Majed Aldayhum

and 3 more

INTRODUCTIONMycosis fungoides: Mycosis fungoides (MF) is a primary cutaneous T-cell lymphoma. It represents the most common type of extranodal non-Hodgkin lymphomas 12. Follicular or folliculotropic MF (F-MF, also known as follicular T-cell lymphoma, follicular MF, MF-associated mucinosis, or pleiotropic MF) is an under-recognized subtype of MF. It is characterized histologically by folliculotropism (histologically analogous to epidermotropism) with malignant T cells, with or without associated follicular mucinosis3. In 2005, The WHO/EORTC considered F-MF as a variant of MF in their classification4-7.Mycosis fungoides in the Kingdom of Saudi Arabia: In the Kingdom of Saudi Arabia, few studies are available about MF and F-MF1,8-12. AlGhamdi et al examined the clinicopathologic features of MF in 43 cases of immunohistologically confirmed MF. The average age was 33.5 years was reviewed. The male-to-female ratio was 2:1. Most of the cases presented in an early-stage (I and II) MF. Most of the patients had classic MF and other subtypes included hypopigmented MF and other variants. The patients were followed up for a duration of 27.6 months. The follow-up data indicated that few patients recovered (9.5%) patients recovered; whereas the majority (83.3%) had persistent MF skin lesions. A single patient had the extracutaneous disease; whereas only two patients died of MF 8.Al-Dawsari et al examined the surgical pathology records of Saudi patients from 1995 to 2014 at the Johns Hopkins Aramco Healthcare Center (Eastern Province of KSA). This medical facility is the healthcare provider for the employees working at Saudi Aramco company and their dependents in the Eastern Province of Saudi Arabia. The total number of primary skin tumors was 204 and out of them 22 cases were MF10. Albasri et al examined the pattern of skin cancer in the Madinah region of KSA. Among the 202 cancer cases examined in their series, there were 14 (6.8%) cases of MF. The mean age of the patients was 30.7 years with a male-to-female ratio of 11:3. The distribution of the MF skin lesions included: 4 cases (head and neck), 3 cases (upper limb), and 7 cases (lower limb) 11.Alsaif et al described the Leser-Trelat sign (sudden appearance of seborrheic keratosis) in a 60-year-old Saudi male patient with MF. The lesions appeared on the face and back 12. Alojail et reported the clinical features and treatment regimens in 34 cases of MF in KSA. The cases included both hyperpigmented (11 cases) and hypopigmented (21 cases) MF, poikilodermatous MF, and pagetoid reticulosis (a single, each). The treatment regimens included phototherapy Narrowband UVB (NB-UVB) in combination with topical corticosteroids, phototherapy NB-UVB in combination with the topical corticosteroid, and phototherapy NB-UVB with topical corticosteroid and systemic acitretin 1.Alghubaywi et al examined all MF cases diagnosed at King Abdulaziz Medical City in KSA (January 2016 to July 2022). A total of 73 patients were included in their study. The incidence of MF was slightly higher in females (male to female ratio: 1: 1.3). The average age was 44 years. The most common MF subtypes included classic MF, followed by hypopigmented MF. Most of the cases were presented at an early-stage MF (IA, IB, and IIA). The treatment option was mainly in the form of topical steroids. Immunohistological staining of CD4+/CD8+ with CD8 predominance was common and this may reason for the favorable disease outcome 13. These authors also indicated that MF was under-recognized with 20% of the patients with MF initially misdiagnosed as atopic dermatitis. The correct diagnosis of MF was rendered within a mean of 33 months from the time of presentation to the Dermatology clinics. The authors reasoned the delay in the diagnosis of MF the fact that chronic dermatitis and MF share several clinical manifestations such as the presence of pruritic, erythematous, scaly plaques 13. A summary of the previous studies is shown in Table 1.Follicular mucinosis: Follicular mucinosis, i.e. accumulation of mucin in the epithelium of the hair follicles can be either a primary idiopathic disorder or an associated event with several inflammatory conditions (such as chronic spongiotic dermatitis), benign or malignant neoplasms14-16. In 1957, Pinkus first described mucin deposits in the follicular epithelium (alopecia mucinosa) that are associated with folliculotropism, i.e., infiltration of the epithelial cells of the outer hair sheath by atypical lymphocytes17. In 1959, the term follicular mucinosis was introduced by Jablonska 18. There are two clinical types of follicular mucinosis: idiopathic follicular mucinosis and lymphoma-related follicular mucinosis, i.e., F-MF. The latter can be separated from the classical MF on a clinical and histological basis2,19,20. Currently, there is no specific therapy for idiopathic follicular mucinosis and the skin lesions can resolve spontaneously within a period of 2 to 24. Some therapeutic options include steroids (topical, intralesional, and systemic steroids), photodynamic therapy, antimalarials, interferon, and dapson21,22.Folliculotropic mycosis fungoides: F-MF represents the most common non-classic variant of MF, representing approximately 10% of the cases of MF 23. It usually shows a male predominance and most of the cases occur in adults 24. Its clinical findings include follicle-based infiltrated erythematous grouped papules resembling acne or keratosis pilaris, patches, plaques, and tumor-like lesions with follicular accentuation 2,20,25. Other clinical presentations include prurigo-like lesions, pseudotumors, lichen spinulosus-like lesions, rosacea-like lesions, and lupus tumidus-like plaques 26. Pruritis is a common complaint and it is aggravated by superinfection by staphylococcus auras resulting in pyoderma 25. The lesions of F-MF vary from solitary lesions to extensive lesions giving the appearance of a leonine face 23. The head and neck region is the most commonly affected site. Other sites include the trunk, and the extremities 17. The features of F-MF are seen in nearly 10% of the patients with MF 2,20,25. The salient histological features of F-MF include perivascular and peri‑adnexal dermal lymphocytic infiltrates with variable infiltration and destruction of the follicular epithelium by small, medium‑sized, or large-sized lymphocytes with hyperconvoluted nuclei (cerebriformnuclei). The epidermis is usually spared. Other histological findings include follicular mucinosis, Pautrier’s Micro-abscess, follicular plugging, tagging of the atypical lymphocytes along the follicular epithelium, peri-eccrine infiltrate of atypical lymphocytes23,26. Immunohistochemistry usually reveals prominent CD3 positive infiltrate, witha predominance of CD4 positive lymphocytes over rare CD8 positive T-cells. CD7 expression is reduced or completely lost. Molecular analysis (T-cell receptor-γ gene rearrangement) reveals a monoclonal T-cell population23,26-28. The therapeutic options in F-MF include photochemotherapy combined with interferon alpha-2a and retinoids. Other options include local radiotherapy, topical imiquimod, and total body electron beam irradiation 23.Several F-MF case reports were presented in the literature. Monopoli et al described two adult patients with F-MF. The clinical manifestations included alopecia and follicular erythematous papules, and comedones and cysts, respectively. The histological findings included folliculotropic atypical T-cell infiltrate that extends to the epidermis. There was no associated mucinosis. Clonality analyses revealed the oligo/monoclonal nature of the T-cell lymphocytic infiltrate 27.Rajalakshmi et al examined four cases of F-MF affecting the skin of the face. The mean age of the patients was 17.5 years and there were no differences in gender distribution. The lesions were in the form of hypopigmented patches or erythematous plaques. Two patients had associated alopecia. The salient histological finding included folliculotropism, mucin deposition around the hair follicles, lymphocyte tagging with haloes, atypical lymphocytes with hyperconvoluted/cerebriform nuclei, and infiltration of the eccrine and sebaceous glands by atypical lymphocytes. Other histological features included the presence of epithelioid cells, parakeratosis, and a prominent infiltrate of eosinophils 29. Magro et al reported 6 cases of unilesional follicular MF. All the patients were males with a mean age of 28 years. The patients were presented with a solitary lesion on the face and scalp (five patients) and trunk (a single patient) that persisted for a few months. There were follicular prominence and alopecia. On histology, there was an atypical folliculotropic lymphocytic infiltrate associated with follicular mucinosis. Immunohistology revealed a high CD4: CD8 ratio and loss of CD7 expression. A single untreated case developed similar lesions over the skin of the thigh and buttock after 3-4 years 28. F-MF usually occurs in adults with an average age at the time of diagnosis of 60 years 30. F-MF is exceptionally rare in children and adolescents. Mantri et al reported a case of F-MF in a 16-year-old boy who presented with 6x7 cm plaque on the forehead for a 2-month duration. Immunohistological examination revealed dense atypical T-cell lymphocytes invading and destroying the hair follicles. Follicular mucinosis was also seen. Most of the neoplastic cells were CD4-positive T- T-lymphocytes with scarce CD8-positive T cells around the hair follicles. Treatment was in the form of a spot electron beam and the lesion completely resolved within 2 months 30. Interestingly, Emge et al reported a case of F-MF in a 6-year-old boy. The lymphoma was associated with idiopathic follicular mucinosis31.Taken collectively, these previous reports indicated that F-MF is a rare and aggressive form of MF that has a worse prognosis compared to conventional MF. It usually affects the head and neck region in adults. It has distinct histological features and may or may not be associated with follicular mucinosis. F-MF has several clinical presentations and many histological faces. Therefore, it is often misdiagnosed, or diagnosed with much delay at an advanced stage as compared to the conventional MF.Herein, we report a case of F-MF in a middle-aged male patient. To date and to be best of our knowledge, this is the first case of F-MF to be reported in the Southern region (Asir region) of the KSA. The clinicopathologic features were discussed.