A strange infiltrative plaque on the face

A 64-year old lady, known to have hypertension and urolithiasis, presented with a 4-month history of a slowly growing, erythematous-edematous and infiltrative plaque with superficial desquamation, involving all the nose skin surface – except for the root – and both zigomatic areas up to the naso-labial folds (Fig. 1).

Fig. 1
Erythematous-edematous-infiltrative plaque involving mainly the right side of the centro-facial region.

The patient came from a remote rural area of the south-eastern Sicily, and was a farmer. Personal and familiar anamnesis was negative for autoimmune diseases or skin disorders.

Routine blood exams were within normal limits; serum autoantibodies (antinuclear, anti-ENA, anti-JO1 and anti-Scl70) were negative. Nailfold capillaroscopy and chest X-ray examination were normal.

Histopathologic examination of a cutaneous biopsy revealed a granulomatous infiltrate in the dermis, consisting of lymphocytes, histiocytes, and multinuclear giant cells with hyperkeratotic overlying epidermis. A touch-imprint preparation of a skin specimen, showed Leishmania amastigotes, within the histiocytes as well as extracellularly.

Lupoid leishmanniasis (LL) is a rare form of cutaneous leishmaniasis (CL) showing a striking resemblance with some other granulomatous skin diseases of inflammatory or infectious origin.¹

In fact, LL is characterized by a typical spreading of the initial lesion leading to an infiltrated plaque with undefined borders, whereas some papules and nodules, often with scaling, may become apparent, presenting a lupoid aspect. The involvement of suggestive areas, as in our case, may further complicate the differential diagnosis.²

Histopathological features are that of epithelioid granulomas, and the detection of amastigotes is often hollow, both in microscopy and cultures.³

It seems that in LL certain strains replicate inside the macrophage, so assuming their ability to evade intracellular destruction or a concomitant defect in the T-cell activation process.^{2 3}Leishmania infantum, the most frequent causative agent of CL in our geographic area, have been rarely linked with LL.^{1 4}

The patient received N-methylglucamine-antimoniate, 1 mL twice-a-week intralesionally (total of 7 doses), with progressive improvement.

Conflicts of interest

The authors declare no conflicts of interest.

References

• ¹
  Guarneri C, Vaccaro M, Cannavò S.P, Borgia F, Guarneri B. Erythematous-edematous-infiltrative plaque on the face: cutaneous angio-lupoid leishmaniasis. Eur J Dermatol. 2002; 12:597-9
• ²
  Ul Bari A, Raza N. Lupoid cutaneous leishmaniasis: a report of 16 cases. Indian J Dermatol Venereol Leprol. 2010; 76:85
• ³
  Khaled A, Goucha S, Trabelsi S, Zermani R, Fazaa B. Lupoid cutaneous leishmaniasis: a case report. Dermatol Ther (Heidelb). 2011; 1:36-41
• ⁴
  Guarneri C, Tchernev G, Bevelacqua V, Lotti T, Nunnari G. The unwelcome trio: HIV plus cutaneous and visceral leishmaniasis. Dermatol Ther. 2015; , http://dx.doi.org/10.1111/dth.12303
  » https://doi.org/10.1111/dth.12303

Publication Dates

• Publication in this collection
  Mar-Apr 2016

History

• Received
  07 Nov 2015
• Accepted
  25 Nov 2015