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Pediatr Allergy Immunol 2022 Jun;33(6):e13804

Pneumology and Thorax Surgery Department, National Institute of Pediatrics, Mexico City, Mexico.

J Cancer Res Clin Oncol 2022 Jun 22. Epub 2022 Jun 22.

Medical Clinic III for Oncology, Hematology, Immune-Oncology and Rheumatology, University Medical Center Bonn (UKB), Venusberg-Campus 1, 53127, Bonn, Germany.

Infection of lymphocytes with the Epstein-Barr virus (EBV) is a well-documented risk factor for developing lymphoma. The incidence of EBV positivity in lymphoma depends on the subtype and can range from 10% in diffuse large B-cell lymphoma (DLBCL) to 100% in endemic Burkitt lymphoma (BL), (Shannon-Lowe and Rickinson, Front Oncol 9:713, 2019). However, in most cases, EBV infection remains unnoticed until diagnosis of lymphoma is made. Read More

Cureus 2022 Mar 27;14(3):e23534. Epub 2022 Mar 27.

Department of Otolaryngology, Showa General Hospital, Tokyo, JPN.

An 89-year-old man presented with the chief complaint of a sore throat and a mass in the right side of his neck that tended to increase in size. He displayed the right Horner's sign, and imaging findings showed a 65-mm mass in the right side of the neck, invading the carotid artery. There were no other obvious lesions. Read More

JAAD Case Rep 2022 Mar 7;21:66-69. Epub 2022 Jan 7.

Department of Dermatology, University of North Carolina School of Medicine, Chapel Hill, North Carolina.

Sarcoidosis Vasc Diffuse Lung Dis 2022 13;38(4):e2021038. Epub 2022 Jan 13.

Ankara Ataturk Chest Disease and Chest Surgery Research and Training Hospital, Department of Chest Disease, Ankara, Turkey.

Lymphomatoid granulomatosis (LG) is Epstein-Barr virus associated and aggressive B cell lymphoproliferative disease. The most common sites of involvement are lungs, skin, kidneys, liver and central nervous system. The clinical presentation of pulmonary LG may mimic infectious diseases, malignancies or vasculitis. Read More

Chest 2022 01;161(1):e35-e41

Department of Cardiothoracic Surgery, Mayo Clinic, Jacksonville, FL. Electronic address:

Case Presentation: A 71-year-old man with history of gastroesophageal reflux disease, chronic sinusitis, arthritis, hypothyroidism, and anemia of chronic disease initially sought treatment with a recurrent left pleural effusion along with other abnormal lung findings on chest CT scan. Before his referral, he was being managed for 3 years at his local hospital for waxing and waning fevers, fatigue, productive cough, chills, and night sweats. He did not report any hemoptysis or chest pain, but reported weight loss of 13 kgs in 15 months. Read More

Cureus 2021 Nov 29;13(11):e19992. Epub 2021 Nov 29.

Internal Medicine, Hospital Prof. Doutor Fernando Fonseca, Amadora, PRT.

Lymphomatoid granulomatosis (LYG) is a rare B-cell lymphoproliferative disorder associated with Epstein-Barr virus (EBV) infection and is frequently associated with immunodeficiency. Pulmonary involvement with angiocentric distribution is the most common clinical manifestation. Diagnosis is confirmed by tissue biopsy, usually from lung lesions. Read More

J Neuroimmunol 2021 12 8;361:577748. Epub 2021 Oct 8.

Department of Neurology, Gifu University Graduate School of Medicine, 1-1 Yanagido, Gifu, Gifu, Japan. Electronic address:

We report two patients with meningoencephalomyelitis without evidence of extra central nervous system (CNS) involvement. Brain MRI showed linear perivascular radial gadolinium enhancement patterns and spinal cord MRI showed longitudinal extensive T2-hyperintensity lesions. Pathological findings from brain biopsies were angiocentric T-cell predominant lymphoid infiltrates that lacked Epstein-Barr virus-positive atypical B cells. Read More

FEBS J 2021 Sep 18. Epub 2021 Sep 18.

Department of Microbiology & Immunology & Lineberger Comprehensive Cancer Center, University of North Carolina, Chapel Hill, NC, USA.

Epstein-Barr virus (EBV; human herpesvirus 4; HHV-4) and Kaposi sarcoma-associated herpesvirus (KSHV; human herpesvirus 8; HHV-8) are human gammaherpesviruses that have oncogenic properties. EBV is a lymphocryptovirus, whereas HHV-8/KSHV is a rhadinovirus. As lymphotropic viruses, EBV and KSHV are associated with several lymphoproliferative diseases or plasmacytic/plasmablastic neoplasms. Read More

Clin Case Rep 2021 Sep 7;9(9):e04808. Epub 2021 Sep 7.

Division of Hematologic Malignancies and Cellular Therapy, Department of Internal Medicine, Harold C. Simmons Comprehensive Cancer Center UT Southwestern Medical Center Dallas TX USA.

Isolated central nervous system lymphomatoid granulomatosis (CNS-LYG) can mimic aggressive glioblastomas. We describe a complex presentation of CNS-LYG coexisting with immune thrombocytopenia successfully managed with rituximab and ultra-low-dose radiation therapy. Read More

Rinsho Shinkeigaku 2021 Sep 26;61(9):618-623. Epub 2021 Aug 26.

Department of Pathology, Ekisaikai Hospital.

A 71-year-old man was hospitalized because of low back pain and weakness in both lower limbs. He presented with fever and stiff neck, and his cerebrospinal fluid sample contained blood. MRI revealed intramedullary and epidural hemorrhages in the spinal cord. Read More

Am J Clin Pathol 2021 Oct;156(5):871-885

Divisions of Hematopathology, University of Pittsburgh Medical Center, Pittsburgh, PA, USA.

Objectives: Follicular hyperplasias (FHs) with light chain-restricted (LCR) plasmacytoid/plasma cells (PCs) within germinal centers (GCs) based on immunohistochemistry (IHC)/in situ hybridization (ISH) can potentially lead to diagnostic error. This study aims to better characterize such cases, including their clinical implications.

Methods: LC expression by IHC/ISH was quantitatively assessed in GCs of 17 FHs with LCRGCs. Read More

Chest 2021 07;160(1):e29-e34

Division of Pulmonary, Critical Care and Sleep Medicine, University of Florida, Gainesville, FL.

Case Presentation: A 67-year-old woman with a medical history significant for hypertension, hyperlipidemia, type 2 diabetes mellitus, OSA, and schizophrenia was admitted multiple times the previous 3 months for generalized abdominal pain. Her most recent admission was unique for new onset bilateral upper and lower extremity weakness with paresthesia. Pertinent review of systems included malaise, fever, cough, left lower quadrant pain without weight loss, and rash. Read More

Neurologia (Engl Ed) 2021 Oct 3;36(8):625-628. Epub 2021 Jul 3.

Servicio de Anatomía Patológica, Hospital Universitario de Cruces, Barakaldo, Spain.

Int J Hematol 2021 Oct 22;114(4):502-508. Epub 2021 Jun 22.

Division of Hematology and Oncology, Department of Medicine, Kurume University School of Medicine, 67 Asahi-machi, Kurume, 830-0011, Japan.

Lymphomatoid granulomatosis (LYG) is a rare Epstein-Barr virus (EBV)-driven B-cell lymphoproliferative disease affecting mainly extranodal sites such as the lung, central nervous system (CNS), skin, kidney, and liver. We report a case of low-grade LYG involving the CNS that was successfully treated with interferon alpha (IFNα). A 69-year-old woman developed necrotic erythema of the skin and was initially diagnosed with pyoderma gangrenosum based on skin biopsy. Read More

World Neurosurg X 2021 Jul 30;11:100106. Epub 2021 Apr 30.

Department of Neurosurgery, Nippon Medical School Hospital, Tokyo, Japan.

Lymphomatoid granulomatosis (LYG) is a rare Epstein-Barr virus-associated systemic angiocentric and angiodestructive lymphoproliferative disorder. It commonly involves the lungs and can also affect the skin, liver, kidney, and central nervous system. It can rarely occur in the spine, however, the details are unclear. Read More

Fam Cancer 2021 10 15;20(4):363-380. Epub 2021 Jun 15.

Department of Pediatric Hematology-Oncology, "Aghia Sophia" Children's Hospital, Thivon 1 & Papadiamantopoulou, 11527, Athens, Greece.

Hematological malignancies (HM) developed on underlying primary immunodeficiencies (PID) are rare and of unusual features. Differentiating between malignant and non-malignant lymphoproliferation in cases of pediatric hematology and oncology and revealing their molecular predisposition demonstrate the complex interplay between PID and HM. We retrospectively studied a case series of seven pediatric patients, all with PID with manifestations raising suspicion for HM or hypereosinophilic syndrome (HES) or confirmed HM of lymphoid origin. Read More

Intern Med 2021 Dec 12;60(23):3795-3799. Epub 2021 Jun 12.

Department of Hematology, Juntendo University School of Medicine, Japan.

The primary central nervous system (CNS) presentation of lymphomatoid granulomatosis (LYG) is rare, and no standard therapy for LYG with primary CNS symptoms exists. CNS-LYG patients usually survive for only less than a year from diagnosis. This is the first report of high-grade primary CNS-LYG with monoclonality that was successfully treated with rituximab monotherapy, resulting in a durable remission for more than 1 year in a 66-year-old woman with pemphigus vulgaris who was also on immunosuppressive therapy. Read More

Respirol Case Rep 2021 Jul 26;9(7):e00789. Epub 2021 May 26.

Department of Respiratory & Sleep Medicine St George Hospital Kogarah NSW Australia.

Pulmonary lymphomatoid granulomatosis (PLG) is a rare multisystem Epstein-Barr virus (EBV)-associated lymphoproliferative disorder. Exact incidence is unknown and, with its variable clinical presentation, making an accurate diagnosis of PLG can be difficult. We present two distinct cases at our tertiary centre that underline PLG's non-specific clinical presentations. Read More

Neurol India 2021 Mar-Apr;69(2):497-499

Department of Neurology, Shandong Provincial Hospital Affiliated to Shandong First Medical University, No. 324 JingWu Road, 250021 Jinan, Shandong, P.R. China.

Lymphomatoid granulomatosis (LYG) is a rare lymphoproliferative disease characterized by angiocentric and angiodestructive infiltrate. It primarily affects the lung and sometimes may also affect the central nervous system (CNS), skin, kidney, liver, etc., but the involvement of lymph nodes and/or bone marrow is extremely rare, and if present, other diagnoses are usually considered. Read More

Brain Tumor Pathol 2021 Jul 30;38(3):263-270. Epub 2021 Mar 30.

Department of Neurosurgery, Keio University School of Medicine, 35 Shinanomachi, Shinjuku-Ku, Tokyo, Japan.

Lymphomatoid granulomatosis (LYG) is a rare lymphoproliferative disease with angiocentric and angiodestructive infiltrates, and by definition, Epstein-Barr virus (EBV)-associated B-cell malignancy. It most frequently involves the lung, and in some cases, the lesions are confined to the central nervous system (isolated CNS-LYG). However, it remains a controversial disease in terms of pathophysiology, especially in those confined to the CNS. Read More

J Hematop 2021 Mar 20:1-5. Epub 2021 Mar 20.

Department of Pathology, Instituto Nacional de Cancerología, México City, México.

Lymphomatoid granulomatosis is a very rare B cell lymphoproliferative disease associated with Epstein-Barr virus infection. It is related to states of immunosuppression and affects the lung in more than 90% of cases, forcing the clinician to establish a differential diagnosis with other diseases such as infections, Wegener's granulomatosis, lymphoma, or lung metastases. There is no standard treatment for this disease. Read More

Ear Nose Throat J 2021 Feb 26:145561321997536. Epub 2021 Feb 26.

22629Saint Vincent Health Center, Erie, PA, USA.

Epstein-Barr virus (EBV) associated lymphoproliferative disorders includes a diverse group of diagnoses, encompassing both B-cell and T-cell lineages. With EBV mucocutaneous ulcers becoming a World health Organization diagnosis in 2018, introduction of the disease entity will be beneficial to the practicing otolaryngologist. We are reporting a case of a 69-year-old male with history of rheumatoid arthritis on methotrexate, recently undergoing dental extractions, who then developed multiple oral ulcerations and bony erosions of his palate and alveolar ridge. Read More

BMC Neurol 2021 Feb 18;21(1):80. Epub 2021 Feb 18.

Department of Neurology, Northern Health, 185 Cooper Street, Epping, Victoria, 3076, Australia.

Background: Chronic lymphocytic infiltration with pontine perivascular enhancement responsive to steroids (CLIPPERS) is a neuro-inflammatory syndrome first described in 2010. It has a relationship with lymphoproliferative disorders that has not been fully elucidated. This case represents an unusual progression of CLIPPERS to Epstein-Barr Virus (EBV)-related lymphomatoid granulomatosis (LYG). Read More

J Vet Diagn Invest 2021 Mar 10;33(2):340-344. Epub 2021 Feb 10.

Kansas State Veterinary Diagnostic Laboratory, College of Veterinary Medicine, Kansas State University, Manhattan, KS.

Lymphomatoid granulomatosis (LYG) is a rare variant of an angioinvasive T-cell lymphoproliferative disorder that primarily affects the lungs, with common sites of metastasis including the skin and subcutis. In humans, it is a B-cell lymphoproliferative disorder associated with Epstein-Barr virus infection. Our case is a 7-y-old, spayed female, domestic longhair cat that decompensated and was euthanized following an initial diagnosis of angioinvasive lymphoma from a skin biopsy. Read More

Cancers (Basel) 2021 Jan 22;13(3). Epub 2021 Jan 22.

Pathology Unit, Azienda Ospedaliera S. Maria di Terni, University of Perugia, 05100 Terni, Italy.

Primary pulmonary B-cell lymphomas (PP-BCLs) comprise a group of extranodal non-Hodgkin lymphomas of B-cell origin, which primarily affect the lung without evidence of extrapulmonary disease at the time of diagnosis and up to 3 months afterwards. Primary lymphoid proliferations of the lung are most often of B-cell lineage, and include three major entities with different clinical, morphological, and molecular features: primary pulmonary marginal zone lymphoma of mucosa-associated lymphoid tissue (PP-MZL, or MALT lymphoma), primary pulmonary diffuse large B cell lymphoma (PP-DLBCL), and lymphomatoid granulomatosis (LYG). Less common entities include primary effusion B-cell lymphoma (PEL) and intravascular large B cell lymphoma (IVLBCL). Read More

Respir Med Case Rep 2021 28;32:101327. Epub 2020 Dec 28.

Department of Pathology, Hakodate National Hospital, 0418512, Hakodate, Hokkaido, Japan.

Lymphomatoid granulomatosis (LYG) is a rare Epstein-Barr virus-associated B-cell lymphoproliferative disorder and was incorporated into the WHO classification of Tumours of the Lung, Pleura, Thymus and Heart in 2015. LYG is known to be associated with the host's immune function, and can be caused by some immunosuppressive agents, including methotrexate. A woman in her sixties with an 18-year history of methotrexate treatment for rheumatoid arthritis visited our hospital after detection of an abnormal chest shadow on her radiograph. Read More

Neurol Sci 2021 04 20;42(4):1587-1590. Epub 2020 Oct 20.

Department of Neurology, Shandong Provincial Hospital Affiliated to Shandong First Medical University, No. 324 JingWu Road, Jinan, 250021, Shandong, People's Republic of China.

Neurologia (Engl Ed) 2020 Oct 16. Epub 2020 Oct 16.

Servicio de Anatomía Patológica, Hospital Universitario de Cruces, Barakaldo, España.

Front Neurol 2020 11;11:901. Epub 2020 Sep 11.

Department of Neurology, Shandong Provincial Hospital Affiliated to Shandong First Medical University, Jinan, China.

Lymphomatoid granulomatosis (LYG) is an infrequent lymphoproliferative disease that typically involves the lungs, but may also affect the central nervous system (CNS). Isolated CNS involvement is very rare, and its clinicopathological features have not been fully elucidated. Here, we systematically reviewed the English literature through PubMed to collect all relevant case reports and small case series with pathologically confirmed primary CNS-LYG. Read More