ABSTRACT
IMPORTANCE: Chronic inflammatory demyelinating polyradiculoneuropathy (CIDP) is an autoimmune neurological disorder that is characterized by symmetrical progressive worsening or relapsing weakness and numbness of the limbs. There are no reliable diagnostic tests or definitive diagnostic criteria, and the diagnosis remains one of excluding other cases of polyneuropathy. Typical treatment for CIDP includes corticosteroids, intravenous immunoglobulin (IVIG), and plasma exchange. Little is known about CIDP as a treatment complication of immune checkpoint inhibitors (ipilimumab and nivolumab). This report will be helpful in increasing awareness and knowledge about this unique entity. OBSERVATIONS: We describe two cases of CIDP secondary to treatment with combined ipilimumab and nivolumab in patients with metastatic melanoma that were successfully treated with prednisone and IVIG. Conclusion and Relevance: This report illustrates that treatment with immune checkpoint inhibitors can lead to CIDP that can be successfully treated with complete resolution of symptoms. CIDP secondary to checkpoint inhibitors may have unique features such as low-grade lymphocytic pleocytosis on CSF evaluation as well as severe neuropathic pain as an early presenting symptom. Additionally, it is interesting to note that both patients presented in this report remained melanoma-free on follow-up more than 16 months later.
Subject(s)
Antineoplastic Agents, Immunological/adverse effects , Antineoplastic Combined Chemotherapy Protocols/adverse effects , Melanoma/drug therapy , Polyradiculoneuropathy, Chronic Inflammatory Demyelinating/chemically induced , Skin Neoplasms/drug therapy , Aged , Female , Humans , Immunoglobulins, Intravenous/therapeutic use , Ipilimumab/adverse effects , Male , Melanoma/immunology , Melanoma/pathology , Middle Aged , Nivolumab/adverse effects , Polyradiculoneuropathy, Chronic Inflammatory Demyelinating/diagnosis , Polyradiculoneuropathy, Chronic Inflammatory Demyelinating/drug therapy , Polyradiculoneuropathy, Chronic Inflammatory Demyelinating/immunology , Prednisone/therapeutic use , Skin Neoplasms/immunology , Skin Neoplasms/pathology , Treatment OutcomeABSTRACT
The development of progressive multifocal leukoencephalopathy (PML) in patients treated with natalizumab is a well-known potential risk. Diagnosis of PML can be confounded in patients with multiple sclerosis (MS) if new demyelinating lesions develop, and the sensitivity of existing diagnostic tests is less than ideal. In the case presented here, four samples of cerebrospinal fluid tested negative for John Cunningham virus (JCV) DNA by polymerase chain reaction, yet brain biopsy eventually proved positive by immunohistochemistry. A review of the limitations of existing clinical diagnostic tests is addressed, and we review the most recent literature on the proper management of natalizumab-treated MS patients.
Subject(s)
Antibodies, Monoclonal, Humanized/adverse effects , JC Virus/isolation & purification , Leukoencephalopathy, Progressive Multifocal/cerebrospinal fluid , Leukoencephalopathy, Progressive Multifocal/chemically induced , Aged , Biomarkers/cerebrospinal fluid , Female , Humans , Leukoencephalopathy, Progressive Multifocal/virology , NatalizumabSubject(s)
Dyspnea/diagnosis , Glycogen Storage Disease Type II/diagnosis , Glycogen Storage Disease Type II/epidemiology , Respiratory Paralysis/diagnosis , Age of Onset , Biopsy , Dyspnea/etiology , Glycogen Storage Disease Type II/complications , Humans , Male , Middle Aged , Muscle, Skeletal/enzymology , Muscle, Skeletal/pathology , Pulmonary Atelectasis/diagnostic imaging , Radiography , Respiratory Paralysis/etiology , alpha-Glucosidases/metabolismABSTRACT
We report a case of acute, painful polyneuropathy in a boy with newly diagnosed type 1 diabetes mellitus associated with a precipitous drop in hemoglobin A1c. After initiation of insulin, the patient's hemoglobin A1c dropped from 14.1 to 7.6%, and he developed severe pain in his feet, which prevented him from walking. Nerve conduction studies were consistent with mild to moderate sensorimotor peripheral neuropathy. Initially, he required opiate analgesics for pain control. Three months after presentation, the patient showed dramatic improvement and regained his ability to walk. Although not well described in the pediatric literature, this case represents insulin neuritis, one of the few diabetic neuropathies that has a favorable outcome.
