalmic Plast Reconstr Surg, Vol. 35, No. 3, 2019 Case Reports

FIG. 1.   A 75-year-old woman with infraorbital areas of firm


mobile fullness. FIG. 2.   MRI demonstrating bilateral subcutaneous infraorbital
tissue masses (white arrows) with homogenous enhancement
after contrast. Coronal image (A) and Axial image (B) shown.
Patient consent was obtained for use of identifiable clinical
photographs. Gross examination of the excised masses revealed ho-
mogenous, firm, tan-white tissue masses. Histologic exami-
CASE PRESENTATION nation of the specimens revealed dense connective tissue with
A 75-year-old woman with history of systemic sarcoid- multiple foreign-body granulomata (Fig. 3A). Within the granu-
osis sought treatment for 10 years of swelling and subcutaneous lomata were numerous empty vacuoles (Fig. 3B), the most of
nodules in the infraorbital area (Fig. 1). This swelling persisted which were enveloped by foreign-body giant cells (Fig. 3C,D).
through multiple flare-ups of sarcoidosis and was unresponsive Notably, noncaseating granulomata of the type seen in sarcoid-
to treatment with numerous immunosuppressive medications osis were not identified in these specimens; and all the granu-
despite resolution of her other clinical manifestations of sarcoid- lomata seen in this patient were associated with the empty
osis. A positron emission tomography scan of her face revealed vacuoles consistent with foreign-body material. Once examined
bilateral infraorbital tissue masses with 18F-fluorodeoxyglucose forpathologic diagnoses, the patient recalled receiving ArteFill
avidity and dimensions of approximately 1.5 × 0.7 cm on the injections many years prior and also noted having had chin and
right and 2.5 × 0.8 cm on the left. breast implants removed due to the development of granulom-
On examination, her visual acuity, visual fields to con- atous reactions to the implants. The histologic evidence, along
frontation, extraocular movements, and pupils were normal. with the resistance of the bilateral infraorbital masses to sar-
There were firm palpable masses in the medial aspect of the coidosis treatment, led to the diagnosis of foreign-body granu-
cheek and lateral nose with extension up to the tear trough on lomata rather than an exacerbation of sarcoidosis.
each side and small masses below the corner of the mouth on
each side. The slit-lamp examination and fundoscopic exami- DISCUSSION
nation were otherwise unremarkable. She denied any history of Complications from permanent filler injections have been
filler injections. well documented in the literature.2,3 Delayed-onset complica-
An MRI of the face revealed bilateral subcutaneous tions are broad, but the more commonly encountered pathologic
infraorbital tissue masses with homogenous enhancement after findings include noninflammatory or low-grade inflammatory
contrast of similar size and location as the prior positron emis- nodules.3,4 Oftentimes, the diagnosis is obvious with few diag-
sion tomography scan (Fig. 2). The patient underwent a bilateral nostic alternatives, but a large delay in presentation following
anterior orbitotomy through a transconjunctival approach with filler injections may mask the underlying diagnosis especially
excision of the subcutaneous nodules in an effort to relieve the when the patient either denies or neglects to mention prior filler
swelling and achieve a definitive diagnosis. injections. Furthermore, in patients with a known autoimmune

FIG. 3.   Hematoxylin and eosin (H&E)–stained sections of the infraorbital masses. These masses demonstrate dense connective tissue
(white asterisks in A) with multiple foreign-body granulomata (black asterisks in A). These granulomata contained numerous empty
vacuoles (B), the most of which were enveloped by foreign-body giant cells (white arrowheads in C;higher magnification in D). Scale
bars = 1.0 mm (A) and 100 μm (B–D).

© 2019 The American Society of Ophthalmic Plastic and Reconstructive Surgery, Inc. e83
Copyright © 2019 The American Society of Ophthalmic Plastic and Reconstructive Surgery, Inc. Unauthorized reproduction of this article is prohibited.
Case Reports Ophthalmic Plast Reconstr Surg, Vol. 35, No. 3, 2019

or inflammatory condition, these nodular reactions may go fillers (especially those with sarcoidosis) should be educated on
misdiagnosed or may be attributed to part of a patient’s sys- the potential for scarring, tissue damage, and need for possible
temic disease rather than a separate process. In fact, patients surgical interventions due to the foreign-body granuloma for-
with certain granulomatous diseases, such as sarcoidosis, may mation. Furthermore, it is imperative to consider foreign-body
have an increased propensity for these types of reactions.3 It has granuloma in the differential diagnosis of tissue masses unre-
been proposed previously that any foreign body inoculated from sponsive to initial immunosuppressive treatments, as this may
minor trauma may serve as a nidus for granulomatous inflam- obviate the need for patients to be treated with immunosuppres-
mation in patients with sarcoidosis, although only one of these sive medications that carry a vast array of side effects including
cases involved a nodule on the face.6 In patients with sarcoid- potentially life-threatening infections.
osis with implantable fillers, there can be mixed inflammatory
lesions, where sarcoidosis and foreign-body granulomas can be REFERENCES
found concurrently within the same region of inflammation.5
1. American Society of Plastic Surgeons. 2017 Plastic surgery statis-
This case further supports the theory that patients with tics report. Available at: https://www.plasticsurgery.org/documents/
sarcoidosis are more prone to developing foreign-body reac- News/Statistics/2017/top-five-cosmetic-plastic-surgery-proce-
tions from implantable materials. Not only did she have foreign- dures-2017.pdf. Accessed February 1, 2019.
body granuloma formation following permanent dermal filler 2. Rayess HM, Svider PF, Hanba C, et al. A cross-sectional analysis
but she also had a history of chin and breast implants requiring of adverse events and litigation for injectable fillers. JAMA Facial
removal and subsequent surgical excision of the foreign-body Plast Surg 2018;20:207–214.
granulomatous tissue. Our patient is unique in that her facial 3. Daines SM, Williams EF. Complications associated with injectable
granulomatous lesions were treated as a manifestation of sys- soft-tissue fillers: a 5-year retrospective review. JAMA Facial Plast
temic sarcoidosis with years of numerous immunosuppressive Surg 2013;15:226–231.
medications without any improvement. The underlying correct 4. Kadouch JA, Kadouch DJ, Fortuin S, et al. Delayed-onset compli-
diagnosis only became apparent following surgical excision and cations of facial soft tissue augmentation with permanent fillers in
85 patients. Dermatol Surg 2013;39:1474–1485.
the patient then recalling prior filler use.
5. Sidwell RU, McL Johnson N, Francis N, et al. Cutaneous sar-
Although the use of permanent implantable materials is coidal granulomas developing after Artecoll facial cosmetic filler
becoming more frequent, this case illustrates the growing neces- in a patient with newly diagnosed systemic sarcoidosis. Clin Exp
sity of increased awareness of inflammatory responses to inject- Dermatol 2006;31:208–211.
able materials and the importance of asking any potential filler 6. Marcoval J, Mañá J, Moreno A, et al. Foreign bodies in granulom-
patient if they have a condition like sarcoidosis.1 Physicians and atous cutaneous lesions of patients with systemic sarcoidosis. Arch
all patients who are being evaluated for permanent implantable Dermatol 2001;137:427–430.

e84 © 2019 The American Society of Ophthalmic Plastic and Reconstructive Surgery, Inc.

Copyright © 2019 The American Society of Ophthalmic Plastic and Reconstructive Surgery, Inc. Unauthorized reproduction of this article is prohibited.

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