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Table of Contents
CASE REPORT
Year : 2020  |  Volume : 24  |  Issue : 1  |  Page : 59-61

Uncommon presentation of lichen planopilaris during paclitaxel chemotherapy: Case report


1 Department of Dermatology, University of Utah, Salt-Lake City, Utah, USA
2 Department of Dermatology, St. Joseph Mercy Hospital Ann Arbor, Michigan, USA
3 Department of Dermatopathology, St. Joseph Mercy Hospital Ann Arbor, Michigan, USA

Date of Submission28-Jul-2019
Date of Acceptance15-Sep-2019
Date of Web Publication27-Mar-2020

Correspondence Address:
Dr. Nwanneka M Okwundu
Hackensack Meridian Health-Palisades Medical Center, 7600 River Road, North Bergen, NJ 07047
USA
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jdds.jdds_44_19

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  Abstract 


Lichen planopilaris (LPP) is an uncommon scalp disorder of unknown etiology and prevalence. It may be an autoimmune process triggered by unknown genetic and/or environmental factors that attack hair follicles of the scalp. LPP can present in association with various autoimmune diseases and immunomodulatory therapies. We present an atypical case of LPP occurring during the treatment of breast cancer with paclitaxel, after complete resolution of alopecia areata and anagen effluvium. Shortly after initiation of the paclitaxel therapy, the patient experienced a pruritic acneiform facial eruption and hair loss that was localized to her frontal scalp with complete loss of eyebrows and significant loss of her eyelashes. The lesions were biopsied revealing LPP. LPP should be considered among other causes of alopecia in patients treated with paclitaxel.

Keywords: Alopecia, cicatricial, folliculotropic mycosis fungoides, frontal fibrosing alopecia, lichen planopilaris, lichen planus, systemic lupus erythematosus


How to cite this article:
Okwundu NM, Ekpo FE, Ghafferi J, Fivenson D. Uncommon presentation of lichen planopilaris during paclitaxel chemotherapy: Case report. J Dermatol Dermatol Surg 2020;24:59-61

How to cite this URL:
Okwundu NM, Ekpo FE, Ghafferi J, Fivenson D. Uncommon presentation of lichen planopilaris during paclitaxel chemotherapy: Case report. J Dermatol Dermatol Surg [serial online] 2020 [cited 2020 Oct 20];24:59-61. Available from: https://www.jddsjournal.org/text.asp?2020/24/1/59/281421




  Introduction Top


Lichen planopilaris (LPP) is a rare progressive form of scarring (cicatricial) alopecia.[1] LPP is a lymphocytic cicatricial alopecia characterized by follicular hyperkeratosis, perifollicular erythema, and loss of follicular orifices.[2],[3],[4] The annual incidence rate varied from 1% to 8% in a survey conducted by four medical centers. The age of onset for LPP was between 25 and 70 years, and the most common reported symptoms of patients with LPP include shedding, pruritus, scale, and scalp tenderness.[5]

The exact pathogenesis of LPP remains unclear; however, it may be due to an autoimmune, cell-mediated cytotoxic immune reaction against follicular antigens.[3],[6] Some consider LPP to be a follicular variant of lichen planus. LPP may have a possible relationship to other autoimmune diseases.[2],[3]

LPP can occur in association with other autoimmune and nonautoimmune disorders. However, studies about its autoimmune and other comorbid conditions are limited. It is more commonly associated with autoimmune disorders such as Sjogren's syndrome and lupus erythematosus.[7],[8] Nonautoimmune comorbid conditions of LPP include hyperlipidemia, metabolic syndrome, hypothyroidism, anxiety, and depression.[9],[10],[11],[12] In addition to its association to autoimmune and its other comorbid condition, LPP continues to be discovered in atypical situations. We present a case of atypical presentation of LPP complicated by comorbid conditions.


  Case Report Top


A 46-year-old African American female presented with a facial rash and alopecia that began 1 year before presentation while receiving treatment for localized breast cancer. The patient was treated with radiotherapy followed by doxorubicin and cyclophosphamide. These were well tolerated without evidence of skin eruptions during the entire duration of treatment. Temporary anagen effluvium resolved 5 months after completion. Paclitaxel was started following completion of initial therapy. Shortly after initiation, there was subsequent development of scalp hair loss and a pruritic acneiform facial eruption over the next 6 months. The patient denied any associated symptoms including fever, chills, nausea, vomiting, diarrhea, urinary symptoms, fatigue, weight loss, or appetite changes. There was no improvement of the eruption with use of topical benzoyl peroxide/clindamycin, clindamycin lotion, tretinoin, or emollients.

The patient's medical history was notable for biopsy-proven alopecia areata that had resolved 5 years before beginning chemotherapy for breast cancer. The paclitaxel-associated hair loss varied from her previous presentations of alopecia areata and anagen effluvium during doxorubicin and cyclophosphamide therapy. Hair loss was localized to her frontal scalp with complete loss of eyebrows and significant loss of her eyelashes.

Physical examination of her scalp revealed diffuse hair loss along the frontal and bitemporal scalp with a 1.5 cm band of recession of the frontal hairline. In addition, frontal scalp follicular tufting and loss of follicular ostia were visible, suggestive of a scarring alopecia. There were few isolated terminal hairs without tufting or perifollicular erythema located on the frontotemporal scalp (positive lonely hair sign). Hair pull test was negative. The physical examination further revealed complete loss of eyebrows and 60% loss of upper and lower eyelashes. Inspection of her face revealed skin-colored, pinpoint, acneiform papules distributed along her forehead, cheeks, and chin [Figure 1].
Figure 1: Recession of frontal hairline with follicular tufting and loss of follicular ostia and lonely hair sign

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Punch biopsies of the forehead and scalp revealed keratin plugged, mildly dilated follicular ostia with adjacent perifollicular fibrosis, chronic inflammation, and melanophages within the papillary dermis. Vertical and horizontal sections from the frontal scalp demonstrated decreased follicular units, loss of sebaceous glands, and a normal anagen/telogen ratio. Eccentric thinning of the follicular epithelium and polytrichia were also visualized. Clinical and histopathologic correlation was consistent with early stages of LPP, specifically frontal fibrosing alopecia (FFA) [Figure 2]a and [Figure 2]b.
Figure 2: (a and b) Mildly dilated follicular ostia with adjacent perifollicular fibrosis, chronic inflammation, eccentric thinning of the follicular epithelium, and polytrichia (a, ×10; and b, ×20)

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Topical minoxidil and betamethasone dipropionate to the affected areas resulted in regrowth of her eyelashes, but only sparse hair regrowth along the frontal scalp. The recession of the frontal scalp hairline was still present. There was no resolution of hair loss of the eyebrows despite daily use of minoxidil [Figure 1].


  Discussion Top


LPP is an inflammatory disorder that is characterized by perifollicular erythema, follicular hyperkeratosis, and scarring alopecia.[13] The permanent loss of hair is postulated to emanate from the site in which inflammation occurs. Inflammatory infiltrate in LPP is mainly concentrated around the follicular infundibulum and isthmus, unlike alopecia areata, in which inflammation is mostly concentrated around the follicular bulb. The follicular “bulge,” which is a site of pluripotent stem cells involved in the regeneration of the lower portion of the hair follicle during follicular cycling, is within the immunologic target for LPP. Damage to the bulge region is postulated to account for the development of scarring alopecia.[14] Pseudopelade of Brocq is used to describe the noninflammatory end-stage patchy cicatricial alopecia that may be seen in LPP.[15]

There are three variants of LPP that can be distinguished based on their clinical patterns and areas of involvement. These variants include classic LPP, FFA, and Graham–Little–Piccardi–Lassueur syndrome.[16],[17] Classic LPP usually involves the vertex and parietal areas of the scalp. The second variant of LPP, FFA, is characterized by frontotemporal band-like loss of both terminal and vellus hairs. Small numbers of isolated hairs are often spared within the band of alopecia, resulting in a finding called the “lonely hair sign.” Similar to classic LPP, erythematous and hyperkeratotic follicles are often found at the peripheral areas of alopecia. Hypopigmentation may be evident in the affected areas. Pruritus or trichodynia are occasionally reported. FFA may involve other scalp sites, such as the periauricular, parietal, or occipital areas.[18]

In our patient, LPP developed shortly after starting paclitaxel chemotherapy for breast cancer. Her initial chemotherapeutic treatment of doxorubicin and cyclophosphamide was tolerated without any skin eruption, but she had anagen effluvium which resolved within 5 months of the completion of the treatment. However, when paclitaxel was introduced, she developed scalp hair loss and pruritic acneiform facial eruptions that was biopsied and found to be LPP. Immune modulators, including tumor necrosis factor inhibitor and tyrosine kinase inhibitor nilotinib, have been reported in drug-induced LPP.[19] However, LPP after use of paclitaxel, an antimicrotubule chemotherapeutic agent or taxane, is uncommon.

The differential diagnosis of LPP secondary to paclitaxel includes fibrosing alopecia in a patterned distribution, central centrifugal cicatricial alopecia, chronic cutaneous lupus erythematosus, long-standing alopecia areata, long-standing traction alopecia, keratosis follicularis spinulosa decalvans, pseudopelade of Brocq, alopecia mucinosa, and familial high frontal hairline.[7] These were ruled out with history, physical examination, dermoscopy, histopathologic evaluation, and normal serum androgens, thyroid hormones, and hematological indices. The majority of chemotherapeutic drugs cause nonscarring alopecia.[20] We believe this is an atypical case of the development of LPP in association with paclitaxel therapy.

LPP remains an interesting disease mimicker and its etiology remains multifactorial. LPP has also been associated with an increased risk of concomitant diseases such as hypothyroidism, hypertension, and metabolic disorders. New associations continue to arise, such as systemic lupus erythematosus and our case of taxol chemotherapy-induced LPP. We suggest that clinicians maintain a high index of suspicion for associated diseases in the presentation of a new LPP pattern. Early initiation of treatment is paramount to preserve the remaining unaffected hair follicles and may be done in tandem with underlying disease.

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.



 
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