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Simultaneous Presentation of Silicone and Silica Granuloma

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Simultaneous Presentation of Silicone and Silica Granuloma

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Page 1: Simultaneous Presentation of Silicone and Silica Granuloma

Case Report

Dermatology 2002;205:162–165

Simultaneous Presentation of Silicone andSilica GranulomaA Case Report

Lucıa Pimentela Maria Barnadasa David Vidala Francisco Sanchob Ramon Fontarnauc

Agustın Alomara

Departments of aDermatology and bPathology, Hospital Santa Creu i Sant Pau, Universitat Autonoma de Barcelona,and cServeis Cientificotècnics, Universitat de Barcelona, España

Received: July 12, 2001Accepted: January 26, 2002

Dr. Lucıa PimentelDepartment of Dermatology, Hospital Santa Creu i Sant PauSan Antonio Ma. Claret 167E–08025 Barcelona (Spain)Fax +34 93 2919136, E-Mail [email protected]

ABCFax + 41 61 306 12 34E-Mail [email protected]

© 2002 S. Karger AG, Basel1018–8665/02/2052–0162$18.50/0

Accessible online at:www.karger.com/journals/drm

Key WordsSilicon W Silica W Silicone W Granuloma W

Scars W Scar sarcoidosis W Foreign-bodygranuloma

AbstractWe report a case of a 45-year-old womanwho presented a simultaneous foreign-body granuloma reaction to silicone in herface and to silica in the elbow and knee. Thepatient had received silicone injections inher face 7 years earlier and had suffered amotorcycle accident when she was young.Changes suggestive of silicone were ob-served in the biopsy obtained from the face,and silica was detected in the biopsy takenfrom the elbow, confirmed by polarized lightand X-ray microanalysis. The presence ofpolarizable foreign matter in cutaneous epi-thelioid granulomas should alert to the diag-nosis of sarcoidosis.

Copyright © 2002 S. Karger AG, Basel

Introduction

Silicone is used in a variety of medicalapplications. It is a derivative of silicon, thesecond most abundant element on theearth’s surface. Silicone was chosen for med-ical applications because it was consideredto be biologically inert. Significant questions

remain with regard to its chemical, physio-logic and immunologic inertness [1]. Multi-ple studies have shown that silicone poly-mers contain multiple contaminants includ-ing fumed silica, platinum, low-molecular-weight polymers and other products. Sili-cone cannot be considered an ideal materialfor medical use if it or its contaminants arenot biologically inert [2]. Since the introduc-tion of dimethicone (injectable siliconefluid), various undesirable local effects havebeen reported, and it has become obviousthat the substance is not as inert as was ini-tially anticipated [3].

Silica granuloma was first described byShattock in 1916 (cited by Mowry et al. [4])when he performed a biopsy on a lip tumorthat had arisen in a scar resulting from lacer-ations suffered 11 years previously. Hetermed the entity pseudotuberculoma silico-ticum. It usually presents as cutaneous nod-ules often associated with scars that show asarcoid granuloma with birefringent brightmaterial under polarized light [4].

We report a case of silicone and silicagranuloma that appeared simultaneously.

Case Report

A 45-year-old woman visited our derma-tology department in May 1999 with papulesand nodules on her face, around the lips, left

eye, submaxillary mentoniane furrow andchin. She had first noticed the lesions 1month earlier, and some days later, she alsonoticed plaques on her right knee and el-bow.

Her past medical history revealed an in-traductal infiltrating breast carcinoma thathad been diagnosed in 1994 and treated withmastectomy, chemotherapy and lymph nodedissection. No reconstructive breast surgerywas made. In 1996, hepatic and bone metas-tases were detected and treated with localradiotherapy and adjuvant therapy withadriamycin and taxol.

In 1993, silicone had been injected intothe soft tissues of the face for cosmetic pur-poses and tattooing had been performed onthe eyebrows in 1998. She also reported thatwhen she was young she had had a motorcy-cle accident and had suffered multiple frac-tures and lacerations on the arms and legs.

Physical examination showed multiplefirm nodules and linear papules on the face,with a normal skin surface in the periocular,peribuccal, submaxillary areas and menton-iane crease. She also had nontender, firm,reddish brown linear papules and plaquesmeasuring 2 ! 1.5 cm on the right knee withanother of 2.5 ! 1 cm on her right elbow.

Laboratory examination revealed abso-lute neutrophils 1.35 ! 109/l, platelet count121 ! 109/l with normal erythrocyte sedi-mentation rate, and a normal proteinogram

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Simultaneous Presentation of Silicone andSilica Granuloma

Dermatology 2002;205:162–165 163

Fig. 1. Clear vacuoles in the dermis and in the cytoplasm of macro-phages with a foamy aspect. HE. !400.

Fig. 2. a Giant cell with a foreign body in the cytoplasm. HE. !400.b Birefringent foreign body under polarized light in the cytoplasm ofa giant cell. HE. !400.

and renal study. Liver function showedSGOT 99 U/l (normal value, NV, !32 U/l),SGPT 171 U/l (NV !31 U/l), BT 23 Ìmol/l(NV !17 Ìmol/l), alkaline phosphatase 270U/l (NV 82–198 U/l) and GGT 94 U/l (NV7–43 U/l). Angiotensin-converting enzymewas increased 76.8 U/l (NV 20–60 U/l).Chest X-ray, Mantoux reaction, Ziehl-Niel-sen staining and culture were negative. Achest-X-ray performed 1 month previouslyhad been normal, as was a pulmonary scanperformed 4 months later. A pulmonarygammagraphic study with gallium reportedan increased captation of this substance atboth pulmonary hila which was interpretedas consistent with the diagnosis of sarcoid-osis. An ultrasonographic abdominal studyrevealed hypercaptation at the hepatic lo-bules compatible with metastatic lesionsfrom breast carcinoma.

We consider that this patient probablyhad an acute transient sarcoidosis because ofthe consistent pulmonary gammagraphicfindings and elevated angiotensin-convert-ing enzyme levels. However, a chest X-rayand a pulmonary scan performed 4 monthslater did not support this diagnosis whichmight indicate a transient evolution of theprocess.

Histopathologic Studies

A 4-mm punch biopsy was performedfrom a nodule on the face. It showed aninflammatory infiltrate composed of lym-phocytes, histiocytes and foreign-body giantcells which surrounded empty cavities of dif-

ferent sizes giving it a ‘Swiss cheese’ appear-ance in the deep dermis and in the subcuta-neous fat. Clear vacuoles were present in thecytoplasm of the giant cells (fig. 1) and mac-rophages that gave a foamy aspect to thesecells. These findings were consistent with thediagnosis of foreign-body reaction to sili-cone.

A 4-mm punch biopsy was taken from apapule on the elbow. In the papillary andreticular dermis (superficial and deep) itshowed well-circumscribed noncaseatingnaked granulomas that were composed ofhistiocytes, giant cells and scant lympho-cytes. Bright structures were observed in themiddle of these granulomas (fig. 2a) whichwere birefringent under polarized light(fig. 2b).

Scanning Electron Microscopy andX-Ray Microanalysis Studies

Five-micrometer sections of paraffin-embedded tissue from an elbow were depos-ited on the specimen holder and coated withabout 30 nm of gold in a sputter coater (Pola-ron E5000).

Observations under a scanning electronmicroscope were carried out using a JeolJSM-840 scanning electron microscope fit-ted with an energy-dispersive X-ray micro-analyzer (EDX, model AN10000, Link Sys-tems).

Using the scanning electron microscopein the backscattered electron imaging mode,it is possible to visualize tissue sections andrecognize their different structures. Back-scattered electron images, or chemical con-

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164 Dermatology 2002;205:162–165 Pimentel/Barnadas/Vidal/Sancho/Fontarnau/Alomar

Fig. 3. Elemental X-ray spectra of dense particles in the granulomafrom the elbow showing the presence of iron (Fe), aluminum (Al),silicon (Si), gold (Au), potassium (K) and calcium (Ca).

trast images, allow one to distinguish struc-tures of different density or average atomicnumber, according to the gray level theypresent.

The structure of granulomas presenteddense particles in their interior. Their ele-mental X-ray spectra showed the presence ofsilicon, aluminum, potassium, calcium andiron, suggesting they belong to clay, silicaand calcium carbonate, which are character-istic soil components (fig. 3) (The gold peaksin the spectra correspond to the specimencoating).

Discussion

We report the case of a 45-year-old wom-an who presented a simultaneous granulo-matous reaction to foreign-body materialcontaining silicon (silicone and soil) but notto other material that probably did not con-tain silicon, such as tattooing. The fact thatboth granulomatous reactions appeared atthe same time suggests that a relationshipexists between the materials detected in theface and elbow. Silicon is an element whichis present in silica and silicone and whichcan explain the link between these two gran-ulomatous reactions.

Dimethylpolysiloxane is the siliconecompound used in medicine. It is an organicsilicone polymer derived from silica (quartz)and it has applications in joint prostheses,breast implants and tissue augmentation.Soft-tissue foreign-body giant cell granulo-

mas at the site of liquid silicone injections in2 patients were first described in 1964 [5].They were subsequently reported at othersites such as the face [6], breast and penis [7].the most common histopathologic findingsare multinucleated giant histiocytes withempty cytoplasmic vacuoles containing oc-casional tiny yellow refractile spicules thatare not birefringent under polarized light [8,9].

The possible human systemic response tolocally injected dimethicone was first sug-gested by Ben-Hur and Balantyne [10] andRees et al. [11], who found silicone fluiddeposited in the spleen, liver, adrenal glandsand kidneys of mice given subcutaneous in-jections of silicone.

The pathogenesis of the silicone granulo-ma is not clear. It is known that repeatedexposure to silicone gel can induce delayedhypersensitivity [12]. In our case, the patientdid not notice the skin change during thefirst few years. The granuloma appearedabout 7 years after the injections and therelation between this and the simultaneousappearance of the silica granulomas is notclear. The patient insists that they appearedas a result of the chemotherapy treatmentthat she received, but no relationship withthis has been demonstrated. Studies in ex-perimental animals have been designed todetermine whether silicone materials affectthe status or function of the immune system.However, the host immune response to sili-cone gel or to silica remains poorly defined.In summary, the present state of knowledge

suggests that silicone gel can enter the reticu-loendothelial system. Immune responses tovarious forms of silicone may occur in a sub-set of patients. Only anecdotal evidence doc-uments adverse immune responses in pa-tients [13].

We consider that in this case silica andother elements that are usually in the soilwere probably present in the linear papulesin the scars on the elbow and knee. Thishypothesis is supported by the history of themotorcycle accident that she had sufferedwhen she was young, by the histopathologicfindings and moreover by the electron-dis-persive X-ray studies.

The demonstration of foreign bodies incutaneous granulomas is generally thoughtto exclude a diagnosis of sarcoidosis. How-ever, there have been recent reports arguingagainst this concept [14, 15]. Some investiga-tors have reported systemic sarcoidosis withcutaneous manifestations in which polariza-ble particles were associated with granulomaformation in the skin. Sarcoidosis has aknown predilection for scars and areas ofprevious trauma, where deposition of for-eign bodies would be an expected finding[16]. The foreign body may serve as an incit-ing stimulus for granuloma formation in se-lected cases of sarcoidosis. Walsh et al. [17]suggested that cutaneous silica granulomasand sarcoidosis are the same disease. Theyhypothesized that silicate particles alone areincapable of generating a cutaneous granulo-matous reaction, whereas they may do so inthe presence of sarcoidosis, explaining whycutaneous silica granulomas are rare eventhough there is frequent exposure to this sub-stance. They noted that this would also ex-plain the reported subsequent developmentof sarcoidosis in patients with silica granulo-mas.

A characteristic latency period betweenthe time of exposure to silica and the onset ofclinically evident granuloma formation hasoften been noted in published reports. Thishas ranged from 6 months to 59 years with amean interval of approximately 10 years [4,18].

Clinically, silica granulomas present aserythematous, firm, nontender, dermal orsubcutaneous nodules, often associated withscars. Cutaneous nodules may occur singlyor concurrently at multiple sites. Routinehistologic examination (HE) of biopsy speci-mens may reveal a foreign-body reactionwith epithelioid histiocytes surrounding nu-merous crystalline structures, or more orga-nized noncaseating epithelioid granulomasthat mimic cutaneous sarcoidosis as in this

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Simultaneous Presentation of Silicone andSilica Granuloma

Dermatology 2002;205:162–165 165

case [4]. It is important, therefore, to beaware of the difficulty existing to distinguishbetween isolated cutaneous silica granulomafrom silica granuloma in the context of sar-coidosis.

The pathogenesis of silica granuloma ispoorly understood. Two major theories existtoday. One, expressed by Shelley and Hurley[19], views these lesions as the result ofchemical hydrolysis of tissue deposits of sili-ca to a colloidal state that, after many yearsof chemical ‘weathering’ of silica by intersti-tial fluids, then triggers a granulomatous re-

sponse. The second theory, proposed byRank et al. [20], postulates that silica granu-lomas represent a delayed hypersensitivityresponse that occurs over time, but only insusceptible individuals. The latter theorymay be more plausible as several case re-ports, including ours, have documented theconcurrent appearance of silica granulomasat different body sites [4].

In our patient, the simultaneous appear-ance of silicone and silica granuloma whichhad been present on the skin for 7 years andfor more than 20 years, respectively, suggests

the existence of a delayed hypersensitivityreaction to silicon. A loss of the immuno-logic equilibrium between the host and for-eign bodies secondary to chemotherapymight be hypothesized.

Acknowledgement

Scanning electron microscopy and X-raymicroanalysis were performed in the ServeisCientificotècnics of the Universitat de Bar-celona.

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14 Kim YC, Triffet MK, Gibson LE: Foreign bod-ies in sarcoidosis. Am J Dermatopathol 2000;22:408–412.

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