scholarly journals Immunoglobulin G4 hypophysitis in a 63-year-old woman with no autoimmune history: a case report

2021 ◽  
Vol 15 (1) ◽  
Author(s):  
Zachary C. Gersey ◽  
Kenan R. Rajjoub ◽  
Thomas M. Pearce ◽  
Scott A. Segel ◽  
Paul A. Gardner ◽  
...  
Keyword(s):  
Autoimmune Disease ◽  
Response To Treatment ◽  
White Female ◽  
High Dose ◽  
Demographic Differences ◽  
Immunoglobulin G4 ◽  
Case Presentation ◽  
Cranial Nerve Palsies ◽  
History Of ◽  
Pituitary Lesion

Abstract Background Immunoglobulin-G4-related hypophysitis is a rare inflammatory disease that can present as a tumefactive pituitary lesion mimicking hypophyseal neoplasms such as pituitary adenoma or craniopharyngioma. The literature on this entity is sparse, with fewer than 100 cases reported across 19 publications; a recent review found only 24 cases published from 2007 to 2018. Previous reports have described demographic differences, with immunoglobulin-G4-related hypophysitis in females tending to present in the second and third decades in association with other autoimmune disease, while males tend to present in the fifth and sixth decades of life without an autoimmune history. Case presentation In contrast to the reported demographic trends, here we describe a unique case of immunoglobulin-G4-related hypophysitis in a 63-year-old white female with no history of autoimmune disease who presented with a rapidly enlarging sellar and hypothalamic mass causing headaches and cranial nerve palsies, prompting biopsy for diagnosis. The patient experienced rapid response to treatment with high-dose steroids and rituximab. Conclusion The case contributes to the growing clinicopathologic description of immunoglobulin-G4-related hypophysitis and illustrates that this diagnosis should be a consideration even outside the conventional demographic setting.

scholarly journals Case Report. Extracorporeal Membrane Oxygenation in Nivolumab Associated Pneumonitis

2017 ◽  
Vol 3 (2) ◽  
pp. 84-88
Author(s):  
Thomas-Michael Schneider ◽  
Friederike Klenner ◽  
Franz Brettner
Keyword(s):  
Case Report ◽  
Extracorporeal Membrane Oxygenation ◽  
Therapeutic Option ◽  
High Dose ◽  
Great Success ◽  
Case Presentation ◽  
Computed Tomographic ◽  
Rehabilitation Hospital ◽  
Membrane Oxygenation ◽  
History Of

Abstract Background: Newly approved immunotherapeutic agents, like CTLA-4 inhibitors and antibodies against PD-1, are a promising therapeutic option in cancer therapy. Case presentation: A 74-year-old man, with a history of advanced stage melanoma and treatment with ipilimumab, pembrolizumab and nivolumab, was admitted to the hospital due to respiratory failure with hypoxemia and dyspnoea. He rapidly developed severe acute respiratory distress syndrome (ARDS), which required treatment in the intensive care unit which included mechanical ventilation and extracorporeal membrane oxygenation (ECMO). Computed tomographic imaging (CT) showed signs of a pneumonitis, with an ARDS pattern related to the use of PD-1 antibodies. Treating the patient with high-dose immunosuppressive steroids led to an overall improvement. He was transferred to a rehabilitation hospital and subsequently to his home. Discussion and conclusion: This is a unique case report of a patient suffering a grade 4 adverse event under nivolumab who survived having been treated with ECMO. It highlights the possibility of associated adverse reactions as well as the use of ECMO in palliative care patients. ECMO can be of great success even in patients with malignancies, but careful decision making should be done on a case by case basis.

scholarly journals Tocilizumab for massive refractory pleural effusion in an adolescent with systemic lupus erythematosus

2021 ◽  
Vol 19 (1) ◽  
Author(s):  
Arianna De Matteis ◽  
Emanuela Sacco ◽  
Camilla Celani ◽  
Andrea Uva ◽  
Virginia Messia ◽  
...  
Keyword(s):  
Pleural Effusion ◽  
Lupus Erythematosus ◽  
High Dose ◽  
Common Symptom ◽  
Systemic Lupus ◽  
Malar Rash ◽  
Case Presentation ◽  
Double Stranded Dna ◽  
First Case ◽  
History Of

Abstract Background Pleural effusion in systemic lupus erythematous (SLE) is a common symptom, and recent studies demonstrated that IL-6 has a pivotal role in its pathogenesis. Case presentation We report a case of a 15 years old Caucasian boy with a history of persistent pleural effusion without lung involvement or fever. Microbiological and neoplastic aetiologies were previously excluded. Based on the presence of pleuritis, malar rash, reduction of C3 and C4 levels and positivity of antinuclear antibody (ANA) and anti-double stranded DNA (dsDNA), the diagnosis of juvenile SLE (JSLE) was performed. Treatment with high dose of intravenous glucocorticoids and mycophenolate mofetil was started with partial improvement of pleural effusion. Based on this and on adults SLE cases with serositis previously reported, therapy with intravenous tocilizumab (800 mg every two weeks) was started with prompt recovery of pleural effusion. Conclusion To the best of our knowledge, this is the first case of JSLE pleuritis successfully treated with tocilizumab.

Avoiding a lumbar puncture may be a rash decision: a case report of varicella-zoster virus-associated radiculopathy in advanced HIV infection

2019 ◽  
Vol 30 (10) ◽  
pp. 1031-1033
Author(s):  
Emma Wallis ◽  
Bahij Al-Hakim ◽  
Paul Holmes ◽  
Sam Douthwaite ◽  
Ranjababu Kulasegaram
Keyword(s):  
Varicella Zoster Virus ◽  
Lumbar Puncture ◽  
Case Reports ◽  
Response To Treatment ◽  
High Dose ◽  
Serum Samples ◽  
Cd4 Cell Count ◽  
Varicella Zoster ◽  
Intravenous Acyclovir ◽  
History Of

A 34-year-old man recently diagnosed with advanced human immunodeficiency virus infection (CD4 cell count of 139 cells/mm3), not yet started on antiretroviral medications, presented to hospital with a ten-day history of left leg weakness and difficulty walking. He described a childhood history of chickenpox with previous shingles over his buttock over three years ago. Examination revealed reduced power in the left hip and knee flexors and absent knee and adductor reflexes. Lumbar punctures were performed and polymerase chain reaction (PCR) detected varicella-zoster virus (VZV) DNA. Concurrent serum samples for VZV PCR were negative. The patient was diagnosed with VZV radiculopathy and treated with high-dose intravenous acyclovir. Within two days, neurological signs improved. Previous case reports define VZV radiculopathy by a temporal and geographical relationship with a zoster rash. Our diagnosis was based on a clinical picture of radiculopathy with virological evidence in CSF and confirmed by a dramatic clinical response to treatment. We propose that lumbar puncture and detection of VZV DNA by PCR in the cerebrospinal fluid (CSF) is an invaluable investigation that should be considered in the workup of immunosuppressed patients presenting with a radiculopathy.

scholarly journals Hypertrophic pachymeningoencephalitis associated with temporal giant cell arteritis

2018 ◽  
pp. bcr-2018-225304
Author(s):  
Gesine Boisch ◽  
Sven Duda ◽  
Christian Hartmann ◽  
Heinrich Weßling
Keyword(s):  
Giant Cell Arteritis ◽  
Giant Cell ◽  
Temporal Arteritis ◽  
Brain Mri ◽  
Neurological Deficits ◽  
High Dose ◽  
Immunoglobulin G4 ◽  
Perifocal Oedema ◽  
History Of ◽  
Dose Corticosteroid

We report the rare case of an 80-year-old male patient with hypertrophic pachymeningoencephalitis that may be associated with temporal arteritis. The patient presented to our neurological department with a 2-week history of latent paresis and ataxia affecting his right hand. He had been diagnosed with temporal arteritis 12 years earlier. Brain MRI showed an enhancement of the left-sided frontoparietal meninges with oedema of the adjacent tissue of the precentral and postcentral cortex. A leptomeningeal biopsy was performed. An autoimmune-mediated immunoglobulin G4-associated hypertrophic pachymeningoencephalitis was diagnosed. The patient received a high-dose corticosteroid therapy and his symptoms gradually improved. Our results suggest that hypertrophic pachymeningoencephalitis may occur as a complication of giant cell arteritis and may cause central neurological deficits by cerebral perifocal oedema.

Pituitary Apoplexy

Neurosurgery ◽  
2005 ◽  
Vol 56 (1) ◽  
pp. 65-73 ◽  
Author(s):  
Patrick L. Semple ◽  
Michael K. Webb ◽  
Jacques C. de Villiers ◽  
Edward R. Laws
Keyword(s):  
Pituitary Apoplexy ◽  
Average Length ◽  
Imaging Modality ◽  
Previous History ◽  
High Dose ◽  
Corticosteroid Administration ◽  
Cranial Nerve Palsies ◽  
History Of ◽  
Dose Corticosteroid ◽  
University Of Cape Town

Abstract OBJECTIVE: Pituitary apoplexy is a rare yet potentially fatal disease. We reviewed the combined experience of the University of Virginia in Charlottesville, VA, and Groote Schuur Hospital, University of Cape Town, South Africa, with 62 cases of pituitary apoplexy. METHODS: An Internet web site with a database was constructed, and the records of 62 patients were entered into the database from both institutions and then systematically analyzed. RESULTS: The average age of patients was 51.1 years; 60% were male, and the average length of follow-up was 55 months. The average time of presentation was 14.2 days after the ictus, and 81% had no previous history of pituitary tumor. Headache was the most common presenting symptom (87%). Diminished visual acuity was found in 56% of patients, bitemporal hemianopia in 34%, ocular palsies in 45%, and diminished level of consciousness in 13%. Seventy-three percent of the patients had laboratory evidence of hypopituitarism, and 8% had diabetes insipidus. Fifty-eight patients underwent surgery, 3 were treated conservatively, and 1 died before intervention. Histological examination revealed hemorrhagic infarction in 47%, simple infarction in 40%, and frank hemorrhage in 8%. Seventy-nine percent had a good outcome, although 83% required subsequent hormonal replacement therapy. CONCLUSION: Pituitary apoplexy is often misdiagnosed because the majority of patients have undetected pituitary adenomas, and the presentation is often mistaken for subarachnoid hemorrhage. Most cases of pituitary apoplexy occur spontaneously, although precipitating factors have been suggested. Magnetic resonance imaging is the imaging modality of choice. Treatment includes high-dose corticosteroid administration and surgery. Transsphenoidal surgery is indicated in patients with diminished levels of consciousness, hypothalamic dysfunction, and visual deterioration. Conservative management for patients with isolated cranial nerve palsies has been advocated but remains controversial.

scholarly journals A peculiar case of psychosis

2021 ◽  
Author(s):  
Lamia Kouba ◽  
Dalia Alhosain
Keyword(s):  
High Dose ◽  
Case Presentation ◽  
Aspartate Receptor ◽  
Nmdar Encephalitis ◽  
Life Threatening ◽  
History Of ◽  
Simplex Virus ◽  
Peculiar Case ◽  
In Pregnancy ◽  
New Onset

Abstract ● Background: Psychosis in pregnancy is rare and could be life-threatening. It requires prompt evaluation and proper management accordingly. Anti-N-methyl-d-aspartate receptor (anti-NMDAr) encephalitis following herpes simplex virus (HSV) infection is a rare cause of psychosis during pregnancy. ● Case presentation: A 20 year old woman at 18 weeks gestation presented with agitation and a 3-day history of hallucinations. She had a prior hospitalization for HSV encephalitis six weeks before. Her laboratory workup was unremarkable except for positive anti-NMDAr antibodies in the CSF. The patient was treated with high dose corticosteroids and plasmapheresis and she was discharged two weeks later fully recovered.● Conclusions: Anti-NMDAr encephalitis can be the culprit behind a new-onset of psychosis in pregnancy. Early diagnosis and treatment are crucial.

scholarly journals Eosinophilic bronchiolitis successfully treated with benralizumab

2021 ◽  
Vol 14 (10) ◽  
pp. e246058
Author(s):  
Keishi Sugino ◽  
Hirotaka Ono ◽  
Akira Hebisawa ◽  
Eiyasu Tsuboi
Keyword(s):  
Plasma Cells ◽  
Final Diagnosis ◽  
Japanese Woman ◽  
Wall Thickening ◽  
High Dose ◽  
Immunoglobulin G4 ◽  
Bronchial Wall ◽  
History Of ◽  
Frequent Exacerbation ◽  
Wet Cough

A 53-year-old non-smoking Japanese woman was admitted to our hospital with a 20-year history of wet cough and dyspnoea on exertion. Bronchial asthma (BA) had been diagnosed 20 years earlier. Although she has been treated with high-dose inhaled corticosteroid, she had experienced frequent exacerbation of BA, and short-term oral corticosteroid bursts were occasionally administered. High-resolution CT of the chest revealed diffuse centrilobular nodules with bronchial wall thickening and patchy ground-glass opacities in both lungs. Lung biopsy specimens showed widespread cellular bronchiolitis with follicle formations in the membranous and respiratory bronchioles, accompanied by marked infiltration of plasma cells and eosinophils. In addition, immunohistochemical immunoglobulin G4 (IgG4) staining revealed many IgG4-positive plasma cells, and the ratio of IgG4-positive cells to IgG-positive cells exceeded 40%. The final diagnosis was eosinophilic bronchiolitis with marked IgG4-positive plasma cell infiltration in association with BA. With benralizumab therapy, her clinical condition dramatically improved.

scholarly journals Disseminated Mycobacterium Sepsis with Bone Marrow, Liver, and Lung Involvement Following Intravesical Bacillus Calmette-Guerin (BCG) Therapy

2019 ◽  
Vol 14 (2) ◽  
pp. 34-37
Author(s):  
Drew Hager ◽  
Zeenib Kohja ◽  
Terry Wuerz ◽  
Arjuna Ponnampalam
Keyword(s):  
Bone Marrow ◽  
High Dose ◽  
Case Presentation ◽  
First Line Therapy ◽  
Elevated Liver Enzymes ◽  
Bcg Therapy ◽  
Intravesical Bcg ◽  
History Of ◽  
Muscle Invasive ◽  
High Index Of Suspicion

Introduction: BCG therapy is first line therapy for high grade non-muscle invasive bladder cancer (NMIBC).Case Presentation: A 54-year-old male presented with fevers, rigors and hematuria one week following intravesical BCG administration for treatment of NMIBC. He developed fever, pancytopenia, elevated liver enzymes and pulmonary infiltrates with progression of symptoms despite broad spectrum antimicrobial therapy. A bone marrow biopsy showed granulomatous infiltration; cultures of urine demonstrated growth of Mycobacterium bovis. A diagnosis of disseminated BCG infection secondary to intravesical administration was made; rifampin, isoniazid, ethambutol, and high dose prednisone were initiated.Conclusion: Adverse events associated with BCG administration have been attributed to both the primary mycobacterium infection and to hypersensitivity reactions. Timely collection of histopathology can lead to early treatment of disseminated BCG with good outcomes. Internists should have a high index of suspicion for patients presenting with organ dysfunction with an immediate or remote history of intravesical BCG administration.

scholarly journals MO309SARS COV2 INFECTION COMPLICATING CRYOGLOBULINEMIC VASCULITIS WITH RENAL INVOLVEMENT

2021 ◽  
Vol 36 (Supplement_1) ◽  
Author(s):  
Hanene Bouafif ◽  
Hajji Mariem ◽  
Samia Barbouch ◽  
Hedri Hafedh ◽  
Imen Gorsane ◽  
...  
Keyword(s):  
Hemorrhagic Cystitis ◽  
Renal Involvement ◽  
Skin Lesions ◽  
Response To Treatment ◽  
Case Presentation ◽  
Nasal Septal Perforation ◽  
History Of ◽  
Peripheral Arterial ◽  
Inflammatory Syndrome

Abstract Background and Aims Cryoglobulinaemic vasculitis (CV) leading to clinically apparent skin lesions, and in some cases also to internal organ involvement such as renal involvement. The SARS Cov2 infection in this field is very serious and in the case of our patient the outcome was fatal. The aim of this work is to highlight the particularities of CV in this case Case presentation We report a case of a 62-year-old woman followed since 2014 for CV type 1 IgG Lambda revealed by vascular purpura and nasal septal perforation. the etiological assessment was negative. Hepatitis C virus serology was negative. A corticosteroid therapy at a dose of 1 mg/kg/day was started with partial improvement. Then the cyclophosphamide at a dose of 150 mg/d was added with a fairly good clinical result but stopped for hemorrhagic cystitis. Chlorominophene was started at a dose of 6 mg/d and then reduced to 4 mg/d for leuconeutropenia but the peripheral arterial manifestations (acrocyanosis of the limbs and ears) were not treated. The evolution was marked by the appearance of renal failure (creatinine 482.33 umol/l) associated with proteinuria at 1.32 g/24h. An outbreak of cryoglobulinemia was suspected. Plasma exchange was discussed as a therapeutic alternative and the patient was scheduled for a renal biopsy. However, the patient became febrile with the appearance of a dry cough and the presence of a biological inflammatory syndrome (CRP at 72). A SARS-CoV-2 PCR was completed which was positive. Therefore, she was put on antibiotic therapy (Azythromycin and Cefotaxime) associated with vitamin therapy and anticoagulant treatment. The patient was stable in terms of respiration and hemodynema, but her renal function worsened and progressed well under hydration. In addition, she presented with a slippage syndrome and she was died Conclusion The natural history of CV is not predictable and strongly depends on concomitant diseases and complications and response to treatment. The SARS Cov2 infection can complicate its evolution. There is no association has been described between them to our knowledge

Pituitary apoplexy and associated cranial nerve palsies secondary to bleeding caused by immune thrombocytopaenia in a patient with known pituitary macroadenoma

2021 ◽  
Vol 14 (5) ◽  
pp. e240105
Author(s):  
Christopher Ambrose ◽  
Sruthi Sarma ◽  
Ritwick Banerjee ◽  
Sam Myers
Keyword(s):  
Pituitary Apoplexy ◽  
Optic Chiasm ◽  
High Dose ◽  
Pituitary Macroadenoma ◽  
Cranial Nerve Palsies ◽  
History Of ◽  
Low Platelet Count ◽  
Background History ◽  
Minor Improvement ◽  
Multidisciplinary Teamwork

An 84-year-old man presented with a frontal headache and easy bruising. He had a background history of a pituitary macroadenoma, diagnosed incidentally a year earlier. Investigations showed haemorrhage into the pituitary macroadenoma leading to a diagnosis of pituitary apoplexy in the context of low platelet count secondary to immune thrombocytopaenia. He was treated with intravenous hydrocortisone, platelet transfusion, intravenous immunoglobulin and high-dose steroid. Neurosurgical intervention was not indicated initially. Five days into his admission, he developed bilateral ptosis and ophthalmoplegia. MRI confirmed further haemorrhage associated with compression of the optic chiasm. He was transferred to a tertiary neurosurgical centre where he underwent urgent surgical decompression. To date, there has been minor improvement in his neurological symptoms. Management of this patient required considerable multidisciplinary teamwork between the clinics of endocrinology, haematology, neurosurgery, ophthalmology and geriatrics.

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