Fordyce-like sebaceous hyperplasia: A case series of panfacial textural change successfully treated with oral isotretinoin

Introduction

To our knowledge, this report describes the first case series in the literature depicting an atypical variant of sebaceous hyperplasia (SH) presenting as global textural change on the face. SH typically presents as small, soft, yellow or skin-colored papules often with a central pit, commonly on the forehead, nose, and cheeks.¹ Panfacial proliferations have been reported with SH, including eruptive SH (acute onset in immunosuppression)2, 3, 4 and diffuse SH (familial or idiopathic).5, 6, 7 The diffuse SH reports describe typical histopathologic findings of SH, including multiple, enlarged, lobulated sebaceous glands emanating from an associated follicular infundibula and at times, opening directly to the epidermis.^5,6

The current case series introduces an atypical subtype of SH, presenting as cosmetic textural concerns rather than the typical single or multiple distinct cutaneous facial papules seen in SH. These cases demonstrated no oily or sebaceous skin type, with striking periorbital involvement—an area rarely affected by SH. Morphologic and histologic features were more consistent with Fordyce spots (FSs), sebaceous glands typically found in mucosal locations such as the vermilion border of the lips, oral cavity, and genitalia.⁸

Clinically, FSs are small (1-5 mm), well–circumscribed white or yellowish papules, affecting up to 80% of adults.⁸ Diagnosis is usually clinical, with biopsy rarely required; histology reveals mature sebaceous glands lacking associated hair follicles.

Although treatment is not generally necessary, successful use of CO₂ laser, electrocautery, and oral isotretinoin has been reported for both FS and SH.⁹ Isotretinoin decreases hyperplastic sebaceous gland development, reduces lesion size, and suppresses sebum production in SH.^1,5 Topical retinoids, including adapalene, have also been reported to successfully treat SH.²

Effective treatment of mucosal FS has been reported in the context of oral isotretinoin therapy for acne vulgaris.10, 11, 12A case series of 2 patients demonstrated successful management of FS of the lip with low-dose oral isotretinoin.¹⁰ Of note, 1 patient had sustained remission following 1 course of treatment, whereas the other patient had FS recurrence after 1 month. A trial of topical tazarotene 0.1% gel and tretinoin 0.04% microgel failed to improve the lesions.¹⁰ A report from 1993 also details incidental improvement of lip FS with oral isotretinoin for acne vulgaris, with recurrence of FS upon treatment cessation.¹²

This case series details 4 patients with panfacial Fordyce-like SH successfully treated with oral isotretinoin at a private dermatology clinic in Melbourne, Victoria, Australia.

Case 1

A 42-year-old woman was referred for a 3-year history of a ‟milia-like” eruption over her face. She had a background of systemic lupus erythematosus treated with hydroxychloroquine. There was no family history of a similar skin complaint. Prior to this presentation, the medical advice provided was that her cutaneous changes reflected the lupus disease process. She was prescribed topical preparations without improvement.

Examination revealed extensive Fordyce-like lesions involving most of the face (Figs 1 and 2), with lesions coalesced into plaques in the central upper face (Fig 1, A). Notably, the lesions involved the eyelids (Fig 1, B).

Fig 1.

Case 1 clinical photographs. Baseline photographs of Fordyce-like sebaceous hyperplasia of the forehead (A) and periorbital skin (B) and following 3 months of oral isotretinoin (C).

Fig 2.

Case 1 clinical photographs. Baseline photographs of Fordyce-like sebaceous hyperplasia of the temple (A) and cheek (C) and following 3 months of oral isotretinoin (B, D).

Punch biopsy histopathology demonstrated an unremarkable epidermis, with mature sebaceous glands in the dermis and no associated hair follicle, in keeping with FS. There was no increase in dermal mucin and no inflammation. There was no evidence of milia cysts.

Isotretinoin was commenced at 10 mg daily, with dramatic improvement at 3 months. Notably, the patient self-discontinued isotretinoin for several months, resulting in the recurrence of textural change. Clear skin was recovered on recommencement (Figs 1 and 2).

Case 2

A 50-year-old woman presented with a 1-year history of skin texture concerns. She had no significant medical history, no regular medications, and no family history of similar cutaneous changes. Prior to presentation, she was diagnosed with rosacea by a dermatologist and underwent multiple sessions of pulsed-dye laser therapy and radiofrequency microneedling without improvement. The reported cost for these treatments was $6000 AUD. Given her ongoing textural concerns, she was then offered full-face resurfacing laser with uncertain guarantee, at a cost of more than $3000 AUD and weeks of recovery.

Clinical examination revealed widespread small skin-colored papules, with notable involvement around the chin, temples, eyelids, and cheeks (Figs 3 and 4).

Fig 3.

Case 2 clinical photographs. Baseline photographs of Fordyce-like sebaceous hyperplasia on the chin (A) and following 3 months of oral isotretinoin treatment (B).

Fig 4.

Case 2 clinical photographs. Baseline photographs of Fordyce-like sebaceous hyperplasia on the temple and chin (A, B) and following 3 months of isotretinoin treatment (C, D).

Punch biopsy histopathology demonstrated an increase in mature sebaceous glands in the papillary dermis, without an associated hair follicle, more consistent with FS.

The patient was commenced on oral isotretinoin 10 mg thrice weekly. After 3 months, there was a dramatic improvement in the skin texture (Figs 3 and 4). The patient was highly satisfied with the results but self-discontinued the isotretinoin to pursue other cosmetic treatments unrelated to skin texture. Upon cessation, the lesions recurred within months but again cleared on restarting isotretinoin.

Case 3

A 40-year-old woman presented with cosmetic concerns about skin texture present for several months. She had no significant medical history, no family history of similar complaints, and had not sought treatment prior to this presentation.

On examination, she had widespread tiny skin-colored papules across the face, including eyelids, which coalesced in some areas (Fig 5).

Fig 5.

Case 3 clinical photographs. Baseline photographs of Fordyce-like sebaceous hyperplasia on the eyelid and chin (A, B) and following 3 months of oral isotretinoin treatment (C, D).

Histology of a punch biopsy of the left jawline demonstrated an increase in mature sebaceous glands in the papillary dermis, again without an associated hair follicle, and mildly inflamed adjacent dermal fibrosis (Fig 6).

Fig 6.

Case 3 histopathology image. Histopathology demonstrates a collection of sebaceous glands in the dermis. There is a lack of associated hair follicle, and a lack of typical lobulated architecture seen in sebaceous hyperplasia. (Original magnification: ×10.)

She was commenced on oral isotretinoin at a dose of 10 mg, thrice weekly, with significant textural improvement at 3 months. A brief cessation of the medication resulted in recurrence. She remains on oral isotretinoin 6 years after diagnosis.

Case 4

A 27-year-old woman presented with a 2-year history of ‟rough” skin texture. Prior to dermatological review, she underwent skin needling and chemical peels without benefit. She had a history of inactive mild atopic dermatitis. There was no family history of similar skin changes.

Clinical examination revealed Fordyce-like lesions over the face, with predominance over cheeks, temples, and eyelids (Fig 7).

Fig 7.

Case 4 clinical photographs. Baseline photographs of Fordyce-like lesions on the temple and eyelid (A) and following 3 months of oral isotretinoin treatment (B).

A biopsy was recommended but not undertaken due to the risk of scarring. She was empirically treated with isotretinoin 5 mg daily, which yielded significant improvement after 3 months, with a minor complaint of lip dryness.

Discussion

This case series highlights a previously unreported panfacial subtype of Fordyce-like SH. The observed clinical presentation in all 4 cases in this series is tiny yellowish or flesh-colored papules, diffusely spread across the face, causing noticeable textural distortion. The aim of this publication is to raise awareness about this atypical presentation, reduce unnecessary and costly treatments, and demonstrate effective treatment with low-dose oral isotretinoin.

Clinically, all 4 cases presented with Fordyce-like hyperplastic sebaceous glands. Histopathologically, findings reflected this in cases 1 to 3. Dermoscopic examination was noncontributory, demonstrating an absence of specific diagnostic features and revealing only uniformly flesh-colored skin. Case 4 was clinically diagnosed and empirically treated due to a very similar presentation and examination findings. Histopathology in the 3 cases demonstrated 1 to 2 mature sebaceous glands in the papillary dermis, without associated hair follicles (Fig 6). Notably, the lesions lacked the typical lobulated architecture seen in classic SH (Fig 8). Although comparative biopsies of clinically unaffected facial skin were not available, the histology findings were consistent across all 3 cases. Thus, the overall impression favored FS despite having previously been reported only to affect mucosal sites.

Fig 8.

Histopathology image of typical sebaceous hyperplasia. Many enlarged, lobulated sebaceous glands are present around the infundibulum of a hair follicle. (Original magnification: ×100.)

Furthermore, there was notable involvement of the periorbital skin in all cases and the perioral skin to a lesser degree. This is distinct from the described presenile familial diffuse SH variant, which has periorificial sparing.^5,6 None of the 4 patients in our study were immunocompromised, which was a common observation in the eruptive subtype of SH.2, 3, 4 Similarly, none of the 4 patients had a positive family history for SH nor a history of significant acne or oily skin, which is typical in classic cases of SH.

As with any atypical lesion, histopathology is required in circumstances of diagnostic uncertainty. This is especially relevant to rule out the differential diagnoses of diffuse facial lesions, which can include Muir-Torre syndrome and Cowden syndrome.^2,5 Other differential diagnoses worthy of consideration include comedones, trichoepitheliomas, syringomas, and milia cysts, all of which can be reliably differentiated on histopathological evaluation.⁵

All 4 cases responded well to oral isotretinoin therapy and were highly satisfied with their treatment. Notably, 2 of 4 cases experienced recurrence of their textural change on ceasing the isotretinoin. This is a known occurrence in the setting of SH and FS of mucosal sites,^6,10,12 and larger-scale studies should aim to inform guidelines and safe isotretinoin treatment protocols.

Patients in this case series experienced no major adverse effects from isotretinoin, with 1 patient complaining of lip dryness. Oral isotretinoin prescribing is limited to specialist dermatologists in Australia, and pretreatment laboratory testing of liver function tests, lipid panel, and pregnancy testing was performed for each patient. Careful counseling took place regarding the teratogenicity risk in pregnancy and the effects on mood.

Atypical SH should be considered in patients presenting with cosmetic skin texture concerns, to reduce costly, invasive treatments and diagnostic delay. Case 2 underwent pulsed-dye laser therapy and radiofrequency with no effect for presumed rosacea, prior to biopsy and diagnosis. She reported treatment costs amounting to $6000 AUD, which offered no benefit. She was recommended to undergo a full-face ablative laser for a further $3000 AUD, but was advised there may not be an improvement and that recovery time would be weeks. Similarly, case 4 underwent skin needling and chemical peels with no effect on skin texture concerns, and case 1 underwent failed topical treatment.

Conclusion

This case series aims to highlight an atypical variant of SH, presenting as diffuse, tiny, Fordyce-like papular lesions and textural concerns. All 4 cases were successfully treated with low-dose oral isotretinoin. Ongoing clinical suspicion is warranted where patients present with cosmetic concerns of diffuse skin textural change, especially when unresponsive to topical therapies.

Conflicts of interest

None disclosed.