Gnathophyma: Two Cases and Review of the Literature

Dear Editor:

Phyma is considered an uncommon presentation of the last stage of rosacea, but it can also appear independent from rosacea. The most common phyma is rhinophyma; however, phyma of the forehead (e.g., metophyma), the eyelids, uni- or bilaterally (e.g., blepharophyma), the ears, uni- or bilaterally (e.g., otophyma) and the chin (e.g., gnathophyma) can occur. Phyma is characterized clinically by telangiectasias and disfiguring, erythematous papules, nodules and plaques. The consistency is parenchymatous and hard. Gnathophyma, in particular, has been very rarely reported in the literature; to our knowledge, only 11 articles including a total of 12 patients have been published (Table 1).1–11 We describe two cases of gnathophyma with an accompanying review of the literature.

TABLE 1.

Cases of gnathophyma reported in the literature

AUTHORS	NUMBER OF CASES	GENDER	AGE*	RACE	THERAPY
Javaid1	1	M	31	White	Surgery
Ilyas et al2	1	F	31	White	Doxycycline, minocycline, topical metronidazole, dermabrasion, CO2 laser, electrocautery
Schintler et al3	1	M	70	White	Surgery
Vidigal et al4	1	F	44	White	Ivermectin, tetracycline, metronidazole gel
Ezra et al5	1	M	56	Black	Surgery
de Macedo et al6	1	F	41	White	Tetracycline, ivermectin, metronidazole cream
Cameli et al7	2	F/F	55/50	White	Azelaic acid
Kumar & Das8	1	F	23	White	NS**
Kola et al9	1	F	54	White	Electrosurgery
Moiin et al10	1	M	65	White	IL triamcinolone, surgery
Bittar et al11	1	M	75	White	Hidrosurgery

*Years; **Not specified

Patient 1. A 33-year-old White male patient was admitted to our clinic with a diagnosis of gnathophyma. The patient stated that he was in good general health and that he was not in therapy with systemic drugs. He also declared that the dermatitis appeared at the age of 28 years and that it was unsuccessfully treated with topical metronidazole and ivermectin for four months, as well as oral doxycycline, 40mg/day, and limecycline, 600mg/day, for three months. The patient did not have a history of acne or rosacea. Dermatological examination revealed an enlargement of the chin with slight erythema (Figure 1), and a parenchymatous, hard consistency. No lesions in other sites were found.

FIGURE 1.

Gnathophyma of patient 1.

General physical examination did not reveal anything pathological. Laboratory tests were within normal ranges, except for high levels of total cholesterol (320mg/dl). Tests for Helicobacter pylori were negative. Microscopic examinations of scrapings from the chin revealed only one Demodex folliculorum/cm2. Histopathologic examination of a 4mm punch biopsy showed numerous hypertrophic sebaceous glands, a perivascular and perifollicular lymphoplasmocytic infiltrate in upper and mid dermis, and fibrosis. It was decided not to prescribe oral isotretinoin because of high total cholesterol levels. The patient did not want to move forward with a surgical approach.

Patient 2. An 18-year-old White female patient was admitted with a diagnosis of gnathophyma. The patient reported being well and that she was not in therapy with systemic drugs. She claimed that the dermatitis appeared one year earlier and that it had not yet been treated. The patient had no history of acne or rosacea. Dermatological examination showed an enlargement of the chin without the presence of abnormal coloring, but with parenchymatous-hard consistency (Figure 2a and 2b). No lesions in other sites were found. General physical examination did not reveal anything pathological. Laboratory tests were within normal ranges. Tests for Helicobacter pylori were not performed. Microscopic examinations of scrapings from the chin were negative for Demodex folliculorum. A biopsy was not performed. The patient’s parents did not want to move forward with any laser or surgical treatment options. The patient was treated with doxycycline 40mg/day for three months; however, clinical results were very poor. The patient was lost to follow up.

FIGURE 2.

Gnathophyma of patient 2.

We identified 12 cases of gnathophyma reported in the literature. All patients were White, except one Black patient.5 Seven patients were female, and five were male. All patients were adults, with an age ranging from 23 to 75 years (mean age: 49.6 years). Suprisingly, gnathophyma appeared to be very rarely associated with rosacea.1,2,4–10 As previously mentioned, both of our patients discussed here had negative medical history and clinical picture for rosacea. In two patients, gnathophyma was associated with severe tooth decay.2,7 In one patient, gnathophyma significantly improved following treatment of bacterial plaque and gingivitis.7 Demodex folliculorum was detected in only 2 out of 12 patients.1,4 Often, pharmacological treatments were found to be inadequate as treatment.2,4 Various laser and surgical approaches appeared to be more effective.1–3,5,9–11 However, no controlled clinical studies have been published so far.

With regard,