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adrenal insufficiency by Adrenoleukodystrophy

Health 2020年第1期12卷 1-13页

作者： Elda Cristinne Mattos Botelho Eduardo Romero Sampaio Botelho Luciana Correa de Souza Rodrigues Tatiana Prado Wanderley Fernanda Navarini Queiroz Pollyana Cirino Gomes Borges Sabrina Arrais Rolim Aragao Ximenes Cruz Janete Pacheco da Silva Larissa Bianca Cunha de Sa Alberto Krayyem Arbex IPEMED Medical School AFYA EducationalBrasíliaBrazil Medical Clinic in Endocrinology and Diabetology Schleswig-FlensburgGermany

The X-linked adrenoleukodystrophy (ALD) is a severe neurodegenerative disorder due to mutations in the ABCD1 gene. Objective: To report a case of a 19-year-old man with adrenal insufficiency due to adrenoleukodystrophy. Method: Case report and literature review. Result: A previously healthy 19-year-old male patient was admitted to the emergency room with nausea and vomiting for 5 days, who progressed to abdominal pain, severe asthenia, and fever (38.5°C). He referred progressive darkening of the skin, oral mucosa, tongue and nail bed of the hands and feet, observed in the last 6 years. Emergency laboratory evaluation showed severe hyponatremia and hyperkalemia, which, together with decreased plasma cortisol, directed the investigation to causes of adrenal insufficiency. High ACTH (Adrenocorticotropic hormone) and very long chain fatty acid levels closed the diagnosis. Discussion: ALD is characterized by progressive demyelination in the central and peripheral nervous system and adrenal insufficiency consequence to the accumulation of very long chain fatty acids (VLCFA) in the adrenal. The overall incidence of ALD is 1:17,000. adrenal insufficiency may be the first symptom of ALD in boys and adults. The diagnosis is based on the measurement of VLCFA plasma levels. Allogeneic bone marrow transplantation is the only treatment that provides a permanent cure when the procedure is performed at an early stage of brain demyelination, i.e. when patients are asymptomatic, although brain magnetic resonance imaging (MRI) is abnormal. Treatment of Addison’s disease is obligatory, but does not change the course of neurological symptoms.

关键词： Adrenoleukodystrophy Adrenomyeloneuropathy adrenal insufficiency Addison’s Disease

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Prolonged adrenal insufficiency after Unilateral adrenalectomy for Cortisolic Adenoma

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Open Journal of Endocrine and Metabolic Diseases 2023年第3期13卷 45-51页

作者： Fatima Akioud Farida Ajdi Ikram Damoune Departements of Endocrinology-Diabetology and Nutrition Medical School University Hospital Center Souss Massa Agadir Morocco

Background: The suppression of the hypothalamic-pituitary-adrenal axis by cortisol-secreting adrenocortical tumors is well recognized and requires peri- and postoperative hydrocortisone substitution. Case Presentation: A 48-year-old female patient with hypertension and progressive weight gain, the clinical signs of hypercorticism motivated a hormonal workup revealing an independent ACTH Cushing’s syndrome: with urinary free cortisol (UFC) at 649 nmol/24h (4× normal), adrenocorticotropin hormone (ACTH) at 1.5 ng/l. The rest of the hormonal workup was not performed due to a lack of financial means. An adrenal CT scan showed a 4 cm right adrenal adenoma. The patient underwent a right adrenalectomy with an adrenal adenoma on pathological examination. The contralateral side was normal. The patient was treated with hydrocortisone 30 mg/d for 6 weeks then 15 mg/d, during the monitoring we noted a persistence of the adrenal insufficiency for now 4 years. Basal cortisol levels during follow-up were very low (adrenal cortisol tumors are recognized by suppression, the duration of hypothalamic-pituitary-adrenal axis suppression is variable from 11 to 60 months depending on the series, which depends on the duration, severity of hypercortisolism, tumor size and other unknown factors. A longer follow-up of these patients is necessary to look for recovery of the contralateral adrenal gland.

关键词： Hypercorticism Hypothalamic-Pituitary-adrenal Axis adrenal insufficiency

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Corticosteroid-Induced adrenal insufficiency in Africa: Report of Nine Cases at the Internal Medicine/Endocrinology-Diabetology Department of Pikine NHC

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Open Journal of Endocrine and Metabolic Diseases 2020年第12期10卷 155-163页

作者： Nafy Ndiaye Ngone Diaba Diack Yakham Mohamed Leye Abdou Dieng Mamadou Ba Abdoulaye Leye Internal Medicine/Endocrinology-Diabetology Department Teaching Hospital of Pikine Dakar Senegal

Introduction: Prolonged corticosteroid therapy is labeled as the main cause of corticotropic adrenal insufficiency. However, the current frequency of this complication remains unknown. The objective of our study was to assess its epidemiologic, diagnostic, therapeutic, and evolutionary aspects of our practice. Methodology: A 60-month retrospective study was performed from May 2014 to April 2020 in the Pikine National Hospital Centre. Data were obtained from the medical records of patients with corticosteroid-induced adrenal insufficiency. Epidemiologic, diagnostic, therapeutic, and evolutionary parameters, and data related to corticotherapy were recorded. Patients with basal plasma cortisol levels Results: Medical records were obtained from nine patients, comprising six women and three men. The mean age of the patients was 48 years. Long-term corticosteroid therapy was used for self-medication in six patients and artificial bleaching in the remaining three patients. Betamethasone was the most commonly used drug in four patients, followed by clobetasol in three patients. Corticotropic insufficiency was revealed as a result of acute decompensation in five patients. All patients presented with a pseudo-cushingoid pattern. Diagnosis of corticotropic adrenal insufficiency was confirmed by measuring basal plasma cortisol levels of adrenal cris

关键词： adrenal insufficiency Corticotherapy Artificial Depigmentation Self-Medication

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Empty Sella Turcica Syndrome: About a Case Revealed by adrenal insufficiency

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Open Journal of Internal Medicine 2021年第2期11卷 68-72页

作者： Sow Maïmouna Diagne Nafissatou Ndao Awa Cheikh Thiam Mbaye Faye Atoumane Dieng Mouhamed Kane Baïdy Sy Djiba Boundia Pouye Abdoulaye Internal Medicine Department Aristide Le Dantec Teaching Hospital Dakar Senegal Radiology Department FANN Teaching Hospital Dakar Senegal

Background: Empty sella turcica corresponds to an arachnocele which may be complete or partial, primary or secondary. The primary form is rarer. Its cause is unknown, but etiological factors have been identified. The prevalence of this condition is variable, depending on techniques of diagnosis. It may be detected fortuitously on imaging or in the presence of neurological, ocular or endocrine manifestations. Aim: We aim to report a case of empty sella turcica syndrome revealed by adrenal insufficiency in a young African black woman. Case presentation: It was an overweight young woman with signs of intracranial hypertension associated with central adrenal insufficiency. The diagnosis of empty sella was confirmed by magnetic resonance imaging. Secondarily, she developed a hypo metabolism syndrome and biology confirmed central hypothyroidism. After correction of the adrenal cortical deficit, the treatment of the thyroid deficit was administered. Evolution was favourable clinically and biologically. Conclusion: The empty sella turcica is rare and its revelation by a central adrenal insufficiency is even more. The case we report is a perfect illustration.

关键词： Empty Sella Turcica adrenal insufficiency Intracranial Hypertension

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The procalcitonin-to-cortisol ratio is a potential prognostic predictor in sepsis with abdominal source:a retrospective observational study

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World Journal of Emergency Medicine 2022年第6期13卷 441-447页

作者： Hui Liu Jie Hu Jian-guo Xiao Hong-jun Kang Fei-hu Zhou Department of Critical Care Medicine the First Medical CenterChinese PLA General HospitalBeijing 100853China

BACKGROUND:The aim of the study was to investigate the procalcitonin-to-cortisol ratio(P/C ratio)as a prognostic predictor among septic patients with abdominal ***:We retrospectively enrolled 132 post-surgery patients between 18 and 90 years old with sepsis of the abdominal *** the second day of sepsis onset,cortisol,procalcitonin(PCT),Acute Physiology and Chronic Health Evaluation(APACHE)II score,Sequential Organ Failure Assessment(SOFA)score,C-response protein(CRP),and other baseline characteristics were *** addition,the length of ICU stay,length of mechanical ventilation(MV)days,length of shock days,and 28-day mortality were also *** analysis was performed to screen potential risk *** analysis was used to identify the interaction among the risk *** analysis was also utilized to demonstrate the relationship between the risk factors and *** receiver operator characteristic(ROC)curve analysis was conducted to evaluate the risk factors.A restricted cubic spline(RCS)demonstrated the association between survival outcome and the P/C ratio ***:A total of twenty-nine patients died,and 103 patients survived within 28 *** were significant differences in cortisol,PCT,P/C ratio,interleukin(IL)-6,SOFA,and APACHE II scores between the survival and non-survival *** significant interaction was observed in the stratified *** regression analysis revealed that P/C ratio(P=0.033)was significantly related to 28-day *** on ROC curves,P/C ratio(AUC=0.919)had a higher AUC value than cortisol or *** analysis depicted a positive relationship between survival possibility and P/C ratio ***:P/C ratio might be a potential prognostic predictor in septic patients with abdominal sources.

关键词： Sepsis Cortisol Procalcitonin Procalcitonin-to-cortisol ratio adrenal insufficiency

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Congenital lipoid adrenal hyperplasia with Graves'disease:A case report

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World Journal of Clinical Cases 2022年第26期10卷 9390-9397页

作者： Yan-Jun Wang Cong Liu Chuan Xing Le Zhang Wan-Feng Xu Hai-Ying Wang Fu-Tao Wang Department of Endocrinology Shengjing Hospital of China Medical UniversityShenyang 110004Liaoning ProvinceChina Department of Endocrinology the Second Hospital of ChaoyangChaoyang 110000Liaoning ProvinceChina Department of Endocrinology Changchun Central HospitalChangchun 110000Jilin ProvinceChina

BACKGROUND Congenital adrenal hyperplasia(CAH),which is caused by a mutation of the steroidogenic acute regulatory(StAR)*** patients are usually characterized by adrenal insufficiency in the first year of life,salt loss,glucocorticoid and mineralocorticoid deficiency,and female external genitalia,regardless of chromosomal *** with non-classical lipoid CAH usually develop glucocorticoid deficiency and mild mineralocorticoid deficiency at 2-4 years of *** SUMMARY Herein,We report the case of a woman with non-classic lipoid CAH combined with Graves’*** chromosome karyotype was 46,XX,and highthroughput sequencing revealed two missense variants in the StAR gene:c.229C>T(p.Q77X)and c.814C>T(p.R272C),which were inherited from both parents(non-close relatives).The patient was treated for Graves’disease in a timely manner and the dosage of glucocorticoid was adjusted during the treatment of Graves’*** This is the first case of non-classic lipoid CAH combined with Graves’disease reported in the Chinese *** addition to conventional glucocorticoid replacement therapy,timely adjustments were made to the dosages of thyroid hormone and glucocorticoid to avoid adrenal crisis as a consequence of the increased demand and accelerated metabolism of glucocorticoids when the patient was diagnosed with Graves’disease.

关键词： Lipoid congenital adrenal hyperplasia Steroidogenic acute regulatory protein adrenal insufficiency Graves’disease Case report

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Relationship between adrenal function and prognosis in patients with severe sepsis

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Chinese Medical Journal 2007年第18期120卷 1578-1582页

作者： YANG Yi LIU Ling ZHAO Bo LI Mao-qin WU Bin YAN Zheng GU Qin SUN Hua QIU Hai-bo Department of Critical Care Medicine Zhong-Da Hospital andSchool of Clinical Medicine Southeast University Nanjing210009 China Department of Critical Care Medicine Forth Hospital of Xuzhouand School of Clinical Medicine Southeast University Xuzhou221006 China Department of Critical Care Medicine Third Hospital of Bengbuand School of Clinical Medicine Southeast University Bengbu233000 China Department of Critical Care Medicine First Hospital of Wuxi_Nanjing Medical University Wuxi 214002 China Department of Critical Care Medicine Gulou Hospital and Schoolof Clinical Medicine Nanjing University Nanjing 210008 China DepatZtment of Critical Care Medicine Second Affiliated Hospitalof Nantong Medical College Nantong 226001 China

Background It is known that the hypothalamic-pituitary-adrenal （HPA） axis is highlighted by stimulation, such as sepsis, trauma, etc, when corticortropin increases and plasma cortisol levels enhance. Relative adrenal insufficiency is not uncommon in critically ill patients and may occur in severe sepsis patients with high plasma cortisol levels. It has been demonstrated that a short corticotropin test has a good prognostic value and is helpful in identifying patients with septic shock at high risk for death, but it has not been established for all severe sepsis patients, especially in China. The aim of this study is to explore the relationship between adrenal function and prognosis in patients with severe sepsis. Methods This prospective study was conducted between July and December 2004 in 6 teaching hospitals. Two hundred and forty patients with severe sepsis were enrolled in this study. A short corticotropin stimulation test was performed in all patients by intravenous injection of 250 pg of corticotropin. Blood samples were taken immediately before the test （TO）, 30 （T30） and 60 （T60） minutes afterward, and the plasma cortisol concentration was measured by radio-immunoassay. At the onset of severe sepsis, the following parameters were recorded： age, sex, Acute Physiology and Chronic Health Evaluation （APACHE） II, heart rate, mean arterial pressure （MAP）, arterial partial pressure of oxygen （PaO2）/fraction of inspired oxygen （FiO2）, peripheral blood of hemoglobin, platelets and leukocyte concentration and the number of organ failure. Patients were designated into two groups （survival and non-survival groups） according to the 28-day mortality. Relative adrenal insufficiency was defined as the difference between TO and the highest value of T30 or T60 （△Tmax） ≤9 μg/dl. Results （1） Two hundred and forty patients with severe sepsis were included in this study, with 134 patients in the survival group and 106 in the non-survival group. The 28-day mortality was 44.2%

关键词： severe sepsis prognosis cortisol corticotropin adrenal insufficiency

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Refractory Arterial Hypotension in a Patient with COVID-19: Could the Hypothalamic-Pituitary-adrenal Axis Be Involved? Case Report and Mini Review

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Advances in Infectious Diseases 2020年第3期10卷 160-167页

作者： Francisco E. Finamor Luciana P. Finamor Frederico J. N. Mancuso Jaquelina S. Ota-Arakaki Marcelo R. S. Moraes Cristina Muccioli Nancy Bellei Discipline of Emergency Medicine Department of Medicine Federal University of S&#227 o Paulo S&#227 o Paulo Brazil Clinical Research Sector Department of Ophthalmology Federal University of S&#227 o Paulo S&#227 o Paulo Brazil Discipline of Pneumology Department of Medicine Federal University of S&#227 o Paulo S&#227 o Paulo Brazil Discipline of Infectious Diseases Federal University of S&#227 o Paulo S&#227 o Paulo Brazil

Neuro-endocrinological manifestations rarely are described in cases of SARS- CoV-2. We describe a case of a previously hypertensive patient who presented COVID-19 and developed refractory arterial hypotension. In the investigation, low levels of ACTH and cortisol were observed, suggesting secondary adrenal insufficiency as the cause of refractory hypotension.

关键词： ACTH Hypocortisolism COVID-19 adrenal insufficiency Arterial Hypotension

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Early Identification and Diagnosis of adrenal Crisis after Retroperitoneal Laparoscopic Unilateral adrenalectomy

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Journal of Endocrinology Research 2020年第2期2卷 22-26页

作者： Li Wan Yong Wang Shubin Wang Jingzhao Cao Zhengjin Yi Xiangyu Liu Chuan Xiao Yun Luo Xupan Wei Department of Urology Pangang Group General HospitalSichuan617023China

The occurrence of adrenal crisis after retroperitoneal laparoscopic unilateral adrenalectomy is usually *** not timely diagnosis and treatment,it may cause shock,and even lead to *** is very difficult to distinguish the clinical manifestations of adrenal crisis from nausea,vomiting,fatigue,gas separation from the lower diaphragm,abdominal pain,hypotension,hypertension,fever and hypothermia after *** makes it very difficult to identify and diagnose adrenal crisis *** article mainly discusses the early recognition,diagnosis and treatment of adrenal crisis after unilateral adrenalectomy by retroperitoneoscope.

关键词： Retroperitoneoscope Unilateral adrenalectomy adrenal crisis adrenal insufficiency Shock Early recognition

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