Dermatology: Practical and Conceptual Research Letter | Dermatol Pract Concept. 2025;15(4):5851 1 Graham-Little-Piccardi-Lassueur Syndrome: Favorable Response to Isotretinoin Treatment Silvia Guerrero-Cornejo1, Vicente Orellana-Westermeyer2, Josefa Catalán-Lobo2 1 Department of Dermatology, Clinical Hospital of the University of Chile, Santiago, Chile 2 Department of Dermatology, Faculty of Medicine, University of Chile, Santiago, Chile Key words: Graham−Little-Piccardi-Lassueur syndrome, Lichen planopilaris, Scarring alopecia, Isotretinoin Citation: Guerrero-Cornejo S, Orellana-Westermeyer V, Catalán-Lobo J. Graham-Little-Piccardi-Lassueur Syndrome: Favorable Response to Isotretinoin Treatment. Dermatol Pract Concept. 2025;15(4):5851. DOI: https://doi.org/10.5826/dpc.1504a5851 Accepted: April 22, 2025; Published: October 2025 Copyright: ©2025 Guerrero-Cornejo et al. This is an open-access article distributed under the terms of the Creative Commons Attribution- NonCommercial License (BY-NC-4.0), https://creativecommons.org/licenses/by-nc/4.0/, which permits unrestricted noncommercial use, distribution, and reproduction in any medium, provided the original authors and source are credited. Funding: None. Competing Interests: None. Authorship: All authors have contributed significantly to this publication. Corresponding Author: Vicente Orellana-Westermeyer, Dr. Carlos Lorca Tobar 999, Independencia, Santiago, Chile. Email: vicente. orellana@ug.uchile.cl. ORCID: 0009-0007-1507-1863 Introduction Graham-Little-Piccardi-Lassueur syndrome (GLPLS) is a rare subtype of lichen planopilaris (LPP), with approxi- mately 50 reported cases [1]. It is characterized by a triad of scarring alopecia on the scalp, non-scarring alopecia in the axillary and pubic regions, and follicular papules on the body [2-3]. Due to its rarity, treatment is based on case re- ports and includes topical and systemic medications, with variable efficacy. Given the lack of strong evidence, alterna- tive therapeutic approaches are needed to prevent progres- sion to cicatricial alopecia. Case Report A 33-year-old female presented with a three-year history of trichodynia, pruritus, and hair loss. Examination revealed reduced hair density at the vertex, and trichoscopy revealed loss of follicular openings, and hyperkeratosis (Figure 1). His- topathology confirmed LPP, showing perifollicular fibrosis, lymphocytic infiltration, and follicular narrowing (Figure 2). Treatment was initiated with hydroxychloroquine (200 mg every 12 hours), oral minoxidil (1 mg/day), topical clo- betasol 0.05%, ketoconazole 2% shampoo, and monthly intralesional corticosteroid injections. Despite improvement in scalp symptoms and partial hair regrowth, new follicular hyperkeratotic papules appeared on the trunk and extremities alongside patches of non-scarring alopecia in the axillary and pubic regions (Figure 1). Trichos- copy revealed yellow dots and hair thinning. These findings led to a diagnosis of GLPLS, prompting a switch from hy- droxychloroquine to isotretinoin (20 mg three times weekly). After three months, the patient showed resolution of scalp symptoms, repopulation of non-scarring alopecia areas, and a decrease in hyperkeratotic papules. Conclusions GLPLS is the rarest form of LPP, primarily affecting Cauca- sian females aged 30 to 70 years [1,4]. Its etiology remains unclear, though associations with vaccines and hormonal changes have been suggested. Clinically, it manifests as a 2 Research Letter | Dermatol Pract Concept. 2025;15(4):5851 triad of scarring alopecia on the scalp, non-scarring alopecia in the axillary and pubic areas, and hyperkeratotic follicular papules [2,3]. Scalp findings include perifollicular erythema, scaling, and progressive multifocal alopecic patches. Trichos- copy typically reveals perifollicular erythema, fibrotic white dots, hair casts, and loss of follicular openings [5]. Histopathologically, early stages show perifollicular lym- phocytic infiltration at the infundibulum and isthmus, with vacuolar degeneration and keratinocyte necrosis. Advanced stages demonstrate perifollicular fibrosis, loss of piloseba- ceous units, and replacement by fibrous tracts. The disease course varies but often leads to irreversible scarring alope- cia [2,3]. Treatment remains challenging and is primarily extrapo- lated from LPP management. The main goal is to halt disease progression and relieve symptoms. Described therapies in- clude topical, intralesional, or systemic corticosteroids, reti- noids, hydroxychloroquine, cyclosporine, methotrexate, and photochemotherapy, all with varying success rates [1,3]. Early recognition of GLPLS is crucial to initiating timely treatment and preventing irreversible alopecia. This case highlights the importance of comprehensive follow-up in LPP patients, as the GLPLS triad may develop after scarring alopecia onset. While acitretin has been used with a favor- able response, to our knowledge, this is the first case where isotretinoin has been reported as a systemic therapy. Its in- clusion in treatment options is valuable, particularly where acitretin availability is limited. References 1. Alkhayal FA, Alsudairy F, Mubarak luluah, Almohanna HM. Graham−Little Piccardi Lassueur syndrome and review of the literature.  Clinical Case Reports. 2021;9(9). DOI: 10.1002/ ccr3.4761. PMID: 34504697. 2. Shahsavari A, Riley CA, Maughan C. Graham Little Piccardi Lasseur Syndrome. PubMed. Published 2022. https://www.ncbi. nlm.nih.gov/books/NBK537330/ 3. Divine J, Rudnick EW, Lien M. Graham-Little-Piccardi-Lassueur syndrome. Cutis. 2019;103(5):E8-E11. PMID: 31233585. 4. Mardones F, Shapiro J. Lichen planopilaris in a Latin American (Chilean) population: demographics, clinical profile and treat- ment experience. Clin Exp Dermatol. 2017;42(7):755-759. DOI: 10.1111/ced.13203. PMID: 28748570. 5. Li X, Chen X, Zhang J, Zhou C. Graham-Little-Piccardi-Lassueur Syndrome: Report of a Chinese Case with Hair Casts.  Int J Trichology. 2020;12(2):97-98. DOI:10.4103/ijt.ijt_27_20. PMID: 32684685. Figure 1. (A) Alopecic patch on the vertex of the scalp (B) Trichoscopy: erythematous base, loss of follicular open- ings, perifollicular scaling, peripilar casts, and tufted hairs (C) Hyperkeratotic follicular papules on the abdomen. Figure 2. Hematoxylin-eosin stain 40x, decreased number of hair follicles with perifollicular fibrosis and lymphocytic infiltrate.