A case of multiple nodular cutaneous B-cell pseudolymphoma successfully treated with glucocorticoid, methotrexate, and hydroxychloroquine.

Cutaneous pseudolymphomas (CPL) is a group of benign, reactive, and polyclonal lymphoproliferative dermatoses that simulate cutaneous lymphomas (CL) clinically and histologically. Based on the predominating component of lymphocytic infiltrate, CPL can be divided into cutaneous B-cell pseudolymphomas (CBPL), cutaneous T-cell pseudolymphomas (CTPL), mixed (T-/B-cell) pseudolymphomas, CD30-positive pseudolymphomas, and non-classifiable pseudolymphomas. Most patients with localized nodular CBPL present with a solitary nodule. However, few patients develop multiple skin lesions, rarely in generalized forms. Here we describe a rare case of multiple nodular CBPL on both sides of the patient's neck, which was treated successfully with intramuscular injection of compound betamethasone, oral methotrexate, and hydroxychloroquine for 4 months. No recurrence was observed in the patient at the one-year follow-up. This combined treatment may be a promising treatment choice for multiple nodular CBPL.

Cutaneous B cell pseudolymphoma treated with rituximab and methotrexate.

Cutaneous B cell pseudolymphoma (CBPL), or cutaneous lymphoid hyperplasia, is the most common pseudolymphoma. It typically responds well to local treatment and follows a benign course. Herein, we describe the unique case of a patient with CBPL that was refractory to a variety of treatments, with subsequent response to rituximab followed by methotrexate. This case explores the complex interplay of T and B lymphocytes, and the potential role of perifollicular T cells in treatment resistant CBPL. Further, it describes the additive therapeutic effect of rituximab and methotrexate to target both B cell and T cell populations in CBPL, a strategy already employed in a number of other conditions.

Use of topical rapamycin in acral pseudolymphomatous angiokeratoma of children (APACHE): A report of two cases and review of the literature.

Acral pseudolymphomatous angiokeratoma of children (APACHE) is a rare, benign disease characterized clinically by multiple, asymptomatic, erythematous papules in the acral regions. We report APACHE in a 12-year-old girl with erythematous-violaceous papules on the lateral dorsum of her foot and toes, and a 3-year-old girl with erythematous papules on the plantar aspect of her foot. Topical rapamycin ointment improved the lesions and both patients tolerated the medication well. Topical rapamycin appears to be a potentially efficacious, well-tolerated, non-invasive therapy in APACHE, although further studies are needed.

Lupus Erythematosus Tumidus with Pseudolymphomatous Infiltrates: A Case Report.

A 39-year-old Japanese woman presented with a pruritic infiltrated erythematous plaque on the right cheek. Histopathologic analysis of the erythema showed dermal edema, separation of collagen bundles, and nodular perivascular and periadnexal infiltration of lymphocytes in the whole dermis, without epidermal changes. Alcian blue staining intensity was elevated between the collagen bundles, indicating dermal mucinosis. The nodular infiltrates consisted of CD3+ T cell clusters and CD20+ B cell clusters (ratio, approximately 3:1) and included numerous CD123+ cells, indicative of plasmacytoid dendritic cells. Blood analysis revealed serum antinuclear antibody at a titer of 1:160 (homogeneous, speckled pattern). Lupus erythematosus tumidus with pseudolymphomatous infiltrates was diagnosed. Hydroxychloroquine treatment partially improved symptoms; however, the addition of prednisolone was required for complete resolution. Lupus erythematosus tumidus is sometimes accompanied by pseudolymphomatous infiltrates. Dermal mucinosis and the presence of numerous plasmacytoid dendritic cells are useful in differentiating lupus erythematosus tumidus from pseudolymphoma.

A case of cutaneous pseudolymphoma with a distinctive appearance treated successfully by intralesional interferon alpha-1b and corticosteroids.

Cutaneous pseudolymphoma (CPL) encompasses various forms of benign lymphocytic proliferative dermatoses that mimic the clinical and/or pathological changes of lymphoma. The clinical manifestations of CPL vary due to differences in the pathogenesis, and accordingly, no specific treatment has been identified. Here, we report a case of CPL on the nose, which had a distinctive appearance and was treated successfully using a combination of intralesional interferon alpha-1b and compound betamethasone (betamethasone sodium phosphate and betamethasone dipropionate). This combination may be a good option for localized CPLs at particular anatomical sites.

Diagnostic Challenges and Treatment Options for Cutaneous T Cell Pseudolymphoma: A Case Study with Rituximab Treatment.

BACKGROUND Pseudolymphoma is a rare disorder that can mimic lymphoma both clinically and histologically. It usually affects middle-aged females. Since pseudolymphoma is a rare disorder not only is diagnosing the condition difficult, but there is also a lack of standardized treatment guidelines. In the literature, anti-CD20 monoclonal antibody rituximab is described as an effective treatment option. CASE REPORT 46-year-old female fell ill suddenly with swelling and enlargement of her chin. Multiple skin biopsies were done, which were re-evaluated multiple times as well. Each ended with a new diagnosis for the patient. Finally, in the last revision of biopsy material, pseudolymphoma was confirmed. The patient received multiple courses of corticosteroid treatments - locally and systemically - without long lasting effect. After diagnosis of pseudolymphoma, the patient was started on intravenous rituximab and this treatment was effective. CONCLUSIONS Cutaneous pseudolymphoma is a diagnostic challenge. Rituximab is a treatment option for refractory pseudolymphoma. Since there are no treatment guidelines for pseudolymphoma, more clinical studies are needed to establish best treatment options for these patients. Therefore, each reported clinical case is important.

Ipilimumab/nivolumab-induced pseudolymphoma in a patient with malignant melanoma.

Combination therapy with ipilimumab and nivolumab is an adjuvant treatment approach for metastatic melanoma that boasts increased 3-year survival when compared with a single immunotherapy agent. Combination therapy, however, is associated with increased toxicities, especially cutaneous side-effects. Here we present a patient with metastatic melanoma and a sudden eruption of painful nodules on the face and arms 10 days after the administration of the fourth dose of combination ipilimumab/nivolumab. Biopsies demonstrated lymphoid hyperplasia, not clinically or pathologically consistent with an infectious, malignant or autoimmune etiology; a diagnosis of pseudolymphoma secondary to ipilimumab/nivolumab was made. After a steroid taper, the lesions resolved, and the patient was restarted on nivolumab monotherapy 2 weeks later without recurrence of symptoms or disease.

IgG4-rich reactive lymphoid hyperplasia of the lacrimal gland.

We describe an otherwise healthy 24-year-old woman with reactive lymphoid hyperplasia of one lacrimal gland. She was found to have modest elevation of serum IgG4 and high density of IgG4+ plasma cells in her lacrimal gland, ranging from 40% to 60% of IgG+ plasma cells. The patient transiently responded to systemic corticosteroids before treatment with rituximab. IgG4-rich reactive lymphoid hyperplasia of the lacrimal gland is neither an established part of IgG4-related disease spectrum nor a known precursor lesion to lymphoma. Although there is a plausible risk of transforming to IgG4-related disease or to lymphoma, the biological potential and natural history of IgG4-rich reactive lymphoid hyperplasia remains to be determined.

Cutaneous lymphoid hyperplasia induced by Hirudo medicinalis (leeches).

For centuries, Hirudo medicinalis (medicinal leech) has been used as a remedy to treat many discomforts, such as muscle and joint pain. Nowadays, using leech in reconstructive surgery, microsurgery, wound and flap healing, venous insufficiency, varicosities and many other disorders has increased. In this study, we report a 45 year old female patient with six pruritic papules on her legs since10 months ago that appeared about 4 weeks after one session of leech application. Skin biopsy was performed on one of the papules, and in histopathology, superficial ulceration with a dense superficial and deep inflammatory infiltrate within the dermis composed of lymphocytes and eosinophils detected that considering history of leech application at this site, these constellation of data was compatible with the diagnosis of cutaneous lymphoid hyperplasia induced by leeches and the patient was cured by a topical steroid and cryotherapy.