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Clinical Nuclear Medicine May 2024Gamma/delta T-cell lymphoma is a rare and aggressive subtype of primary cutaneous lymphoma. Clinical manifestations typically include the development of subcutaneous...
Gamma/delta T-cell lymphoma is a rare and aggressive subtype of primary cutaneous lymphoma. Clinical manifestations typically include the development of subcutaneous nodules and ulcerated plaques. Some forms present as panniculitis with hemophagocytic syndrome. Prognosis is bleak, with a 10% 5-year survival rate. In this report, we present the case of a 20-year-old man from French Polynesia, referred for 18 F-FDG PET/CT because of the progressive worsening of febrile cutaneous-mucosal infiltration on the face persisting for 1 month. PET examination guided a biopsy from the right deltoid muscle, and expert histological analysis confirmed a CD8 + not otherwise specified T-cell lymphoma, granzyme+ and TCR gamma/delta.
Topics: Humans; Male; Young Adult; Fluorodeoxyglucose F18; Lymphoma, T-Cell; Lymphoma, T-Cell, Cutaneous; Panniculitis; Positron Emission Tomography Computed Tomography; Skin Neoplasms; T-Lymphocytes
PubMed: 38389221
DOI: 10.1097/RLU.0000000000005135 -
Medicina Clinica Jun 2024
Topics: Humans; Panniculitis, Peritoneal; Diagnosis, Differential; Neoplasm Recurrence, Local; Male; Middle Aged; Female
PubMed: 38378333
DOI: 10.1016/j.medcli.2023.11.045 -
Medicina Clinica Jun 2024
Topics: Humans; Panniculitis, Peritoneal; Diagnosis, Differential; Male; Peritoneal Neoplasms; Middle Aged
PubMed: 38378327
DOI: 10.1016/j.medcli.2023.11.041 -
Journal of Diabetes Investigation Jun 2024The potential liability to hypercatabolism in lipodystrophy remains to be fully elucidated. Here we report a 28-year-old Japanese woman with acquired generalized...
The potential liability to hypercatabolism in lipodystrophy remains to be fully elucidated. Here we report a 28-year-old Japanese woman with acquired generalized lipodystrophy, who presented with recurrence of panniculitis and anemia. After corticosteroid treatment was started, she showed rapid reductions in body weight and lean mass by 15% at maximum, accompanied by an elevated urea nitrogen/creatinine ratio, which recovered almost fully as the corticosteroid treatment was tapered and discontinued. She had multiple risk factors for hypercatabolism: lack of metabolic reserves, insulin resistance, and hyperglycemia due to lipodystrophy, lowered daily activity due to anemia, persistent inflammation, and wasting associated with panniculitis, and relatively insufficient energy and protein intake during hospitalization. More attention should be paid to the potential liability to hypercatabolism in patients with lipodystrophy, and to skeletal muscle loss as an adverse effect of corticosteroid treatment in patients at high risk, such as those with diabetes or decreased metabolic reserves.
Topics: Humans; Female; Adult; Lipodystrophy; Muscle, Skeletal; Adrenal Cortex Hormones
PubMed: 38372649
DOI: 10.1111/jdi.14158 -
Dermatology Practical & Conceptual Jan 2024
PubMed: 38364433
DOI: 10.5826/dpc.1401a33 -
The Journal of Dermatology Feb 2024
Panniculitis on the trunk as a possible characteristic feature of anti-SAE1/2 antibody-positive dermatomyositis: A possible cutaneous manifestation of treatment resistance.
PubMed: 38362647
DOI: 10.1111/1346-8138.17128 -
Clinical and Experimental Dermatology Feb 2024Erythema induratum (EI) is a tuberculid associated with Mycobacterium tuberculosis (TB) infection. TB has been identified in Taiwan with a high percentage of EI using...
BACKGROUND
Erythema induratum (EI) is a tuberculid associated with Mycobacterium tuberculosis (TB) infection. TB has been identified in Taiwan with a high percentage of EI using PCR. However, this pathogen has rarely been detected recently.
OBJECTIVE
To explore the association between EI, annual incidence of tuberculosis in Taiwan, and treatment outcomes over the past two decades.
METHODS
Patients diagnosed with EI between 2001 and 2020 were enrolled based on histopathology, tissue culture, and TB-PCR tests. Other cases of panniculitis with TB-PCR results were used as controls. Their clinical information was obtained. The results were correlated with the annual incidence of tuberculosis and compared between groups.
RESULTS
Fifty-five specimens from EI patients fulfilled the inclusion criteria; three (3/55, 5%) had positive TB PCR. One patient diagnosed with erythema nodosum in the control group had positive TB PCR (1/27, 4%). There was no significant relationship between TB and EI (odds ratio = 1.5, 95% confidence interval = -0.964 to 3.964, p = 1). Correlation between the incidence of TB and number of EI cases was not significant (r = -0.185, p = 0.447). Only four patients received anti-tuberculosis therapy. They all showed clinical improvement without recurrence. One patient with PCR-positive EI was not treated with anti-tuberculosis therapy; however, the skin lesion improved 3 months later. No other patients in the EI group were diagnosed with TB infection in the follow up of 508 person-years.
CONCLUSIONS
Most EI in Taiwan are nodular vasculitis and not tuberculids owing to well-controlled tuberculosis. This condition can be alleviated without anti-tuberculosis therapy.
PubMed: 38320211
DOI: 10.1093/ced/llae048 -
Leukemia & Lymphoma May 2024T cell dyscrasias that demonstrate a proclivity for the subcutaneous fat include atypical lymphocytic lobular panniculitis, lupus profundus, and primary subcutaneous T...
T cell dyscrasias that demonstrate a proclivity for the subcutaneous fat include atypical lymphocytic lobular panniculitis, lupus profundus, and primary subcutaneous T cell lymphoma, including subcutaneous panniculitis-like T cell lymphoma (SPTCL). We encountered two patients who developed fever and indurated abdominal erythema at their peginterferon alfa-2a injection sites. Biopsies showed an atypical CD8 positive, granzyme positive, CD5 negative, MXA negative lymphocytic lobular panniculitis, diagnostic of SPTCL. Peginterferon alfa-2a was held in both patients. One patient received chemotherapy with an excellent response, while the other continued to have progressive disease. Peginterferon alfa-2a is known to significantly elevate serum MXA, which may induce high levels of MXA expression at the injection site, creating a microenvironment for the development of lupus profundus, which may eventuate into SPTCL. In summation, a potential risk of peginterferon alfa-2a injections is the development of SPTCL potentially arising in a background of an exogenous interferon triggered lymphocytic panniculitis.
Topics: Humans; Polyethylene Glycols; Interferon-alpha; Recombinant Proteins; Panniculitis; Female; Lymphoma, T-Cell; Middle Aged; Male; Biopsy; Adult
PubMed: 38315613
DOI: 10.1080/10428194.2024.2310139 -
Journal of Veterinary Science Jan 2024The anti-programmed death 1 (PD-1) antibody has led to durable clinical responses in a wide variety of human tumors. We have previously developed the caninized...
BACKGROUND
The anti-programmed death 1 (PD-1) antibody has led to durable clinical responses in a wide variety of human tumors. We have previously developed the caninized anti-canine PD-1 antibody (ca-4F12-E6) and evaluated its therapeutic properties in dogs with advance-staged oral malignant melanoma (OMM), however, their therapeutic effects on other types of canine tumors remain unclear.
OBJECTIVE
The present clinical study was carried out to evaluate the safety profile and clinical efficacy of ca-4F12-E6 in dogs with advanced solid tumors except for OMM.
METHODS
Thirty-eight dogs with non-OMM solid tumors were enrolled prospectively and treated with ca-4F12-E6 at 3 mg/kg every 2 weeks of each 10-week treatment cycle. Adverse events (AEs) and treatment efficacy were graded based on the criteria established by the Veterinary Cooperative Oncology Group.
RESULTS
One dog was withdrawn, and thirty-seven dogs were evaluated for the safety and efficacy of ca-4F12-E6. Treatment-related AEs of any grade occurred in 13 out of 37 cases (35.1%). Two dogs with sterile nodular panniculitis and one with myasthenia gravis and hypothyroidism were suspected of immune-related AEs. In 30 out of 37 dogs that had target tumor lesions, the overall response and clinical benefit rates were 6.9% and 27.6%, respectively. The median progression-free survival and overall survival time were 70 days and 215 days, respectively.
CONCLUSIONS
The present study demonstrated that ca-4F12-E6 was well-tolerated in non-OMM dogs, with a small number of cases showing objective responses. This provides evidence supporting large-scale clinical trials of anti-PD-1 antibody therapy in dogs.
Topics: Dogs; Animals; Humans; Melanoma; Programmed Cell Death 1 Receptor; Skin Neoplasms; Treatment Outcome; Dog Diseases
PubMed: 38311328
DOI: 10.4142/jvs.23144 -
Pathology Apr 2024Kikuchi-Fujimoto disease (KFD) is a benign self-limiting condition primarily affecting young females. It usually presents with fever and cervical lymphadenopathy of...
Kikuchi-Fujimoto disease (KFD) is a benign self-limiting condition primarily affecting young females. It usually presents with fever and cervical lymphadenopathy of unknown aetiology with a preponderance of the Asian population. Histopathology is critical in making an accurate diagnosis. While the typical microscopic features include paracortical necrosis with debris, histiocytosis with immunoblasts, and absent neutrophils, rarely, KFD can show atypical features like marked immunoblastic proliferation mimicking lymphoma, demonstrate vasculitis mimicking lupus erythematosus, etc. The diagnosis is extremely challenging if such features occur in cases with generalised lymphadenopathy, which is infrequent in KFD. The study aims to describe the morphological, clinical, and immunohistochemical features of KFD and determine the frequency of the atypical features. We also analysed the subtle histological and immunohistochemical features that aid in the diagnosis of atypical cases. Cases reported as KFD over a period of 6 years were retrieved from the archives of histopathology. The morphological features were categorised as typical and atypical. In the atypical cases, the features that aided in the correct diagnosis of KFD were analysed. Out of the 42 cases evaluated, 23.9% (n=10) had generalised lymphadenopathy; 57.2% (n=24) were women with a median age of 25 years. Leukopenia was observed in 42% (n=13) of patients. Typical features were present in 76.2% (n=32) cases and 23.8% (n=10) presented with atypical features. Eight cases were antinuclear antibody-positive. Atypical features included five (50%) cases with vasculitis and panniculitis, and three (30%) cases with large, atypical cells for which immunohistochemistry (IHC) was performed. In two of these cases, the patent sinuses, absence of neutrophils, and IHC with CD68 aided the diagnosis. There is an overlap of clinical and histopathological features between KFD and malignant lymphomas and systemic lupus erythematosus. Given the fact that the atypical features (23.8%) are not rare occurrences in KFD, correlations with clinical findings and ancillary studies are essential to avoid misdiagnosis and inadvertent therapy.
Topics: Humans; Female; Adult; Male; Histiocytic Necrotizing Lymphadenitis; Diagnosis, Differential; Lymphadenopathy; Leukopenia; Vasculitis
PubMed: 38296677
DOI: 10.1016/j.pathol.2023.10.017