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PubMed2002

Treatment of childhood mycosis fungoides with topical PUVA.

Mycosis fungoides is the most common type of cutaneous T-cell lymphoma, which is usually observed in mid to late adulthood. We report 5 cases of mycosis fungoides in children, all presenting as patch- and plaque-stage disease most commonly involving the buttocks. Histologic examination showed in every case the typical features of mycosis fungoides. In 4 of the 5 cases, the infiltrating lymphocytes were characterized by the T-cell phenotype CD3(+), CD4(+), CD8(+); and in 3 cases, a monoclonal rearrangement of the T-cell receptor gamma (TCR-gamma) gene was found. Three children received topical PUVA treatment, and the other two were treated with mid-potency topical corticosteroids, resulting in complete clinical remission. A management approach to mycosis fungoides with topical PUVA may be appropriate for children.

Buttocks↗
PubMed2003

Juvenile mycosis fungoides diagnosed before 18 years of age.

The literature regarding mycosis fungoides in children is sparse. To shed further light on the characteristics of mycosis fungoides in the paediatric population we analysed the clinicopathological features of 10 patients in whom this malignancy was diagnosed before the age of 18 years. All were Jews and Arabs with histologically proven patch/early plaque stage disease: 4 in stage IA, 4 in IB and 2 with unilesional disease. Seven patients had hypopigmented lesions either constituting the sole manifestation (2 patients) or in combination with classic lesions (5 patients); of these, 3 had light skin and 4 pigmented skin. Most patients had immunohistochemical features characteristic of mycosis fungoides, with a predominance of CD4+ T cells. Some had deletion of CD7+ cells. In 3 patients, however, the epidermotropic cells were exclusively or predominantly CD8+ cells. All patients responded to conventional therapy and during an average follow-up of 3.4 years only one patient showed stage progression, but without extracutaneous involvement. It is concluded that juvenile mycosis fungoides is characterized by early stage disease, occasionally with unilesional disease, usually with hypopigmented lesions irrespective of skin colour, and a good response to therapy. On the basis of our experience and review of the literature, it appears that the CD8+ phenotype is over-represented in juvenile disease.

Adolescent↗
PubMed2005

Cytokine gene polymorphisms in patch-stage mycosis fungoides.

Cytokine production is under genetic control and certain allelic variants of cytokine genes are associated with lower or higher cytokine production in vitro and in vivo. The general concept is that a shift from a Th1 to a Th2 cytokine profile accompanies disease progression from patch-stage mycosis fungoides to tumour stage, although the results of the studies carried out have not been entirely conclusive. We aimed to investigate whether certain cytokine polymorphisms might represent a risk factor for developing patch-stage mycosis fungoides. Genotyping for IFN-gamma (Th1 cytokine), IL-6, IL-10 (Th2 cytokines), TNF-alpha and TGF-beta 1 was undertaken for 33 patients with patch-stage mycosis fungoides and the results were compared with those in a control group. Genotype distribution showed no significant differences between the patients and the controls for any of the five cytokines studied. Our study suggests that patch-stage mycosis fungoides is not determined by a specific genotype polymorphism. However, further studies on larger numbers of cases are needed before definite conclusions can be drawn.

Adolescent↗
PubMed2005

A retrospective study of the probability of the evolution of parapsoriasis en plaques into mycosis fungoides.

Parapsoriasis en plaque has been suggested to be an early manifestation of mycosis fungoides (cutaneous T-cell lymphoma). We explored the disease course of patients with small plaque or large plaque parapsoriasis in a 26-year retrospective cohort analysis of 105 parapsoriasis patients, who were clinically and histopathologically followed up in Helsinki and Tampere University Hospitals. Eventual later cancers of these patients were verified from the Finnish Cancer Registry. In the small plaque parapsoriasis group, 7 patients (10%) and in the large plaque parapsoriasis group 12 patients (35%), developed histologically confirmed mycosis fungoides during a median of 10 and 6 years, respectively. No significant differences were found regarding the risk of developing mycosis fungoides or the tendency to remission in patients treated with or without phototherapy. Our results show that not only large plaque parapsoriasis, but also small plaque parapsoriasis, as currently defined in textbooks, can progress to mycosis fungoides. The benefits of phototherapy are equivocal in parapsoriasis treatment as far as progression to cancer is concerned.

Adult↗
PubMed1984

Death due to splenic rupture in suppressor cell mycosis fungoides: a case report.

A case of mycosis fungoides in which pathologic rupture of the spleen led to intraperitoneal hemorrhage and death is described. To our knowledge, splenic rupture has not been reported previously as a cause of death in mycosis fungoides. Immunologic studies demonstrated that the neoplastic cell was a suppressor/cytotoxic T-cell. In most cases of mycosis fungoides or the Sézary syndrome, the neoplastic cell has been a helper/inducer T-cell. This case was very aggressive clinically with prominent visceral involvement and suggests that mycosis fungoides may be clinically diverse as well as immunologically heterogeneous.

Aged↗
PubMed1999

Biliary tract obstruction secondary to mycosis fungoides: a case report.

Mycosis fungoides is a cutaneous T-cell lymphoma that can disseminate to multiple organs. We report a patient who presented with obstructive jaundice caused by isolated involvement of the extrahepatic biliary tree by mycosis fungoides. Initially, endoscopic examinations and biopsies of the biliary tree and liver failed to reveal a cause for the obstructive symptoms. Finally, surgical resection of the gallbladder and extrahepatic ducts was performed. Examination revealed a dense, mixed lymphocytic infiltrate with atypical cells within the mucosa. Gene rearrangement studies confirmed the presence of a monoclonal T-cell population. The pattern of the gene rearrangement in the biliary tree was identical to that found in a previous skin biopsy that showed mycosis fungoides. Although liver involvement by mycosis fungoides is not uncommon, disease isolated to the extrahepatic biliary tree has not previously been reported. This case should alert clinicians and pathologists to yet another cause of obstructive jaundice.

Adult↗
PubMed2005

Pseudotumorous folliculotropic mycosis fungoides.

Fungating nodules and infiltrated plaques are usually equated with advanced tumor stage mycosis fungoides. We report an 85-year-old man who presented in this way but multiple skin biopsies revealed that the bulk of his nodules were due to marked follicular hyperplasia as a result of folliculotropic mycosis fungoides. This clinical presentation may be best described as a pseudotumorous form of mycosis fungoides dominated by follicular epithelial hyperplasia rather than lymphocytic proliferation characteristic of true tumor stage disease. Similar presentations have been described as a verrucous and hyperplastic variant of mycosis fungoides due to the presence of prominent epidermal hyperplasia.

Aged↗
PubMed1985

Expression of Leu-8 antigen, a majority T-cell marker, is uncommon in mycosis fungoides.

Predominance of mature helper T cells with the Leu-1+, 2-, 3+, 4+, 5+ phenotype was confirmed in 22 biopsy specimens of mycosis fungoides from 15 patients. Dissection of the T helper/inducer cells into phenotypically distinct subsets was performed with the use of a new monoclonal antibody, anti-Leu-8. One might predict a predominance of Leu-8+ in mycosis fungoides, as the known ratio of Leu-8+/Leu-8- cells is approximately 70/30 in the peripheral blood. Unexpectedly, a deficiency of Leu-8 was demonstrated in 18 of the 22 specimens from 13 of 15 patients. This finding could not be attributed to an artifact of the staining method or to therapy, and was present in early- as well as late-stage disease. Whether neoplastic cells in mycosis fungoides derive from Leu-8-subset of T cells at risk for malignant transformation, or whether there is antigen loss with malignant transformation remains to be determined. Implications of our finding with regard to etiopathogenesis of mycosis fungoides are discussed.

Antibodies, Monoclonal↗
PubMed1991

Cell-bound IgE and increased expression of Fc epsilon-receptors on dendritic cells in cutaneous infiltrates of mycosis fungoides.

Skin biopsies of 31 non-atopic patients, 20 with mycosis fungoides, six with psoriasis and five with contact dermatitis, and of five non-atopic healthy controls were compared for the presence of cell-bound IgE and vacant IgE binding sites. IgE+ cells were demonstrated in the cutaneous infiltrate of nine (45%) patients with mycosis fungoides, two (33%) with psoriasis and one (20%) with contact dermatitis. Following pre-incubation of skin sections with IgE myeloma protein to saturate vacant IgE-binding sites, 14 out of 16 patients (88%) with stage I mycosis fungoides, five (83%) patients with psoriasis and one (20%) with contact dermatitis showed an increase in the number of IgE+ cells. While cell-bound IgE was positively related to serum IgE levels the expression of IgE-binding sites was not. All IgE+ cells were HLA-DR+ dendritic cells identified as either macrophages (CD68+, CD14+) or Langerhans cells (CD1+). Skin biopsies of non-atopic healthy controls or clinically uninvolved skin in mycosis fungoides had neither any IgE+ cells nor any vacant binding sites. Inhibition studies with IgG1, IgG4 and IgE myeloma proteins as well as with several enzymatic fragments of IgE demonstrated that IgE interacted with Fc epsilon-receptors through isotype-specific structures on the Fc epsilon-fragment. Four anti-CD23 monoclonal antibodies, however, were unable to stain vacant Fc epsilon-receptors nor could they block IgE-binding. We hypothesize that locally-secreted lymphokines, like IL-4 or interferon-gamma, induce Fc epsilon-receptors on dendritic cells in the cutaneous infiltrate and that these receptors become occupied in parallel with elevated serum IgE levels.

Antigens, CD↗
PubMed2004

Milia in regressing plaques of mycosis fungoides: provoked by topical nitrogen mustard or not?

OBJECTIVE: To report three cases of mycosis fungoides with milia formation in the regressing lesions. PATIENTS AND SETTING: Dermatology clinic of a university hospital (referral center). Three patients with mycosis fungoides with body surface involvement of 10% in one case (stage IIb) and exceeding 30% in two cases (stages IIb and III). All patients were treated with photochemotherapy and topical nitrogen mustard ointment in a concentration of 0.01%. After approximately 3 months multiple milia erupted on regressing plaques. RESULTS: The presence of milia was evident and was confirmed by histopathology. Regression of mycosis fungoides was noted in these plaques both clinically and in comparison with the pretreatment histologic appearance. Two of the patients showed a histological picture of follicular mucinosis. CONCLUSIONS: We do not know the significance of milia in mycosis fungoides (MF). However, we suggest that follicular rupture or a degenerative process might result in milia formation.

Administration, Cutaneous↗
PubMed2005

Mycosis fungoides--a retrospective study of 40 cases in Hong Kong.

BACKGROUND: Mycosis fungoides is rare in Hong Kong and oriental data on the disease are lacking. METHODS: This is a multiclinic, 13-year, retrospective study to determine the clinicopathologic characteristics, treatment, and disease outcomes of 40 patients with mycosis fungoides/Sézary syndrome seen in the Social Hygiene Service, Hong Kong. RESULTS: There were 27 males and 13 females with a mean age at diagnosis of 56.4 years. Based on figures in the Social Hygiene Service alone, the incidence in Hong Kong was estimated to be 0.044 per 100,000. Eighty-five per cent of patients presented with skin-limited disease, and pruritus was absent in 40% of patients. A mean of 1.48 biopsies was needed to establish the diagnosis, and only 58% of the mycosis fungoides skin biopsies were reported to be histologically diagnostic of the disease. Atypical lymphocytes, epidermotropism, interface changes, and Pautrier's microabscesses were the four most frequently encountered features that attained statistical significance. Sixty-nine per cent of patients were treated with psoralen-UVA as their initial therapy, and the complete response and relapse rates were 78.3% and 66.6%, respectively. Disease progression to more advanced stages was only seen in 15% of patients. The 5-year survival rates for the whole group and for stage IA and IB patients were 88.8% and 100%, respectively. CONCLUSION: Mycosis fungoides is rare amongst Hong Kong Chinese and the majority present with skin-limited nonprogressive disease.

Aged↗
PubMed2005

Multiple squamous cell carcinomas in a patient with mycosis fungoides.

A 51-year-old man with type IV skin presented for evaluation of a generalized rash associated with multiple ulcerated, nodular lesions on his legs. The nodular lesions occurred approximately 18 months after the initial onset of generalized rash, which had been diagnosed as plaque/patch stage mycosis fungoides. He continued to develop further nodular lesions on his trunk in the weeks following presentation. The nodular lesions were shown to be squamous-cell carcinoma on histopathology. He had received only topical hydrocortisone prior to the development of the second cutaneous malignancy and had no past exposure to carcinogens. His squamous cell carcinomas were treated with surgical excision and split-skin grafting. He received total skin electron-beam therapy to treat the mycosis fungoides. Second malignancy in mycosis fungoides is a recognized phenomenon and usually occurs after potentially carcinogenic therapy. This case demonstrates the occurrence of second malignancy in the absence of a precipitating factor, suggesting that there are innate, immune-mediated mechanisms in the development of cancer in patients with mycosis fungoides.

Acitretin↗
PubMed2006

Retrospective 5-year review of 131 patients with mycosis fungoides and Sézary syndrome seen at the National Skin Centre, Singapore.

A total of 131 new cases of mycosis fungoides and Sézary syndrome were diagnosed clinically and histopathologically at our centre over a 5-year period. There were 87 males and 44 females with a mean age of 36.3 years (range 3-87 years) and no racial predilection. Of the 62 patients (47.3%) with classical mycosis fungoides, the majority were male (male : female = 4.2:1). There was one patient with Sézary syndrome. Patients aged older than 50 years were more likely to present with a longer duration of symptoms and advanced disease. In contrast to classical mycosis fungoides, the 47 patients diagnosed with hypopigmented mycosis fungoides had early stage disease, were younger, and no gender predilection was noted. The mean duration of follow up was 19.7 months (range 0.2-54.8 months). Complete remission was achieved in 24.7% and 53.8% of patients followed up at 1 and 3 years, respectively, using skin-directed and systemic treatment modalities appropriate for the stage of disease. There were five patients with progressive disease and three patients with advanced disease who died from disease-related complications. The most significant prognostic factors for 1-year and 3-year outcomes were the patient's duration of symptoms and stage of disease at presentation.

Adolescent↗
PubMed1982

Variably acid-fast pleomorphic bacteria as a possible cause of mycosis fungoides. A report of a necropsied case and two living patients.

Variably acid-fast coccoid forms, suggestive of acid-fast, cell-wall-deficient forms of mycobacteria, were observed in microscopy of sections of skin from three patients with mycosis fungoides. Similar coccoid forms were also seen in sections of the heart, lung, liver, lymph nodes, connective tissue, and other organs of one of them who died, and in biopsy material from lymph nodes obtained one year antemortem in that patient. The present findings of microbes in mycosis fungoides may be related to the previous findings of acid-fast bacteria in mycosis fungoides and other malignancies as reported by other investigators. The present histopathologic findings also suggest that mycosis fungoides may be an infectious disease of mycobacterial origin.

Adult↗
PubMed1981

Pagetoid reticulosis and solitary mycosis fungoides. Distinct clinicopathological entities.

The two cases reported in this paper allow the differentiation of pagetoid reticulosis from solitary lesions of mycosis on the basis of clinical, histological ultrastructural, immunohistochemical and enzyme histochemical studies. The first patient presented with a slowly growing asymptomatic scaly plaque of 30 years duration. Histology of the lesion was typical of pagetoid reticulosis. Immunohistochemical studies showed that the abnormal cells present in the cutaneous infiltrate were not T lymphocytes and ultrastructural studies suggested these were of histocytic origin. The second patient presented with a poikilodermatous lesion present for 14 years with histology of plaque stage mycosis fungoides. Enzyme and immunohistochemistry showed the predominant T cell nature of the cutaneous infiltrate and electron microscopy showed numerous mycosis fungoides cells. In contrast to generalized forms of mycosis fungoides, no peripheral blood monocyte chemotactic defect was found in either case. Both lesions responded to radiotherapy.

Adult↗
PubMed1999

Mycosis fungoides and cutaneous Hodgkin's disease in the same patient: a case report.

We describe a case of a 34-year-old man who presented with a 10-year history of mycosis fungoides and recent onset of inguinal lymphadenopathy, fever, and cutaneous nodules on the right hip. Biopsy of the inguinal lymph node showed mixed cellularity Hodgkin's lymphoma. Biopsy of the pre-existing skin lesions showed mycosis fungoides, while that of the nodules on the hip revealed cutaneous Hodgkin's lymphoma. Immunohistochemical and molecular analysis demonstrated a null phenotype in the Reed-Sternberg cells of the Hodgkin's disease lesions, and a T-cell phenotype in the lymphoid cells of the mycosis fungoides lesions. He was treated with chemotherapy for Hodgkin's disease, which is in remission 11 years later. Topical nitrogen mustard and maintenance PUVA therapy have been used for the mycosis fungoides, which is also in remission.

Adult↗
PubMed1999

Mycosis fungoides with mucinosis follicularis in childhood.

Mycosis fungoides is a cutaneous T-cell lymphoma (CTCL) usually observed in mid to late adulthood. It occurs only rarely during childhood. Follicular mucinosis is a chronic dermatosis involving the sebaceous glands and outer root sheaths. It is normally differentiated into a juvenile benign form and an adult form possibly associated with mycosis fungoides. We report a 12-year-old boy who presented with an 8-month history of erythematous mucinous plaques on the scalp. Three months later, he developed erythematous patches and plaques on his whole body, accompanied by cervical lymphadenopathy. A biopsy showed follicular mucinosis and epidermotropism of the lymphocytic infiltrate. Immunophenotyping and a PCR clonality test were consistent with CTCL. The patient received PUVA treatment and local steroids, resulting in partial remission. Mycosis fungoides should be considered in the differential diagnosis of chronic, scaling dermatoses in childhood. Moreover, follicular mucinosis in childhood can be associated with mycosis fungoides.

Child↗
PubMed2003

Mycosis fungoides with follicular mucinosis displaying aggressive tumor-stage transformation : successful treatment using radiation therapy plus oral bexarotene combination therapy.

Follicular mucinosis is a tissue reaction pattern characterized by mucin deposition with follicular sebaceous units and is found as an idiopathic, primary, benign process (alopecia mucinosa), or as a secondary process due to inflammatory and neoplastic disorders (mycosis fungoides). When associated with follicular mucinosis, mycosis fungoides commonly pursues an aggressive course, often undergoing large-cell transformation, which is associated with resistance to therapy and poor prognosis. We present a case of mycosis fungoides with follicular mucinosis that was treated with incomplete courses of interferon, isotretinoin, and polychemotherapy with subsequent rapid progression to tumor-stage mycosis fungoides with large cell transformation and nodal and bone marrow involvement. In this setting, the patient was treated with local radiation therapy, total-skin electron beam therapy, and therapy and maintenance with the oral retinoid-X-receptor retinoid bexarotene, and achieved a durable complete remission.

Administration, Oral↗