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CORRESPONDENCE
Year : 2021  |  Volume : 39  |  Issue : 2  |  Page : 101-102

Disseminated eruption of ectopic sebaceous glands after toxic epidermal necrolysis with good treatment response to oral isotretinoin


1 Department of Dermatology, Veterans General Hospital, Kaohsiung, Taiwan
2 Department of Dermatology and Allergy, Technical University of Munich, Munich, Germany

Date of Submission12-Jul-2020
Date of Decision23-Sep-2020
Date of Acceptance07-Oct-2020
Date of Web Publication23-Jun-2021

Correspondence Address:
Dr. Kai-Che Wei
Department of Dermatology, Veterans General Hospital, No. 386, Dazhong 1st Rd., Zuoying Dist., Kaohsiung 81362
Taiwan
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ds.ds_50_20

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How to cite this article:
Fang YH, Chen WC, Wei KC. Disseminated eruption of ectopic sebaceous glands after toxic epidermal necrolysis with good treatment response to oral isotretinoin. Dermatol Sin 2021;39:101-2

How to cite this URL:
Fang YH, Chen WC, Wei KC. Disseminated eruption of ectopic sebaceous glands after toxic epidermal necrolysis with good treatment response to oral isotretinoin. Dermatol Sin [serial online] 2021 [cited 2021 Jul 24];39:101-2. Available from: https://www.dermsinica.org/text.asp?2021/39/2/101/319151



Dear Editor,

We report a case of a 22-year-old male with numerous skin-colored, partly confluent papules over whole face [Figure 1]a and [Figure 1]d, 3 months after an attack of toxic epidermal necrolysis (TEN), induced by Ibuprofen and Tenoxicam, which involved face and >90% of total body surface area. Treatment for TEN included systemic corticosteroids, intravenous immunoglobulin and Etanercept, in addition to supportive management and wound care, without debridement, skin grafts or other surgical procedures. He recovered with ophthalmic and dermatologic sequelae including corneal erosions, nail dystrophy, postinflammatory hypopigmentation, and hypohydrosis. Dermoscopic examination showed numerous white-yellowish globules [Figure 1]b. Histopathology from the facial lesions revealed multiple sebaceous lobules that directly open to the epidermis without connection to hair follicles, indicating the diagnosis of ectopic sebaceous glands [Figure 1]c. Treatment with oral isotretinoin 20 mg daily led to gradual improvement by the 2nd month [Figure 1]e. After 10 weeks, isotretinoin was tapered to 20 mg every 2 days and further improvement with obvious flattening of the papular lesions was observed after 6 months of treatment [Figure 1]f.
Figure 1: (a) Disseminated skin-colored, partly confluent papules over whole face in a 22-year-old man (b) Dermoscopic findings of his face (c) histopathology revealed numerous sebaceous lobules that directly open to the epidermis without connection to hair follicles (H and E, ×100) (d) before treatment (e) after one month of oral isotretinoin treatment (f) after 6 months of oral isotretinoin treatment.

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Dermatologic sequelae are the most common long-term complications of Stevens–Johnson syndrome (SJS) and TEN, with post-inflammatory dyspigmentation, abnormal scarring, eruptive nevi, nail dystrophy and chronic pruritus among the most frequently reported changes.[1] Disseminated eruption of ectopic sebaceous glands is a rare complication after SJS/TEN, with only three reported cases in the literature.[2],[3],[4] The affected patients, including the current one, were young people, aged 17, 27, 21, 21, respectively, when TEN occurred, and developed the skin changes 3–4 months after skin lesions healed. The diagnosis was confirmed by histopathology, which distinguishes from sebaceous gland hyperplasia. Numerous enlarged gland lobules with the association to a hair follicle can be seen in sebaceous gland hyperplasia, but ectopic sebaceous glands open directly onto the epidermis with no connection to hair follicles.[5] No gender or ethnic predilection was found. The offending drug of TEN varied from antibiotics, analgesics to anti-epileptic drugs [Table 1].
Table 1: Cases of disseminated ectopic sebaceous glands eruption after SJS/TEN reported in literature

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The pathogenesis for the disseminated eruption of ectopic sebaceous glands after TEN is yet unknown. Damage to the pilosebaceous unit and destruction of hair follicles occur during epidermal necrolysis. It is suggested that cytokines and growth factors related to skin healing following SJS/TEN could contribute to sebaceous gland proliferation as well as to the development of eruptive nevi.[2],[6] Due to abundant sebaceous glands in the face, this could explain the facial predilection of the disseminated eruption of ectopic sebaceous glands.[3]

Treatment is so far empirical. In the three previous reports, invasive procedures were attempted, such as resurfacing laser or dermabrasion treatment, with only mild improvement and frequent recurrence.[2],[3],[4] Oral isotretinoin has been tried, but the results were unclear.[2] Our case showed a rapid satisfying response to oral isotretinoin up to 6 months. Further observation of more cases is required to see the long-term effect.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient has given his consent for his images and other clinical information to be reported in the journal. The patient understands that name and initials will not be published and that efforts will be made to conceal identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

Prof. Wen-Chieh Chen, an editorial board member of Dermatologica Sinica, had no role in the peer review process of or decision to publish this article. The other authors declared no conflicts of interest in writing this paper.



 
  References Top

1.
Lee HY, Walsh SA, Creamer D. Long-term complications of Stevens-Johnson syndrome/toxic epidermal necrolysis (SJS/TEN): The spectrum of chronic problems in patients who an episode of SJS/TEN necessitates multidisciplinary follow-up. Br J Dermatol 2017;177:924-35.  Back to cited text no. 1
    
2.
Moosbrugger EA, Adams BB. Disseminated eruption of ectopic sebaceous glands following Stevens-Johnson syndrome. J Am Acad Dermatol 2011; 65: 446-8.  Back to cited text no. 2
    
3.
Park J, Hwang SR, Woo SH, Yun SK, Kim HU. Disseminated eruptive free sebaceous glands on the face as a rare sequel of toxic epidermal necrolysis. Eu J Dermatol 2016;26:92-4.  Back to cited text no. 3
    
4.
Habre M, Ortonne N, Colin A, Meningaud JP, Chosidow O, Wolkenstein P, et al. Facial scars following toxic epidermal necrolysis: Role of adnexal involvement? Dermatology 2016;232:220-3.  Back to cited text no. 4
    
5.
Tumors and related lesions of the sebaceous glands. In: Calonje E, Brenn T, Lazal AJ, Billings SD, editors. McKee's Pathology of the Skin with Clinical Correlations. 5th ed. United States: Elsevier 2020. p. 1589.  Back to cited text no. 5
    
6.
Shoji T, Cockerell CJ, Koff AB, Bhawan J. Eruptive melanocytic nevi after Stevens-Johnson syndrome. J Am Acad Dermatol 1997;37:337-9.  Back to cited text no. 6
    


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