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Chakroun S, Faucher A, Gueguen A, Fadlallah J, Corvilain E, Kubis N, Lozeron P. Primary Neurolymphomatosis: A Literature Review. Eur J Neurol 2025; 32:e70173. [PMID: 40265708 PMCID: PMC12015973 DOI: 10.1111/ene.70173] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 12/03/2024] [Revised: 03/04/2025] [Accepted: 04/10/2025] [Indexed: 04/24/2025]
Abstract
BACKGROUND Primary neurolymphomatosis (PNL) is a rare clinical entity resulting from direct lymphomatous infiltration into the peripheral nervous system. Its diagnosis is challenging as the hematological condition is unknown at the onset of neurological symptoms. METHODS We report two of our own cases and the first extensive review of published cases of PNL to delineate its clinical features, paraclinical investigation results, progression, and treatment response more precisely. We extracted demographic data, clinical presentation, results of the investigations performed, type and number of treatments, overall survival, and progression-free survival. RESULTS We describe 301 cases of PNL in patients with a mean age of 57.9 years, 61% of whom were men. The most common clinical presentation was an often painless asymmetric neuropathy. Other presentations included multifocal neuropathy preferentially affecting the sciatic and peroneal nerves, radiculopathy, brachial plexus lesions, cauda equina syndrome, and cranial nerve palsy. Systemic signs and deterioration of clinical status were uncommon. Diagnosis was established after a median of 8 months, based on histological results (76%) or a cluster of elements in cases of positive PET findings. A B-cell lymphoma was diagnosed in 73% of cases. Systemic chemotherapy (90%) and rituximab (60%) were the most common treatments, with a response rate of 45%. Relapse occurred in 24% of patients, and 55% ultimately died from PNL. Overall survival was 28 months. Type of treatment was not associated with survival. CONCLUSIONS This literature review provides an overview of the available data concerning PNL presentation and progression.
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Affiliation(s)
- Sahar Chakroun
- Service de Physiologie Clinique—Explorations FonctionnellesAP‐HP, DMU DREAM, Hôpital LariboisièreParisFrance
| | - Alice Faucher
- Service de Physiologie Clinique—Explorations FonctionnellesAP‐HP, DMU DREAM, Hôpital LariboisièreParisFrance
| | - Antoine Gueguen
- Service de NeurologieHôpital Fondation Adolphe de RothschildParisFrance
| | - Jehane Fadlallah
- Service D'immuno Pathologie CliniqueHôpital Saint Louis, APHPParisFrance
| | - Emilie Corvilain
- Service D'immuno Pathologie CliniqueHôpital Saint Louis, APHPParisFrance
| | - Nathalie Kubis
- Service de Physiologie Clinique—Explorations FonctionnellesAP‐HP, DMU DREAM, Hôpital LariboisièreParisFrance
- Université Paris Cité, INSERM UMR‐S1144, Optimisation Thérapeutique en NeuropsychopharmacologieFaculté de Pharmacie de ParisParisFrance
- Centre de Compétence Neuropathies Rares et Amyloïdes Ile‐de France Caraïbes (CERAMIC), FILNEMUS, AP‐HP, DMU DREAM, Hôpital LariboisièreParisFrance
| | - Pierre Lozeron
- Service de Physiologie Clinique—Explorations FonctionnellesAP‐HP, DMU DREAM, Hôpital LariboisièreParisFrance
- Université Paris Cité, INSERM UMR‐S1144, Optimisation Thérapeutique en NeuropsychopharmacologieFaculté de Pharmacie de ParisParisFrance
- Centre de Compétence Neuropathies Rares et Amyloïdes Ile‐de France Caraïbes (CERAMIC), FILNEMUS, AP‐HP, DMU DREAM, Hôpital LariboisièreParisFrance
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Madduluri B, Shaik AJ, Kamani NB, Sultana RS, Uppin MS. Clinical Reasoning: An Unusual Etiology of Lumbosacral Plexopathy in a Patient With HIV. Neurology 2024; 103:e209930. [PMID: 39331848 DOI: 10.1212/wnl.0000000000209930] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 09/29/2024] Open
Abstract
We present a compelling case of uncontrolled diabetes, who initially presented as diabetic lumbosacral radiculoplexus neuropathy (DLRPN), with radicular pain in the right lower limb (LL) followed by asymmetric weakness of both LLs (right greater than left) with wasting in the medial compartment of the right thigh and significant sensory loss in the bilateral sural and right saphenous nerve distribution. Electrophysiology was suggestive of right lumbosacral radiculoplexus neuropathy. Incidentally, the patient tested positive for HIV-1 at our tertiary care center. CSF analysis revealed markedly elevated protein levels (>400 mg/dL) with lymphocytosis, a red flag for DLRPN. This observation led to further workup. Nerve biopsy showed large collections of perivascular endoneurial and epineurial lymphoid inflammatory cells, which favored an alternative diagnosis. This case highlights the intricate interplay between HIV infection, diabetes, and neurologic manifestations, challenging the initial clinical suspicion of DLRPN. This study emphasizes the importance of considering atypical presentations of neuropathy, especially in the context of coexisting medical conditions, and emphasizes the significance of comprehensive diagnostic workup, including CSF studies and nerve biopsy, for an accurate diagnosis.
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Affiliation(s)
- Bhavani Madduluri
- From the Departments of Neurology (B.M., A.J.S., N.B.K., R.S.S.), and Pathology (M.S.U.), Nizam's Institute of Medical Sciences
| | - Afshan J Shaik
- From the Departments of Neurology (B.M., A.J.S., N.B.K., R.S.S.), and Pathology (M.S.U.), Nizam's Institute of Medical Sciences
| | - Naresh Babu Kamani
- From the Departments of Neurology (B.M., A.J.S., N.B.K., R.S.S.), and Pathology (M.S.U.), Nizam's Institute of Medical Sciences
| | - Reshma S Sultana
- From the Departments of Neurology (B.M., A.J.S., N.B.K., R.S.S.), and Pathology (M.S.U.), Nizam's Institute of Medical Sciences
| | - Megha S Uppin
- From the Departments of Neurology (B.M., A.J.S., N.B.K., R.S.S.), and Pathology (M.S.U.), Nizam's Institute of Medical Sciences
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Bakshi N, Chand V, Anand R, Duggal R. Primary neurolymphomatosis presenting as foot drop: A rare case report. INDIAN J PATHOL MICR 2024; 67:918-920. [PMID: 38391312 DOI: 10.4103/ijpm.ijpm_731_22] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 09/08/2022] [Accepted: 11/14/2022] [Indexed: 02/24/2024] Open
Abstract
ABSTRACT Neurolymphomatosis (NL) is the direct infiltration of the peripheral nervous system (PNS) by lymphoma cells and represents the least common form of PNS involvement by lymphoma. Clinical presentation is varied, and early diagnosis remains challenging. Nerve biopsy remains the diagnostic gold standard, use of magnetic resonance imaging (MRI) and fluorodeoxyglucose-positron emission tomography (FDG-PET) may help in diagnosis and selecting targets for biopsy. We report an account of an older male patient who presented with subacute onset multifocal neuropathy involving bilateral lower limbs and left foot drop. The patient was initially misdiagnosed as chronic inflammatory demyelinating polyneuropathy; however radiological assessment revealed fusiform thickening of the L5 nerve root and biopsy from the site revealed extensive infiltration by diffuse large B cell type non-Hodgkin lymphoma (NHL). High index of suspicion is vital to ensure correct diagnosis, timely treatment, and to improve patient survival.
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Affiliation(s)
- Neha Bakshi
- Department of Pathology (Histopathology Division), Sir Ganga Ram Hospital, New Delhi, India
| | - Vineeta Chand
- Department of Histopathology and Cytopathology, BLK-MAX Hospital, New Delhi, India
| | - Rajiv Anand
- Department of Neurology, BLK-MAX Hospital, New Delhi, India
| | - Rajan Duggal
- Department of Histopathology and Cytopathology, BLK-MAX Hospital, New Delhi, India
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4
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Choi K, Jung S, Jung G, Kim D, Oh J. Incidence of cancer in chronic inflammatory demyelinating polyneuropathy: a nationwide cohort study in South Korea. Front Neurol 2024; 15:1456835. [PMID: 39268061 PMCID: PMC11390450 DOI: 10.3389/fneur.2024.1456835] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/29/2024] [Accepted: 08/02/2024] [Indexed: 09/15/2024] Open
Abstract
Background Chronic inflammatory demyelinating polyneuropathy (CIDP) is a rare disease, and the potential risk of cancer in patients with CIDP remains an important concern during treatment. However, a comprehensive epidemiological study examining this association is yet to be conducted. This study aimed to investigate the incidence of cancer in patients with CIDP in South Korea using data from the Korean Health Insurance Review and Assessment Service (HIRA) database. Methods Data from the HIRA database between January 2016 and June 2021 were analyzed. The actual incidence of cancer in patients with CIDP was compared with the expected incidence based on the general population statistics in South Korea, with adjustments for age. Results In total, 888 patients with CIDP were included in the analysis, of whom 50 (5.63% of malignancy incidence) were newly diagnosed with cancer during the study period. Among the patients with CIDP diagnosed with cancer, 32 (64.00%) were aged 60 years or older, and 36 (72.00%) were male. The observed number of cancer diagnoses corresponded to an incidence rate of 5.63%, with a standardized incidence ratio (SIR) of 2.83 (95% confidence interval [CI]: 1.89-4.39) compared to the expected cancer incidence rate of 2.00%. Notably, the SIR for malignancies of lymphoid, hematopoietic, and related tissues, excluding malignant immunoproliferative diseases, multiple myeloma, and plasma cell neoplasms (C81-96, except C88 and C90), was the highest at 8.51 (95% CI: 4.18-19.83). Conclusion Our study shows a potential association between CIDP and an increased risk of hematological malignancies, which is consistent with previous investigations. Further studies are required to better understand the relationship between CIDP and cancer.
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Affiliation(s)
- Kyomin Choi
- Department of Neurology, College of Medicine, Soonchunhyang University Cheonan Hospital, Cheonan-si, Republic of Korea
| | - Sohee Jung
- Department of Medical Artificial Intelligence, Deepnoid Inc., Seoul, Republic of Korea
| | - Gucheol Jung
- Department of Medical Artificial Intelligence, Deepnoid Inc., Seoul, Republic of Korea
| | - Dayoung Kim
- Department of Neurology, College of Medicine, Konkuk University Medical Center, Seoul, Republic of Korea
| | - Jeeyoung Oh
- Department of Neurology, College of Medicine, Konkuk University Medical Center, Seoul, Republic of Korea
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Shelly S, Dubey D, Mills JR, Klein CJ. Paraneoplastic neuropathies and peripheral nerve hyperexcitability disorders. HANDBOOK OF CLINICAL NEUROLOGY 2024; 200:239-273. [PMID: 38494281 DOI: 10.1016/b978-0-12-823912-4.00020-7] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Subscribe] [Scholar Register] [Indexed: 03/19/2024]
Abstract
Peripheral neuropathy is a common referral for patients to the neurologic clinics. Paraneoplastic neuropathies account for a small but high morbidity and mortality subgroup. Symptoms include weakness, sensory loss, sweating irregularity, blood pressure instability, severe constipation, and neuropathic pain. Neuropathy is the first presenting symptom of malignancy among many patients. The molecular and cellular oncogenic immune targets reside within cell bodies, axons, cytoplasms, or surface membranes of neural tissues. A more favorable immune treatment outcome occurs in those where the targets reside on the cell surface. Patients with antibodies binding cell surface antigens commonly have neural hyperexcitability with pain, cramps, fasciculations, and hyperhidrotic attacks (CASPR2, LGI1, and others). The antigenic targets are also commonly expressed in the central nervous system, with presenting symptoms being myelopathy, encephalopathy, and seizures with neuropathy, often masked. Pain and autonomic components typically relate to small nerve fiber involvement (nociceptive, adrenergic, enteric, and sudomotor), sometimes without nerve fiber loss but rather hyperexcitability. The specific antibodies discovered help direct cancer investigations. Among the primary axonal paraneoplastic neuropathies, pathognomonic clinical features do not exist, and testing for multiple antibodies simultaneously provides the best sensitivity in testing (AGNA1-SOX1; amphiphysin; ANNA-1-HU; ANNA-3-DACH1; CASPR2; CRMP5; LGI1; PCA2-MAP1B, and others). Performing confirmatory antibody testing using adjunct methods improves specificity. Antibody-mediated demyelinating paraneoplastic neuropathies are limited to MAG-IgM (IgM-MGUS, Waldenström's, and myeloma), with the others associated with cytokine elevations (VEGF, IL6) caused by osteosclerotic myeloma, plasmacytoma (POEMS), and rarely angiofollicular lymphoma (Castleman's). Paraneoplastic disorders have clinical overlap with other idiopathic antibody disorders, including IgG4 demyelinating nodopathies (NF155 and Contactin-1). This review summarizes the paraneoplastic neuropathies, including those with peripheral nerve hyperexcitability.
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Affiliation(s)
- Shahar Shelly
- Department of Neurology, Mayo Clinic, Rochester, MN, United States; Department of Laboratory Medicine and Pathology, Mayo Clinic, Rochester, MN, United States; Department of Neurology, Rambam Health Care Campus, Haifa, Israel; Faculty of Medicine, Bruce Rappaport Faculty of Medicine, Technion-Israel Institute of Technology, Haifa, Israel
| | - Divyanshu Dubey
- Department of Neurology, Mayo Clinic, Rochester, MN, United States; Department of Laboratory Medicine and Pathology, Mayo Clinic, Rochester, MN, United States
| | - John R Mills
- Department of Laboratory Medicine and Pathology, Mayo Clinic, Rochester, MN, United States
| | - Christopher J Klein
- Department of Neurology, Mayo Clinic, Rochester, MN, United States; Department of Laboratory Medicine and Pathology, Mayo Clinic, Rochester, MN, United States.
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Gao X, Murphy MM, Peyer JG, Ni Y, Yang M, Zhang Y, Guo J, Kara N, Embree C, Tasdogan A, Ubellacker JM, Crane GM, Fang S, Zhao Z, Shen B, Morrison SJ. Leptin receptor + cells promote bone marrow innervation and regeneration by synthesizing nerve growth factor. Nat Cell Biol 2023; 25:1746-1757. [PMID: 38012403 PMCID: PMC10709146 DOI: 10.1038/s41556-023-01284-9] [Citation(s) in RCA: 23] [Impact Index Per Article: 11.5] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 01/25/2023] [Accepted: 10/09/2023] [Indexed: 11/29/2023]
Abstract
The bone marrow contains peripheral nerves that promote haematopoietic regeneration after irradiation or chemotherapy (myeloablation), but little is known about how this is regulated. Here we found that nerve growth factor (NGF) produced by leptin receptor-expressing (LepR+) stromal cells is required to maintain nerve fibres in adult bone marrow. In nerveless bone marrow, steady-state haematopoiesis was normal but haematopoietic and vascular regeneration were impaired after myeloablation. LepR+ cells, and the adipocytes they gave rise to, increased NGF production after myeloablation, promoting nerve sprouting in the bone marrow and haematopoietic and vascular regeneration. Nerves promoted regeneration by activating β2 and β3 adrenergic receptor signalling in LepR+ cells, and potentially in adipocytes, increasing their production of multiple haematopoietic and vascular regeneration growth factors. Peripheral nerves and LepR+ cells thus promote bone marrow regeneration through a reciprocal relationship in which LepR+ cells sustain nerves by synthesizing NGF and nerves increase regeneration by promoting the production of growth factors by LepR+ cells.
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Affiliation(s)
- Xiang Gao
- National Institute of Biological Sciences, Beijing, China
- Tsinghua Institute of Multidisciplinary Biomedical Research, Tsinghua University, Beijing, China
- Academy for Advanced Interdisciplinary Studies, Peking University, Beijing, China
| | - Malea M Murphy
- Children's Research Institute and the Department of Pediatrics, University of Texas Southwestern Medical Center, Dallas, TX, USA
- Integrated Microscopy and Imaging Laboratory, Texas A&M Health Science Center, Texas A&M University, College Station, TX, USA
| | - James G Peyer
- Children's Research Institute and the Department of Pediatrics, University of Texas Southwestern Medical Center, Dallas, TX, USA
- Cambrian Bio, Inc., New York, NY, USA
| | - Yuehan Ni
- National Institute of Biological Sciences, Beijing, China
- College of Life Sciences, Beijing Normal University, Beijing, China
| | - Min Yang
- National Institute of Biological Sciences, Beijing, China
- College of Life Sciences, Beijing Normal University, Beijing, China
| | - Yixuan Zhang
- National Institute of Biological Sciences, Beijing, China
| | - Jiaming Guo
- National Institute of Biological Sciences, Beijing, China
- Academy for Advanced Interdisciplinary Studies, Peking University, Beijing, China
| | - Nergis Kara
- Children's Research Institute and the Department of Pediatrics, University of Texas Southwestern Medical Center, Dallas, TX, USA
- Ensoma, Inc., Boston, MA, USA
| | - Claire Embree
- Children's Research Institute and the Department of Pediatrics, University of Texas Southwestern Medical Center, Dallas, TX, USA
| | - Alpaslan Tasdogan
- Children's Research Institute and the Department of Pediatrics, University of Texas Southwestern Medical Center, Dallas, TX, USA
- Department of Dermatology, University Hospital Essen and German Cancer Consortium, Essen, Germany
| | - Jessalyn M Ubellacker
- Children's Research Institute and the Department of Pediatrics, University of Texas Southwestern Medical Center, Dallas, TX, USA
- Department of Molecular Metabolism, Harvard T.H. Chan School of Public Health, Boston, MA, USA
| | - Genevieve M Crane
- Robert J. Tomsich Pathology and Laboratory Medicine Institute, Cleveland Clinic, Cleveland, OH, USA
| | - Shentong Fang
- School of Biopharmacy, China Pharmaceutical University, Nanjing, China
| | - Zhiyu Zhao
- Children's Research Institute and the Department of Pediatrics, University of Texas Southwestern Medical Center, Dallas, TX, USA
| | - Bo Shen
- National Institute of Biological Sciences, Beijing, China.
- Tsinghua Institute of Multidisciplinary Biomedical Research, Tsinghua University, Beijing, China.
- Children's Research Institute and the Department of Pediatrics, University of Texas Southwestern Medical Center, Dallas, TX, USA.
| | - Sean J Morrison
- Children's Research Institute and the Department of Pediatrics, University of Texas Southwestern Medical Center, Dallas, TX, USA.
- Howard Hughes Medical Institute, UT Southwestern Medical Center, Dallas, TX, USA.
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Ponzo G, Umana GE, Valastro M, Giuffrida M, Tranchina MG, Nicoletti GF, Scalia G. Cervical radiculopathy as first presentation of CD3-positive diffuse large B-cell lymphoma of the cervico-thoracic junction. Br J Neurosurg 2023; 37:1190-1193. [PMID: 33012208 DOI: 10.1080/02688697.2020.1828278] [Citation(s) in RCA: 2] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/14/2020] [Accepted: 09/21/2020] [Indexed: 12/27/2022]
Abstract
BACKGROUND Diffuse large B-cell lymphoma (DLBCL) in rare cases can presents an unusual expression of CD3 T-cell specific antigen. We report the first case of a CD3-positive DLBCL of the cervico-thoracic junction presenting with persistent cervical radiculopathy. CASE PRESENTATION A 74-years-old male patient presented a severe and persistent right C8 radiculopathy associated with right-sided neck pain, progressive numbness and weakness of the right arm. The symptoms prominent during the night interfering with sleep and were resistant to anti-inflammatory drugs and cervical orthosis. Spine MRI showed a solid hypointense lesion on T2-weighted images and hyperintense on STIR sequences involving the epidural space at C7, T1 and T2. The patient underwent a C7-T1 decompressive laminectomy and left T2 hemilaminectomy with resection of the epidural tissue resulting in subtotal removal. Histology showed a DLBCL germinal center B-cell lymphoma with expression of CD3 T-cell specific antigen. Then the patient underwent adjuvant radiotherapy and chemotherapy consisting of R-CHOP protocol. At last follow-up (2 years) the patient is still in good clinical status (KPS = 80) with almost complete recovery of the cervical radiculopathy. CONCLUSIONS To our knowledge this is the first case of DLBLC GCB-like CD3 positive to present with radiculopathy.
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Affiliation(s)
- Giancarlo Ponzo
- Neurosurgery Unit, Highly Specialized Hospital and of National Importance "Garibaldi", Catania, Italy
| | - Giuseppe Emmanuele Umana
- Department of Neurosurgery, Cannizzaro Hospital, Trauma Center, Gamma Knife Center, Catania, Italy
| | - Maurizio Valastro
- Radiodiagnostic Unit, Highly Specialized Hospital of National Importance "Garibaldi", Catania, Italy
| | - Massimiliano Giuffrida
- Neurosurgery Unit, Highly Specialized Hospital and of National Importance "Garibaldi", Catania, Italy
| | | | | | - Gianluca Scalia
- Neurosurgery Unit, Highly Specialized Hospital and of National Importance "Garibaldi", Catania, Italy
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Yao X, Zhang C, Zhang Y, Geng J, Bai S, Hao Y, Guan Y. Amphiphysin-IgG autoimmune sciatic neuropathy and facial neuropathy related to primary central nervous system lymphoma: A case report. J Neuroimmunol 2023; 382:578156. [PMID: 37556888 DOI: 10.1016/j.jneuroim.2023.578156] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/28/2023] [Revised: 06/16/2023] [Accepted: 07/25/2023] [Indexed: 08/11/2023]
Abstract
We reported a 61-year-old man presented with 10-month progressing left sciatic neuropathy and 10-day right facial neuropathy. Serum amphiphysin-IgG was positive. 18F-FDG PET/CT of the whole body showed no signs of malignancy. Treatment with plasma exchange and oral prednisone relieved the symptoms. Nine months later, right hemiparesis and seizure of right limbs developed. 18F-FDG and 18F-PBR06 (18 kDa translocator protein, TSPO) radioligand PET/MRI of the whole body revealed intense uptake in the intracranial lesions. Intracranial lymphoma was diagnosed by stereotactic needle brain biopsy. Mononeuropathies could be paraneoplastic syndromes. TSPO shows high uptake in intracranial lymphoma on 18F-PBR06 PET images.
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Affiliation(s)
- Xiaoying Yao
- Department of Neurology, Ren Ji Hospital, Shanghai Jiao Tong University School of Medicine, Shanghai 200127, China
| | - Chenpeng Zhang
- Department of Nuclear Medicine, Ren Ji Hospital, Shanghai Jiao Tong University School of Medicine, Shanghai 200127, China
| | - Ying Zhang
- Department of Neurology, Ren Ji Hospital, Shanghai Jiao Tong University School of Medicine, Shanghai 200127, China
| | - Jieli Geng
- Department of Neurology, Ren Ji Hospital, Shanghai Jiao Tong University School of Medicine, Shanghai 200127, China
| | - Shuwei Bai
- Department of Neurology, Ren Ji Hospital, Shanghai Jiao Tong University School of Medicine, Shanghai 200127, China
| | - Yong Hao
- Department of Neurology, Ren Ji Hospital, Shanghai Jiao Tong University School of Medicine, Shanghai 200127, China
| | - Yangtai Guan
- Department of Neurology, Ren Ji Hospital, Shanghai Jiao Tong University School of Medicine, Shanghai 200127, China.
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9
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Chen Y, Wang Y, Corrigan J, Memon AB. B-Cell Lymphoma Presenting With Seventh Cranial Nerve Palsy and Mononeuritis Multiplex: A Case Report and Comprehensive Literature Review. Cureus 2023; 15:e44983. [PMID: 37822434 PMCID: PMC10564262 DOI: 10.7759/cureus.44983] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Accepted: 09/10/2023] [Indexed: 10/13/2023] Open
Abstract
Diagnosing B-cell lymphoma-associated mononeuritis multiplex is challenging due to its rarity and the potential co-existence of other causes of mononeuritis multiplex. Here, we report a case of a 74-year-old male who initially presented with left cranial neuropathies followed by right-sided extremity weakness with hyporeflexia, right facial involvement, and subsequently asymmetric weakness and multifocal muscle wasting. Minor improvements were observed with multiple rounds of steroid treatment. The diffuse large B-cell lymphoma diagnosis was eventually established six months later upon a repeat mediastinal lymph node biopsy and cerebrospinal fluid cytology. A nerve biopsy demonstrated severe axonal neuropathy with loss of axons in all fascicles without evidence of vasculitis. A muscle biopsy showed atrophy in both type 1 and type 2 fibers. A presentation of mononeuritis multiplex warrants concern for B-cell lymphoma, mainly when other mechanisms of peripheral neuropathy are less likely.
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Affiliation(s)
- Yongzhen Chen
- School of Medicine, Saint Louis University, Saint Louis, USA
| | - Yilun Wang
- School of Medicine, Texas Agricultural and Mechanical (A&M) University, Bryan, USA
| | - John Corrigan
- Department of Radiology, Henry Ford Health System, Detroit, USA
- School of Medicine, Wayne State University, Detroit, USA
| | - Anza B Memon
- Department of Neurology, John D. Dingell Veterans Affairs Medical Center, Detroit, USA
- School of Medicine, Wayne State University, Detroit, USA
- Department of Neurology, Henry Ford Health System, Detroit, USA
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10
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Fargeot G, Gitiaux C, Magy L, Pereon Y, Delmont E, Viala K, Echaniz-Laguna A. French recommendations for the management of adult & pediatric chronic inflammatory demyelinating polyradiculoneuropathy (CIDP). Rev Neurol (Paris) 2022; 178:953-968. [PMID: 36182621 DOI: 10.1016/j.neurol.2022.06.004] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/06/2021] [Revised: 05/31/2022] [Accepted: 06/05/2022] [Indexed: 11/22/2022]
Abstract
Chronic inflammatory demyelinating polyradiculoneuropathy (CIDP) is a rare autoimmune disorder of the peripheral nervous system, primarily affecting the myelin sheath. The pathophysiology of CIDP is complex, involving both humoral and cellular immunity. The diagnosis of CIDP should be suspected in patients with symmetrical proximal and distal motor weakness and distal sensory symptoms of progressive onset, associated with decreased/abolished tendon reflexes. Treatments include intraveinous immunoglobulins, steroids and plasma exchange, with usually an induction phase followed by a maintenance therapy with progressive weaning. Treatment should be rapidly initiated to prevent axonal degeneration, which may compromise recovery. CIDP outcome is variable, ranging from mild distal paresthesiae to complete loss of ambulation. There have been several breakthroughs in the diagnosis and management of CIDP the past ten years, e.g. discovery of antibodies against the node of Ranvier, contribution of nerve ultrasound and magnetic resonance imaging to diagnosis, and demonstration of subcutaneous immunoglobulins efficiency. This led us to elaborate French recommendations for the management of adult & pediatric CIDP patients. These recommendations include diagnosis assessment, treatment, and follow-up.
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Affiliation(s)
- G Fargeot
- Neurophysiology Department, Pitié-Salpêtrière Hospital, AP-HP, Paris, France.
| | - C Gitiaux
- Department of Paediatric Neurophysiology, Necker-Enfants-Malades Hospital, AP-HP, Paris University, Paris, France
| | - L Magy
- Department of Neurology, National Reference Center for 'Rare Peripheral Neuropathies', University Hospital of Limoges, Limoges, France
| | - Y Pereon
- CHU Nantes, Centre de Référence Maladies Neuromusculaires AOC, Filnemus, Euro-NMD, Explorations Fonctionnelles, Hôtel-Dieu, Nantes, France
| | - E Delmont
- Reference Center for Neuromuscular Diseases and ALS Timone University Hospital, Aix-Marseille University, Marseille, France
| | - K Viala
- Neurophysiology Department, Pitié-Salpêtrière Hospital, AP-HP, Paris, France
| | - A Echaniz-Laguna
- Neurology Department, CHU de Bicêtre, AP-HP, Le-Kremlin-Bicêtre, France; French National Reference Center for Rare Neuropathies (NNERF), Le-Kremlin-Bicêtre, France; Inserm U1195, Paris-Saclay University, Le-Kremlin-Bicêtre, France
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11
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Cerri F, Gentile F, Clarelli F, Santoro S, Falzone YM, Dina G, Romano A, Domi T, Pozzi L, Fazio R, Podini P, Sorosina M, Carrera P, Esposito F, Riva N, Briani C, Cavallaro T, Filippi M, Quattrini A. Clinical and pathological findings in neurolymphomatosis: Preliminary association with gene expression profiles in sural nerves. Front Oncol 2022; 12:974751. [PMID: 36226068 PMCID: PMC9549065 DOI: 10.3389/fonc.2022.974751] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/21/2022] [Accepted: 08/22/2022] [Indexed: 11/13/2022] Open
Abstract
Although inflammation appears to play a role in neurolymphomatosis (NL), the mechanisms leading to degeneration in the peripheral nervous system are poorly understood. The purpose of this exploratory study was to identify molecular pathways underlying NL pathogenesis, combining clinical and neuropathological investigation with gene expression (GE) studies. We characterized the clinical and pathological features of eight patients with NL. We further analysed GE changes in sural nerve biopsies obtained from a subgroup of NL patients (n=3) and thirteen patients with inflammatory neuropathies as neuropathic controls. Based on the neuropathic symptoms and signs, NL patients were classified into three forms of neuropathy: chronic symmetrical sensorimotor polyneuropathy (SMPN, n=3), multiple mononeuropathy (MN, n=4) and acute motor-sensory axonal neuropathy (AMSAN, n=1). Predominantly diffuse malignant cells infiltration of epineurium was present in chronic SMPN, whereas endoneurial perivascular cells invasion was observed in MN. In contrast, diffuse endoneurium malignant cells localization occurred in AMSAN. We identified alterations in the expression of 1266 genes, with 115 up-regulated and 1151 down-regulated genes, which were mainly associated with ribosomal proteins (RP) and olfactory receptors (OR) signaling pathways, respectively. Among the top up-regulated genes were actin alpha 1 skeletal muscle (ACTA1) and desmin (DES). Similarly, in NL nerves ACTA1, DES and several RPs were highly expressed, associated with endothelial cells and pericytes abnormalities. Peripheral nerve involvement may be due to conversion towards a more aggressive phenotype, potentially explaining the poor prognosis. The candidate genes reported in this study may be a source of clinical biomarkers for NL.
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Affiliation(s)
- Federica Cerri
- Experimental Neuropathology Unit, Institute of Experimental Neurology, Division of Neuroscience, IRCCS Ospedale San Raffaele Scientific Institute, Milan, Italy
- Department of Neurology, IRCCS Ospedale San Raffaele Scientific Institute, Milan, Italy
| | - Francesco Gentile
- Experimental Neuropathology Unit, Institute of Experimental Neurology, Division of Neuroscience, IRCCS Ospedale San Raffaele Scientific Institute, Milan, Italy
- Department of Neurology IRCCS Istituto Auxologico Italiano, Milan, Italy
| | - Ferdinando Clarelli
- Laboratory of Human Genetics of Neurological Disorders, Institute of Experimental Neurology, Division of Neuroscience, IRCCS Ospedale San Raffaele Scientific Institute, Milan, Italy
| | - Silvia Santoro
- Laboratory of Human Genetics of Neurological Disorders, Institute of Experimental Neurology, Division of Neuroscience, IRCCS Ospedale San Raffaele Scientific Institute, Milan, Italy
| | - Yuri Matteo Falzone
- Experimental Neuropathology Unit, Institute of Experimental Neurology, Division of Neuroscience, IRCCS Ospedale San Raffaele Scientific Institute, Milan, Italy
- Department of Neurology IRCCS Istituto Auxologico Italiano, Milan, Italy
| | - Giorgia Dina
- Experimental Neuropathology Unit, Institute of Experimental Neurology, Division of Neuroscience, IRCCS Ospedale San Raffaele Scientific Institute, Milan, Italy
| | - Alessandro Romano
- Experimental Neuropathology Unit, Institute of Experimental Neurology, Division of Neuroscience, IRCCS Ospedale San Raffaele Scientific Institute, Milan, Italy
| | - Teuta Domi
- Experimental Neuropathology Unit, Institute of Experimental Neurology, Division of Neuroscience, IRCCS Ospedale San Raffaele Scientific Institute, Milan, Italy
| | - Laura Pozzi
- Experimental Neuropathology Unit, Institute of Experimental Neurology, Division of Neuroscience, IRCCS Ospedale San Raffaele Scientific Institute, Milan, Italy
| | - Raffaella Fazio
- Department of Neurology, IRCCS Ospedale San Raffaele Scientific Institute, Milan, Italy
| | - Paola Podini
- Experimental Neuropathology Unit, Institute of Experimental Neurology, Division of Neuroscience, IRCCS Ospedale San Raffaele Scientific Institute, Milan, Italy
| | - Melissa Sorosina
- Laboratory of Human Genetics of Neurological Disorders, Institute of Experimental Neurology, Division of Neuroscience, IRCCS Ospedale San Raffaele Scientific Institute, Milan, Italy
| | - Paola Carrera
- Division of Genetics and Cell Biology and Laboratory of Clinical Molecular Biology and Cytogenetics, Unit of Genomics for Human Disease Diagnosis, IRCCS Ospedale San Raffaele Scientific Institute, Milan, Italy
| | - Federica Esposito
- Department of Neurology, IRCCS Ospedale San Raffaele Scientific Institute, Milan, Italy
- Laboratory of Human Genetics of Neurological Disorders, Institute of Experimental Neurology, Division of Neuroscience, IRCCS Ospedale San Raffaele Scientific Institute, Milan, Italy
| | - Nilo Riva
- Experimental Neuropathology Unit, Institute of Experimental Neurology, Division of Neuroscience, IRCCS Ospedale San Raffaele Scientific Institute, Milan, Italy
- Department of Neurology, IRCCS Ospedale San Raffaele Scientific Institute, Milan, Italy
- *Correspondence: Nilo Riva, ; Angelo Quattrini,
| | - Chiara Briani
- Department of Neuroscience , University of Padova, Padova, Italy
| | - Tiziana Cavallaro
- Department of Neurology, Azienda Ospedaliera Universitaria Integrata, University Hospital G.B. Rossi, Verona, Italy
| | - Massimo Filippi
- Department of Neurology, IRCCS Ospedale San Raffaele Scientific Institute, Milan, Italy
| | - Angelo Quattrini
- Experimental Neuropathology Unit, Institute of Experimental Neurology, Division of Neuroscience, IRCCS Ospedale San Raffaele Scientific Institute, Milan, Italy
- *Correspondence: Nilo Riva, ; Angelo Quattrini,
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12
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Hattori K, Matsuda N, Yoshizawa M, Ugawa Y, Kanai K. [Oculomotor nerve palsy with preserved pupillary reaction in two cases of neurolymphomatosis]. Rinsho Shinkeigaku 2022; 62:552-557. [PMID: 35753787 DOI: 10.5692/clinicalneurol.cn-001727] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 06/15/2023]
Abstract
Case 1: A 64-year-old woman with acute ptosis and diplopia was admitted to our hospital. She had right oculomotor nerve palsy with preserved pupillary reaction without any other neurological deficits. MRI showed abnormal enhancement in the right oculomotor nerve. An ovarian tumor was detected on CT examination, and was pathologically diagnosed as diffuse large B-cell lymphoma (DLBCL). Cerebrospinal fluid cytology disclosed malignant lymphoma cells. Based on the above findings, we concluded that she had neurolymphomatosis (NL) of the right oculomotor nerve. Case 2: A 63-year-old woman was admitted to our hospital due to weakness of the bilateral lower extremities and gait disturbance. Lumbar MRI showed enhanced lesions in the cauda equina, and we diagnosed her as having DLBCL based on bone marrow aspiration study. She later developed right oculomotor nerve palsy with preserved pupillary reaction together with the right abducens and hypoglossal nerve palsies, which were caused by NL. Our cases suggest that oculomotor nerve palsy with preserved pupillary reaction can be a clinical feature of NL. Although NL mainly affects the subperinerium, as parasympathetic fibers are located in the periphery of the oculomotor nerve and supplied by pia matar blood vessels, patients with NL may shows this clinical feature.
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Affiliation(s)
| | | | | | - Yoshikazu Ugawa
- Department of Human Neurophysiology, Fukushima Medical University
| | - Kazuaki Kanai
- Department of Neurology, Fukushima Medical University
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13
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Lee YC, Juhng SK, Kang HJ. Neurolymphomatosis of the Ulnar Nerve Presenting as Cubital Tunnel Syndrome: A Case Report. J Hand Surg Asian Pac Vol 2022; 27:394-397. [PMID: 35443886 DOI: 10.1142/s242483552272016x] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/18/2022]
Abstract
Lymphoma is a malignant tumour of the lymphatic system. Neurolymphomatosis occurs when lymphoma cells invade the peripheral nervous system (PNS). Neurolymphomatosis has been reported involving the sciatic nerve, nerve roots and within the axilla and the upper arm. It can cause nerve dysfunction depending on the involved nerve. Neurolymphomatosis involving the ulnar nerve is rare. We report a patient with neurolymphomatosis of the ulnar nerve around the elbow that presented with features of cubital tunnel syndrome. A high index of suspicion is necessary in patients with cubital tunnel syndrome that present with uncommon symptoms such as sudden onset, rapid progression and/or severe pain. Level of Evidence: Level V (Therapeutic).
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Affiliation(s)
- Yeong Chang Lee
- Department of Orthopedic Surgery, Wonkwang University Hospital, Iksan, South Korea
| | - Seon Kwan Juhng
- Department of radiology, Wonkwang University Hospital, Iksan, South Korea
| | - Hong Je Kang
- Department of Orthopedic Surgery, Wonkwang University Hospital, Iksan, South Korea
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14
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De Vitis R, D'Orio M, Fiorentino V, Passiatore M. Primary lymphoma of the medial cutaneous nerve of the forearm: the first case in a pure sensory nerve of the upper limb. BMJ Case Rep 2022; 15:e248597. [PMID: 35354569 PMCID: PMC8968523 DOI: 10.1136/bcr-2021-248597] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Accepted: 03/17/2022] [Indexed: 11/04/2022] Open
Abstract
Primary lymphoma of the peripheral nerve is very rare and occurs most frequently in sciatic nerves. We describe the first patient reported in literature with a primary lymphoma in a pure sensory peripheral nerve of the upper limb. A woman in her 40s, with painful swelling and dysaesthesias in her left forearm in the past 3 months, was presented at our unit. Clinical MRI and ultrasound findings revealed a lesion that showed signs of a peripheral nerve sheath tumour. After complete excision, morpho-pathological evaluation revealed a primary B-cell lymphoma. The patient underwent radiotherapy and at the last follow-up there were no signs of residual pathology. Peripheral neuropathy may be caused by a lymphoma involving the nerve. Hand surgeons have to distinguish primary lymphoma of the peripheral nerves from schwannoma for their different clinical behaviour.
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Affiliation(s)
- Rocco De Vitis
- Department of Geriatrics Neurosciences and Orthopedics, Fondazione Policlinico Universitario Agostino Gemelli IRCCS, Roma, Lazio, Italy
| | - Marco D'Orio
- Department of Geriatrics Neurosciences and Orthopedics, Fondazione Policlinico Universitario Agostino Gemelli IRCCS, Roma, Lazio, Italy
| | - Vincenzo Fiorentino
- Department of Pathology, Fondazione Policlinico Universitario Agostino Gemelli IRCCS, Roma, Lazio, Italy
| | - Marco Passiatore
- Department of Geriatrics Neurosciences and Orthopedics, Fondazione Policlinico Universitario Agostino Gemelli IRCCS, Roma, Lazio, Italy
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15
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Zajc AL, Warwick H, Rosa C, Grant J, Marrington M. Bilateral trigeminal neuropathy associated with multicentric lymphoma in two dogs: Clinical presentation, MRI findings and flow cytometry characteristics. VETERINARY RECORD CASE REPORTS 2022. [DOI: 10.1002/vrc2.358] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 01/15/2023]
Affiliation(s)
| | - Harry Warwick
- Northwest Veterinary Specialists Sutton Weaver Runcorn UK
| | - Chantal Rosa
- Northwest Veterinary Specialists Sutton Weaver Runcorn UK
| | - Jessica Grant
- Northwest Veterinary Specialists Sutton Weaver Runcorn UK
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16
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Vizcaino MA, Kurtin PJ, King RL, Spinner RJ, Giannini C. Diffuse Large B-Cell Lymphoma of Peripheral Nerve with Distinctive Pathological Features Resembling Primary CNS Lymphoma. J Neuropathol Exp Neurol 2021; 81:76-78. [DOI: 10.1093/jnen/nlab117] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/14/2022] Open
Affiliation(s)
- M Adelita Vizcaino
- Department of Laboratory of Medicine and Pathology, Mayo Clinic Rochester, Minnesota, USA
| | - Paul J Kurtin
- Department of Laboratory of Medicine and Pathology, Mayo Clinic Rochester, Minnesota, USA
| | - Rebecca L King
- Department of Laboratory of Medicine and Pathology, Mayo Clinic Rochester, Minnesota, USA
| | - Robert J Spinner
- Department of Neurosurgery, Mayo Clinic Rochester, Minnesota, USA
| | - Caterina Giannini
- Department of Laboratory of Medicine and Pathology, Mayo Clinic Rochester, Minnesota, USA
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17
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Jiravichitchai T, Chira-Adisai W, Panuwannakorn M, Kiertiburanakul S. Claw Hand Deformity: A Rare Complication of Herpes Zoster. Orthop Res Rev 2021; 13:209-214. [PMID: 34819756 PMCID: PMC8607187 DOI: 10.2147/orr.s339064] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 09/13/2021] [Accepted: 11/03/2021] [Indexed: 11/23/2022] Open
Abstract
Distal upper extremity weakness is a rare complication after herpes zoster, which can be easily misdiagnosed by other nerve entrapment syndromes. We present a 31-year-old immunocompromised woman who developed a claw-like deformity of her right hand after full medical treatment and resolution of herpes zoster. The electrodiagnostic finding was compatible with right multiple mononeuropathies of the median and ulnar nerves, ongoing axonal loss, unlike the nerve entrapment patterns. The early recognition and early electrodiagnosis of herpes zoster-induced distal motor weakness, especially in the upper extremity, is necessary to exclude the nerve entrapment syndrome and to be the baseline for functional motor recovery prediction. The prognosis of functional motor recovery is considered good but may take months to years to accomplish. Rehabilitation management plays an important role after full medical treatment.
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Affiliation(s)
- Tachit Jiravichitchai
- Department of Rehabilitation Medicine, Faculty of Medicine Ramathibodi Hospital, Mahidol University, Bangkok, Thailand
| | - Waree Chira-Adisai
- Department of Rehabilitation Medicine, Faculty of Medicine Ramathibodi Hospital, Mahidol University, Bangkok, Thailand
| | - Monratta Panuwannakorn
- Department of Rehabilitation Medicine, Faculty of Medicine Ramathibodi Hospital, Mahidol University, Bangkok, Thailand
| | - Sasisopin Kiertiburanakul
- Division of Infectious Diseases, Department of Medicine, Faculty of Medicine Ramathibodi Hospital, Mahidol University, Bangkok, Thailand
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18
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Kajtazi NI, Khalid E, AlGhamdi J, Altaf S, AlHameed MH. Chronic inflammatory demyelinating polyneuropathy evolving to primary CNS lymphoma. BMJ Case Rep 2021; 14:e244767. [PMID: 34728507 PMCID: PMC8565527 DOI: 10.1136/bcr-2021-244767] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Accepted: 09/19/2021] [Indexed: 11/04/2022] Open
Abstract
A 56-year-old woman presented with 3 months history of all four limbs' numbness in glove and stocking distribution. A week before admission, she developed all four limbs' weakness and numbness. Brain and whole spine imaging revealed no lesions, and cerebrospinal fluid showed high protein. A nerve conduction study revealed severe sensorimotor polyneuropathy, and she was diagnosed with chronic inflammatory demyelinating polyneuropathy. We treated her with plasma exchange and later developed bilateral pulmonary embolism, deep venous thrombosis and worsening of weakness. The second set of seven sessions of plasma exchange gave her improvement in muscle strength. However, after a chest infection, another neurological deterioration occurred. The third set of plasma exchanges resulted in excellent response. Nineteen months into her illness she developed a headache followed by left upper limb weakness and focal seizures with left side involvement. Brain imaging revealed a right frontal enhancing lesion that required resection, and biopsy showed diffuse large B-cell lymphoma. She was treated with chemotherapy and whole-brain radiation therapy and remained with left-sided weakness.
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Affiliation(s)
- Naim Izet Kajtazi
- Department of Neurology, King Fahad Medical City, Riyadh, Saudi Arabia
| | - Ehtesham Khalid
- Department of Neurology, Ideal Medicare Clinic, Multan, Pakistan
| | - Juman AlGhamdi
- Medical Imaging Administration, Intervention Neuroradiology, King Fahad Medical City, Riyadh, Saudi Arabia
| | - Syed Altaf
- Department of Hematology, King Fahad Medical City, Riyadh, Saudi Arabia
| | - Majed H AlHameed
- Department of Neurology, King Fahad Medical City, Riyadh, Saudi Arabia
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19
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Zhang S. Chemotherapy-induced peripheral neuropathy and rehabilitation: A review. Semin Oncol 2021; 48:193-207. [PMID: 34607709 DOI: 10.1053/j.seminoncol.2021.09.004] [Citation(s) in RCA: 48] [Impact Index Per Article: 12.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 11/01/2020] [Revised: 09/02/2021] [Accepted: 09/03/2021] [Indexed: 12/29/2022]
Abstract
Chemotherapy-induced peripheral neuropathy (CIPN) is a common complication after chemotherapy that can damage the sensory, motor, autonomic, or cranial nerves in approximately 30%-60% of patients with cancer. CIPN can lead to detrimental dose modifications and/or premature chemotherapy discontinuation due to patient intolerance. The long-term impact of CIPN is particularly challenging and can have a profound impact on the quality of life (QoL) and survivorship. However, this condition is often underdiagnosed. No agents have been established to prevent CIPN. Pre-chemotherapy testing is recommended for high-risk patients. Duloxetine is considered a first-line treatment, whereas gabapentin, pregabalin, tricyclic antidepressants, and topical compounding creams may be used for neuropathic pain control. Home-based, low-to-moderate walking, and resistance exercise during chemotherapy can reduce the severity and prevalence of CIPN symptoms, especially in older patients. Pre-habilitation and rehabilitation should be recommended for all patients receiving cytotoxic chemotherapies. The purpose of this article is to review common chemotherapeutic drugs causing CIPN, risk factors, diagnosis and treatment of CIPN, and evidence of the benefits of rehabilitation.
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Affiliation(s)
- Shangming Zhang
- Department of Physical Medicine and Rehabilitation, Penn State Health Milton S. Hershey medical Center, Hershey, PA.
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20
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Pacoureau L, Labeyrie C, Catalan P, Echaniz-Laguna A, Henriquez S, Laparra A, Cauquil C, Chrétien P, Hacein-Bey-Abina S, Goujard C, Adam C, Lambotte O, Adams D, Noël N. Neuropathies périphériques associées aux syndromes lymphoprolifératifs : spectre clinique et démarche diagnostique. Rev Med Interne 2021; 42:844-854. [PMID: 34373143 DOI: 10.1016/j.revmed.2021.06.013] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 01/26/2021] [Revised: 06/17/2021] [Accepted: 06/20/2021] [Indexed: 10/20/2022]
Abstract
Lymphoproliferative syndromes (multiple myeloma, Waldenström's disease, chronic lymphocytic leukemia, lymphomas) may be associated with peripheral neuropathies. The mechanism can be dysimmune, associated or not with monoclonal gammopathies; paraneoplastic; infiltrative; or more commonly, iatrogenic or due to vitamin deficiency. The diagnosis can be complex, especially when the neuropathy is the presenting manifestation, requiring a close cooperation between internists and neurologists. The positive diagnosis of the neuropathy is based on a systematic electro-clinical investigation, which specifies the topography and the mechanism of the nerve damage, sometimes reinforced by imaging examinations, in particular, nerve and/or plexus MRI. The imputability of the neuropathy to a lymphoproliferative syndrome is based on a set of arguments including the clinical context (B signs, tumour syndrome), first-line laboratory tests (hemogram, protein electrophoresis, viral serologies, complement), auto-antibodies discussed according to the neuropathy (anti-MAG, anti-gangliosides) and sometimes more invasive examinations (bone marrow or neuro-muscular biopsies).
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Affiliation(s)
- L Pacoureau
- Service de médecine interne et immunologie clinique, Assistance publique-Hôpitaux Paris Saclay, Hôpital Bicêtre, 94275 Le Kremlin-Bicêtre cedex, France
| | - C Labeyrie
- Service de neurologie, Assistance publique-Hôpitaux de Paris, Groupe hospitalier universitaire Paris Sud, Hôpital Bicêtre, 94275 Le Kremlin-Bicêtre cedex, France; Centre de référence neuropathies amyloïdes familiales et autres neuropathies rares (NNERF), Groupe hospitalier universitaire Paris Sud, Hôpital Bicêtre, 94275 Le Kremlin-Bicêtre cedex, France
| | - P Catalan
- Service de médecine interne et immunologie clinique, Assistance publique-Hôpitaux Paris Saclay, Hôpital Bicêtre, 94275 Le Kremlin-Bicêtre cedex, France; Université Paris Saclay, Faculté de médecine, Le Kremlin Bicêtre, France
| | - A Echaniz-Laguna
- Université Paris Saclay, Faculté de médecine, Le Kremlin Bicêtre, France; Service de neurologie, Assistance publique-Hôpitaux de Paris, Groupe hospitalier universitaire Paris Sud, Hôpital Bicêtre, 94275 Le Kremlin-Bicêtre cedex, France; Centre de référence neuropathies amyloïdes familiales et autres neuropathies rares (NNERF), Groupe hospitalier universitaire Paris Sud, Hôpital Bicêtre, 94275 Le Kremlin-Bicêtre cedex, France
| | - S Henriquez
- Service de médecine interne et immunologie clinique, Assistance publique-Hôpitaux Paris Saclay, Hôpital Bicêtre, 94275 Le Kremlin-Bicêtre cedex, France; Université Paris Saclay, Faculté de médecine, Le Kremlin Bicêtre, France
| | - A Laparra
- Service de médecine interne et immunologie clinique, Assistance publique-Hôpitaux Paris Saclay, Hôpital Bicêtre, 94275 Le Kremlin-Bicêtre cedex, France; Université Paris Saclay, Faculté de médecine, Le Kremlin Bicêtre, France
| | - C Cauquil
- Service de neurologie, Assistance publique-Hôpitaux de Paris, Groupe hospitalier universitaire Paris Sud, Hôpital Bicêtre, 94275 Le Kremlin-Bicêtre cedex, France; Centre de référence neuropathies amyloïdes familiales et autres neuropathies rares (NNERF), Groupe hospitalier universitaire Paris Sud, Hôpital Bicêtre, 94275 Le Kremlin-Bicêtre cedex, France
| | - P Chrétien
- INSERM, UTCBS, Unité des technologies chimiques et biologiques pour la Santé, Université de Paris, CNRS, 75006 Paris, France; Service d'immunologie biologique, Assistance publique-Hôpitaux de Paris, Groupe hospitalier universitaire Paris Sud, Hôpital Bicêtre, 94275 Le Kremlin-Bicêtre cedex, France
| | - S Hacein-Bey-Abina
- INSERM, UTCBS, Unité des technologies chimiques et biologiques pour la Santé, Université de Paris, CNRS, 75006 Paris, France; Service d'immunologie biologique, Assistance publique-Hôpitaux de Paris, Groupe hospitalier universitaire Paris Sud, Hôpital Bicêtre, 94275 Le Kremlin-Bicêtre cedex, France
| | - C Goujard
- Service de médecine interne et immunologie clinique, Assistance publique-Hôpitaux Paris Saclay, Hôpital Bicêtre, 94275 Le Kremlin-Bicêtre cedex, France; Université Paris Saclay, Faculté de médecine, Le Kremlin Bicêtre, France
| | - C Adam
- Service d'anatomie pathologique et neuropathologie, Assistance publique-Hôpitaux de Paris, Groupe Hospitalier Universitaire Paris Sud, Hôpital Bicêtre, 94275 Le Kremlin-Bicêtre cedex, France
| | - O Lambotte
- Service de médecine interne et immunologie clinique, Assistance publique-Hôpitaux Paris Saclay, Hôpital Bicêtre, 94275 Le Kremlin-Bicêtre cedex, France; Université Paris Saclay, Faculté de médecine, Le Kremlin Bicêtre, France; Inserm UMR 1184, Immunologie des maladies virales et auto-immunes (IMVA), Université Paris Saclay, 94275 Le Kremlin-Bicêtre cedex, France; CEA, DSV/iMETI, Division of Immuno-Virology, IDMIT, Université Paris Saclay, 94275 Le Kremlin-Bicêtre cedex, France
| | - D Adams
- Université Paris Saclay, Faculté de médecine, Le Kremlin Bicêtre, France; Service de neurologie, Assistance publique-Hôpitaux de Paris, Groupe hospitalier universitaire Paris Sud, Hôpital Bicêtre, 94275 Le Kremlin-Bicêtre cedex, France; Centre de référence neuropathies amyloïdes familiales et autres neuropathies rares (NNERF), Groupe hospitalier universitaire Paris Sud, Hôpital Bicêtre, 94275 Le Kremlin-Bicêtre cedex, France
| | - N Noël
- Service de médecine interne et immunologie clinique, Assistance publique-Hôpitaux Paris Saclay, Hôpital Bicêtre, 94275 Le Kremlin-Bicêtre cedex, France; Université Paris Saclay, Faculté de médecine, Le Kremlin Bicêtre, France; Inserm UMR 1184, Immunologie des maladies virales et auto-immunes (IMVA), Université Paris Saclay, 94275 Le Kremlin-Bicêtre cedex, France; CEA, DSV/iMETI, Division of Immuno-Virology, IDMIT, Université Paris Saclay, 94275 Le Kremlin-Bicêtre cedex, France.
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21
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Lefland A, Proia AD, Shah S. Clinical Reasoning: A 64-Year-Old Man With Multiple Cranial Neuropathies. Neurology 2021; 97:e215-e221. [PMID: 33893198 DOI: 10.1212/wnl.0000000000012081] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/15/2022] Open
Affiliation(s)
- Ariel Lefland
- From the Departments of Neurology (A.L., S.S.) and Pathology and Ophthalmology (A.D.P.), Duke University Medical Center, Durham; and Department of Pathology (A.D.P.), Campbell University School of Osteopathic Medicine, Lillington, NC.
| | - Alan D Proia
- From the Departments of Neurology (A.L., S.S.) and Pathology and Ophthalmology (A.D.P.), Duke University Medical Center, Durham; and Department of Pathology (A.D.P.), Campbell University School of Osteopathic Medicine, Lillington, NC
| | - Suma Shah
- From the Departments of Neurology (A.L., S.S.) and Pathology and Ophthalmology (A.D.P.), Duke University Medical Center, Durham; and Department of Pathology (A.D.P.), Campbell University School of Osteopathic Medicine, Lillington, NC
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22
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Murphy D, Lester D, Clay Smither F, Balakhanlou E. Peripheral neuropathic pain. NeuroRehabilitation 2021; 47:265-283. [PMID: 32986619 DOI: 10.3233/nre-208002] [Citation(s) in RCA: 18] [Impact Index Per Article: 4.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 01/05/2023]
Abstract
Neuropathic pain (NP) can have either central nervous system causes or ones from the peripheral nervous system. This article will focus on the epidemiology, classifications, pathology, non-invasive treatments and invasive treatments as a general review of NP involving the peripheral nervous system. NP has characteristic symptomatology such as burning and electrical sensations. It occurs in up to 10% of the general population. Its frequency can be attributed to its occurrence in neck and back pain, diabetes and patients receiving chemotherapy. There are a wide range of pharmacologic options to control this type of pain and when such measures fail, numerous interventional methods can be employed such as nerve blocks and implanted stimulators. NP has a cost to the patient and society in terms of emotional consequences, quality of life, lost wages and the cost of assistance from the medical system and thus deserves serious consideration for prevention, treatment and control.
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Affiliation(s)
- Douglas Murphy
- Regional Amputation Center, Central Virginia Veterans Health Center, Richmond, VA, USA.,Physical Medicine & Rehabilitation, VCU Medical Center, Richmond, VA, USA
| | - Denise Lester
- Physical Medicine & Rehabilitation, VCU Medical Center, Richmond, VA, USA.,Peripheral Nerve Stimulation and Interventional Pain Research, Central Virginia Veterans Health Center, Richmond, VA, USA.,Anesthesiology, VCU Medical Center, Richmond, VA, USA
| | - F Clay Smither
- Polytrauma and Amputation, MSK/Ultrasound Fellow, Department of Physical Medicine and Rehabilitation, Central Virginia Veterans Health Center, Richmond, VA, USA
| | - Ellie Balakhanlou
- Physical Medicine & Rehabilitation, VCU Medical Center, Richmond, VA, USA
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Al-Attas AA, Aldayel AY, Al Najjar SA, Alkhonezan SM. Guillain-Barré Syndrome Heralding the Diagnosis of Hodgkin Lymphoma: A Case Report. Case Rep Neurol 2020; 12:365-372. [PMID: 33250750 PMCID: PMC7670385 DOI: 10.1159/000509681] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/13/2020] [Accepted: 06/21/2020] [Indexed: 01/08/2023] Open
Abstract
Lymphoma is a prevalent type of lymphoid tissue malignancy that is seldom associated with Guillain-Barré syndrome (GBS). In the majority of instances, both Hodgkin’s and non-Hodgkin’s lymphoma are not proceeded by GBS. Here, we report on a case of a young patient with a manifestation and investigation suggestive of GBS, signaling an unconfirmed diagnosis of Hodgkin’s lymphoma. A cerebrospinal fluid test revealed an albuminocytological dissociation with a noteworthy rise in protein (2.32 g/L). The patient was initiated on intravenous immunoglobulin (IVIG) treatment and then showed dramatic improvement after the third dose of IVIG. His constitutional presentation alongside high inflammatory labs prompted further investigation. An enhanced pan-computed tomography scan showed multiple enlarged mediastinal and hilar lymph nodes that were confirmed as Hodgkin’s lymphoma after biopsy. Brentuximab was initiated immediately after IVIG therapy. This case highlights consideration of Hodgkin’s lymphoma as a differential diagnosis under the auspices of GBS.
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Affiliation(s)
- Alawi Aqel Al-Attas
- Department of Neurology, Prince Sultan Military Medical City, Riyadh, Saudi Arabia
| | - Abdulrahman Yousef Aldayel
- Department of Neurology, Prince Sultan Military Medical City, Riyadh, Saudi Arabia
- *Abdulrahman Yousef Aldayel, Department of Neurology, Prince Sultan Military Medical City (PSMMC), Makkah Al Mukarramah Road, As Sulimaniyah, PO Box 7897, Riyadh (Saudi Arabia),
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24
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Vallat JM, Duchesne M, Magy L. Biopsia del nervo periferico. Neurologia 2020. [DOI: 10.1016/s1634-7072(20)44225-4] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 10/23/2022] Open
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25
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Stino AM, Naddaf E, Dyck PJ, Dyck PJB. Chronic inflammatory demyelinating polyradiculoneuropathy-Diagnostic pitfalls and treatment approach. Muscle Nerve 2020; 63:157-169. [PMID: 32914902 DOI: 10.1002/mus.27046] [Citation(s) in RCA: 42] [Impact Index Per Article: 8.4] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 05/06/2020] [Revised: 08/07/2020] [Accepted: 08/11/2020] [Indexed: 12/19/2022]
Abstract
Chronic inflammatory demyelinating polyradiculoneuropathy (CIDP) is characterized by progressive weakness and sensory loss, often affecting patients' ability to walk and perform activities of daily living independently. With the lack of a diagnostic biomarker, the diagnosis relies on clinical suspicion, clinical findings, and the demonstration of demyelinating changes on electrodiagnostic (EDx) testing and nerve pathology. As a result, patients can often be misdiagnosed with CIDP and unnecessarily treated with immunotherapy. Interpreting the EDx testing and cerebrospinal fluid findings in light of the clinical phenotype, recognizing atypical forms of CIDP, and screening for CIDP mimickers are the mainstays of the approach to patients suspected of having CIDP, and are detailed in this review. We also review the currently available treatment options, including intravenous immunoglobulin (IVIg), corticosteroids (CCS), and plasma exchange (PE), and discuss how to approach treatment-refractory cases. Finally, we emphasize the need to adopt objective outcome measures to monitor treatment response.
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Affiliation(s)
- Amro M Stino
- Division of Neuromuscular Medicine, Department of Neurology, University of Michigan School of Medicine, Ann Arbor, Michigan, USA
| | - Elie Naddaf
- Department of Neurology, Mayo Clinic, Rochester, Minnesota, USA
| | - Peter J Dyck
- Department of Neurology, Mayo Clinic, Rochester, Minnesota, USA
| | - P James B Dyck
- Department of Neurology, Mayo Clinic, Rochester, Minnesota, USA
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26
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Li T, Timmins HC, Lazarus HM, Park SB. Peripheral neuropathy in hematologic malignancies – Past, present and future. Blood Rev 2020; 43:100653. [DOI: 10.1016/j.blre.2020.100653] [Citation(s) in RCA: 8] [Impact Index Per Article: 1.6] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/27/2019] [Revised: 11/13/2019] [Accepted: 01/14/2020] [Indexed: 02/06/2023]
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Kawahigashi T, Teshima S, Tanaka E. Intravascular lymphoma with hypopituitarism: A case report. World J Clin Oncol 2020; 11:673-678. [PMID: 32879852 PMCID: PMC7443836 DOI: 10.5306/wjco.v11.i8.673] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 01/20/2020] [Revised: 05/07/2020] [Accepted: 06/27/2020] [Indexed: 02/06/2023] Open
Abstract
BACKGROUND Intravascular lymphoma (IVL) is a rare subtype of lymphoma involving the growth of lymphoma cells within the vessel lumina without lymphadenopathy. Because of various modes of presentation and its rarity, IVL is often diagnosed postmortem. Herein, we report a case of intravascular B-cell lymphoma with hypopituitarism, an extremely rare complication, that was successfully treated with chemotherapy.
CASE SUMMARY An 80-year-old Japanese woman presented with a 7-mo history of a tingling sensation in the lower limbs. She also presented with various other symptoms such as pancytopenia, high fever daily, and unconsciousness with hypoglycemia. Although the doctor who previously treated her diagnosed hypoglycemia as being due to hypopituitarism, the cause of the other symptoms remained uncertain despite a 7-mo evaluation period. We performed bone marrow aspiration to evaluate pancytopenia and found that she had hemophagocytic lymphohistiocytosis (HLH). On the basis of a random skin biopsy for assessing the cause of HLH, she was diagnosed with intravascular B-cell lymphoma. HLH and hypopituitarism were considered secondary to IVL. All her clinical findings matched the presentations of IVL. She was immediately treated with chemotherapy and achieved complete response. She was relapse free two years after treatment.
CONCLUSION IVL should be included in the differential diagnosis of hypopituitarism, which although life-threatening, is treatable through prompt diagnosis and appropriate chemotherapy.
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Affiliation(s)
- Teiko Kawahigashi
- Department of General Internal Medicine, Shonan Kamakura General Hospital, Kanagawa 247-8533, Japan
| | - Shinichi Teshima
- Department of Histopathology, Shonan Kamakura General Hospital, Kanagawa 247-8533, Japan
| | - Eri Tanaka
- Department of Hematology, Shonan Kamakura General Hospital, Kanagawa 247-8533, Japan
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Rozłucka L, Semik-Grabarczyk E, Pietrukaniec M, Żak-Gołąb A, Grabarczyk M, Grosicki S, Holecki M. Demyelinating polyneuropathy and lymphoplasmacytic lymphoma coexisting in 36-year-old man: A case report. World J Clin Cases 2020; 8:2566-2573. [PMID: 32607333 PMCID: PMC7322434 DOI: 10.12998/wjcc.v8.i12.2566] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 02/24/2020] [Revised: 05/20/2020] [Accepted: 05/23/2020] [Indexed: 02/05/2023] Open
Abstract
BACKGROUND Lymphoplasmacytic lymphoma is a rare non-Hodgkin's lymphoma, occurring mostly in the elderly. It develops slowly and leads to malignant proliferation of lymphoid line cells in the bone marrow, lymph nodes and spleen. It may also affect nerve roots and meninges; some patients develop sensorimotor polyneuropathy which may precede general symptoms of lymphoma. CASE SUMMARY We present a case of a 36-year-old man diagnosed in 2012 with chronic inflammatory demyelinating polyneuropathy (CIDP), then he was hospitalized in 2019 due to progressive symptoms of heart failure and significant weight loss over the previous four months. Based on clinical and laboratory findings a diagnosis of lymphoplasmacytic lymphoma was suspected and confirmed by bone marrow flow cytometry. There was no improvement in the results of laboratory tests and the patient's condition after immediate implementation of chemotherapy. Patient died on the fifth day of treatment. CONCLUSION While CIDP and malignant disease co-occurrence is rare, it should be suspected and investigated in patients with atypical neuropathy symptoms.
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Affiliation(s)
- Lesia Rozłucka
- Department of Internal Medicine, Allergology and Clinical Immunology, Faculty of Medical Sciences in Zabrze, Medical University of Silesia, Katowice 40-752, Poland
| | - Elżbieta Semik-Grabarczyk
- Department of Internal Medicine, Autoimmune and Metabolic Diseases, Faculty of Medical Sciences in Katowice, Medical University of Silesia, Katowice 40-752, Poland
| | - Marta Pietrukaniec
- Department of Internal Medicine, Autoimmune and Metabolic Diseases, Faculty of Medical Sciences in Katowice, Medical University of Silesia, Katowice 40-752, Poland
| | - Agnieszka Żak-Gołąb
- Department of Internal Medicine, Autoimmune and Metabolic Diseases, Faculty of Medical Sciences in Katowice, Medical University of Silesia, Katowice 40-752, Poland
| | - Małgorzata Grabarczyk
- Student Scientific Society at the Department of Internal Medicine, Autoimmune and Metabolic Diseases, Faculty of Medical Sciences in Katowice, Medical University of Silesia, Katowice 40-752, Poland
| | - Sebastian Grosicki
- Department of Hematology and Cancer Prevention, Faculty of Health Sciences in Bytom, Medical University of Silesia, Bytom, 41-902, Poland
| | - Michał Holecki
- Department of Internal Medicine, Autoimmune and Metabolic Diseases, Faculty of Medical Sciences in Katowice, Medical University of Silesia, Katowice 40-752, Poland
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29
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Deb B, Pandey MR, Torka P, Sundaram S. Diffuse large B-Cell lymphoma associated with paraneoplastic Guillain-Barré syndrome: A diagnostic and therapeutic challenge. Hematol Oncol Stem Cell Ther 2020:S1658-3876(20)30106-0. [PMID: 32470333 DOI: 10.1016/j.hemonc.2020.05.008] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/11/2020] [Accepted: 05/04/2020] [Indexed: 11/19/2022] Open
Abstract
Paraneoplastic neurological syndromes are a rare manifestation of non-Hodgkin lymphoma and can make treatment of these patients more challenging. We report the case of a 67-year-old man with high grade diffuse large B-cell lymphoma who presented with severe paraneoplastic Guillain-Barré syndrome. He was treated with intravenous immunoglobulin therapy and definitive chemoimmunotherapy, and achieved a full neurological recovery. In this report, we discuss various mechanisms of neurological dysfunction seen in lymphomas. Prompt oncologic treatment and immunotherapy for Guillain-Barré syndrome if instituted concurrently and early in the course of the disease can be associated with the best outcomes.
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Affiliation(s)
- Brototo Deb
- Roswell Park Comprehensive Cancer Centre, Buffalo, NY, USA
| | - Manu R Pandey
- Roswell Park Comprehensive Cancer Centre, Buffalo, NY, USA
| | - Pallawi Torka
- Roswell Park Comprehensive Cancer Centre, Buffalo, NY, USA
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30
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Bouattour N, Kamoun F, Charfi S, Nsir SB, Bouchaala W, Boudawara T, Triki C. Neurological Nuance: Sporadic Burkitt's Lymphoma Presenting with Guillain–Barre Syndrome. JOURNAL OF PEDIATRIC NEUROLOGY 2020. [DOI: 10.1055/s-0040-1710515] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 10/24/2022]
Abstract
AbstractBurkitt's lymphoma (BL) is a high-grade mature B cell neoplasm. Primary lymphoma of the central nervous system (CNS) is very rare. In contrast, BL can be localized in any part of the body and may spread to the CNS. Peripheral nervous system impairment caused by BL has been thoroughly described. However, Guillain–Barre syndrome (GBS) does not commonly appear as an initial manifestation of BL. We presented the case of a young child with sporadic BL with neuromeningeal involvement and spinal cord compression whose first clinical manifestation was the appearance of GBS. Lymphoma should be taken into account, especially in children presenting with the clinical features of acute polyradiculoneuropathy.
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Affiliation(s)
- Nadia Bouattour
- Department of Child Neurology, Hedi Chaker Hospital, Sfax, Tunisia
| | - Fatma Kamoun
- Department of Child Neurology, Hedi Chaker Hospital, Sfax, Tunisia
- Research Laboratory “Neuropediatrics,” University of Sfax, Sfax, Tunisia
| | - Slim Charfi
- Department of Pathology, Habib Bourguiba Hospital, Sfax, Tunisia
| | - Siham Ben Nsir
- Department of Child Neurology, Hedi Chaker Hospital, Sfax, Tunisia
- Research Laboratory “Neuropediatrics,” University of Sfax, Sfax, Tunisia
| | - Wafa Bouchaala
- Department of Child Neurology, Hedi Chaker Hospital, Sfax, Tunisia
- Research Laboratory “Neuropediatrics,” University of Sfax, Sfax, Tunisia
| | - Tahya Boudawara
- Department of Pathology, Habib Bourguiba Hospital, Sfax, Tunisia
| | - Chahnez Triki
- Department of Child Neurology, Hedi Chaker Hospital, Sfax, Tunisia
- Research Laboratory “Neuropediatrics,” University of Sfax, Sfax, Tunisia
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31
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Fukami Y, Koike H, Iijima M, Hagita J, Niwa H, Nishi R, Kawagashira Y, Katsuno M. Demyelinating Neuropathy Due to Intravascular Large B-cell Lymphoma. Intern Med 2020; 59:435-438. [PMID: 31588080 PMCID: PMC7028418 DOI: 10.2169/internalmedicine.3228-19] [Citation(s) in RCA: 7] [Impact Index Per Article: 1.4] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/25/2022] Open
Abstract
We herein report the case of a 67-year-old man who presented with the acute onset of limb weakness. Brain magnetic resonance imaging revealed multiple abnormal-signal-intensity lesions. Steroids were administered, and the patient initially responded. Nerve conduction testing findings were consistent with demyelinating polyneuropathy. A sural nerve biopsy specimen revealed fascicles with extensive onion-bulb formation. Although skin and sural nerve biopsies showed no atypical cellular infiltration, the histopathological diagnosis of intravascular large B-cell lymphoma was obtained by a brain biopsy. The neuropathy in this patient may be attributed to a demyelinating process independent of ischemic damage by lymphoma.
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Affiliation(s)
- Yuki Fukami
- Department of Neurology, Nagoya University Graduate School of Medicine, Japan
| | - Haruki Koike
- Department of Neurology, Nagoya University Graduate School of Medicine, Japan
| | - Masahiro Iijima
- Department of Neurology, Nagoya University Graduate School of Medicine, Japan
- Center for Advanced Medicine and Clinical Research, Department of Advanced Medicine, Nagoya University Hospital, Japan
| | - Junichirou Hagita
- Department of Internal Medicine, Kariya Toyota General Hospital, Japan
| | - Hisayoshi Niwa
- Department of Neurology, Kariya Toyota General Hospital, Japan
| | - Ryoji Nishi
- Department of Neurology, Nagoya University Graduate School of Medicine, Japan
| | - Yuichi Kawagashira
- Department of Neurology, Nagoya University Graduate School of Medicine, Japan
| | - Masahisa Katsuno
- Department of Neurology, Nagoya University Graduate School of Medicine, Japan
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32
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Gupta A, Kumar PS, Puri V, Saran RK, Narang P, Chowdhury D. 51-year old man with tingling, burning and progressive limb weakness. Pract Neurol 2019; 18:382-388. [PMID: 31091191 DOI: 10.1136/practneurol-2017-001877] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Accepted: 04/08/2018] [Indexed: 11/04/2022]
Abstract
Peripheral neuropathy is a common reason for referral to neurology. Chronic acquired demyelinating neuropathies are an important and varied group with overlapping presentations, and may have an immune-mediated cause. Their correct diagnosis is important as they respond to different treatments; timely intervention can prevent irreversible axonal degeneration. We present a case that highlights the approach to an adult presenting with a chronic demyelinating neuropathy.
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Affiliation(s)
- Anu Gupta
- Department of Neurology, Govind Ballabh Pant Institute of Post Graduate Medical Education and Research (GIPMER), New Delhi, India
| | - Pappula Santhosh Kumar
- Department of Neurology, Govind Ballabh Pant Institute of Post Graduate Medical Education and Research (GIPMER), New Delhi, India
| | - Vinod Puri
- Department of Neurology, Max Super Speciality Hospital, New Delhi, India
| | - Ravindra Kumar Saran
- Department of Pathology, Govind Ballabh Pant Institute of Post Graduate Medical Education and Research (GIPMER), New Delhi, India
| | - Poonam Narang
- Department of Radiology, Govind Ballabh Pant Institute of Post Graduate Medical Education And Research (GIPMER), New Delhi, India
| | - Debashish Chowdhury
- Department of Neurology, Govind Ballabh Pant Institute of Post Graduate Medical Education and Research (GIPMER), New Delhi, India
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Hsueh CS, Tsai CY, Lee JCS, Kao CL, Wang FI, Jeng CR, Chang HW, Pang VF, Liu CH. CD56 + B-cell Neurolymphomatosis in a Cat. J Comp Pathol 2019; 169:25-29. [PMID: 31159947 PMCID: PMC7094645 DOI: 10.1016/j.jcpa.2019.03.004] [Citation(s) in RCA: 6] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/18/2019] [Revised: 03/10/2019] [Accepted: 03/15/2019] [Indexed: 11/26/2022]
Abstract
A 16-year-old male Russian blue cat was presented with acute onset of paraparesis of the forelimbs that progressed to tetraparesis. Neurological examination revealed non-ambulatory tetraparesis with decreased postural reactions in all four limbs. Magnetic resonance imaging revealed multifocal nerve root swelling on the right at C6/C7 and C7/T1, while ultrasonography demonstrated swelling of the right brachial plexus. To understand the cause of the nerve swelling, the right musculocutaneous nerve arising from the brachial plexus and the pectoralis muscle were biopsied. Histologically, there was evidence of neurolymphomatosis (neurotropic lymphoma) with Wallerian degeneration and denervation atrophy of myofibres. The neoplastic lymphoid cells expressed CD79a, CD20 and CD56. Based on these findings, a diagnosis of B-cell neurolymphomatosis was made. Expression of CD56, synonymous with neural cell adhesion molecule, is rare in B-cell lymphomas and has not been reported in feline B-cell lymphomas or feline neurolymphomatosis. CD56 expression was suspected to have played an important role in neurotropism of the neoplastic cells in this case.
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Affiliation(s)
- C-S Hsueh
- Graduate Institute of Molecular and Comparative Pathobiology, School of Veterinary Medicine, Taipei, Taiwan
| | - C-Y Tsai
- National Taiwan University Veterinary Hospital, College of Bioresources and Agriculture, Taipei, Taiwan
| | - J C-S Lee
- National Taiwan University Veterinary Hospital, College of Bioresources and Agriculture, Taipei, Taiwan
| | - C-L Kao
- National Taiwan University Veterinary Hospital, College of Bioresources and Agriculture, Taipei, Taiwan
| | - F-I Wang
- Graduate Institute of Molecular and Comparative Pathobiology, School of Veterinary Medicine, Taipei, Taiwan; School of Veterinary Medicine, National Taiwan University, Taipei, Taiwan
| | - C-R Jeng
- Graduate Institute of Molecular and Comparative Pathobiology, School of Veterinary Medicine, Taipei, Taiwan; School of Veterinary Medicine, National Taiwan University, Taipei, Taiwan
| | - H-W Chang
- Graduate Institute of Molecular and Comparative Pathobiology, School of Veterinary Medicine, Taipei, Taiwan; School of Veterinary Medicine, National Taiwan University, Taipei, Taiwan
| | - V F Pang
- Graduate Institute of Molecular and Comparative Pathobiology, School of Veterinary Medicine, Taipei, Taiwan; School of Veterinary Medicine, National Taiwan University, Taipei, Taiwan
| | - C-H Liu
- Graduate Institute of Molecular and Comparative Pathobiology, School of Veterinary Medicine, Taipei, Taiwan; School of Veterinary Medicine, National Taiwan University, Taipei, Taiwan.
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Hasannejad F, Ansar MM, Rostampour M, Mahdavi Fikijivar E, Khakpour Taleghani B. Improvement of pyridoxine-induced peripheral neuropathy by Cichorium intybus hydroalcoholic extract through GABAergic system. J Physiol Sci 2019; 69:465-476. [PMID: 30712095 PMCID: PMC10718042 DOI: 10.1007/s12576-019-00659-8] [Citation(s) in RCA: 8] [Impact Index Per Article: 1.3] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/21/2018] [Accepted: 01/05/2019] [Indexed: 02/07/2023]
Abstract
Pyridoxine (vitamin B6) toxicity is a well-known model for peripheral neuropathy. GABA and glutamate are two neurotransmitters in neural pathways involved in the peripheral neuropathy. Cichorium intybus (Chicory) contains glycosides and triterpenoids, which inhibit glutamatergic transmission and enhance GABAergic transmission. The present study was aimed at studying the effect of chicory extract (CE) on the pyridoxine-induced peripheral neuropathy with a particular focus on glutamatergic and GABAergic systems. In this experimental study, a high dose of pyridoxine (800 mg/kg, i.p.) was injected for 14 days to induce neuropathy in male rats. To evaluate the behavioral symptoms, three tests including rotarod, hot plate, and foot fault were used. After the induction of neuropathy, CE (50 mg/kg i.p.) was injected intraperitoneally for 10 consecutive days. Morphologically, the sciatic nerve and the DRG neurons were evaluated in the control, neuropathy, and chicory groups by H&E staining. For evaluating the mechanism, picrotoxin (1 mg/kg) and MK-801 (0.1 mg/kg) were also individually injected 15 min before the extract administration. The concentration of TNF-α in rat sciatic nerve and DRG neurons were also measured by enzyme-linked-immunoassay (ELISA). Morphological and physiological changes occurred in the DRG and sciatic nerve following pyridoxine intoxication. The CE exerted an anti-neuropathic effect on the sciatic nerve and DRG neurons and also decreased reaction time in hot plate test (p < 0.05), increased balance time in rotarod test (p < 0.001), and improved foot fault performance (p < 0.01). Moreover, CE administration reduced TNF-α level in DRG (p < 0.001) and sciatica nerve (p < 0.001). Picrotoxin, unlike MK-801, showed a significant difference in all three behavioral tests and reduced TNF-α content in comparison with group received extraction alone (with p < 0.001 for all three tests). Our results showed beneficial effects of CE on pyridoxine-induced peripheral neuropathy. Modulating of the GABAergic system mediated by TNF-α may be involved in the anti-neurotoxic effect of CE.
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Affiliation(s)
| | - Malek Moein Ansar
- Neuroscience Research Center, School of Medicine, Guilan University of Medical Sciences, Rasht, Iran
- Department of Biochemistry, School of Medicine, Guilan University of Medical Sciences, Rasht, Iran
| | - Mohammad Rostampour
- Neuroscience Research Center, School of Medicine, Guilan University of Medical Sciences, Rasht, Iran
- Department of Physiology, School of Medicine, Guilan University of Medical Sciences, Rasht, Iran
| | | | - Behrooz Khakpour Taleghani
- Neuroscience Research Center, School of Medicine, Guilan University of Medical Sciences, Rasht, Iran.
- Department of Physiology, School of Medicine, Guilan University of Medical Sciences, Rasht, Iran.
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Jiang Z, Ju W, Luo S, Yang Y. Diffuse large B-cell lymphoma involving peripheral nervous system with IgM antibodies against GM1 and GD1b: A case report. Medicine (Baltimore) 2019; 98:e15049. [PMID: 30985655 PMCID: PMC6485863 DOI: 10.1097/md.0000000000015049] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 11/25/2022] Open
Abstract
RATIONALE The occurrence of peripheral neuropathy associated with non-Hodgkin's lymphoma (NHL) is uncommon. And autoimmunity may play an important role. We report a case of the patient with NHL, has sensorimotor demyelinating polyneuropathy. PATIENT CONCERNS The patient presented with a 1-month history of progressive numbness at the distal extremities and motor weakness of the lower limbs. Meanwhile, patient also endorsed a painful lump on her right cheek. And then the enlarged cervical and supra clavicular lymph nodes were observed on admission. Biopsy of the lymph nodes showed NHL. Serum IgM antibodies against GM1 and GD1b were also positive. DIAGNOSIS Biopsy of the lymph nodes showed NHL. Serum IgM antibodies against GM1 and GD1b were also positive. Thus, the patience was diagnosed with lymphoma and sensorimotor polyneuropathy. INTERVENTIONS Patient refused the further treatment. OUTCOMES After 11-month follow-up, the weakness of bilateral lower limbs worsens. LESSONS We have presented a case of NHL involving peripheral polyneuropathy with IgM antibodies against GM1 and GD1b. Patients may initially present with peripheral nerve complications or develop them during the course of lymphoma, even when in remission. This could complicate the diagnosis of peripheral polyneuropathy secondary to NHL.
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36
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Montejo C, Navarro‐Otano J, Mayà‐Casalprim G, Campolo M, Casanova‐Mollá J. Acute lumbar polyradiculoneuropathy as early sign of methotrexate intrathecal neurotoxicity: Case report and literature review. Clin Case Rep 2019; 7:638-643. [PMID: 30997053 PMCID: PMC6452470 DOI: 10.1002/ccr3.2026] [Citation(s) in RCA: 7] [Impact Index Per Article: 1.2] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/21/2018] [Revised: 12/30/2018] [Accepted: 01/16/2019] [Indexed: 12/12/2022] Open
Abstract
Acute paraplegia after treatment with intrathecal methotrexate requires a complete spinal cord neuroimaging as well as electrodiagnostic examination. The absence of lumbosacral F waves motor responses without demyelinating findings may indicate early direct root toxicity. Early electromyography (EMG) screening could be a valuable tool for detecting peripheral neurotoxicity.
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Affiliation(s)
- Carmen Montejo
- Department of Neurology, EMG and Neuropathic Pain UnitHospital ClínicBarcelonaSpain
| | - Judith Navarro‐Otano
- Department of Neurology, EMG and Neuropathic Pain UnitHospital ClínicBarcelonaSpain
| | | | - Michela Campolo
- Department of Neurology, EMG and Neuropathic Pain UnitHospital ClínicBarcelonaSpain
| | - Jordi Casanova‐Mollá
- Department of Neurology, EMG and Neuropathic Pain UnitHospital ClínicBarcelonaSpain
- Institut d´Investigacions Biomèdiques August Pi i Sunyer (IDIBAPS)University of BarcelonaBarcelonaSpain
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Xu M, Pinto M, Sun C, Engelstad JK, James Dyck P, Dyck PJ, Klein CJ. Expanded teased nerve fibre pathological conditions in disease association. J Neurol Neurosurg Psychiatry 2019; 90:138-140. [PMID: 30385486 DOI: 10.1136/jnnp-2018-319077] [Citation(s) in RCA: 11] [Impact Index Per Article: 1.8] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 06/21/2018] [Revised: 08/29/2018] [Accepted: 08/31/2018] [Indexed: 11/04/2022]
Abstract
OBJECTIVE To describe an expanded teased nerve fibre classification in disease association. METHODS We reviewed four newly proposed teased nerve fibre types (Types J-M): Type J, rope-like fibres; K, fibril-like clumps of osmium positivity; L, cellular debris along and within nerve fibres; M, circular axonal inclusions surrounded by thin myelin. Different clinical pathological entities were studied for these fibre types including chronic inflammatory demyelinating polyradiculoneuropathy (CIDP: N=20); amyloid polyneuropathy (N=20); intraneural B-cell lymphoma (N=20) or adult-onset polyglucosan body disease (APBD: N=6) in comparison with 112 disease controls. Student's t-test was used to test significance of association between the identified fibre types and the specific clinical diagnosis. RESULTS Each fibre type significantly associated (p<0.001) with particular disease categories: Type J, 60% of CIDP cases; Type K, 75% of amyloid cases; Type L, 75% of intraneural lymphoma cases; Type M, 100% of APBD cases. Rarely were these fibres found in the other disease control cases ≤3% of cases. In three cases, the teased fibre findings were so striking additional paraffin nerve preparations were made to make the pathological diagnosis when initial paraffin sections were non-diagnostic. CONCLUSIONS Teased nerve fibre Types J-M associate with commonly seen pathological diagnosis and are helpful in the consideration of specific neuropathy diagnoses.
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Affiliation(s)
- Min Xu
- Department of Neurology, Mayo Clinic, Rochester, Minnesota, USA.,Department of Neurology, Xuan Wu Hospital, Capital Medical University, Beijing, China
| | - Marcus Pinto
- Department of Neurology, Mayo Clinic, Rochester, Minnesota, USA
| | - Chenjing Sun
- Department of Neurology, Mayo Clinic, Rochester, Minnesota, USA.,Navy Teaching Hospital, Beijing, China
| | | | - P James Dyck
- Department of Neurology, Mayo Clinic, Rochester, Minnesota, USA
| | - Peter J Dyck
- Department of Neurology, Mayo Clinic, Rochester, Minnesota, USA
| | - Christopher J Klein
- Department of Neurology, Mayo Clinic, Rochester, Minnesota, USA .,Laboratory Medicine and Pathology, Mayo Clinic, Rochester, Minnesota, USA
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38
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Briani C, Visentin A, Campagnolo M, Salvalaggio A, Ferrari S, Cavallaro T, Manara R, Gasparotti R, Piazza F. Peripheral nervous system involvement in lymphomas. J Peripher Nerv Syst 2019; 24:5-18. [PMID: 30556258 DOI: 10.1111/jns.12295] [Citation(s) in RCA: 40] [Impact Index Per Article: 6.7] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/08/2018] [Revised: 12/01/2018] [Accepted: 12/08/2018] [Indexed: 12/11/2022]
Abstract
The peripheral nervous system may be involved at any stage in the course of lymphoproliferative diseases. The different underlying mechanisms include neurotoxicity secondary to chemotherapy, direct nerve infiltration (neurolymphomatosis), infections, immune-mediated, paraneoplastic or metabolic processes and nutritional deficiencies. Accordingly, the clinical features are heterogeneous and depend on the localization of the damage (ganglia, roots, plexi, and peripheral nerves) and on the involved structures (myelin, axon, and cell body). Some clinical findings, such a focal or diffuse involvement, symmetric or asymmetric pattern, presence of pain may point to the correct diagnosis. Besides a thorough medical history and neurological examination, neurophysiological studies, cerebrospinal fluid analysis, nerve biopsy (in selected patients with suspected lymphomatous infiltration) and neuroimaging techniques (magnetic resonance neurography and nerve ultrasound) may be crucial for a proper diagnostic workup.
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Affiliation(s)
- Chiara Briani
- Department of Neuroscience, University of Padova, Padova, Italy
| | - Andrea Visentin
- Hematology and Clinical Immunology Unit, Department of Medicine, University of Padova, Padova, Italy
| | | | | | - Sergio Ferrari
- Department of Neurology, Azienda Ospedaliera Universitaria Integrata, University Hospital G.B. Rossi, Verona, Italy
| | - Tiziana Cavallaro
- Department of Neurology, Azienda Ospedaliera Universitaria Integrata, University Hospital G.B. Rossi, Verona, Italy
| | - Renzo Manara
- Neuroradiology, Department of Medicine and Surgery, University of Salerno, Fisciano, Italy
| | - Roberto Gasparotti
- Department of Medical and Surgical Specialties, Radiological Sciences, and Public Health, University of Brescia, Brescia, Italy
| | - Francesco Piazza
- Hematology and Clinical Immunology Unit, Department of Medicine, University of Padova, Padova, Italy
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39
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Mustafa R, Klein CJ, Martinez-Thompson J, Johnson AC, Engelstad JK, Spinner RJ, Crum BA. Recurrent Brachial Neuritis Attacks in Presentation of B-Cell Lymphoma. Mayo Clin Proc Innov Qual Outcomes 2018; 2:382-386. [PMID: 30560241 PMCID: PMC6260480 DOI: 10.1016/j.mayocpiqo.2018.10.002] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.7] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/17/2018] [Revised: 09/28/2018] [Accepted: 10/05/2018] [Indexed: 12/03/2022] Open
Abstract
We describe a 51-year-old woman who over 5 years had 9 painful monophasic attacks affecting the brachial plexus before a fascicular plexus biopsy diagnosed large B-cell lymphoma. The initial attacks were responsive to steroids with clinical resolution. At last attack, magnetic resonance imaging showed multifocal T2 hyperintensities and nodular gadolinium enhancement in the right brachial plexus not seen previously. Also seen were similar changes in the thoracic spinal cord, basal ganglia, cerebellum, and brainstem. Positron emission tomography revealed marked hypermetabolic activity of the plexus facilitating targeted fascicular brachial plexus biopsy, making the pathological diagnosis. Neurolymphomatosis affecting the peripheral nervous system typically presents with insidious painful progressive infiltration of nerves, roots, or plexi. Recurrent idiopathic brachial neuritis attacks (ie, Parsonage-Turner syndrome) in contrast most commonly are seen in persons with a family history and a discoverable genetic cause by SEPT9 mutations, which tested negative in this patient. This case illustrates how neurolymphomatosis, which represents a malignant transformation of B cells within peripheral nerves, can sometimes present with paraneoplastic immune-responsive neuritis mimicking Parsonage-Turner syndrome. Recurrence, an immune-refractory course or insidious progressive involvement of the nervous system, should raise suspicion of neurolymphomatosis.
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Affiliation(s)
- Rafid Mustafa
- Department of Neurology, Mayo Clinic College of Medicine & Science, Rochester, MN
| | - Christopher J Klein
- Department of Neurology, Mayo Clinic College of Medicine & Science, Rochester, MN
| | | | - Adam C Johnson
- Department of Radiology, Mayo Clinic College of Medicine & Science, Rochester, MN
| | - JaNean K Engelstad
- Department of Neurology, Mayo Clinic College of Medicine & Science, Rochester, MN
| | - Robert J Spinner
- Department of Neurologic Surgery, Orthopedics, and Anatomy, Mayo Clinic College of Medicine & Science, Rochester, MN
| | - Brian A Crum
- Department of Neurology, Mayo Clinic College of Medicine & Science, Rochester, MN
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40
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Barp A, Gilardin L, Afanasiev V, Delorme C, Viala K, Bernard S, Brice P, Psimaras D, Lenglet T. Subacute inflammatory demyelinating polyradiculoneuropathy complicating relapsing Hodgkin lymphoma: another immune-related adverse event of the anti-PD1 therapy? Leuk Lymphoma 2018; 60:547-549. [DOI: 10.1080/10428194.2018.1485912] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 01/04/2023]
Affiliation(s)
- Andrea Barp
- Département de Neurophysiologie, Hôpital de la Pitié-Salpêtrière, APHP, Paris, France
| | - Laurent Gilardin
- Service d’Onco-Hématologie, Hôpital Saint-Louis, APHP, Paris, France
- Institut National de la Santé et de la Recherche Médicale (INSERM), Unité Mixte de Recherche en Santé (UMR S) 1138, Centre de Recherche des Cordeliers, Equipe Immunopathology and Therapeutic Immunointervention, Paris, France
| | - Vadim Afanasiev
- Centre d’Evaluation et Traitement des Neuropathies, Hôpital de la Pitié-Salpêtrière, APHP, Paris, France
| | - Cécile Delorme
- Département de Neurophysiologie, Hôpital de la Pitié-Salpêtrière, APHP, Paris, France
- Centre d’Evaluation et Traitement des Neuropathies, Hôpital de la Pitié-Salpêtrière, APHP, Paris, France
| | - Karine Viala
- Département de Neurophysiologie, Hôpital de la Pitié-Salpêtrière, APHP, Paris, France
- Centre d’Evaluation et Traitement des Neuropathies, Hôpital de la Pitié-Salpêtrière, APHP, Paris, France
| | - Sophie Bernard
- Service d’Onco-Hématologie, Hôpital Saint-Louis, APHP, Paris, France
| | - Pauline Brice
- Service d’Onco-Hématologie, Hôpital Saint-Louis, APHP, Paris, France
| | - Dimitri Psimaras
- OncoNeuroTox group: Center for investigations of patients with neurological complications after cancer treatment, Hôpitaux universitaires Pitié-Salpêtrière-Charles Foix et Val-de-Grâce, Paris, France
| | - Timothée Lenglet
- Département de Neurophysiologie, Hôpital de la Pitié-Salpêtrière, APHP, Paris, France
- OncoNeuroTox group: Center for investigations of patients with neurological complications after cancer treatment, Hôpitaux universitaires Pitié-Salpêtrière-Charles Foix et Val-de-Grâce, Paris, France
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41
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Mahmoud RA, Abrams CK. Acute demyelinating neuropathy in a patient with neurolymphomatosis. BMJ Case Rep 2018; 2018:bcr-2017-222814. [PMID: 29507018 DOI: 10.1136/bcr-2017-222814] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/04/2022] Open
Abstract
Peripheral neurological complications of lymphomas are rare and much less frequent than central complications. Nonetheless, on occasion, systemic non-Hodgkin's lymphoma may directly infiltrate the peripheral nervous system at various levels. This report describes a man with non-Hodgkin's lymphoma and leptomeningeal disease who developed progressive areflexic quadraparesis. Initial electromyography (EMG) was consistent with a polyradiculopathy and a repeat EMG performed 1 month later for worsening symptoms showed evidence of demyelination. The patient expired due to systemic complications of his illness. Autopsy of the sural nerve showed moderately severe distal sensory axonal loss, direct infiltration of the brachial plexus by malignant lymphocytes and demyelination in brachial and lumbar plexus, most prominent in areas of neoplastic infiltration. Based on this patient's course and pathology, we suggest that widespread demyelination may accompany neurolymphomatosis and the clinical presentation may be indistinguishable from an acute demyelinating neuropathy.
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Affiliation(s)
- Rola A Mahmoud
- Department of Neurology, UMKC School of Medicine Saint Luke's Hospital, Kansas City, Missouri, USA
| | - Charles K Abrams
- Department of Neurology and Rehabilitation, University of Illinois at Chicago College of Medicine, Chicago, Illinois, USA
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42
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Park HJ, Shin HY, Kim SH, Jeong HN, Choi YC, Suh BC, Park KD, Kim SM. Partial Conduction Block as an Early Nerve Conduction Finding in Neurolymphomatosis. J Clin Neurol 2018; 14:73-80. [PMID: 29629543 PMCID: PMC5765259 DOI: 10.3988/jcn.2018.14.1.73] [Citation(s) in RCA: 9] [Impact Index Per Article: 1.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/14/2017] [Revised: 09/20/2017] [Accepted: 09/25/2017] [Indexed: 12/17/2022] Open
Abstract
BACKGROUND AND PURPOSE Neurolymphomatosis is a rare manifestation of hematological malignancy and is characterized by direct infiltration of the peripheral nervous system. The objective of this study was to identify the clinical and electrophysiological features of neurolymphomatosis. METHODS We retrospectively analyzed the medical records of 13 patients with neurolymphomatosis. Seven (54%) of the patients were men, and the median age at symptom onset was 60.0 years. RESULTS The most common type of underlying malignancy was diffuse large B-cell lymphoma (69%). Twelve patients had painful asymmetric neuropathies. The median survival time after diagnosis was 7 months, and 12 patients died during the study period. Thirty-eight motor nerve conduction studies (NCSs) were performed in the affected nerves. Ten and 28 motor nerves were classified into the conduction-block and simple-axon-degeneration groups, respectively. The median time interval between symptom onset and the NCS was significantly shorter in the conduction-block group than in the simple-axon-degeneration group (p=0.032). However, no significant differences in the motor nerve conduction velocities, terminal latencies, and distal compound muscle action potential amplitudes were identified between the conduction-block and simple-axon-degeneration groups. The conduction-block group showed excessive temporal dispersion in only five of the ten NCSs (50%). Follow-up NCSs revealed that partial conduction blocks had changed into axonal degeneration patterns. CONCLUSIONS This is the first study to analyze the electrophysiological features of patients with neurolymphomatosis. Our findings showed that a partial conduction block is not rare and is an early nerve conduction abnormality in neurolymphomatosis.
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Affiliation(s)
- Hyung Jun Park
- Department of Neurology, Mokdong Hospital, College of Medicine, Ewha Womans University, Seoul, Korea.,Department of Neurology, Yonsei University College of Medicine, Seoul, Korea
| | - Ha Young Shin
- Department of Neurology, Yonsei University College of Medicine, Seoul, Korea
| | - Se Hoon Kim
- Department of Pathology, Yonsei University College of Medicine, Seoul, Korea
| | - Ha Neul Jeong
- Department of Neurology, Yonsei University College of Medicine, Seoul, Korea
| | - Young Chul Choi
- Department of Neurology, Yonsei University College of Medicine, Seoul, Korea
| | - Bum Chun Suh
- Department of Neurology, Kangbuk Samsung Hospital, Sungkyunkwan University School of Medicine, Seoul, Korea
| | - Kee Duk Park
- Department of Neurology, Mokdong Hospital, College of Medicine, Ewha Womans University, Seoul, Korea.
| | - Seung Min Kim
- Department of Neurology, Yonsei University College of Medicine, Seoul, Korea.
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43
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Rajabally YA, Attarian S. Chronic inflammatory demyelinating polyneuropathy and malignancy: A systematic review. Muscle Nerve 2017; 57:875-883. [PMID: 29194677 DOI: 10.1002/mus.26028] [Citation(s) in RCA: 22] [Impact Index Per Article: 2.8] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Accepted: 11/28/2017] [Indexed: 12/19/2022]
Abstract
A systematic review of the literature was performed on the association of chronic inflammatory demyelinating polyneuropathy (CIDP) with malignancy. Hematological disorders are the most common association, particulalry non-Hodgkin lymphoma. CIDP frequently precedes the malignancy diagnosis, and there is a favorable CIDP response to treatment more than 70% of the time. Melanoma is the second most common association and may be accompanied by antiganglioside antibodies; CIDP shows a good response to immunotherapy. Other cancers are rare, with variable timings and presentations but good responses to immunomodulation and/or cancer therapy. Unusual neurological features such as ataxia, distal/upper limb predominance, or cranial/respiratory/autonomic involvement may suggest associated malignancy as may abdominal pain, diarrhea/constipation, poor appetite/weight loss, dermatological lesions, and lymphadenopathy. In the appropriate clinical and electrophysiological setting, CIDP associated with cancer should be considered. Immunomodulatory therapy, cancer treatment alone, or a combination may be effective. Muscle Nerve 57: 875-883, 2018.
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Affiliation(s)
- Yusuf A Rajabally
- School of Life and Health Sciences, Aston Brain Centre, Aston University, Aston Triangle, Birmingham, B4 7ET, United Kingdom.,Regional Neuromuscular Service, University Hospitals Birmingham, Birmingham, United Kingdom
| | - Shahram Attarian
- Reference Centre for Neuromuscular Diseases and ALS, Centre Hospitalier Universitaire La Timone, Marseille, France.,Aix-Marseille University, Inserm, GMGF, Marseille, France
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44
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Khandelwal S, Saxena S, Hansalia DJ. Neurolymphomatosis: A Surreal Presentation of Lymphoma. Indian J Med Paediatr Oncol 2017; 38:287-290. [PMID: 29200675 PMCID: PMC5686968 DOI: 10.4103/ijmpo.ijmpo_151_16] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/18/2022] Open
Abstract
Background: Neurolymphomatosis is a neurologic complication poorly recognized by neurologists and oncologists and presents usually several months after successful treatment of systemic lymphoma. Other disorders that must be differentiated from these entities include peripheral-nerve or nerve root compression and paraneoplastic neuropathy. Aim: To describe the unusual occurrence of neurolymphomatosis in a patient of B-cell lymphoma. Method: Diagnosis was made by demonstration of enhancement of nerve roots on Magnetic Resonance Imaging of the brachial, lumbosacral plexus, peripheral nerves or by increased hyper-metabolic activity along the course of affected nerves on fluorodeoxyglucose positron emission tomography (FDG-PET). Results and Conclusion: MRI and PET-CT are imaging modalities of choice for evaluation of patients with lymphoma and suspected neural involvement. Treatment of neurolymphomatosis consists of focal radiotherapy and high-dose methotrexate therapy.
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Affiliation(s)
- Shikha Khandelwal
- Gujarat Imaging Centre, Post Graduate Institute of Radiology, Samved Hospital, Ahmedabad, Gujarat, India
| | - Suvinay Saxena
- Department of Radiodiagnosis, Gujarat Imaging Centre, Post Graduate Institute of Radiology, Samved Hospital, Ahmedabad, Gujarat, India
| | - Disha J Hansalia
- Gujarat Imaging Centre, Post Graduate Institute of Radiology, Samved Hospital, Ahmedabad, Gujarat, India
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45
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Bakogeorgos M, Georgoulias V. Risk-reduction and treatment of chemotherapy-induced peripheral neuropathy. Expert Rev Anticancer Ther 2017; 17:1045-1060. [PMID: 28868935 DOI: 10.1080/14737140.2017.1374856] [Citation(s) in RCA: 18] [Impact Index Per Article: 2.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 01/05/2023]
Abstract
INTRODUCTION Chemotherapy-induced peripheral neuropathy (CIPN), a common adverse effect of several chemotherapeutic agents, has a significant impact on quality of life and may even compromise treatment efficacy, requiring chemotherapy dose reduction or discontinuation. CIPN is predominantly related with sensory rather than motor symptoms and the most common related cytotoxic agents are platinum compounds, taxanes and vinca alkaloids. CIPN symptoms may resolve after treatment cessation, but they can also be permanent and continue for years. Areas covered: We present an overview of CIPN pathophysiology, clinical assessment, prevention and treatment identified through a Pubmed search. Expert commentary: No substantial progress has been made in the last few years within the field of prevention and/or treatment of CIPN, in spite of remarkable efforts. Continuous research could expand our knowledge about chemotherapeutic-specific neuropathic pathways and eventually lead to the conception of innovative and targeted agents for the prevention and/or treatment of this debilitating chemotherapy adverse effect.
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46
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Milton J, Renner J, Awuor V. B-cell lymphoma presenting as multiple nerve sheath tumors. Surg Neurol Int 2017; 8:142. [PMID: 28781919 PMCID: PMC5523469 DOI: 10.4103/sni.sni_60_17] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/09/2017] [Accepted: 05/06/2017] [Indexed: 11/04/2022] Open
Abstract
BACKGROUND Diffuse Large B-cell Lymphoma (DLBCL) is the most common form of Non-Hodgkin lymphoma (NHL), accounting for 25-30 percent of cases in the United States.1 Extranodal sites are involved in approximately 40% of cases of DLBCL. CASE DESCRIPTION In this report, we discuss the case of a patient with extranodal DLBCL within the cervical nerve roots that underwent surgical intervention due to the presence of cervical radiculopathy. CONCLUSION The diagnosis of DLBCL was surprising given the appearance of the masses on MRI being similar to that of a neurofibroma or schwannoma. Surgical decompression provided a tissue sample for biopsy as well as an opportunity for decompression of the nerve roots and restoration of function of the patient's left upper extremity.
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Affiliation(s)
- Jason Milton
- Grant Medical Center, Ohio Health, Columbus, USA
| | - Julie Renner
- Department of Neurosurgery, Genesis Healthcare System, Zanesville, Ohio, USA
| | - Victor Awuor
- Grant Medical Center, Ohio Health, Columbus, USA.,Department of Neurosurgery, Genesis Healthcare System, Zanesville, Ohio, USA
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47
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Staff NP, Grisold A, Grisold W, Windebank AJ. Chemotherapy-induced peripheral neuropathy: A current review. Ann Neurol 2017; 81:772-781. [PMID: 28486769 PMCID: PMC5656281 DOI: 10.1002/ana.24951] [Citation(s) in RCA: 522] [Impact Index Per Article: 65.3] [Reference Citation Analysis] [Abstract] [MESH Headings] [Grants] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 11/03/2016] [Revised: 04/30/2017] [Accepted: 05/01/2017] [Indexed: 12/16/2022]
Abstract
Chemotherapy-induced peripheral neuropathy (CIPN) is a common dose-limiting side effect experienced by patients receiving treatment for cancer. Approximately 30 to 40% of patients treated with neurotoxic chemotherapy will develop CIPN, and there is considerable variability in its severity between patients. It is often sensory-predominant with pain and can lead to long-term morbidity in survivors. The prevalence and burden of CIPN late effects will likely increase as cancer survival rates continue to improve. In this review, we discuss the approach to peripheral neuropathy in patients with cancer and address the clinical phenotypes and pathomechanisms of specific neurotoxic chemotherapeutic agents. Ann Neurol 2017;81:772-781.
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Affiliation(s)
| | - Anna Grisold
- Department of Neurology, Medical University of Vienna, Austria
| | - Wolfgang Grisold
- Ludwig Boltzmann Institute for Experimental und Clinical
Traumatology, Vienna, Austria
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48
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Byun JM, Kim KH, Kim M, Kim TM, Jeon YK, Park JH, Paik JH, Lee JM, Lee HY, Lee JS, Heo DS, Lee JO. Diagnosis of secondary peripheral neurolymphomatosis: a multi-center experience. Leuk Lymphoma 2017; 58:2624-2632. [PMID: 28482727 DOI: 10.1080/10428194.2017.1312376] [Citation(s) in RCA: 4] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 10/19/2022]
Abstract
Here, we describe our experience with secondary neurolymphomatosis (NL) in non-Hodgkin lymphoma patients, with the emphasis on the diagnosis process. A retrospective chart review of 12 patients from 3 tertiary academic centers between January 2005 and December 2015 was conducted. Secondary NL was diagnosed within a median interval of 10 months (range 5-41 months) after initial diagnosis of NHL. Painful neuropathy was present in 66.7%, but the diagnosis of NL was delayed in nine out of 12 patients (75.0%) by median of 2 months. Diagnostic modalities included CSF analysis performed in nine patients (75.0%), electrodiagnostic studies in seven (58.3%), radiologic studies in all, and nerve biopsy in two (16.7%). The diagnostic yield was 100.0% (15/15) for FDG-PET/CT and 75.0% (9/12) for MRI. Our experience emphasizes the importance of clinical suspicion of NL in patients with previous history of NHL and selection of diagnostic modality with the greatest clinical utility.
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Affiliation(s)
- Ja Min Byun
- a Department of Internal Medicine , Seoul National University College of Medicine, Seoul National University Hospital , Seoul , Korea.,b Department of Internal Medicine , Seoul Metropolitan Government Seoul National University Boramae Medical Center , Seoul , Korea.,c Department of Internal Medicine , Seoul National University Bundang Hospital , Seongnam , Korea
| | - Ki Hwan Kim
- b Department of Internal Medicine , Seoul Metropolitan Government Seoul National University Boramae Medical Center , Seoul , Korea
| | - Miso Kim
- a Department of Internal Medicine , Seoul National University College of Medicine, Seoul National University Hospital , Seoul , Korea
| | - Tae Min Kim
- a Department of Internal Medicine , Seoul National University College of Medicine, Seoul National University Hospital , Seoul , Korea
| | - Yoon Kyung Jeon
- d Department of Pathology, College of Medicine , Seoul National University , Seoul , Korea
| | - Jeong Hwan Park
- d Department of Pathology, College of Medicine , Seoul National University , Seoul , Korea
| | - Jin Ho Paik
- e Department of Pathology , Seoul National University Bundang Hospital , Seongnam , Korea
| | - Jung Min Lee
- f Department of Nuclear Medicine , Seoul National University Bundang Hospital , Seongnam , Korea
| | - Ho-Young Lee
- g Department of Nuclear Medicine , Seoul National University, College of Medicine , Seoul , Korea
| | - Jong Seok Lee
- c Department of Internal Medicine , Seoul National University Bundang Hospital , Seongnam , Korea
| | - Dae Seog Heo
- a Department of Internal Medicine , Seoul National University College of Medicine, Seoul National University Hospital , Seoul , Korea
| | - Jeong-Ok Lee
- c Department of Internal Medicine , Seoul National University Bundang Hospital , Seongnam , Korea
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49
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Incomplete Miller—Fisher syndrome with advanced stage Burkitt lymphoma. Indian Pediatr 2017; 54:413-415. [DOI: 10.1007/s13312-017-1116-0] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 10/19/2022]
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50
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Illes Z, Blaabjerg M. Cerebrospinal fluid findings in Guillain-Barré syndrome and chronic inflammatory demyelinating polyneuropathies. HANDBOOK OF CLINICAL NEUROLOGY 2017; 146:125-138. [PMID: 29110767 DOI: 10.1016/b978-0-12-804279-3.00009-5] [Citation(s) in RCA: 24] [Impact Index Per Article: 3.0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Subscribe] [Scholar Register] [Indexed: 06/07/2023]
Abstract
The classic immunologic alteration of the cerebrospinal fluid (CSF) in Guillain-Barré syndrome (GBS), albuminocytologic dissociation, has been known since the original paper by Guillain, Barré, and Strohl. Albuminocytologic dissociation has been also described in other forms of the GBS spectrum, such as axonal motor or motor-sensory forms (AMAN, AMSAN), the anti-GQ1b spectrum of Miller Fisher syndrome, and Bickerstaff brainstem encephalitis. Cytokines, chemokines, antibodies, complement components, and molecules with a putative neuroprotective role or indicating axonal damage have also been examined using different methods. Besides these candidate approaches, proteomics has been recently applied to discover potential biomarkers. The overall results support the immunopathogenesis of GBS, but albuminocytologic dissociation remained the only consistent CSF biomarker supporting the diagnosis of GBS. Chronic inflammatory neuropathies also comprise a heterogeneous group of diseases. Increased protein in the CSF is a supportive factor of chronic inflammatory demyelinating polyneuropathy, especially in the absence of definite electrophysiologic criteria. A number of other markers have also been investigated in the CSF of patients with chronic inflammatory neuropathies, similar to GBS. However, none has been used in supporting diagnosis, differentiating among syndromes, or predicting the clinical course and treatment responses.
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Affiliation(s)
- Zsolt Illes
- Department of Neurology, Odense University Hospital, Odense, Denmark; Institute of Clinical Research, University of Southern Denmark, Odense, Denmark.
| | - Morten Blaabjerg
- Department of Neurology, Odense University Hospital, Odense, Denmark; Institute of Clinical Research, University of Southern Denmark, Odense, Denmark
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