The role of infliximab in the treatment of superficial granulomatous pyoderma of the head and neck
Authors: Omer Ibrahim, Christopher G Bunick, Bhaskar Srivastava, Rossitza Lazova, Christine J Ko, Kalman L Watsky
Journal: Journal of the American Academy of Dermatology (2014)

To the Editor: Superficial granulomatous pyoderma (SGP) is a rare, chronic inflammatory disorder.1Ormaechea-Perez N. Lopez-Pestana A. Lobo-Moran C. Tuneu-Valls A. Superficial granulomatous pyoderma. Report of 2 cases treated with topical tacrolimus.Actas Dermosifiliogr. 2013; 104: 721-724Crossref PubMed Scopus (6) Google Scholar Although considered a superficial, vegetative variant of pyoderma gangrenosum (PG), SGP exhibits important differences (Fig 1, A).1Ormaechea-Perez N. Lopez-Pestana A. Lobo-Moran C. Tuneu-Valls A. Superficial granulomatous pyoderma. Report of 2 cases treated with topical tacrolimus.Actas Dermosifiliogr. 2013; 104: 721-724Crossref PubMed Scopus (6) Google Scholar, 2Winkelmann R.K. Wilson-Jones E. Gibson L.E. Quimby S.R. Histopathologic features of superficial granulomatous pyoderma.J Dermatol. 1989; 16: 127-132Crossref PubMed Scopus (25) Google Scholar Moreover, SGP responds favorably to therapy, except in cases of SGP involving the head and neck.1Ormaechea-Perez N. Lopez-Pestana A. Lobo-Moran C. Tuneu-Valls A. Superficial granulomatous pyoderma. Report of 2 cases treated with topical tacrolimus.Actas Dermosifiliogr. 2013; 104: 721-724Crossref PubMed Scopus (6) Google Scholar We present a patient with SGP on the face, neck, and trunk treated with combined infliximab and topical tacrolimus, and review all systemic therapies reported, to our knowledge, in the treatment of SGP of the head and neck. An 83-year-old Caucasian man with extensive history of cardiovascular disease presented for nonhealing ulcers of more than 1 year's duration on the right temple, right postauricular neck, and central chest (Fig 1, B). Each lesion evolved after a surgical procedure as slowly growing ulcerative plaques for which multiple therapies were attempted but unsuccessful, including surgical debridement, skin grafting, and hyperbaric oxygen. The patient was on oral prednisone 20 mg daily upon presentation in early November 2010 and had failed several oral antibiotics. Prior biopsies of the right temple and chest were read by an outside hospital as “ulcerative skin with underlying acute and chronic inflammation.” Our dermatopathology review of those biopsies demonstrated fragments of cystic spaces in the dermis lined by well-differentiated squamous epithelium and the presence of zonation, a pathologic hallmark of SGP, with neutrophils and necrosis surrounded by granulomatous inflammation rimmed by plasma cells (Fig 1, C). Diagnosed with SGP, the patient, starting in November 2010, experienced partial relief with continued oral prednisone 20 mg daily, oral tetracycline 500 mg twice daily, and topical clobetasol 0.05% ointment (changed to tacrolimus 0.1% ointment twice daily after 1 month) and intralesional Kenalog (10 mg/mL) to some areas. He began monthly infliximab infusions (5 mg/kg; 2 loading doses 2 weeks apart) as a steroid-sparing agent in January 2011 (without concomitant oral antibiotic) and tapered off prednisone by May 2011. He experienced complete resolution of the ulcers over 3 months (Fig 1, D). Infliximab, as well as tacrolimus, was discontinued in September 2011 after 10 infusions. He has remained in complete remission since. Multiple treatments for SGP have been used: topical and systemic corticosteroids, topical tacrolimus, tetracyclines, dapsone, cyclosporine, infliximab, and intravenous immunoglobulin.1Ormaechea-Perez N. Lopez-Pestana A. Lobo-Moran C. Tuneu-Valls A. Superficial granulomatous pyoderma. Report of 2 cases treated with topical tacrolimus.Actas Dermosifiliogr. 2013; 104: 721-724Crossref PubMed Scopus (6) Google Scholar Systemic treatments reported in the management of SGP of the face, the location most difficult to treat, are characterized in Table I; 81% of patients (9 of 11) had systemic glucocorticoids in their regimen, and 63% (7 of 11) required combination systemic therapy, usually with cyclosporine. Only 3 patients, including the present case, had used anti−tumor necrosis factor therapy3Akhras V. Sarkany R. Walsh S. Hyde N. Marsden R.A. Superficial granulomatous pyoderma treated preoperatively with infliximab.Clin Exp Dermatol. 2009; 34: e183-e185Crossref PubMed Scopus (20) Google Scholar, 4Marzano A.V. Tourlaki A. Alessi E. Caputo R. Widespread idiopathic pyoderma gangrenosum evolved from ulcerative to vegetative type: a 10-year history with a recent response to infliximab.Clin Exp Dermatol. 2008; 33: 156-159Crossref PubMed Scopus (36) Google Scholar; our patient ultimately responded to systemic infliximab combined with topical tacrolimus. The approach of combining systemic therapy with topical therapy was used in approximately 50% of the cases (6 of 11). Overall, the time to complete response using these different therapies for SGP of the face ranged from 2 to 8 months. SGP must be considered in the differential diagnosis of a chronic nonhealing wound that lacks an infectious or systemic etiology. Prompt recognition of SGP enables early treatment and the opportunity to avoid permanent disfigurement.5D'Epiro S, Salvi M, Mattozzi C, Giancristoforo S, Faina V, Macaluso L, et al. Facial superficial granulomatous pyoderma. Int Wound J doi: http://dx.doi.org/10.1111/iwj.12195. Published online November 28, 2013.Google ScholarTable ISystemic and topical therapies reported in the management of superficial granulomatous pyoderma of the facePatient agePatient sexTreatmentResponseSource83MGcort + TCN∗Topical clobetasol 0.05% ointment, intralesional triamcinolone, or topical tacrolimus 0.1% ointment was administered concomitantly.Ifx†Topical tacrolimus 0.1% ointment was administered concomitantly.PR, 2-3 mCR, 3 mPresent case16FCy†Topical tacrolimus 0.1% ointment was administered concomitantly.CR, 4 mD'Epiro S, et al.5D'Epiro S, Salvi M, Mattozzi C, Giancristoforo S, Faina V, Macaluso L, et al. Facial superficial granulomatous pyoderma. Int Wound J doi: http://dx.doi.org/10.1111/iwj.12195. Published online November 28, 2013.Google Scholar37FGcort‡Topical mupirocin 2% ointment was administered concomitantly.PR, 1 mPersing SM, Laub D. Eplasty 2012;12:e56.71FGcort + IfxCR, 3 mAkhras V, et al.3Akhras V. Sarkany R. Walsh S. Hyde N. Marsden R.A. Superficial granulomatous pyoderma treated preoperatively with infliximab.Clin Exp Dermatol. 2009; 34: e183-e185Crossref PubMed Scopus (20) Google Scholar65MGcort + CyCR, 5 mCheung ST, et al. Acta Derm Venereol 2006;86:362-4.85MGcort + IVIG§Topical clobetasol 0.05% ointment or topical tacrolimus 0.1% ointment was administered concomitantly.CR, 3 mDobson CM, et al. J Am Acad Dermatol 2003;48:456-60.44FCyHydrocortisone-17-butyrate and an ointment containing 3% salicylic acid and 4% boric acid were applied concomitantly.CR, 8 mLachapelle JM, et al. Dermatology 2001;202:155-7.29MGcort + CyPR, 3 mWildfeuer T, Albrecht G. Hautarzt 1999;50:217-20.79MGcortIntralesional triamcinolone was administered concomitantly.PR, 1 mMurata J, et al. Clin Exp Dermatol 2006;31:74-6.44FDapsone + GcortCR, 6 mPeretz E, et al. Int J Dermatol 1999;38:703-6.34MGcort + Cy, IfxGcort + Cy were administered separately from Ifx.CR, 2-4 mMarzano AV, et al.4Marzano A.V. Tourlaki A. Alessi E. Caputo R. Widespread idiopathic pyoderma gangrenosum evolved from ulcerative to vegetative type: a 10-year history with a recent response to infliximab.Clin Exp Dermatol. 2008; 33: 156-159Crossref PubMed Scopus (36) Google ScholarCR, Complete response; Cy, cyclosporine; Gcort, glucocorticoids; Ifx, infliximab; IVIG, intravenous immunoglobulin; m, months; PR, partial response; TCN, tetracycline.∗ Topical clobetasol 0.05% ointment, intralesional triamcinolone, or topical tacrolimus 0.1% ointment was administered concomitantly.† Topical tacrolimus 0.1% ointment was administered concomitantly.‡ Topical mupirocin 2% ointment was administered concomitantly.§ Topical clobetasol 0.05% ointment or topical tacrolimus 0.1% ointment was administered concomitantly.∗∗ Hydrocortisone-17-butyrate and an ointment containing 3% salicylic acid and 4% boric acid were applied concomitantly.†† Intralesional triamcinolone was administered concomitantly.‡‡ Gcort + Cy were administered separately from Ifx. Open table in a new tab CR, Complete response; Cy, cyclosporine; Gcort, glucocorticoids; Ifx, infliximab; IVIG, intravenous immunoglobulin; m, months; PR, partial response; TCN, tetracycline.