|Year : 2020 | Volume
| Issue : 4 | Page : 252-253
Case of a Taiwanese patient with granuloma faciale improved with colchicine
Ya-Tang Yang1, Yang Lo1, Chen-Yu Wu1, Shiou-Hwa Jee2, Hsien-Ching Chiu3, Yi-Teng Tseng1
1 Department of Dermatology, Cathay General Hospital, Taipei, Taiwan
2 Department of Dermatology, Cathay General Hospital; Department of Dermatology, National Taiwan University Hospital, National Taiwan University College of Medicine, Taipei, Taiwan
3 Department of Dermatology, National Taiwan University Hospital, National Taiwan University College of Medicine, Taipei, Taiwan
|Date of Submission||01-May-2020|
|Date of Decision||02-Jun-2020|
|Date of Acceptance||03-Jun-2020|
|Date of Web Publication||16-Dec-2020|
Dr. Yi-Teng Tseng
No. 280, Sec. 4, Renai Road, Taipei 106216
Source of Support: None, Conflict of Interest: None
|How to cite this article:|
Yang YT, Lo Y, Wu CY, Jee SH, Chiu HC, Tseng YT. Case of a Taiwanese patient with granuloma faciale improved with colchicine. Dermatol Sin 2020;38:252-3
|How to cite this URL:|
Yang YT, Lo Y, Wu CY, Jee SH, Chiu HC, Tseng YT. Case of a Taiwanese patient with granuloma faciale improved with colchicine. Dermatol Sin [serial online] 2020 [cited 2021 Aug 5];38:252-3. Available from: https://www.dermsinica.org/text.asp?2020/38/4/252/303699
Granuloma faciale (GF) is a rare disease with distinctive clinical and pathological features, yet it lacks well-established, effective treatment modalities. Considering that the nature of GF suggests that it is a variant of leukocytoclastic vasculitis, the administration of colchicine is a reasonable treatment option; however, literature discussing the efficacy of such a treatment is limited. Here, we report the case of a Taiwanese patient with GF who exhibited a significant response after treatment with colchicine for 3 weeks.
The 26-year-old woman, who did not have any underlying systemic diseases, presented with an asymptomatic erythematous lump on her left cheek for 2 years. Physical examination revealed a coin-sized, firm, erythematous plaque with prominent follicular openings on the left cheek [Figure 1]a. The patient reported that the lesion initially looked like a pimple, but gradually grew. The initial differential diagnoses included lupus erythematosus tumidus, sarcoidosis, and hypertrophic scar. Serum autoantibody screening, including antinuclear antibody, anti-extractable nuclear antigens antibody screening, and anti-double-stranded DNA antibody, all yielded negative results. Intralesional triamcinolone acetonide injection (5 mg/mL) and alexandrite laser were administered, but both had limited effects. A skin biopsy revealed a dense inflammatory infiltrate in the mid-dermis. The upper dermis was spared from inflammation, forming a grenz zone [Figure 2]a. The polymorphic infiltrate composed of lymphocytes, neutrophils, scattered eosinophils, and a few plasma cells. Leukocytoclastic change with fibrinoid debris was also noted, although it was not prominent [Figure 2]b. The results of periodic acid–Schiff staining, Alcian blue staining, and direct immunofluorescence study were all negative. Based on the physical and histopathological findings, the patient was diagnosed as having GF. Because of the patient's concerns regarding the potential adverse effects of dapsone, treatment with colchicine (1 mg/day) was initiated. The lesion markedly improved after 3 weeks of treatment [Figure 1]b, and no adverse reaction was noted.
|Figure 1: Clinical image of granuloma faciale: (a) One coin-sized, firm, erythematous plaque with prominent follicular openings on the left cheek; (b) the lesion exhibited significant improvement after 3 weeks of colchicine treatment|
Click here to view
|Figure 2: Histopathology of granuloma faciale: (a) A grenz zone separated the epidermis from the dense infiltrate in the upper/mid-dermis. A keratin plug with follicular dilatation was also observed (H and E, ×40); (b) the infiltrate was polymorphic, consisting of lymphocytes, neutrophils, scattered eosinophils, along with an admixture of plasma cells. Leukocytoclastic change with fibrinoid debris was also observed (H and E, ×400)|
Click here to view
GF is a localized form of leukocytoclastic vasculitis of uncertain pathogenesis. Patients with GF typically present with one or multiple red-brown papules, nodules, or plaques on their face. Dilated follicles, which were observed in the present case, and fine telangiectasia, are often present. Histologically, GF is characterized by a mixed, dense, inflammatory infiltrate of lymphocytes, neutrophils, plasma cells, and eosinophils occupying the mid-dermis, although the deep dermis and subcutaneous fat may be involved. Generally, the epidermis and follicles are separated from the underlying inflammation by an infiltrate-spared grenz zone. Typical features of leukocytoclastic vasculitis are usually observed, but they may be difficult to be identified in some lesions.
The primary histologic differential diagnosis is erythema elevatum diutinum (EED), another form of localized vasculitis. The histologic features of GF and EED are similar and overlapping. The two diseases cannot be solely distinguished by the presence of a grenz zone or patterned fibrosis. Ziemer et al. had suggested that a predominance of eosinophils infiltrates favors a diagnosis of GF while granulomatous nodules may be seen in EED more frequently. Clinically, EED tends to be located on the extensor surfaces of the extremities.
Treatment is often provided to address cosmetic concerns. Nevertheless, GF is usually resistant to therapy and tends to recur. Topical steroids or intralesional triamcinolone acetonide suspension (2.5–5 mg/mL) are often a first-line therapeutic option. Other therapies include dapsone, oral clofazimine, topical psoralen plus ultraviolet A photochemotherapy, topical calcineurin inhibitors, cryotherapy, pulsed-dye laser therapy, carbon dioxide laser therapy, dermabrasion, electrosurgery, surgical excision, and tumor necrosis factor-alpha inhibitors.
Dapsone is the most well-documented systemic treatment for GF, but responses vary from complete remission to no effect. A variety of adverse reactions, including hemolytic anemia, methemoglobinemia, neuropathy, agranulocytosis, and severe cutaneous adverse reaction, may occur after dapsone therapy. In a retrospective study in Taiwan, the incidence of dapsone hypersensitivity syndrome in nonleprosy patients was 1.66%.
Colchicine has been described in the literature as an alternative for treating GF, and Ohata and Nakama reported it to be effective in a patient with GF. Colchicine is also used in other inflammatory skin diseases and vasculitis. The mechanisms of action include interfering with microtubule formation and increasing intracellular cyclic adenosine monophosphate. Colchicine suppresses leukocyte function, inhibits DNA synthesis, and interrupts the degranulation of mast cells. Moreover, colchicine has fewer or milder adverse effects than dapsone does.
In summary, this case report presents a rare case of GF treated with colchicine. The patient exhibited an excellent response and had no adverse reaction to the treatment. Our experience may provide insights regarding the use of colchicine as an alternative to dapsone for treating GF.
Declaration of patient consent
The authors certify that they have obtained all appropriate patient consent forms. In the form the patient has given her consent for her images and other clinical information to be reported in the journal. The patient understands that her name and initials will not be published and due efforts will be made to conceal identity, but anonymity cannot be guaranteed.
Financial support and sponsorship
Conflicts of interest
There are no conflicts of interest.
| References|| |
Oliveira CC, Ianhez PE, Marques SA, Marques ME. Granuloma faciale: Clinical, morphological and immunohistochemical aspects in a series of 10 patients. An Bras Dermatol 2016;91:803-7.
Ziemer M, Koehler MJ, Weyers W. Erythema elevatum diutinum-a chronic leukocytoclastic vasculitis microscopically indistinguishable from granuloma faciale? J Cutan Pathol 2011;38:876-83.
Lindhaus C, Elsner P. Granuloma faciale treatment: A systematic review. Acta Derm Venereol 2018;98:14-8.
Chen WT, Wang CW, Lu CW, Chen CB, Lee HE, Hung SI, et al
. The function of HLA-B*13:01 involved in the pathomechanism of dapsone-induced severe cutaneous adverse reactions. J Invest Dermatol 2018;138:1546-54.
Sheen YS, Chu CY, Wang SH, Tsai TF. Dapsone hypersensitivity syndrome in non-leprosy patients: A retrospective study of its incidence in a tertiary referral center in Taiwan. J Dermatolog Treat 2009;20:340-3.
Ohata C, Nakama T. Granuloma faciale treated successfully with colchicine. Acta Derm Venereol 2019;99:833-4.
Anzengruber F, Graf V, Hafner J, Meienberger N, Guenova E, Dummer R. Efficacy and safety of colchicine in inflammatory skin diseases: A retrospective, monocentric study in a large tertiary center [Published online ahead of print, 2019 Nov 20]. J Dermatolog Treat. 2019;1-6. doi:10.1080/09546634.2019.1690621
[Figure 1], [Figure 2]