Severe Ectopic Cushing Syndrome Caused by Adenoid Cystic Carcinoma of a Salivary Gland |
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| Institution: | 1. Endocrinology Department, EDUAB-HSP Hospital de la Santa Creu I Sant Pau, Barcelona, Spain;2. Clinical Biochemistry Department, EDUAB-HSP Hospital de la Santa Creu I Sant Pau, Barcelona, Spain;3. Pathology Department, EDUAB-HSP Hospital de la Santa Creu I Sant Pau, Barcelona, Spain;4. CIBER-BBN, EDUAB-HSP Hospital de la Santa Creu I Sant Pau, Barcelona, Spain;1. Department of Endocrinology, Tan Tock Seng Hospital, Singapore;2. Yong Loo Lin School of Medicine, Singapore;3. Duke-NUS Graduate Medical School, Singapore;4. Brenner Centre for Molecular Medicine, National University of Singapore, Singapore;1. Internal Medicine Department, Carlos Haya Hospital, Malaga, Spain;2. CIBER Fisiopatologia de la Obesidad y la Nutricion (CB06/003), Malaga, Spain;3. Preventive Medicine Department, Malaga University, Malaga, Spain;4. Health Center “Ciudad Jardin,” Malaga, Spain;5. Research Laboratory, Internal Medicine Department, Carlos Haya Hospital, Malaga, Spain;6. Endocrinology and Nutrition Department, Virgen de la Victoria Hospital, Malaga, Spain;7. Biomedical Research Laboratory, Endocrinology and Nutrition Department, Virgen de la Victoria Hospital, Malaga, Spain;1. Division of Endocrinology, Department of Medicine, College of Physicians and Surgeons, Columbia University, New York, New York;2. Department of Surgery, College of Physicians and Surgeons, Columbia University, New York, New York;3. Division of Pediatric Otolaryngology, The Children’s Hospital of Philadelphia, Philadelphia, Pennsylvania;4. Department of Radiology, Jacobi Medical Center, Bronx, New York. |
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| Abstract: | ObjectiveWe present a rare case of Cushing syndrome due to ectopic adrenocorticotropic hormone (ACTH) secretion (EAS). To our knowledge only two similar cases have been previously reported.MethodsThis is a case report of EAS by a metastatic lingual adenoid cystic carcinoma (ACC).ResultsThe patient was diagnosed of a Cushing syndrome caused by tumoral EAS two years after initial cancer diagnosis. Clinical presentation included asthenia, insomnia, hypertension, acne, and hyperpigmentation developing in a period of two months. Laboratory and imaging testing revealed hypokalemic metabolic alkalosis, hypercortisole- mia, high ACTH, nonsuppresion by 8 mg dexamethasone, and a normal pituitary magnetic resonance imaging (MRI). With a high clinical suspicion of EAS, combined medical treatment was started but was unsuccessful. Bilateral adrenalectomy could not be performed given the patient’s rapid deterioration. Immunostained tissue from the original tumor was positive for synaptophysin.ConclusionThis rare case of EAS illustrates the challenge that this condition may confer regarding diagnosis and management. (Endocr. Pract. 2013;19:e118-e121) |
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