Hepatic reactive lymphoid hyperplasia-associated primary biliary cholangitis masquerading as a neoplastic liver lesion.

Hepatic reactive lymphoid hyperplasia is an uncommon benign condition, often found incidentally as a solitary liver lesion. The chronic inflammatory reaction associated with autoimmune conditions and malignancies has been postulated as a possible aetiology. The diagnosis is challenging as it often mimics various malignancies radiologically and histologically, hence the diagnosis being made only after surgical resection. Lymphadenopathy is common with primary biliary cholangitis, though rarely reported with reactive lymphoid hyperplasia. We report a case of hepatic reactive lymphoid hyperplasia associated with portacaval lymphadenopathy in a patient with primary biliary cholangitis, diagnosed after surgical resection. We propose lesional biopsy be considered in patients with primary biliary cholangitis found to have a solitary lesion with supporting low-risk clinical and radiological features.

A case of hepatic reactive lymphoid hyperplasia: the review of 23 cases from the literatures.

A 70-year-old woman was referred to our hospital because of slight elevation of soluble interleukin-2 receptor (sIL-2R) and accumulation of 18F-fluorodeoxyglucose (FDG) in S8 of the liver on positron emission tomography. The mass was strongly suspected to be malignant because of contrast enhancement and enlargement in size of the mass, and suspicion of portal vein invasion. Hepatic S8 subsegmentectomy was performed for diagnostic and therapeutic purposes. Hematoxylin and eosin staining of the resected specimen showed small lymphocytes with no atypia and no formation of lymphoid follicles. Immunostaining showed CD3-positive cells in the interfollicular region and CD20-positive cells in the lymphoid follicles. Both CD10 and BCL-2 were negative in the follicular germinal center. CD138-positive plasma cells were observed and there was no light chain restriction. Based on polyclonal growth pattern of lymphocytes in the lymphoid follicles and interfollicular region, she was diagnosed with hepatic reactive lymphoid hyperplasia (RLH).Review of the English literature of hepatic RLH which referred to imaging findings yielded 23 cases, including this case. As a result, we suggest that liver biopsy should be performed for definitive diagnosis, when hepatic RLH is suspected by imaging findings and backgrounds.

Multippel erythema migrans, borrelialymfocytom og nevroborreliose hos eit barn.

BACKGROUND: Lyme disease after a tick bite often presents as erythema migrans, yet less frequent variants of this disease, such as Borrelia lymphocytoma, multiple erythema migrans and neuroborreliosis, are also seen occasionally. CASE PRESENTATION: We report a case of a tick-bitten child who first presented with an indistinct macular erythema around the left eye and a more distinct macular erythema on and around the left ear. The next day, she developed a facial palsy. INTERPRETATION: The case was interpreted as facial multiple erythema migrans and Borrelia lymphocytoma on the ear, followed by neuroborreliosis. The diagnosis of lymphocytoma was made from clinical findings and PCR of skin biopsy. She recovered quickly after intravenous ceftriaxone and is now healthy.

Borrelial pseudolymphoma of the eyebrow in an adult.

Borrelial pseudolymphoma, more commonly known as Borrelia lymphocytoma and previously also as lymphadenosis benigna cutis, is a rare manifestation of Lyme borreliosis, which occurs nearly always in children after an infection caused by Borrelia afzelii; this pathogen is transmitted exclusively by the Ixodes ricinus tick in our region. The most common body locations of this lymphocytoma include the earlobe, scrotum, nipples and the areomamillary complex. Therefore, the case of our patient was unexpected and quite rare. The aim of this article is to point out the high incidence of Lyme disease and its atypical manifestations which can be cured without surgical intervention in most cases. The authors describe the case of a 58-year-old healthy female patient with a very rare manifestation of Lyme disease.

Clinical features, natural history and outcomes of pseudolymphoma of liver: A case-series and systematic review.

BACKGROUND: Pseudolymphoma is a rare, benign, nonspecific condition that forms a mass-like lesion characterized by the proliferation of non-neoplastic lymphocytes. Lacking of specific clinical symptoms, serological markers, and imaging features, the diagnosis is difficult. We reporte five cases of hepatic pseudolymphoma and provide a systematic review of existing literatures to improve our understanding of this rare liver disease. METHODS: We followed-up five cases of hepatic pseudolymphoma in West China Hospital from January 2002 to January 2022. We also summarized the cases of hepatic pseudolymphoma from January 1981 to December 2021 through the PubMed database and comprehensively analyzed the characteristics of the cases. RESULTS: The pathologic features of the five cases were characterized by benign lymphoid tissue hyperplasia, lymphoid follicle formation, and a polarized germinal center. Immunohistochemistry, in situ hybridization, and gene rearrangement revealed non-malignant lymphoma. Besides, a total of 116 cases have been reported in the PubMed database from 1981 to 2021. The incidence of hepatic pseudolymphoma is higher in middle-aged and elderly women and has been reported more frequently in Asia. All cases were pathologically diagnosed, among which 85.95% of the patients were treated by surgery. CONCLUSIONS: Hepatic pseudolymphoma is an extremely rare benign disease, mainly in middle-aged and elderly women. Without distinctive clinical and imaging characteristics, pathological diagnosis is the highly reliable method at present. Thus, in the absence of risk factors for a primary liver tumor or metastatic tumor in middle-aged and elderly women, the possibility of pseudolymphoma should be considered to avoid extensive treatments.

Lymphoproliferative disorders in the lacrimal caruncle: report of three cases and review of literature.

This report included three cases of lymphoproliferative disorders developing from the lacrimal caruncle. The first case was an 11-year-old boy with reactive lymphoid hyperplasia in the left lacrimal caruncle. The second case was an 80-year-old woman with reactive lymphoid hyperplasia in the right lacrimal caruncle. The third case was a 77-year-old man with follicular lymphoma in the left lacrimal caruncle. Our literature review of cases with lacrimal caruncular lesions showed 11 reported cases with reactive lymphoid hyperplasia and 17 with malignant lymphoma. There had been no previous report on follicular lymphoma in the lacrimal caruncle.

Drug-induced cutaneous pseudolymphoma: A systematic review of the literature.

Drug-induced cutaneous pseudolymphoma (CPL) is a common form of pseudolymphoma and there are numerous drugs associated with it. In this study, we performed a systematic review of the literature by searching PubMed/Medline and Embase databases to determine the most common drugs responsible for CPL and to define the demographic, clinical, histopathological and immunopathological characteristics of patients (updated on 30 December 2020). From 883 initially found articles, 56 studies (89 reported cases) were included. The mean age of patients was 54.4 ± 17.7 (ranging 8-86) years, and 46 (51.7%) were men. The median time interval between drug intake and CPL occurrence was 120 days (range 1-7300 days). The shortest median time interval between taking the drug and the onset of the disease was observed among patients taking antidepressants (60 days) (range 7-540) and the longest median time interval was observed in individuals using immunomodulators (300 days) (range 3-7300). The most-reported drug categories causing CPL were anti-hypertensives (17.9%), anticonvulsants (14.6%), monoclonal antibodies (13.4%) and antidepressants (11.2%). Moreover, the most common drugs were phenytoin (6.7%), amlodipine (5.6%), fluoxetine (5.6%) and carbamazepine (4.4%). Histopathological evaluation of 76 cases revealed 62 (81.5%) reports of T-cell infiltrations. Furthermore, positive reports of CD4 (94.0%), CD8 (93.0%) and CD30 (87.5%) were noted. The lowest prevalence of CD30-positive reports was observed among monoclonal antibodies. In conclusion, anti-hypertensives, anti-convulsants, monoclonal antibodies and anti-depressants are the most common drugs responsible for CPL. It mostly presents in middle-aged patients with almost no gender difference as pruritic papules, nodules and plaques.

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.

Inflammatory lobular hemangioma (T-cell-rich angiomatoid polypoid pseudolymphoma)-Assessment of FOS/FOSB and lymphoid markers and comparison with epithelioid hemangioma.

BACKGROUND: T-cell-rich angiomatoid polypoid pseudolymphoma (TRAPP) and inflammatory lobular hemangioma (ILH) encompass a spectrum of cutaneous vascular lesions in which a prominent lymphoplasmacytic component may impart a pattern highly reminiscent of low-grade cutaneous lymphoma (pseudolymphoma). Epithelioid hemangioma, including its most common variant angiolymphoid hyperplasia with eosinophilia (ALHE), is a distinct entity associated with FOS and/or FOSB expression detected by immunohistochemistry (IHC). These entities can show significant morphological overlap. METHODS: We performed IHC for FOSB, FOS, and lymphoid markers in a series of TRAPP/ILH and ALHE. RESULTS: We identified 13 cases of ILH/TRAPP, which showed a predominance in CD8+ T-cells (CD8>CD4: 11/13) while FOSB and FOS were expressed in 36% (4/11) and 27% (3/11) of cases, respectively. ALHE (n = 9) showed a predominance in CD4+ T-cell (67%) with FOSB and FOS co-expression in 78% (seven of nine) of the cases. CONCLUSION: We showed, based on FOS and/or FOSB immunohistochemical expression, that there is a possible link between ILH/TRAPP and epithelioid hemangioma/ALHE. The use of FOS and FOSB IHC in the routine diagnostic setting of cutaneous vascular lesions will help to redefine cases of ILH/TRAPP as a subset of these may represent inflammatory variants of epithelioid hemangioma.

Cutaneous reactive B-cell lymphoid proliferations.

Cutaneous lymphoid hyperplasia (CLH), also known as cutaneous pseudolymphoma, is a spectrum of benign conditions characterized by reactive B- and T-cell cutaneous lymphocytic infiltrates. B-cell lymphoid proliferations are a heterogenous group of non-neoplastic cutaneous diseases that must be histopathologically distinguished from cutaneous B-cell lymphomas. These proliferations can be observed as reactive phenomena to infections, medications, allergens, neoplasms, and more. Furthermore, there are many inflammatory conditions that present with reactive B-cell infiltrates, including actinic prurigo, Zoon balanitis, Rosai-Dorfman disease, and cutaneous plasmacytosis. This review summarizes multiple cutaneous B-cell lymphoid proliferations within the major categories of reactive and disease-associated CLH. Further we discuss major discriminating features of atypical CLH and malignancy. Understanding the specific patterns of B-cell CLH is essential for the proper diagnosis and treatment of patients presenting with such lesions.