Diagnosis and Treatment > Medication > Methylprednisolone
You are looking at 1 - 10 of 21 items
Search for other papers by Agnieszka Łebkowska in
Google Scholar
PubMed
Search for other papers by Anna Krentowska in
Google Scholar
PubMed
Search for other papers by Agnieszka Adamska in
Google Scholar
PubMed
Search for other papers by Danuta Lipińska in
Google Scholar
PubMed
Search for other papers by Beata Piasecka in
Google Scholar
PubMed
Search for other papers by Otylia Kowal-Bielecka in
Google Scholar
PubMed
Search for other papers by Maria Górska in
Google Scholar
PubMed
Search for other papers by Robert K Semple in
Google Scholar
PubMed
Search for other papers by Irina Kowalska in
Google Scholar
PubMed
Summary
Type B insulin resistance syndrome (TBIR) is characterised by the rapid onset of severe insulin resistance due to circulating anti-insulin receptor antibodies (AIRAs). Widespread acanthosis nigricans is normally seen, and co-occurrence with other autoimmune diseases is common. We report a 27-year-old Caucasian man with psoriasis and connective tissue disease who presented with unexplained rapid weight loss, severe acanthosis nigricans, and hyperglycaemia punctuated by fasting hypoglycaemia. Severe insulin resistance was confirmed by hyperinsulinaemic euglycaemic clamping, and immunoprecipitation assay demonstrated AIRAs, confirming TBIR. Treatment with corticosteroids, metformin and hydroxychloroquine allowed withdrawal of insulin therapy, with stabilisation of glycaemia and diminished signs of insulin resistance; however, morning fasting hypoglycaemic episodes persisted. Over three years of follow-up, metabolic control remained satisfactory on a regimen of metformin, hydroxychloroquine and methotrexate; however, psoriatic arthritis developed. This case illustrates TBIR as a rare but severe form of acquired insulin resistance and describes an effective multidisciplinary approach to treatment.
Learning points:
-
We describe an unusual case of type B insulin resistance syndrome (TBIR) in association with mixed connective tissue disease and psoriasis.
-
Clinical evidence of severe insulin resistance was corroborated by euglycaemic hyperinsulinaemic clamp, and anti-insulin receptor autoantibodies were confirmed by immunoprecipitation assay.
-
Treatment with metformin, hydroxychloroquine and methotrexate ameliorated extreme insulin resistance.
Search for other papers by Mike Lin in
Google Scholar
PubMed
Cancer Genetics Unit, Kolling Institute of Medical Research, New South Wales, Australia
Faculty of Health and Medicine, University of Sydney, New South Wales, Australia
Search for other papers by Venessa Tsang in
Google Scholar
PubMed
Search for other papers by Janice Brewer in
Google Scholar
PubMed
Cancer Genetics Unit, Kolling Institute of Medical Research, New South Wales, Australia
Faculty of Health and Medicine, University of Sydney, New South Wales, Australia
Search for other papers by Roderick Clifton-Bligh in
Google Scholar
PubMed
Cancer Genetics Unit, Kolling Institute of Medical Research, New South Wales, Australia
Faculty of Health and Medicine, University of Sydney, New South Wales, Australia
Search for other papers by Matti L Gild in
Google Scholar
PubMed
Summary
Lymphocytic hypophysitis is a rare neuroendocrine disease characterised by an autoimmune inflammatory disorder of the pituitary gland. We report a 50-year-old woman who presented with headaches and bilateral sixth cranial nerve palsies. MRI of the pituitary revealed extensive fibrosis involving the sellar and extending into both cavernous sinuses causing bilateral occlusion of the internal carotid arteries (ICA). Transphenoidal biopsy confirmed the diagnosis of infiltrative fibrotic lymphocytic hypophysitis. Symptoms resolved with high dose of oral steroids but relapsed on tapering, requiring several treatments of i.v. pulse steroids over 8 months. Rituximab combined with mycophenolate mofetil was required to achieve long-term symptom relief. Serial MRI pituitary imaging showed stabilisation of her disease without reduction in sellar mass or regression of ICA occlusion. The patient’s brain remained perfused solely by her posterior circulation. This case demonstrates an unusual presentation of a rare disease and highlights a successful steroid-sparing regimen in a refractory setting.
Learning points:
-
Lymphocytic hypophysitis is a rare inflammatory disorder of the pituitary gland. In exceptional cases, there is infiltration of the cavernous sinus with subsequent occlusion of the internal carotid arteries.
-
First-line treatment of lymphocytic hypophysitis is high-dose glucocorticoids. Relapse after tapering or discontinuation is common and its use is limited by long-term adverse effects.
-
There is a paucity of data for treatment of refractory lymphocytic hypophysitis. Goals of treatment should include improvement in symptoms, correction of hormonal insufficiencies, reduction in lesion size and prevention of recurrence.
-
Steroid-sparing immunosuppressive drugs such as rituximab and mycophenolate mofetil have been successful in case reports. This therapeutic combination represents a viable alternative treatment for refractory disease.
Search for other papers by Mariana Barbosa in
Google Scholar
PubMed
Search for other papers by Sílvia Paredes in
Google Scholar
PubMed
Search for other papers by Maria João Machado in
Google Scholar
PubMed
Pituitary Consult, Hospital de Braga, Braga, Portugal
Search for other papers by Rui Almeida in
Google Scholar
PubMed
Pituitary Consult, Hospital de Braga, Braga, Portugal
Search for other papers by Olinda Marques in
Google Scholar
PubMed
Summary
Gonadotropin-releasing hormone (GnRH) agonists, currently used in the treatment of advanced prostate cancer, have been described as a rare cause of pituitary apoplexy, a potentially life-threatening clinical condition. We report the case of a 69-year-old man with a known pituitary macroadenoma who was diagnosed with prostate cancer and started treatment with GnRH agonist leuprorelin (other hormones were not tested before treatment). Few minutes after drug administration, the patient presented with acute-onset severe headache, followed by left eye ptosis, diplopia and vomiting. Pituitary MRI revealed tumor enlargement and T1-hyperintense signal, compatible with recent bleeding sellar content. Laboratory endocrine workup was significant for low total testosterone. The patient was managed conservatively with high-dose steroids, and symptoms significantly improved. This case describes a rare phenomenon, pituitary apoplexy induced by GnRH agonist. We review the literature regarding this condition: the pathophysiological mechanism involved is not clearly established and several hypotheses have been proposed. Although uncommon, healthcare professionals and patients should be aware of this complication and recognize the signs, preventing a delay in diagnosis and treatment.
Learning points:
-
Pituitary apoplexy (PA) is a potentially life-threatening complication that can be caused by gonadotropin-releasing hormone agonist (GnRHa) administration for the treatment of advanced prostate cancer.
-
This complication is rare but should be taken into account when using GnRHa, particularly in the setting of a known pre-existing pituitary adenoma.
-
PA presents with classic clinical signs and symptoms that should be promptly recognized.
-
Patients should be instructed to seek medical care if suspicious symptoms occur.
-
Healthcare professionals should be aware of this complication, enabling its early recognition, adequate treatment and favorable outcome.
Search for other papers by Aishah Ekhzaimy in
Google Scholar
PubMed
Search for other papers by Afshan Masood in
Google Scholar
PubMed
Search for other papers by Seham Alzahrani in
Google Scholar
PubMed
Search for other papers by Waleed Al-Ghamdi in
Google Scholar
PubMed
Search for other papers by Daad Alotaibi in
Google Scholar
PubMed
Search for other papers by Muhammad Mujammami in
Google Scholar
PubMed
Summary
Central diabetes insipidus (CDI) and several endocrine disorders previously classified as idiopathic are now considered to be of an autoimmune etiology. Dermatomyositis (DM), a rare autoimmune condition characterized by inflammatory myopathy and skin rashes, is also known to affect the gastrointestinal, pulmonary, and rarely the cardiac systems and the joints. The association of CDI and DM is extremely rare. After an extensive literature search and to the best of our knowledge this is the first reported case in literature, we report the case of a 36-year-old male with a history of CDI, who presented to the hospital’s endocrine outpatient clinic for evaluation of a 3-week history of progressive facial rash accompanied by weakness and aching of the muscles.
Learning points:
-
Accurate biochemical diagnosis should always be followed by etiological investigation.
-
This clinical entity usually constitutes a therapeutic challenge, often requiring a multidisciplinary approach for optimal outcome.
-
Dermatomyositis is an important differential diagnosis in patients presenting with proximal muscle weakness.
-
Associated autoimmune conditions should be considered while evaluating patients with dermatomyositis.
-
Dermatomyositis can relapse at any stage, even following a very long period of remission.
-
Maintenance immunosuppressive therapy should be carefully considered in these patients.
Search for other papers by Misaki Aoshima in
Google Scholar
PubMed
Search for other papers by Koji Nagayama in
Google Scholar
PubMed
Search for other papers by Kei Takeshita in
Google Scholar
PubMed
Search for other papers by Hiroshi Ajima in
Google Scholar
PubMed
Search for other papers by Sakurako Orikasa in
Google Scholar
PubMed
Search for other papers by Ayana Iwazaki in
Google Scholar
PubMed
Search for other papers by Hiroaki Takatori in
Google Scholar
PubMed
Search for other papers by Yutaka Oki in
Google Scholar
PubMed
Summary
Patients treated with immunosuppressive drugs, especially methotrexate (MTX), rarely develop lymphoproliferative disorders (LPDs), known as MTX-related LPD (MTX–LPD). The primary site of MTX–LPD is often extranodal. This is the first reported case of MTX–LPD in the pituitary. A 65-year-old woman was admitted to our hospital with symptoms of oculomotor nerve palsy and multiple subcutaneous nodules. She had been treated with MTX for 11 years for rheumatoid arthritis. Computed tomography showed multiple masses in the orbit, sinuses, lung fields, anterior mediastinum, kidney, and subcutaneous tissue. Brain magnetic resonance imaging revealed a sellar mass. She was diagnosed with hypopituitarism and central diabetes insipidus based on endocrine examination. Although pituitary biopsy could not be performed, we concluded that the pituitary lesion was from MTX–LPD, similar to the lesions in the sinuses, anterior mediastinum, and subcutaneous tissue, which showed polymorphic LPD on biopsy. MTX was discontinued, and methylprednisolone was administered to improve the neurologic symptoms. After several weeks, there was marked improvement of all lesions, including the pituitary lesion, but the pituitary function did not improve. When pituitary lesions are caused by MTX–LPD, the possibility of anterior hypopituitarism and central diabetes insipidus needs to be considered. Further studies are needed to investigate the effectiveness of early diagnosis and treatment of MTX–LPD in restoring pituitary dysfunction.
Learning points
-
Pituitary lesions from MTX–LPD may cause hypopituitarism and central diabetes insipidus.
-
Pituitary metastasis of malignant lymphoma and primary pituitary lymphoma, which have the same tissue types with MTX–LPD, have poor prognosis, but the lesions of MTX–LPD can regress only after MTX discontinuation.
-
In cases of pituitary lesions alone, a diagnosis of MTX–LPD may be difficult, unless pituitary biopsy is performed. This possibility should be considered in patients treated with immunosuppressive drugs.
-
Pituitary hypofunction and diabetes insipidus may persist, even after regression of the lesions on imaging due to MTX discontinuation.
Search for other papers by Charlotte Delcourt in
Google Scholar
PubMed
Search for other papers by Halil Yildiz in
Google Scholar
PubMed
Search for other papers by Alessandra Camboni in
Google Scholar
PubMed
Search for other papers by Eric Van den Neste in
Google Scholar
PubMed
Search for other papers by Véronique Roelants in
Google Scholar
PubMed
Search for other papers by Alexandra Kozyreff in
Google Scholar
PubMed
Search for other papers by Jean Paul Thissen in
Google Scholar
PubMed
Search for other papers by Dominique Maiter in
Google Scholar
PubMed
Search for other papers by Raluca Maria Furnica in
Google Scholar
PubMed
Summary
A 26-year-old woman presented with persistent headache and tiredness. Biological investigations disclosed a moderate inflammatory syndrome, low PTH-hypercalcemia and complete anterior hypopituitarism. A magnetic resonance imaging (MRI) of the pituitary gland was performed and revealed a symmetric enlargement with a heterogeneous signal. Ophthalmological examination showed an asymptomatic bilateral anterior and posterior uveitis, and a diagnosis of pituitary sarcoidosis was suspected. As the localization of lymphadenopathies on the fused whole-body FDG-PET/computerized tomography (CT) was not evoking a sarcoidosis in first instance, an excisional biopsy of a left supraclavicular adenopathy was performed showing classic nodular sclerosis Hodgkin’s lymphoma (HL). A diagnostic transsphenoidal biopsy of the pituitary gland was proposed for accurate staging of the HL and surprisingly revealed typical granulomatous inflammation secondary to sarcoidosis, leading to the diagnosis of a sarcoidosis–lymphoma syndrome. The co-existence of these diseases constitutes a diagnostic challenge and we emphasize the necessity of exact staging of disease in order to prescribe adequate treatment.
Learning points:
-
The possibility of a sarcoidosis–lymphoma syndrome, although rare, should be kept in mind during evaluation for lymphadenopathies.
-
In the case of such association, lymphoma usually occurs after sarcoidosis. However, sarcoidosis and lymphoma can be detected simultaneously and development of sarcoidosis in a patient with previous lymphoma has also been reported.
-
An accurate diagnosis of the disease and the respective organ involvements, including biopsy, is necessary in order to prescribe adequate treatment.
Search for other papers by Jonathan Brown in
Google Scholar
PubMed
Search for other papers by Luqman Sardar in
Google Scholar
PubMed
Summary
A 68-year-old previously independent woman presented multiple times to hospital over the course of 3 months with a history of intermittent weakness, vacant episodes, word finding difficulty and reduced cognition. She was initially diagnosed with a TIA, and later with a traumatic subarachnoid haemorrhage following a fall; however, despite resolution of the haemorrhage, symptoms were ongoing and continued to worsen. Confusion screen blood tests showed no cause for the ongoing symptoms. More specialised investigations, such as brain imaging, cerebrospinal fluid analysis, electroencephalogram and serology also gave no clear diagnosis. The patient had a background of hypothyroidism, with plasma thyroid function tests throughout showing normal free thyroxine and a mildly raised thyroid-stimulating hormone (TSH). However plasma anti-thyroid peroxidise (TPO) antibody titres were very high. After discussion with specialists, it was felt she may have a rare and poorly understood condition known as Hashimoto’s encephalopathy (HE). After a trial with steroids, her symptoms dramatically improved and she was able to live independently again, something which would have been impossible at presentation.
Learning points:
-
In cases of subacute onset confusion where most other diagnoses have already been excluded, testing for anti-thyroid antibodies can identify patients potentially suffering from HE.
-
In these patients, and under the guidance of specialists, a trial of steroids can dramatically improve patient’s symptoms.
-
The majority of patients are euthyroid at the time of presentation, and so normal thyroid function tests should not prevent anti-thyroid antibodies being tested for.
-
Due to high titres of anti-thyroid antibodies being found in a small percentage of the healthy population, HE should be treated as a diagnosis of exclusion, particularly as treatment with steroids may potentially worsen the outcome in other causes of confusion, such as infection.
Search for other papers by Valeria de Miguel in
Google Scholar
PubMed
Search for other papers by Andrea Paissan in
Google Scholar
PubMed
Search for other papers by Patricio García Marchiñena in
Google Scholar
PubMed
Search for other papers by Alberto Jurado in
Google Scholar
PubMed
Search for other papers by Mariana Isola in
Google Scholar
PubMed
Search for other papers by José Alfie in
Google Scholar
PubMed
Search for other papers by Patricia Fainstein-Day in
Google Scholar
PubMed
Summary
We present the case of a 25-year-old male with a history of neurofibromatosis type 1 and bilateral pheochromocytoma 4 years after kidney transplantation that was successfully treated with simultaneous bilateral posterior retroperitoneoscopic adrenalectomy.
Learning points:
-
Hypertensive patients with NF1 should always be screened for pheochromocytoma.
-
Pheochromocytoma is rarely associated with transplantation, but it must be ruled out in patients with genetic susceptibility.
-
Posterior retroperitoneoscopic adrenalectomy (PRA) allows more direct access to the adrenal glands, especially in patients with previous abdominal surgeries.
Search for other papers by H Joshi in
Google Scholar
PubMed
Search for other papers by M Hikmat in
Google Scholar
PubMed
Search for other papers by A P Devadass in
Google Scholar
PubMed
Search for other papers by S O Oyibo in
Google Scholar
PubMed
Search for other papers by S V Sagi in
Google Scholar
PubMed
Summary
IgG4-related disease (IgG4-RD) is an immune-mediated fibro-inflammatory condition which can affect various organs including the pituitary gland. The true annual incidence of this condition remains widely unknown. In addition, it is unclear whether IgG4 antibodies are causative or the end result of a trigger. With no specific biomarkers available, the diagnosis of IgG4-related hypophysitis remains a challenge. Additionally, there is a wide differential diagnosis. We report a case of biopsy-proven IgG4-related hypophysitis in a young man with type 2 diabetes mellitus.
Learning points:
-
IgG4-related hypophysitis is part of a spectrum of IgG4-related diseases.
-
Clinical manifestations result from anterior pituitary hormone deficiencies with or without diabetes insipidus, which can be temporary or permanent.
-
A combination of clinical, radiological, serological and histological evidence with careful interpretation is required to make the diagnosis.
-
Tissue biopsy remains the gold standard investigation.
-
Disease monitoring and long-term management of this condition is a challenge as relapses occur frequently.
Search for other papers by Raluca Maria Furnica in
Google Scholar
PubMed
Search for other papers by Julie Lelotte in
Google Scholar
PubMed
Search for other papers by Thierry Duprez in
Google Scholar
PubMed
Search for other papers by Dominique Maiter in
Google Scholar
PubMed
Search for other papers by Orsalia Alexopoulou in
Google Scholar
PubMed
Summary
A 26-year-old woman presented with severe postpartum headaches. Magnetic resonance imaging (MRI) revealed a symmetric, heterogeneous enlargement of the pituitary gland. Three months later, she developed central diabetes insipidus. A diagnosis of postpartum hypophysitis was suspected and corticosteroids were prescribed. Six months later, the pituitary mass showed further enlargement and characteristics of a necrotic abscess with a peripheral shell and infiltration of the hypothalamus. Transsphenoidal surgery was performed, disclosing a pus-filled cavity which was drained. No bacterial growth was observed, except a single positive blood culture for Staphylococcus aureus, considered at that time as a potential contaminant. A short antibiotic course was, however, administered together with hormonal substitution for panhypopituitarism. Four months after her discharge, severe headaches recurred. Pituitary MRI was suggestive of a persistent inflammatory mass of the sellar region. She underwent a new transsphenoidal resection of a residual abscess. At that time, the sellar aspiration fluid was positive for Staphylococcus aureus and she was treated with antibiotics for 6 weeks, after which she had complete resolution of her infection. The possibility of a pituitary abscess, although rare, should be kept in mind during evaluation for a necrotic inflammatory pituitary mass with severe headaches and hormonal deficiencies.
Learning points:
-
The possibility of a pituitary abscess, although rare, should be kept in mind during evaluation for a necrotic inflammatory pituitary mass with severe headaches and hormonal deficiencies.
-
In a significant proportion of cases no pathogenic organism can be isolated.
-
A close follow-up is necessary given the risk of recurrence and the high rate of postoperative pituitary deficiencies.
