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LETTER TO THE EDITOR
Year : 2018  |  Volume : 9  |  Issue : 1  |  Page : 68-70  

Granulomatous periorificial dermatitis effectively managed with oral isotretinoin


Department of Dermatology, MGM Medical College and General Hospital, Kamothe, Navi Mumbai, Maharashtra, India

Date of Web Publication22-Jan-2018

Correspondence Address:
Swagata Tambe
19/558, Udyan Society, Nehru Nagar, Kurla East, Mumbai - 400 024, Maharashtra
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/idoj.IDOJ_129_17

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How to cite this article:
Tambe S, Jerajani H, Pund P. Granulomatous periorificial dermatitis effectively managed with oral isotretinoin. Indian Dermatol Online J 2018;9:68-70

How to cite this URL:
Tambe S, Jerajani H, Pund P. Granulomatous periorificial dermatitis effectively managed with oral isotretinoin. Indian Dermatol Online J [serial online] 2018 [cited 2020 Sep 27];9:68-70. Available from: http://www.idoj.in/text.asp?2018/9/1/68/223713



Sir,

Perioral dermatitis is an uncommon facial eruption of women and children presenting as tiny papules and pustules often in a perioral distribution.[1] It is now more correctly referred to as “periorificial dermatitis” as it frequently involves perinasal and periocular skin. Granulomatous periorificial dermatitis (GPD) is a distinct facial eruption in prepubertal children characterized by a monomorphic papular eruption occurring in the perinasal, perioral, and periocular areas.

A 30-year-old unmarried female, saleswoman by occupation, presented with appearance of reddish, scaly lesions on the face since 2 months. There was history of mild photosensitivity associated with the lesions. She did not give a history of introduction of new creams or cosmetic products, drug ingestion, or parlour procedures prior to appearance of lesions. Patient received treatment with oral doxycycline and topical antibacterial creams for 1 month without significant improvement. She had also received topical steroids for 7 days in the recent past with worsening of lesions requiring discontinuation.

Cutaneous examination revealed multiple erythematous, scaly papules and plaques on the supra orbital, periorbital, perioral, and perinasal area with classical sparing of upper forehead and butterfly area of the face (nose and corresponding area on the cheeks) [Figure 1]a and [Figure 1]b. A provisional diagnosis of periorificial dermatitis was considered.
Figure 1: (a and b) Erythematous, scaly papules, and plaques on the supraorbital, periorbital, perioral, and perinasal area with classical sparing of upper forehead and butterfly area of the face

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Laboratory investigations of the patient did not reveal any abnormality. Skin biopsy from the inflammatory papule revealed perifollicular and perivascular granulomatous inflammatory infiltrate composed of lymphocytes, epithelioid cells, and giant cells suggesting the diagnosis of granulomatous type of periorificial dermatitis [Figure 2]a and [Figure 2]b.
Figure 2: (a) Histopathology ×100, H and E stains: Periappendageal perivascular and interstitial inflammatory infiltrate with Langhans giant cells. (b) Histopathology ×400, H and E stain: Perifollicular and periappendageal granulomatous inflammatory infiltrate composed of lymphocytic, epithelioid cells with multiple Langhans giant cells

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Considering unresponsiveness to doxycycline in the past, patient was started on oral isotretinoin 20 mg daily with topical application of metronidazole cream. The patient followed up after 5 days with acute exacerbation of lesions [Figure 3]a and [Figure 3]b. She was counselled and advised to continue the treatment. After 3 weeks of treatment with isotretinoin, there was remarkable improvement in the lesions with disappearance of papules and significant reduction in the erythema [Figure 4]a, [Figure 4]b, [Figure 4]c.
Figure 3: (a and b) Acute exacerbation of the lesions after 5 days of isotretinoin

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Figure 4: (a) Before treatment. (b) Exacerbation of lesions after 5 days of starting isotretinoin. (c) Resolution of lesions with mild persistent erythema after 3 weeks of isotretinoin

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Periorificial dermatitis (POD) is an acneiform eruption of unknown origin, most commonly found around the orifices.[1] POD occurs worldwide, especially in the fair-skinned population; predominantly women between the ages of 15 and 45 years are affected. Peak incidence is in the second and third decade of life. POD is also observed in children, where in contrast to the adult form, males are predominantly affected.[2] The exact etiology is unknown; however, it is related to the impairment of barrier function and dryness of the skin as well as proliferation of the skin flora. It may be induced by topical application [3] or inhalation of corticosteroids, allergic response to amalgam, and mercury in dental fillings,[4] toothpaste containing fluorides,[5] cosmetics, fusiform bacteria,[6]Candida albicans,[7] and Demodex folliculorum.[8] Nikkels and Pierard noted POD in females after stopping oral contraceptives. These patients also had premenstrual flares.[9]

It usually presents as grouped follicular reddish papules, papulovesicles, and papulopustules on an erythematous base with a possible prospect of confluence of lesions, predominantly in perioral distribution with classical sparing of vermilion border of lips.

Granulomatous variant of periorificial dermatitis (GPD) is a well-recognized entity, presenting most commonly in prepubertal children as monomorphic small papules limited to the perioral, perinasal, and periocular regions. The primary lesion is a discrete 1–3 mm dome-shaped flesh-colored, yellow-brown, or red papule. Mild scaling of the lesions or surrounding erythema may occur. Extra facial lesions had been reported, involving the trunk, extremities, and labia majora and could be generalized. Extra facial involvement does not appear to adversely affect the duration and response to therapy.[10]

Diagnosis of POD is often established by histopathology which reveals spongiosis, primarily of the outer root sheath of hair follicles and epidermis of the skin. In addition, variable degree of perifollicular or perivascular lymphohistiocytic infiltrate is also evident. GPD has a characteristic histopathology, with epidermal spongiosis and upper dermal and perifollicular granulomas surrounded by lymphocytes.[11],[12]

Differential diagnosis of GPD includes POD, granulomatous rosacea (GR), lupus miliaris disseminatus faciei (LMDF), and sarcoidosis.

GR and POD are inflammatory skin conditions characterized by erythematous papules most commonly affecting the face. These entities have been the topic of controversy because of similarity in clinical presentation but they still have variable causes and prognoses. They are both benign and self-limiting conditions. Some clinicians consider POD and its clinical variants on the same spectrum of GR. Clinically and histologically, GPD can be similar to GR but typically has a less granulomatous histology, responds better to treatment, and has a shorter clinical course.[13]

For mild cases, in most children and pregnant women, topical therapy is sufficient. Topical anti-inflammatory agents commonly used in the treatment of POD include metronidazole,[14] erythromycin, or clindamycin in a nongreasy base (e.g., gel, lotion, cream). Pimecrolimus cream seems to be most effective in steroid-induced perioral dermatitis.[15] Topical anti-acne medications such as adapalene [16] and Azelaic acid [17] can be considered as effective alternatives. Ointments should be avoided due to their greasy base.

Moderate-to-severe forms require systemic treatment. The drugs of choice are doxycycline (or tetracycline) and minocycline. In pediatric patients, management consists of topical metronidazole, oral erythromycin, oral tetracyclines (only in children older than 8 years), or cotrimoxazole. Oral isotretinoin may be considered in unresponsive and granulomatous type of POD.[12],[18]

The prognosis of GPD is good, and spontaneous resolution usually occurs by a few months to 3 years after onset. Although it is asymptomatic, patients seek for medical help due to cosmetic disfigurement.

Our case is unique in regards to the appearance of GPD in a young female, unresponsiveness to tetracyclines which are usually the first line of management, excellent response to isotretinoin, and initial worsening of the symptoms after isotretinoin similar to that reported in acne vulgaris.

This phenomenon of initial worsening of lesions of periorificial dermatitis after isotretinoin ingestion has never been reported; hence, patient should be informed about this so that treatment can be continued. Secondly, in cases of preceding long-term use of topical steroids, steroid weaning with low-dose 0.1–0.5% hydrocortisone cream can be tried initially to prevent serious rebound phenomenon.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
Kihiczak GG, Cruz MA, Schwartz RA. Periorificial dermatitis in children: An update and description of a child with striking features. Int J Dermatol 2009;48:304-6.  Back to cited text no. 1
    
2.
Hafeez ZH. Perioral dermatitis: An update. Int J Dermatol 2003;42:514-7  Back to cited text no. 2
    
3.
Wells K, Brodell RT. Topical corticosteroid 'addiction': A cause of perioral dermatitis. Postgrad Med 1993;93:225-30.  Back to cited text no. 3
    
4.
Brehler R, Panzer B, Forck G, Bertram HP. Mercury sensitization in amalgam fillings. Assessment from a dermatologic viewpoint. Dtsch Med Wochenschr 1993;118:451-6.  Back to cited text no. 4
    
5.
Mellette JR, Aeling JL, Nuss DD. Fluoride tooth paste: A cause of perioral dermatitis. Arch Dermatol 1976;112:730-1.  Back to cited text no. 5
    
6.
Berardi P, Benvenuti S, Genga A, Cecchini F. Demonstration of fusobacteria in eruptions of perioral dermatitis using the tape stripping toluidine blue (TSTB) method. J Eur Acad Dermatol Venereol 1994;3:495-9.  Back to cited text no. 6
    
7.
Bradford LG, Montes LF. Perioral dermatitis and Candida albicans. Arch Dermatol 1972;105:892-95.  Back to cited text no. 7
    
8.
Rufli T, Mumcuoglu Y, Cajacob A, Büchner S. Demodex folliculorum: Zur čtiopathogenese und Therapie der Rosazea und perioralen Dermatitis. Dermatologica 1981;162:12-26.  Back to cited text no. 8
    
9.
Nikkels AF, Pierard GE. Control of perimenstrual flares of perioral dermatitis by isotretinoin. J Dermatol Treat 2000;11:97-9.  Back to cited text no. 9
    
10.
Urbatsch AJ, Frieden I, Willialms ML, Elevaski BE, Mancini AJ, Paller AS. Extarfacial and generalized granulomatous periorificial dermatitis. Arch Dermatol 2002;138:1354-8.  Back to cited text no. 10
    
11.
Lucas CR, Korman NJ, Gilliam AC. Granulomatous periorificial dermatitis: A variant of granulomatous rosacea in children? J Cutan Med Surg 2009;13:115-8.  Back to cited text no. 11
    
12.
Smith KW. Perioral dermatitis with histopathologic features of granulomatous rosacea: Successful treatment with isotretinoin. Cutis 1990;46:413-5.  Back to cited text no. 12
    
13.
Lee GL, Zirwas MJ. Granulomatous Rosacea and Periorificial Dermatitis: Controversies and Review of Management and Treatment. Dermatol Clin 2015;33:447-55.  Back to cited text no. 13
    
14.
Miller SR, Shalita AR. Topical metronidazole gel (0.75%) for the treatment of perioral dermatitis in children. J Am Acad Dermatol 1994;31:847-8.  Back to cited text no. 14
    
15.
Oppel T, Pavicic T, Kamann S, Brautigam M, Wollenberg A. Pimecrolimus cream (1%) efficacy in perioral dermatitis - Results of a randomized, double-blind, vehicle-controlled study in 40 patients. J Eur Acad Dermatol Venereol 2007;21:1175-80.  Back to cited text no. 15
    
16.
Jansen T. Perioral dermatitis successfully treated with topical adapalene. J Eur Acad Dermatol Venereol 2002;16:175-7.  Back to cited text no. 16
    
17.
Jansen T. Azelaic acid as a new treatment for perioral dermatitis: Results from an open study. Br J Dermatol 2004;151:933-4.  Back to cited text no. 17
    
18.
Brunner M, Megahed M, Hölzle E, Ruzicka T. Granulomatous perioral dermatitis in childhood. Treatment with isotretinoin. Akt Dermatol 1995;21:60-2.  Back to cited text no. 18
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4]



 

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