Cutaneous Lymphoid Hyperplasia With T-Cell Clonality and Monotypic Plasma Cells Secondary to a Tick Bite: A Hidden Critter and the Power of Deeper Levels.

ABSTRACT: Cutaneous lymphoid hyperplasia (CLH) is a benign reactive process with T-cell or B-cell lymphocytic infiltration in the skin, which can simulate cutaneous lymphomas both clinically and histologically. Various antigenic stimuli have been implicated in the development of CLH, including tick bites. Finding histologic evidence of such triggering factors, however, is often difficult. Moreover, the presence of clonality in CLH can potentially be interpreted as a neoplastic process, posing a further diagnostic challenge to dermatopathologists, if one is not aware of such peculiar phenomena. Herein, we describe a case of CLH secondary to a tick bite, featuring both T-cell clonality and monotypic plasma cells with lambda light chain restriction; the diagnostic clue being tick parts, which became evident on assessment of deeper levels. To the best of our knowledge, this is the first reported case of a tick-associated clonal CLH with simultaneous detection of monoclonal T cells and monotypic lambda light chain restriction, mimicking primary cutaneous CD4+ small/medium T-cell lymphoproliferative disorder and Borrelia-associated primary cutaneous marginal zone B-cell lymphoma, respectively.

Cutaneous B-Cell Pseudolymphoma (Lymphocytoma Cutis) of the Earlobe: A Poorly Recognized Complication of Ear Piercing in Children.

Background: Cutaneous pseudolymphoma (CPL) refers to a group of benign, reactive processes that mimic cutaneous lymphoma and are associated with a variety of triggering immune stimuli, including arthropod bites, drugs, and foreign bodies. In children, most cases of CPL are due to a variant of Borreliosis that is specific to Eurasia. Cutaneous pseudolymphoma secondary to ear piercing has only been documented in adults. Case Reports: We present the clinical and pathological findings of cutaneous Bcell psuedolymphoma in two adolescent patients (11-year-old female and 15-year-old male) secondary to ear piercing. Conclusion: Our report expands the clinico-pathological spectrum of CPL associated with ear piercing by documenting its occurrence in children.

Histopathologic Spectrum of Findings Associated With Tattoos: Multicenter Study Series of 230 Cases.

IMPORTANCE: Reactions to tattoo may simulate common dermatosis or skin neoplasms. Histopathology allows diagnosis and helps determining the level and degree of inflammation associated, consequently orientating treatment. OBJECTIVE: To describe the histological features found in biopsies of cutaneous reactions to tattoo. DESIGN: This study was designed as a multicenter case series. SETTING: All consecutive histopathological samples of tattoos referred from 1992 to 2019 to the Hospital General de Catalunya, Hospital Germans Trias i Pujol, and a private practice, all in Barcelona, Spain, and from the Kempf und Pfaltz Histologische Diagnostik in Zurich, Switzerland were retrieved from the files. PARTICIPANTS AND EXPOSURE: The inclusion criteria were all cosmetic/permanent makeup, artistic/professional, and traumatic tattoos associated with either inflammatory reactions alone and/or with tumors and/or infections. Exclusion criteria were cases without any associated pathologic finding in the place of the ink, amalgam tattoos, and medical or temporary tattoos. MAIN OUTCOMES AND MEASURES: In all patients, clinical features (age, sex, location, tattoo color, and presentation) were recorded. Histological features evaluated included ink color, associated tumors or infections, and inflammatory reaction pattern. Inflammation was graded in low to moderate or severe. RESULTS: From 477 biopsies diagnosed as tattoos, 230 cases from 226 patients met the inclusion criteria. Samples corresponded to 107 male and 120 female subjects and 3 of unknown gender. Median age was 39 years (ranging from 9 to 84 years). Fifty-three samples were referred from centers in Spain and 177 from the center in Switzerland. The series was analyzed in 2 parts: tattoos associated only with inflammatory reactions (117/230) and tattoos associated with tumors or infections (113/230). The most common form of inflammatory pattern associated with tattoo was the fibrosing reaction (79/117, 68%), followed by granulomatous reaction (56/117, 48%), lichenoid reaction (33/117, 28%), epithelial hyperplasia (28/117, 24%), pseudolymphoma (27/117, 23%) and spongiotic reaction (27/117, 23%). Combined features of 2 or more types of inflammatory patterns were seen in 64% cases. CONCLUSIONS AND RELEVANCE: Our series confirms that cutaneous reactions to tattoos are polymorphous. Inflammation tends to present with combined patterns. Infections are tending to decline, and pathologic findings are not specific to ink color or clinical features.

Recurrent episodes of palpable migratory arciform erythema associated with IVIg infusions.

Palpable migratory arciform erythema (PMAE) is an uncommon T cell pseudolymphoma characterized by erythematous, annular-to-arciform papules and plaques. Although the eruption is self-limited in most cases, recurrences are routine. Diagnosis requires attention to clinical history as well as histopathologic analysis, which allow for differentiation from other T cell pseudolymphomas and gyrate erythemas. A common triggering factor has not been identified. We report a 60-year-old man who developed PMAE after IVIg infusion. Interestingly, although the individual eruptions were self-limited and resolved after several weeks, subsequent infusions predictably resulted in recurrence of PMAE, confirming the association. To our knowledge, this is the first reported case of recurrent PMAE in association with IVIg infusions.

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.

Aggressive large B-cell lymphoma triggered by a parvovirus B19 infection in a previously healthy child.

In absence of red blood cells disease or immune defect, parvovirus B19 (PVB-19) is usually considered as a benign condition. Here, we report the case of a 10-year-old boy, previously healthy, presenting with a PVB-19 infection revealed by a bicytopenia and a voluminous axillary adenopathy. Pathophysiology examination showed reactional lymphoid population. Nine months later and in the absence of remission, a new biopsy of the same adenopathy revealed a Hodgkin lymphoma with area of T-cell rich aggressive large B-cell lymphoma. This case suggests PVB-19 as potential trigger of this malignant childhood hemopathy. Although no definitive conclusion can be drawn, our clinical case questions the role of PVB-19 in lymphomagenesis.

Cutaneous Pseudolymphoma Due to Hirudotherapy

Pseudolymphoma, also known as Jessner's lymphocytic infiltration, is a benign but usually chronic, T-cell infiltrating disease with erythematous papules and plaques usually seen on the skin of the face, neck, and back. The use of leech therapy also known as hirudotherapy has increased in recent years. Here, we report a 52-year-old male patient who had undergone hirudotherapy in his neck and developed infiltrating plaques after four months. A skin biopsy confirmed the diagnosis of Jessner's lymphocytic infiltration. In parallel with the increasing use of hirudotherapy in recent years, the side-effect reports will likely to increase. Indications and contraindications of hirudotherapy, which is being used officially in hospitals, should be taken into consideration.

Lymphoplasmacytic plaque in children: Case report and literature review.

We report a case of benign lymphoplasmacytic plaque (LPP) in a child. These asymptomatic erythematous papulonodular lesions are an emerging clinicopathological entity. Herein, we describe a previously unreported site for LPP lesions, namely, the volar wrist and the distal ipsilateral palm.

Overview of fever of unknown origin in adult and paediatric patients.

Fever of unknown origin (FUO) can be caused by a wide group of diseases, and can include both benign and serious conditions. Since the first definition of FUO in the early 1960's, several updates to the definition, diagnostic and therapeutic approaches have been proposed. This review outlines a case report of an elderly Italian male patient with high fever and migrating arthralgia who underwent many procedures and treatments before a final diagnosis of Adult-onset Still's disease was achieved. This case report highlights the difficulties in diagnosing certain causes of FUO that requires a very high index of suspicion. The main causes of FUO in paediatric and adult patients will be reviewed here, underlying the fact that a physician should also consider the possibility that a patient with FUO may have a monogenic autoinflammatory disease (AID). The identification of AIDs requires a careful evaluation of both history and clinical details that may reveal important clues to identify the correct aetiology. We also provide a comprehensive account of specific signs and symptoms that could suggest possible diagnoses and guide the work-up of FUO and non-genetic periodic fevers in children.

Borrelia Lymphocytoma Mimicking Butterfly Rash in a Pediatric Patient.

A 5-year-old girl presented with a facial butterfly rash that persisted for 5 months without arthralgia, fever, malaise, photosensitivity, or other symptoms. Lupus erythematosus was clinically suspected. All blood tests were negative or within normal values. Skin biopsy showed a dense nodular superficial and deep inflammatory infiltrate of lymphocytes that reaches subcutaneous tissue. The most striking histopathological finding was plasma cells with some perifollicular accentuation. Borrelia polymerase chain reaction assay of the tissue was positive, and we made the diagnoses of borrelia lymphocytoma mimicking butterfly rash of lupus erythematosus. The lesions disappeared with amoxicillin followed by cefuroxime for 28 days. In children, a variety of diseases including lupus erythematous may lead to a butterfly rash that is usually short lasting and commonly associated with systemic symptoms. Borreliosis may be related with long-lasting facial erythema in children and may mimic lupus and present high titers of antinuclear antibodies. In any case, borrelial lymphocytoma has not been previously reported as a cause of butterfly rash, thus mimicking acute lupus in a child, as in our case. From a histopathological point of view, the presence of plasma cells in a pseudolymphomatous infiltrate is a clue for the right diagnosis. Therefore, we suggest that borrelia serology should be done in children with butterfly rash lasting more than a month and empirical antibiotic treatment should be tried even in cases with negative serology.

Mixed Lichenoid and Follicular T- and B-Cell Lymphoid Reaction to Red Tattoos With Monoclonal T Cells.

Pseudolymphomatous reactions have been described to occur in tattoos. Most cases have exhibited T-cell predominance and polyclonal T-cell receptor gene rearrangements. One case with monoclonal IgH gene rearrangements progressed into B-cell lymphoma. Lichenoid infiltrates are commonly described but lymphoid follicles much less frequently. We report a case with mixed lichenoid and follicular T- and B-cell reaction to red tattoos. The histopathology and the immunohistochemical studies were constant with a mixed T- and B-cell pseudolymphoma, the IgH gene rearrangement study was polyclonal, but the T-cell receptor gene rearrangement study was monoclonal. The patient who responded to intralesional corticosteroid injections remains under close scrutiny.

Areolar lymphocytoma in a child: A rare cutaneous presentation of borreliosis.

Lyme disease is a common tick-borne infection caused by Borrelia burgdorferi in the United States, where infection is most prevalent in the northeastern and mid-Atlantic states. Although classically associated with erythema migrans, Lyme disease caused by Borrelia species found in Europe may also present with other cutaneous findings. Here we report the case of a girl who was clinically diagnosed with Lyme disease based on her history of recent travel and the appearance of an areolar lymphocytoma; this was confirmed by testing. Testing for European Lyme disease does not follow the testing algorithm that the Centers for Disease Control and Prevention recommends and may be easily missed. Our case serves as an important reminder that common infections can have varying presentations depending on their region of acquisition and may require specialized testing for accurate diagnosis.