Generic selectors
Exact matches only
Search in title
Search in content
Search in posts
Search in pages
Filter by Categories
15th National Conference of the IAOMFP, Chennai, 2006
Abstract
Abstracts from current literature
Acne in India: Guidelines for management - IAA Consensus Document
Addendum
Announcement
Art & Psychiatry
Article
Articles
Association Activities
Association Notes
Award Article
Book Review
Brief Report
Case Analysis
Case Letter
Case Letters
Case Notes
Case Report
Case Reports
Clinical and Laboratory Investigations
Clinical Article
Clinical Studies
Clinical Study
Commentary
Conference Oration
Conference Summary
Continuing Medical Education
Correspondence
Corrigendum
Cosmetic Dermatology
Cosmetology
Current Best Evidence
Current View
Derma Quest
Dermato Surgery
Dermatopathology
Dermatosurgery Specials
Dispensing Pearl
Do you know?
Drug Dialogues
e-IJDVL
Editor Speaks
Editorial
Editorial Remarks
Editorial Report
Editorial Report - 2007
Editorial report for 2004-2005
Errata
Erratum
Focus
Fourth All India Conference Programme
From Our Book Shelf
From the Desk of Chief Editor
General
Get Set for Net
Get set for the net
Guest Article
Guest Editorial
History
How I Manage?
IADVL Announcement
IADVL Announcements
IJDVL Awards
IJDVL AWARDS 2015
IJDVL Awards 2018
IJDVL Awards 2019
IJDVL Awards 2020
IJDVL International Awards 2018
Images in Clinical Practice
In Memorium
Inaugural Address
Index
Knowledge From World Contemporaries
Leprosy Section
Letter in Response to Previous Publication
Letter to Editor
Letter to the Editor
Letter to the Editor - Case Letter
Letter to the Editor - Letter in Response to Published Article
LETTER TO THE EDITOR - LETTERS IN RESPONSE TO PUBLISHED ARTICLES
Letter to the Editor - Observation Letter
Letter to the Editor - Study Letter
Letter to the Editor - Therapy Letter
Letter to the Editor: Articles in Response to Previously Published Articles
Letters to the Editor
Letters to the Editor - Letter in Response to Previously Published Articles
Letters to the Editor: Case Letters
Letters to the Editor: Letters in Response to Previously Published Articles
Medicolegal Window
Messages
Miscellaneous Letter
Musings
Net Case
Net case report
Net Image
Net Letter
Net Quiz
Net Study
New Preparations
News
News & Views
Obituary
Observation Letter
Observation Letters
Oration
Original Article
ORIGINAL CONTRIBUTION
Original Contributions
Pattern of Skin Diseases
Pearls
Pediatric Dermatology
Pediatric Rounds
Perspective
Presedential Address
Presidential Address
Presidents Remarks
Quiz
Recommendations
Regret
Report
Report of chief editor
Report of Hon : Treasurer IADVL
Report of Hon. General Secretary IADVL
Research Methdology
Research Methodology
Resident page
Resident's Page
Resident’s Page
Residents' Corner
Residents' Corner
Retraction
Review
Review Article
Review Articles
Revision Corner
Self Assessment Programme
SEMINAR
Seminar: Chronic Arsenicosis in India
Seminar: HIV Infection
Short Communication
Short Communications
Short Report
Special Article
Specialty Interface
Studies
Study Letter
Supplement-Photoprotection
Supplement-Psoriasis
Symposium - Contact Dermatitis
Symposium - Lasers
Symposium - Pediatric Dermatoses
Symposium - Psoriasis
Symposium - Vesicobullous Disorders
SYMPOSIUM - VITILIGO
Symposium Aesthetic Surgery
Symposium Dermatopathology
Symposium-Hair Disorders
Symposium-Nails Part I
Symposium-Nails-Part II
Tables
Technology
Therapeutic Guidelines
Therapeutic Guidelines - IADVL
Therapeutics
Therapy
Therapy Letter
View Point
Viewpoint
What’s new in Dermatology
View/Download PDF
Letter to the Editor - Therapy Letter
2017:83:6;710-713
doi: 10.4103/ijdvl.IJDVL_1077_16
PMID: 28984625

Low-dose methotrexate combined with superficial X-ray in the treatment of folliculotropic mycosis fungoides: A case report

Yanan Jiang1 , Ruzeng Xue2 , Weijia Wang2 , Yongfeng Chen2
1 Graduate School, Guangdong Medical University, Zhanjiang; Dermatology Department, Guang Dong Provincial Dermatology Hospital, Guangzhou, China
2 Dermatology Department, Guang Dong Provincial Dermatology Hospital, Guangzhou, China

Correspondence Address:
Yongfeng Chen
Guang Dong Provincial Dermatology Hospital, No 2, LuJing Road, Guangzhou
China
Published: 06-Oct-2017
How to cite this article:
Jiang Y, Xue R, Wang W, Chen Y. Low-dose methotrexate combined with superficial X-ray in the treatment of folliculotropic mycosis fungoides: A case report. Indian J Dermatol Venereol Leprol 2017;83:710-713
Copyright: (C)2017 Indian Journal of Dermatology, Venereology, and Leprology

Sir,

According to the World Health Organization-European Organization for Research and Treatment of Cancer (WHO-EORTC), folliculotropic mycosis fungoides (FMF) is categorized as a variant of mycosis fungoides (MF) because of its distinctive clinicopathologic features.[1] Skin findings include grouped follicular or acneiform papules, such as clustered comedones, nodulocystic lesions, pustules, milia, as well as indurated plaques or tumors. Representative skin lesions typically involve the head and neck, especially the eyebrow, showing plaques and alopecia with severe pruritus.[2] Histopathologically, the follicular epithelium is infiltrated with atypical lymphocytes, with relative epidermal sparing.[3] This variant is associated with a worse prognosis than classical mycosis fungoides because of its resistance to conventional therapies.[4]

A 51-year-old female was admitted to our outpatient department, with a 10-year history of erythematous papules and plaques associated with severe pruritus. Clinical examination revealed alopecia and erythematous, scaly papules, and plaques scattered on the head, neck, trunk, and extremities [Figure - 1] and [Figure - 2]. Alopecia only involved the scalp. Lesions of the head and neck showed infiltration and erosion [Figure - 1]. There was no palpable lymphadenopathy or hepatosplenomegaly. Laboratory investigations such as blood count, urinalysis, biochemical profile, serum protein electrophoresis levels, and bone marrow aspiration were all within normal limits. Imaging studies including chest X-ray, computed tomographic scan, and echocardiography were normal. Skin biopsy revealed intrafollicular or perifollicular infiltration of atypical lymphocytes with minimal epidermal involvement [Figure - 3]. In addition, intrafollicular mucin deposits [Figure - 4] were observed on Alcian blue stain [Figure - 5]. The overlying epidermis showed focal epidermotropism, and few lymphocytes showed mild cytological atypia [Figure - 6]. Immunohistochemically, the atypical lymphocytes were positive for CD2, CD3, CD4, CD7, and CD8, with a normal CD4:CD8 ratio, which favored T-cell proliferation. Staining for CD20, CD79a, CD30, and CD68 showed negative results. T-cell receptor gamma (TCR gamma) rearrangement confirmed a monoclonal proliferation of the atypical T lymphocytes. Considering the clinical and histopathological examination, diagnosis of stage IIA folliculotropic mycosis fungoides was made.

Figure 1: Erythematous papules and plaques, scales, and erosion involving the face and neck
Figure 2: Erythematous papules, plaques, and scales scattered on the trunk
Figure 3: Intrafollicular and perifollicular infiltration with atypical lymphocytes and minimal epidermal involvement (H and E, ×100)
Figure 4: Follicular infiltration with mucin deposition (H and E, ×100)
Figure 5: Alcian blue staining showing intrafollicular mucin (×200)
Figure 6: Epidermis showing focal epidermotropism and few lymphocytes showing mild cytological atypia (H and E, ×200)

Initially, the patient was treated with interferon α-2b, isotretinoin, nitrogen mustard, and psoralen-UVA for 1 month, resulting in minimal improvement. Subsequently, methotrexate was added for 1 month, which resulted in an excellent response [Table - 1]. The initial dose of methotrexate was 10 mg per week and the maximal dose was 15 mg per week. Most lesions on the trunk and extremities disappeared after these treatments. However, some persistent plaques remained on the head and neck. The patient was then treated with a combination of methotrexate 10 mg weekly plus superficial X-ray (100 kV, 400 cGy~500 cGy, a total dose of 40 Gy for 9 cycles) per week for 3 months [Table - 2], which resulted in dramatic remission [Figure - 7]. The patient continues to be in clinical remission after 10 months.

Figure 7: Lesions on the face and neck almost disappeared
Table 1: Initial treatments
Table 2: Treatment plan used from September 2015 November 2016

Folliculotropic mycosis fungoides, a distinct variant of mycosis fungoides, is characterized by the presence of folliculotropic infiltrates, that often spare the epidermis and preferentially involve the head and neck area.[1] It is associated with a worse prognosis than classical mycosis fungoides, due to resistance to conventional therapies such as isotretinoin, methotrexate, PUVA, and interferon.[4] In our patient, most lesions responded well to conventional therapies, except for persistent lesions on the head and neck area. Agar et al.[5] reported that a patient with localized thick plaques on the head, was treated successfully with superficial radiotherapy. Demirkesen et al.[4] reported that patients with folliculotropic mycosis fungoides, who received more aggressive and combined treatments such as PUVA, methotrexate, interferon, and radiotherapy, achieved a higher survival rate. Kelly et al.[6] reported that patients in stages I–III, who were treated with topical carmustine as monotherapy, with or without an added systemic agent demonstrated a high degree of response. In addition, a 16-year-old patient who presented with a single, firm, well-circumscribed erythematous infiltrated plaque over the forehead was successfully treated with local electron beam therapy.[7] Since the persistent lesions were localized in our patient, we chose X-ray as an additional treatment. She showed dramatic remission-a fact which may help direct future treatment guidelines of this condition. We were unable to find previous reports of the combination treatment of methotrexate with superficial X-ray in our literature search.

This regimen – methotrexate with superficial X-ray therapy – may be a new viable therapeutic option for recalcitrant mycosis fungoides, especially the folliculotropic variant.

Acknowledgment

The authors would like to thank the pathology department of Guang Dong Provincial Dermatology Hospital for their contribution to the histopathology images and thank Tiange Zhang for his help of revision.

Declaration of patient consent

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

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

References
1.
Hodak E, Amitay-Laish I, Atzmony L, Prag-Naveh H, Yanichkin N, Barzilai A, et al. New insights into folliculotropic mycosis fungoides (FMF): A single-center experience. J Am Acad Dermatol 2016;75:347-55.
[Google Scholar]
2.
Spence-Shishido A, Streicher JL, George RP, Parker SR, Lawley LP. Folliculotropic mycosis fungoides as a posttransplant lymphoproliferative disorder. Pediatrics 2015;136:e701-5.
[Google Scholar]
3.
Amitay-Laish I, Feinmesser M, Ben-Amitai D, Fenig E, Sorin D, Hodak E. Unilesional folliculotropic mycosis fungoides: A unique variant of cutaneous lymphoma. J Eur Acad Dermatol Venereol 2016;30:25-9.
[Google Scholar]
4.
Demirkesen C, Esirgen G, Engin B, Songur A, Oguz O. The clinical features and histopathologic patterns of folliculotropic mycosis fungoides in a series of 38 cases. J Cutan Pathol 2015;42:22-31.
[Google Scholar]
5.
Agar N, Whittaker SJ. Folliculotropic mycosis fungoides (stage IIA) progressing to Sézary syndrome: A case report. Br J Dermatol 2008;159:1197-9.
[Google Scholar]
6.
MacArthur KM, Jariwala N, Kim EJ, Rook AH. Topical carmustine as monotherapy or as multimodality therapy for folliculotropic mycosis fungoides. Acta Derm Venereol 2017;97:373-4.
[Google Scholar]
7.
Mantri MD, Khadke MP, Ameet DL, Rachita DS. Folliculotropic mycosis fungoides in an adolescent: A rare case. Indian J Dermatol 2016;61:467.
[Google Scholar]

Fulltext Views
188

PDF downloads
46
Show Sections