British fournal of Dermatoiogy (19*)]) 125, 183-185.
ADONIS 0007096391001926
Recurrent facial oedema associated with a silicone-rubber implant MARION I.WHITE. LOUISE M.SMART,* DIANA M.MACGREGOR. C.R.W.RAYNERt AND F.W.SMITHt * Department of Patholofiy. ilniversitii of Aberdeen. Aberdeen. U.K. IX'ifartmenls of l^krinalohgif. t Plastic Surgerii and % Nuclear Medicine. Aberdeen Royal Infirmary. Aberdeen Accepted for publication 1 i February 1991
Summary
A 40-year-oId man presented with facial oedema that resulted from an inflammatory reaction to a silicone-rubber implant inserted 16 years previously for an augmentation rhinoplasty.
Case report A 40-year-oId man presented with a history of intermittent periorbitai oedema over the preceding 3 years. At the onset he had had a severe upper respiratory tract infection and had been treated with systemic antibiotics. Initially both eyes became intermittently uncomfortable, but gradually there was a worsening of his periorbitai oedema which was always more marked on wakening. The condition was specifically aggravated by stooping or entering a hot atmosphere and he was also aware that the flushing became more severe after hot drinks, alcoholic beverages and spicy foods. He had received systemic antihistamine treatment without benefit and both specialist ophthalmological and ENT examinations were normal. He gave a history of acne vulgaris as a teenager. During childhood, he had injured his nose at football and at the age of 1(S years had undergone a submucous resection of part of the nasal septum, followed 5 months later by an augmentation rhinoplasty using a siliconerubber implant to correct bridge-line depression. On examination he had obvious oedema of his face most prominently in his cheeks and forehead. There was telangiectasia and evidence of previous acne scarring. There was no lymphadenopathy (Fig. 1). Investigations The following investigations were normal: full blood count. ESR. ASO titre. serum IgE and RAST: chest X-ray, sinus X-ray, and CT scan of skull: antinuclear factor; Corrt-spondence: Ur Marion I.White, Ward 29. Foresterhill. Aberdeen Royal Inlirmary. Aberdeen AB9 2ZB, U.K.
Figure 1. Oedema of the face.
creatinine kinase: FHaria complement fixation test: TrichineUa CIE; 99 mTc, HM-PAO labelled white-cell scan of nasal passages or paranasal sinuses: and urinary porphyrins and 5-hydroxyindoIeacetic acid. 183
184
M.r.WHITE et al
Discussion
Figure 2. Transverse calculated T1 magnetic resonante image through both orbits demonstrating bilateral periorbital i>edema (white) lying in front of the eyes in the medial aspect of the eyelids and marked anterior nasal mucosal thickening and oedema. No ocular or orbital abnormality is seen.
A skin biopsy of the cheek revealed alcianophilic solar elastosis. a perivascular chronic inflammatory infiltrate and upper dermal oedema. A nasal swab was positive for Staphylococcus aureus. Nuclear magnetic resonance imaging was performed and the images were made using an 0.08 Tesla. resistive MR iraager designed and built at theUniversity of Aberdeen' with a binocular surface coll specifically designed for imaging the orbits.- Bilateral periorbilal oedema was revealed with marked anterior nasal mucosal thickening and oedema {Fig. 2). Treatment and progress
Systemic antihistamine therapy and an 8-week course of tetracycline (1 g per day) were ineffective. Oral prednisolone temporarily reduced the swelling but only at a dose >12-5 mg/day. Minocycline at a dose of 100 mg/day was commenced but discontintied ufter b weeks because it appeared to provoke a borderline photosensitivity at 400 nm. Oral isotretinoin 0-5 mg/kg was prescribed for 3 months without benefit. U was then decided to remove the implant. An incision was made vertically through the columella of the nose which was scarred. The strut of the hand-carved implant was removed and the cavity biopsied. The swab from this area also yielded a scanty growth of S. aureus on culture. On histologicai examination it was noted that there was a mild, predominantly lymphocytic. inflammatory giant cell reaction in relation to amorphous granular material. Post-operatively there was no problem of infection. In the 2 years since his operation the oedema has not totally resolved but his condition is less troublesome.
This was an unusual presentation of facial oedema. The differential diagnoses considered initially were solid facial oedema and lymphoedematous rosacea. Solid facial oedema has been noted as a complication of acne vulgaris,^ usually occurring at least 4 years after the onset of the acne. The pathogenesis is so far unknown although it has been speculated that the chronic cutaneous infiammation from acne can produce lymphatic damage and subsequent progressive oedema of the face. Improvement after treatment with isotretinoin and clofazimine, both of which are known to stimulate macrophage function and phagocytosis, suggests an immunological response to the mediators of acne.'' It has been reported that solid facial oedema in acne does not respond to antibiotics, but does respond to steroids in doses in excess of 15 mg/day.^ Our patient did not respond to tetracycline and minocycline but was improved temporarily with oral prednisolone. Similar inflammation, oedema and pilosebaceous changes occur in rare cases of rosacea.'' Oedema of the face in rosacea may be similar to the acne/solid oedema syndrome but responds to antibiotic treatment. The possible role of the prosthetic graft material in the aetiology of the facial oedema was considered. The adherence of bacteria to prosthetic graft material is thought to play an important role in the ultimate development of infections. It has been demonstrated that coaguiase negative Staphnhioici may be pathogenic in this situation/ Both Escherkhia coli and S. aureus have been demonstrated to adhere to silicone. Proplast 11. and Goretex.** Interestingly, in this study, the adherence of S. aureus was at a faster rate than E. coli, and the bacterial attachment continued to increase through the incubation time. This heips to explain the foreign-body effect of increased susceptibility to infection of foreign materials. Our patient had S. aureus isolated from nasal swabs and frotn the biopsy of the cavity after the retnoval of the silicone implant. Long-term problems after insertion of silicone breast and joint prostheses have been recognized. Siiicone lymphadenopathy has been noted in association with silicone mastitis**'" and after insertion of joint prostheses.^'"" Silicone is now a wcll-rccognized inducer of localized granulomatous inflammation but there are less common associations with more complex clinicopathological entities. Three patients developed unilateral proptosis 13-20 years after repair of orbital lloor fractures with teflon implants.'"* A woman who had subcutaneous liquid silicone injections in the forehead
RECURRENT FACIAL OEDEMA
for cosmetic reasons developed an acute inflammation of the eyelids and orbits 20 years later, which settled over 2-5 months with topical application of cortisone and heparin.' ^ There is evidence of a death from a giant cell myocarditis associated with silicone rubber in dialysis tubing."' A silicone-induced foreign-body reaction with dental prostheses has also been reported.'" Both scleroderma"^ and human adjuvant disease'**'^" have been noted after silicone augmentation mammoplasty. There are reports in which removal of the implanted prostheses led to some clinical improvement.'** The mechanisms by which silicone can induce tissue damage have been reviewed.-^'••^Our patient was assumed to have lymphoedematous rosacea. and solid facial oedema was also considered because of his resemblance to patients previously described.-' There is. however, no doubt that the siliconerubber implant was a crucial factor. The presence of local infection and the probability of migration of silicone particles causing a localized granulomatous inflammation, would appear to have resulted in his facial oedema. Modern radiological and bioengineering tests did not disclose this problem. Foreign-body implants should be added to the differential diagnosis of recurrent facial oedema in dermatological textbooks.
References 1 Redpiith TW. Hutchison JMS. Eastwood LM el al. A low-field NMR imager for clinical use. fournal of Physics E: Scientific Instruments 1987; 20: 1228-J4. 2 Deans HK. Redpath TW. Smith FW et al. Magnetic resonance Imaging of the orbits using a binocular surface coil. Br / Radhl 1988:61:665-72. ? Connelly Mtl. Winkeimann RK, Solid facial edema as a complicatiem of ucne vulgaris. Arch Dernuitol 1985: 121: H7-90. 4 Helander I. Aho ll|. Solid facial edema as a complication of acne vulgaris: treatment with isotretinoin and clofazimine. Acta Demi Vviwreol IStofkh) 1987; 67: 5 55-7. 5 Camacho-Martinen I', Winkeimann RK. Solid facial edema as a manifestation of acne. } Am Acad Dermatol 1990: 22: 129-30.
185
6 Marks R. Wilkinson DS. Rosacea and peri-oral dermatitis. In: Textbook of Derminoloftij. (Rook A. Wilkinson DS. Ebling FIG. Champion RII, Burton IR. eds). 4th edn. 1986: 1605-10. 7 Wong PTL. Nicld DV. Kh(K> CTK. The pathogenicity of roagulase negative staphylotoccus in the presence of silicone rubber implants. Br } Plast Surti 1987: 40: 94-6. 8 Brewer AR. Stromberg BV. In vitro adherence of bacteria to prosthetic grafting materials. Ann Plant Surg 1990; 24: 1 34-8. 9 Symmers WSC. Silicone mastitis in 'topless' waitresses and some other variations of foreign body mastitis. B Med j 1968; 3: 19-22. 10 Tniong LD. Cartwright 1. (loodman MD. Woznicki D. Siticone lymphcdenopathy asst>ciated with augmentation mammoplasty. Am ] Surg Pathol 1988: 12:484-91. 11 Christie A|. Weinberger RA. Dietrich M. Silicone lymphadenopathy and synovitis. Complications of silicone elastomer finger joint prostheses. JAMA 1977: 237: 1463-4. 12 lasin KA. Weerasinghe BD. Silicone lymphadenopathy. synovitis and osteitis complicating big toe silastic prosthesis. / R Col! Surg Edin 1986; 32:29-33. 1 3 Rogers LA, Longtine |A. Garnick MB. Pinkus GS. Silicone lymphadenopathy in a long distance runner: complications of a silastic prosthesis. Hum Pathol 1988: 19: 12 57-9. 14 Mauriello | A. Flanagan JC. Peyster RG. An unusual late complication of orbital floor fracture repair. Op/ilha/mofogH 1984:91: 1027. 15 Hennekes R. Garus HJ. I^te orbital complications following subcutaneous silicone injection. I'ortschr Ophthalmol 1989: 86: 249-50. 16 Kossovsky N. Cole P. Zackson DA. Giant cell myocarditis associated with silicone. An unusual case of biomaterials pathology discovered at autopsy using X-ray energy spectroscopic techniques. Am / C/in PfllM 1990: 93: 14«-52. 17 Acton C. Hoffman G. McKenna H. Moloney K. Silicone-induced foreign-body reaction after tempomandibular joint arthroplasty. Aust Dent I 1989; 34: 228-J2. 18 Spiera H. Scleroderma after silicone augmentation mammoplasty. lAMA 1988:260: 216-8. 19 Brozena SJ. Fenske NA, Cruse L'W et al. Human adjuvant disease following augmentation mammopiasty. An?T Oermafd/ 1988: 124: 1383-6. 20 Varga |, Schumacher HR. [imenez SA. Systemic sclerosis after augmentation mammoplasty with silicone implants. Am ItHern M P ( / 1 9 8 9 : 111: 177-8i.
21 Travis WD, Balogh K. Abraham IL. Silicone granulomas: Report of three cases and review of the literature. Human Piithol 1985: 16: 19-27. 22 Meigel W. Winzer M. Berg A. Wolff HH. Silikonom. Z. Hautkr 1989; 64:815-16.
ADONIS 0007096391001926
Recurrent facial oedema associated with a silicone-rubber implant MARION I.WHITE. LOUISE M.SMART,* DIANA M.MACGREGOR. C.R.W.RAYNERt AND F.W.SMITHt * Department of Patholofiy. ilniversitii of Aberdeen. Aberdeen. U.K. IX'ifartmenls of l^krinalohgif. t Plastic Surgerii and % Nuclear Medicine. Aberdeen Royal Infirmary. Aberdeen Accepted for publication 1 i February 1991
Summary
A 40-year-oId man presented with facial oedema that resulted from an inflammatory reaction to a silicone-rubber implant inserted 16 years previously for an augmentation rhinoplasty.
Case report A 40-year-oId man presented with a history of intermittent periorbitai oedema over the preceding 3 years. At the onset he had had a severe upper respiratory tract infection and had been treated with systemic antibiotics. Initially both eyes became intermittently uncomfortable, but gradually there was a worsening of his periorbitai oedema which was always more marked on wakening. The condition was specifically aggravated by stooping or entering a hot atmosphere and he was also aware that the flushing became more severe after hot drinks, alcoholic beverages and spicy foods. He had received systemic antihistamine treatment without benefit and both specialist ophthalmological and ENT examinations were normal. He gave a history of acne vulgaris as a teenager. During childhood, he had injured his nose at football and at the age of 1(S years had undergone a submucous resection of part of the nasal septum, followed 5 months later by an augmentation rhinoplasty using a siliconerubber implant to correct bridge-line depression. On examination he had obvious oedema of his face most prominently in his cheeks and forehead. There was telangiectasia and evidence of previous acne scarring. There was no lymphadenopathy (Fig. 1). Investigations The following investigations were normal: full blood count. ESR. ASO titre. serum IgE and RAST: chest X-ray, sinus X-ray, and CT scan of skull: antinuclear factor; Corrt-spondence: Ur Marion I.White, Ward 29. Foresterhill. Aberdeen Royal Inlirmary. Aberdeen AB9 2ZB, U.K.
Figure 1. Oedema of the face.
creatinine kinase: FHaria complement fixation test: TrichineUa CIE; 99 mTc, HM-PAO labelled white-cell scan of nasal passages or paranasal sinuses: and urinary porphyrins and 5-hydroxyindoIeacetic acid. 183
184
M.r.WHITE et al
Discussion
Figure 2. Transverse calculated T1 magnetic resonante image through both orbits demonstrating bilateral periorbital i>edema (white) lying in front of the eyes in the medial aspect of the eyelids and marked anterior nasal mucosal thickening and oedema. No ocular or orbital abnormality is seen.
A skin biopsy of the cheek revealed alcianophilic solar elastosis. a perivascular chronic inflammatory infiltrate and upper dermal oedema. A nasal swab was positive for Staphylococcus aureus. Nuclear magnetic resonance imaging was performed and the images were made using an 0.08 Tesla. resistive MR iraager designed and built at theUniversity of Aberdeen' with a binocular surface coll specifically designed for imaging the orbits.- Bilateral periorbilal oedema was revealed with marked anterior nasal mucosal thickening and oedema {Fig. 2). Treatment and progress
Systemic antihistamine therapy and an 8-week course of tetracycline (1 g per day) were ineffective. Oral prednisolone temporarily reduced the swelling but only at a dose >12-5 mg/day. Minocycline at a dose of 100 mg/day was commenced but discontintied ufter b weeks because it appeared to provoke a borderline photosensitivity at 400 nm. Oral isotretinoin 0-5 mg/kg was prescribed for 3 months without benefit. U was then decided to remove the implant. An incision was made vertically through the columella of the nose which was scarred. The strut of the hand-carved implant was removed and the cavity biopsied. The swab from this area also yielded a scanty growth of S. aureus on culture. On histologicai examination it was noted that there was a mild, predominantly lymphocytic. inflammatory giant cell reaction in relation to amorphous granular material. Post-operatively there was no problem of infection. In the 2 years since his operation the oedema has not totally resolved but his condition is less troublesome.
This was an unusual presentation of facial oedema. The differential diagnoses considered initially were solid facial oedema and lymphoedematous rosacea. Solid facial oedema has been noted as a complication of acne vulgaris,^ usually occurring at least 4 years after the onset of the acne. The pathogenesis is so far unknown although it has been speculated that the chronic cutaneous infiammation from acne can produce lymphatic damage and subsequent progressive oedema of the face. Improvement after treatment with isotretinoin and clofazimine, both of which are known to stimulate macrophage function and phagocytosis, suggests an immunological response to the mediators of acne.'' It has been reported that solid facial oedema in acne does not respond to antibiotics, but does respond to steroids in doses in excess of 15 mg/day.^ Our patient did not respond to tetracycline and minocycline but was improved temporarily with oral prednisolone. Similar inflammation, oedema and pilosebaceous changes occur in rare cases of rosacea.'' Oedema of the face in rosacea may be similar to the acne/solid oedema syndrome but responds to antibiotic treatment. The possible role of the prosthetic graft material in the aetiology of the facial oedema was considered. The adherence of bacteria to prosthetic graft material is thought to play an important role in the ultimate development of infections. It has been demonstrated that coaguiase negative Staphnhioici may be pathogenic in this situation/ Both Escherkhia coli and S. aureus have been demonstrated to adhere to silicone. Proplast 11. and Goretex.** Interestingly, in this study, the adherence of S. aureus was at a faster rate than E. coli, and the bacterial attachment continued to increase through the incubation time. This heips to explain the foreign-body effect of increased susceptibility to infection of foreign materials. Our patient had S. aureus isolated from nasal swabs and frotn the biopsy of the cavity after the retnoval of the silicone implant. Long-term problems after insertion of silicone breast and joint prostheses have been recognized. Siiicone lymphadenopathy has been noted in association with silicone mastitis**'" and after insertion of joint prostheses.^'"" Silicone is now a wcll-rccognized inducer of localized granulomatous inflammation but there are less common associations with more complex clinicopathological entities. Three patients developed unilateral proptosis 13-20 years after repair of orbital lloor fractures with teflon implants.'"* A woman who had subcutaneous liquid silicone injections in the forehead
RECURRENT FACIAL OEDEMA
for cosmetic reasons developed an acute inflammation of the eyelids and orbits 20 years later, which settled over 2-5 months with topical application of cortisone and heparin.' ^ There is evidence of a death from a giant cell myocarditis associated with silicone rubber in dialysis tubing."' A silicone-induced foreign-body reaction with dental prostheses has also been reported.'" Both scleroderma"^ and human adjuvant disease'**'^" have been noted after silicone augmentation mammoplasty. There are reports in which removal of the implanted prostheses led to some clinical improvement.'** The mechanisms by which silicone can induce tissue damage have been reviewed.-^'••^Our patient was assumed to have lymphoedematous rosacea. and solid facial oedema was also considered because of his resemblance to patients previously described.-' There is. however, no doubt that the siliconerubber implant was a crucial factor. The presence of local infection and the probability of migration of silicone particles causing a localized granulomatous inflammation, would appear to have resulted in his facial oedema. Modern radiological and bioengineering tests did not disclose this problem. Foreign-body implants should be added to the differential diagnosis of recurrent facial oedema in dermatological textbooks.
References 1 Redpiith TW. Hutchison JMS. Eastwood LM el al. A low-field NMR imager for clinical use. fournal of Physics E: Scientific Instruments 1987; 20: 1228-J4. 2 Deans HK. Redpath TW. Smith FW et al. Magnetic resonance Imaging of the orbits using a binocular surface coil. Br / Radhl 1988:61:665-72. ? Connelly Mtl. Winkeimann RK, Solid facial edema as a complicatiem of ucne vulgaris. Arch Dernuitol 1985: 121: H7-90. 4 Helander I. Aho ll|. Solid facial edema as a complication of acne vulgaris: treatment with isotretinoin and clofazimine. Acta Demi Vviwreol IStofkh) 1987; 67: 5 55-7. 5 Camacho-Martinen I', Winkeimann RK. Solid facial edema as a manifestation of acne. } Am Acad Dermatol 1990: 22: 129-30.
185
6 Marks R. Wilkinson DS. Rosacea and peri-oral dermatitis. In: Textbook of Derminoloftij. (Rook A. Wilkinson DS. Ebling FIG. Champion RII, Burton IR. eds). 4th edn. 1986: 1605-10. 7 Wong PTL. Nicld DV. Kh(K> CTK. The pathogenicity of roagulase negative staphylotoccus in the presence of silicone rubber implants. Br } Plast Surti 1987: 40: 94-6. 8 Brewer AR. Stromberg BV. In vitro adherence of bacteria to prosthetic grafting materials. Ann Plant Surg 1990; 24: 1 34-8. 9 Symmers WSC. Silicone mastitis in 'topless' waitresses and some other variations of foreign body mastitis. B Med j 1968; 3: 19-22. 10 Tniong LD. Cartwright 1. (loodman MD. Woznicki D. Siticone lymphcdenopathy asst>ciated with augmentation mammoplasty. Am ] Surg Pathol 1988: 12:484-91. 11 Christie A|. Weinberger RA. Dietrich M. Silicone lymphadenopathy and synovitis. Complications of silicone elastomer finger joint prostheses. JAMA 1977: 237: 1463-4. 12 lasin KA. Weerasinghe BD. Silicone lymphadenopathy. synovitis and osteitis complicating big toe silastic prosthesis. / R Col! Surg Edin 1986; 32:29-33. 1 3 Rogers LA, Longtine |A. Garnick MB. Pinkus GS. Silicone lymphadenopathy in a long distance runner: complications of a silastic prosthesis. Hum Pathol 1988: 19: 12 57-9. 14 Mauriello | A. Flanagan JC. Peyster RG. An unusual late complication of orbital floor fracture repair. Op/ilha/mofogH 1984:91: 1027. 15 Hennekes R. Garus HJ. I^te orbital complications following subcutaneous silicone injection. I'ortschr Ophthalmol 1989: 86: 249-50. 16 Kossovsky N. Cole P. Zackson DA. Giant cell myocarditis associated with silicone. An unusual case of biomaterials pathology discovered at autopsy using X-ray energy spectroscopic techniques. Am / C/in PfllM 1990: 93: 14«-52. 17 Acton C. Hoffman G. McKenna H. Moloney K. Silicone-induced foreign-body reaction after tempomandibular joint arthroplasty. Aust Dent I 1989; 34: 228-J2. 18 Spiera H. Scleroderma after silicone augmentation mammoplasty. lAMA 1988:260: 216-8. 19 Brozena SJ. Fenske NA, Cruse L'W et al. Human adjuvant disease following augmentation mammopiasty. An?T Oermafd/ 1988: 124: 1383-6. 20 Varga |, Schumacher HR. [imenez SA. Systemic sclerosis after augmentation mammoplasty with silicone implants. Am ItHern M P ( / 1 9 8 9 : 111: 177-8i.
21 Travis WD, Balogh K. Abraham IL. Silicone granulomas: Report of three cases and review of the literature. Human Piithol 1985: 16: 19-27. 22 Meigel W. Winzer M. Berg A. Wolff HH. Silikonom. Z. Hautkr 1989; 64:815-16.
