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      Corticotroph adenoma and pituitary fungal infection: a rare association

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      1 , 1 , 1 , 1
      Endocrinology, Diabetes & Metabolism Case Reports
      Bioscientifica Ltd
      Adult, Female, White, Portugal, Pituitary, Pituitary, ACTH, Prolactin, Thyroxine (T4), TSH, LH, FSH, GH, Cortisol, IGF1, Oestradiol (E2), Corticotrophic adenoma, Pituitary apoplexy, Diabetes mellitus type 2, Pituitary adenoma, Hypopituitarism, Cushing's syndrome, Headache, Ptosis, Agitation, Anisocoria, Fatigue, Hypercortisolaemia, Diabetes mellitus type 2, Hypertension, Hypopituitarism, CT scan, Histopathology, Haemoglobin A1c, Blood pressure, Prolactin, TSH, ACTH, FT4, LH, FSH, GH, MRI, Cortisol (serum), IGF1, Groccots methenamine stain*, Immunohistochemistry, Haematoxylin and eosin staining, Oestradiol (E2), Transsphenoidal surgery, Resection of tumour, Spironolactone, Metformin, Gliclazide, Hydrocortisone, Glucocorticoids, Thyroxine (T4), Acyclovir*, Sitagliptin, DPP4 inhibitors, Insulin glargine, Infectiology, Unique/unexpected symptoms or presentations of a disease, March, 2020

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          Abstract

          Summary

          Pituitary infections, particularly with fungus, are rare disorders that usually occur in immunocompromised patients. Cushing’s syndrome predisposes patients to infectious diseases due to their immunosuppression status. We report the case of a 55-year-old woman, working as a poultry farmer, who developed intense headache, palpebral ptosis, anisocoria, prostration and psychomotor agitation 9 months after initial diabetes mellitus diagnosis. Cranioencephalic CT scan showed a pituitary lesion with bleeding, suggesting pituitary apoplexy. Patient underwent transsphenoidal surgery and the neuropathologic study indicated a corticotroph adenoma with apoplexy and fungal infection. Patient had no preoperative Cushing’s syndrome diagnosis. She was evaluated by a multidisciplinary team who decided not to administer anti-fungal treatment. The reported case shows a rare association between a corticotroph adenoma and a pituitary fungal infection. The possible contributing factors were hypercortisolism, uncontrolled diabetes and professional activity. Transsphenoidal surgery is advocated in these infections; however, anti-fungal therapy is still controversial.

          Learning points:
          • Pituitary infections are rare disorders caused by bacterial, viral, fungal and parasitic infections.

          • Pituitary fungal infections usually occur in immunocompromised patients.

          • Cushing’s syndrome, as immunosuppression factor, predisposes patients to infectious diseases, including fungal infections.

          • Diagnosis of pituitary fungal infection is often achieved during histopathological investigation.

          • Treatment with systemic anti-fungal drugs is controversial.

          • Endocrine evaluation is recommended at the time of initial presentation of pituitary manifestations.

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          Most cited references7

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          Opportunistic infections in endogenous Cushing's syndrome.

          The cases of 6 patients with endogenous Cushing's syndrome and opportunistic infections were studied, and compared with those of 17 similar patients reported in the literature. Cushing's syndrome was caused by ectopic adrenocorticotrophic hormone production or adrenal tumors in most patients, and hypercortisolism was extreme. Four infectious processes were preponderant: Cryptococcosis, aspergillosis, nocardiosis, or Pneumocystis carinii pneumonia occurred in 21 patients. Signs and symptoms of infection were often masked by the hypercortisolism. Morning plasma cortisol levels correlated with the infection type (rank-order Spearman correlation coefficient = 0.78, p less than 0.01): Levels of less than 70 micrograms/dL or greater than 121 micrograms/dL were associated with cryptococcosis or pneumocystis, respectively, by discriminant analysis. Of the 9 patients who survived their infection, 8 had evidence that cortisol production was reduced to near normal. In contrast, all 14 patients died in whom cortisol production went uncontrolled. In patients with hypercortisolism from endogenous Cushing's syndrome (especially of nonpituitary origin), opportunistic infections should be anticipated and prompt control of cortisol overproduction should be initiated.
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            Cushing’s syndrome complicated by multiple opportunistic infections

            The case history of a 56-year-old man is described who suffered from severe adrenocorticotrophic hormone (ACTH)-dependent Cushing’s syndrome. The clinical course was complicated by simultaneous infections with Pneumocystis carinii, Staphylococcus aureus, Candida albicans, Aspergillus fumigatus and Herpes simplex, which proved to be fatal. A study of the literature shows that opportunistic infections in endogenous Cushing’s syndrome are associated with severe cortisol excess and carry a high mortality. Opportunistic infections are most prevalent in the ectopic ACTH syndrome, explained by the very high plasma cortisol concentrations in this condition. Infections with Aspergillus species, Cryptoccus neoformans, Pneumocystis carinii and Nocardia asteroides predominated. Cushing’s syndrome with a very high plasma cortisol concentration causes a severe immunocompromized state. Prompt evaluation of the cause of the hypercortisolism, initiation of cortisol lowering therapy, primary prophylaxis for Pneumocystis carinii infection when plasma cortisol exceeds 2500 nmol L−1 and a search for concomitant infectious disease is recommended.
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              DIAGNOSIS OF ENDOCRINE DISEASE: Expanding the cause of hypopituitarism.

              Hypopituitarism is defined as one or more pituitary hormone deficits due to a lesion in the hypothalamic-pituitary region. By far, the most common cause of hypopituitarism associated with a sellar mass is a pituitary adenoma. A high index of suspicion is required for diagnosing hypopituitarism in several other conditions such as other massess in the sellar and parasellar region, brain damage caused by radiation and by traumatic brain injury, vascular lesions, infiltrative/immunological/inflammatory diseases (lymphocytic hypophysitis, sarcoidosis and hemochromatosis), infectious diseases and genetic disorders. Hypopituitarism may be permanent and progressive with sequential pattern of hormone deficiencies (radiation-induced hypopituitarism) or transient after traumatic brain injury with possible recovery occurring years from the initial event. In recent years, there is increased reporting of less common and less reported causes of hypopituitarism with its delayed diagnosis. The aim of this review is to summarize the published data and to allow earlier identification of populations at risk of hypopituitarism as optimal hormonal replacement may significantly improve their quality of life and life expectancy.

                Author and article information

                Journal
                Endocrinol Diabetes Metab Case Rep
                Endocrinol Diabetes Metab Case Rep
                EDM
                Endocrinology, Diabetes & Metabolism Case Reports
                Bioscientifica Ltd (Bristol )
                2052-0573
                25 March 2020
                2020
                : 2020
                : 20-0010
                Affiliations
                [1 ]Endocrinology , Diabetes and Metabolism Department, Centro Hospitalar e Universitário de Coimbra EPE, Coimbra, Portugal
                Author notes
                Correspondence should be addressed to D Catarino; Email: diana_catarino@ 123456hotmail.com
                Author information
                http://orcid.org/0000-0002-0158-3952
                Article
                EDM200010
                10.1530/EDM-20-0010
                7159253
                32213650
                662cffd2-586d-4bea-83fa-e1745abae50e
                © 2020 The authors

                This work is licensed under a Creative Commons Attribution-NonCommercial-NoDerivatives 4.0 International License..

                History
                : 28 February 2020
                : 04 March 2020
                Categories
                Adult
                Female
                White
                Portugal
                Pituitary
                Pituitary
                ACTH
                Prolactin
                Thyroxine (T4)
                TSH
                LH
                FSH
                GH
                Cortisol
                IGF1
                Oestradiol (E2)
                Corticotrophic adenoma
                Pituitary apoplexy
                Diabetes mellitus type 2
                Pituitary adenoma
                Hypopituitarism
                Cushing's syndrome
                Headache
                Ptosis
                Agitation
                Anisocoria
                Fatigue
                Hypercortisolaemia
                Diabetes mellitus type 2
                Hypertension
                Hypopituitarism
                CT scan
                Histopathology
                Haemoglobin A1c
                Blood pressure
                Prolactin
                TSH
                ACTH
                FT4
                LH
                FSH
                GH
                MRI
                Cortisol (serum)
                IGF1
                Groccots methenamine stain*
                Immunohistochemistry
                Haematoxylin and eosin staining
                Oestradiol (E2)
                Transsphenoidal surgery
                Resection of tumour
                Spironolactone
                Metformin
                Gliclazide
                Hydrocortisone
                Glucocorticoids
                Thyroxine (T4)
                Acyclovir*
                Sitagliptin
                DPP4 inhibitors
                Insulin glargine
                Infectiology
                Unique/Unexpected Symptoms or Presentations of a Disease
                Unique/Unexpected Symptoms or Presentations of a Disease

                adult,female,white,portugal,pituitary,acth,prolactin,thyroxine (t4),tsh,lh,fsh,gh,cortisol,igf1,oestradiol (e2),corticotrophic adenoma,pituitary apoplexy,diabetes mellitus type 2,pituitary adenoma,hypopituitarism,cushing's syndrome,headache,ptosis,agitation,anisocoria,fatigue,hypercortisolaemia,hypertension,ct scan,histopathology,haemoglobin a1c,blood pressure,ft4,mri,cortisol (serum),groccots methenamine stain*,immunohistochemistry,haematoxylin and eosin staining,transsphenoidal surgery,resection of tumour,spironolactone,metformin,gliclazide,hydrocortisone,glucocorticoids,acyclovir*,sitagliptin,dpp4 inhibitors,insulin glargine,infectiology,unique/unexpected symptoms or presentations of a disease,march,2020

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