scholarly journals Postoperative expression of Cushing disease in a young male: metamorphosis of silent corticotroph adenoma?

Author(s):  
Sharmin Jahan ◽  
M A Hasanat ◽  
Tahseen Mahmood ◽  
Shahed Morshed ◽  
Raziul Haq ◽  
...  

Summary Silent corticotroph adenoma (SCA) is an unusual type of nonfunctioning pituitary adenoma (NFA) that is silent both clinically and biochemically and can only be recognized by positive immunostaining for ACTH. Under rare circumstances, it can transform into hormonally active disease presenting with severe Cushing syndrome. It might often produce diagnostic dilemma with difficult management issue if not thoroughly investigated and subtyped accordingly following surgery. Here, we present a 21-year-old male who initially underwent pituitary adenomectomy for presumed NFA with compressive symptoms. However, he developed recurrent and invasive macroadenoma with severe clinical as well as biochemical hypercortisolism during post-surgical follow-up. Repeat pituitary surgery was carried out urgently as there was significant optic chiasmal compression. Immunohistochemical analysis of the tumor tissue obtained on repeat surgery proved it to be an aggressive corticotroph adenoma. Though not cured, he showed marked clinical and biochemical improvement in the immediate postoperative period. Anticipating recurrence from the residual tumor, we referred him for cyber knife radio surgery. Learning points: Pituitary NFA commonly present with compressive symptoms such as headache and blurred vision. Post-surgical development of Cushing syndrome in such a case could be either drug induced or endogenous. In the presence of recurrent pituitary tumor, ACTH-dependent Cushing syndrome indicates CD. Rarely a SCA presenting initially as NFA can transform into an active corticotroph adenoma. Immunohistochemical marker for ACTH in the resected tumor confirms the diagnosis.

Author(s):  
R. B. Chavan ◽  
V. A. Belgaumkar ◽  
N. S. Deshmukh ◽  
A. M. Pawar

<p>Systemic lupus erythematosus (SLE) is an autoimmune disorder causing microvascular inflammation with generation of antinuclear auto-antibodies. It can have varied presentations and its coexistence with other disorders makes diagnosis and management all the more challenging. We describe a young male, known case of SLE and pulmonary tuberculosis on anti-tubercular treatment presenting with sudden onset diffuse maculopapular dusky rash, oral lesions, fever, joint pain and photosensitivity. Positive Nikolsky’s sign on clinical examination, epidermal necrosis on histopathology, negative direct immunofluorescence and Naranjo’s causality assessment clinched the diagnosis of streptomycin-induced toxic epidermal necrolysis (TEN) in SLE. Rash responded rapidly to systemic steroids and discontinuation of anti-tubercular drugs. Rifampicin, ethambutol and isoniazid have been previously incriminated in TEN but streptomycin- induced toxic epidermal necrolysis remains an extremely rare event.<strong> </strong>TEN like rash of lupus is a rare entity clinically indistinguishable from drug induced TEN. Moreover, TEN is known to occur with increased frequency in connective tissue disorders.</p>


Author(s):  
Soler Guillermo Serra ◽  
Barceló Carlos Antich ◽  
Cubas Javier Bodoque ◽  
Fernández Honorato García ◽  
Bonet Antonio Mas ◽  
...  

2021 ◽  
Vol 13 (1) ◽  
pp. 233-238
Author(s):  
Seyed Mohammad Mousavi Mirzaei ◽  
Zahra Ahmadi

Drug-induced aseptic meningitis (DIAM) is a rare complication of certain drugs, most commonly reported with ibuprofen use. The present study reports on a male adolescent with intracranial hypertension and visual impairment accompanied by DIAM. We present a 16-year-old male patient who after ibuprofen consumption displayed headache, fever, photophobia, and blurred vision following heavy exercises. Examination of cerebrospinal fluid showed a mononuclear pleocytosis and an increase in protein concentration. Other examinations had normal results. The development of common clinical signs following ibuprofen use reflected DIAM. The patient’s vision was found to improve with supportive care and stopping of the drug during follow-up. Given the widespread use of nonsteroidal anti-inflammatory drugs and the fact that these drugs are the most common cause of DIAM, the probability of occurrence of this event should be always kept in mind, and screening for autoimmune diseases in these patients is of great importance.


2021 ◽  
Vol 14 ◽  
pp. 117955142199410
Author(s):  
Alice Y Chang ◽  
Sasan Mirfakhraee ◽  
Elizabeth E King ◽  
Jennifer U Mercado ◽  
Diane M Donegan ◽  
...  

Establishing a definitive diagnosis of Cushing disease (CD), given its clinical and biochemical heterogeneity, initiating effective treatment to control the effects of hypercortisolism, and managing recurrence are challenging disease aspects to address. Mifepristone is a competitive glucocorticoid receptor antagonist that is approved in the US by the Food and Drug Administration to control hyperglycemia secondary to endogenous hypercortisolism (Cushing syndrome) in patients who have glucose intolerance or type 2 diabetes mellitus and have failed surgery or are not candidates for surgery. Herein, we describe 6 patients with CD who received mifepristone as adjunct/bridge therapy in the following clinical settings: to assess clinical benefits of treatment for suspected recurrent disease, to control hypercortisolism preoperatively for severe disease, to control hypercortisolism during the COVID-19 pandemic, and to provide adjunctive treatment to radiation therapy. The patients were treated at multiple medical practice settings. Mifepristone treatment in each of the described cases was associated with clinical improvements, including improvements in overall glycemia, hypertension, and weight loss. In addition, in one case where biochemical and radiological evidence of disease recurrence was uncertain, clinical improvement with mifepristone pointed toward likely disease recurrence. Adverse events associated with mifepristone reported in the 6 cases were consistent with those previously reported in the pivotal trial and included cortisol withdrawal symptoms, antiprogesterone effects (vaginal bleeding), hypothyroidism (treated with levothyroxine), and hypokalemia (treated with spironolactone). These cases show how mifepristone can potentially be utilized as a therapeutic trial in equivocal cases of CD recurrence; as a presurgical treatment strategy, particularly during the COVID-19 pandemic; and as bridge therapy, while awaiting the effects of radiation.


2017 ◽  
Author(s):  
Naomi D.L. Fisher ◽  
Gail K Adler

The secondary causes of hypertension are associated with the excess of the principal hormones produced by the adrenal glands: cortisol, epinephrine, and aldosterone. Excess aldosterone production is recognized as primary hyperaldosteronism, or primary aldosteronism (PA). Individuals with PA are at increased risk for a variety of disorders, including atrial fibrillation, coronary artery disease, myocardial infarction, and stroke. Pheochromocytoma is a very rare tumor (accounting for fewer than one in 10,000 hypertension cases) and is marked by high secretions of catecholamines, mostly epinephrine as well as norepinephrine. Cushing disease and Cushing syndrome are addressed in a separate review. This review contains 5 highly rendered figures, 4 tables, and 39 references.


Author(s):  
Matthew Seymour ◽  
Thomas Robertson ◽  
Jason Papacostas ◽  
Kirk Morris ◽  
Jennifer Gillespie ◽  
...  

Summary A 34-year-old woman presented 18 months post-partum with blurred vision, polyuria, amenorrhoea, headache and general malaise. Comprehensive clinical examination showed left superior temporal visual loss only. Initial investigations revealed panhypopituitarism and MRI demonstrated a sellar mass involving the infundibulum and hypothalamus. Lymphocytic hypophysitis was suspected and high dose glucocorticoids were commenced along with desmopressin and thyroxine. However, her vision rapidly deteriorated. At surgical biopsy, an irresectable grey amorphous mass involving the optic chiasm was identified. Histopathology was initially reported as granulomatous hypophysitis. Despite the ongoing treatment with glucocorticoids, her vision worsened to light detection only. Histopathological review revised the diagnosis to partially treated lymphoma. A PET scan demonstrated avid uptake in the pituitary gland in addition to splenic involvement, lymphadenopathy above and below the diaphragm, and a bone lesion. Excisional node biopsy of an impalpable infraclavicular lymph node confirmed nodular lymphocyte-predominant Hodgkin lymphoma. Hyper-CVAD chemotherapy was commenced, along with rituximab; fluid-balance management during chemotherapy (with its requisite large fluid volumes) was extremely complex given her diabetes insipidus. The patient is now in clinical remission. Panhypopituitarism persists; however, her vision has recovered sufficiently for reading large print and driving. To the best of our knowledge, this is the first reported case of Hodgkin lymphoma presenting initially as hypopituitarism. Learning points Lymphoma involving the pituitary is exceedingly rare and, to the best of our knowledge, this is the first reported case of nodular lymphocyte-predominant Hodgkin lymphoma presenting as hypopituitarism. There are myriad causes of a sellar mass and this case highlights the importance of reconsidering the diagnosis when patients fail to respond as expected to appropriate therapeutic intervention. This case highlights the difficulties associated with managing panhypopituitary patients receiving chemotherapy, particularly when this involves large volumes of i.v. hydration fluid.


2019 ◽  
Vol 62 (1) ◽  
pp. 114-122 ◽  
Author(s):  
Junhyung Kim ◽  
Seon Jin Yoon ◽  
Ju Hyung Moon ◽  
Cheol Ryong Ku ◽  
Se Hoon Kim ◽  
...  

2018 ◽  
Vol 4 (3) ◽  
Author(s):  
Salman Yahya ◽  
Sonia Zafar ◽  
Hafsa S. Babar

Diaphragmatic hernia post esophagectomy is a rare complication but a reality in its existence. It is typically difficult to diagnose but highly depends on keeping high index of suspicion. In our case report, the young male who underwent esophagectomy for esophageal carcinoma, remained disease free and stable in his 9 months follow up, suddenly presented in the emergency department with the symptoms of shortness of breath, chest pain, vomiting and tachycardia. The case was typically complicated by the initial treatment given for acute ischemia and cardiogenic shock. Radiological findings proved to be helpful and turning point in the diagnosis and overall management.


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