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Jia XY, Guo XP, Yao Y, Deng K, Lian W, Xing B. Surgical management of pituitary adenoma during pregnancy. World J Clin Cases 2023; 11:2694-2707. [PMID: 37214566 PMCID: PMC10198099 DOI: 10.12998/wjcc.v11.i12.2694] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 11/22/2022] [Revised: 03/01/2023] [Accepted: 03/24/2023] [Indexed: 04/25/2023] Open
Abstract
BACKGROUND Although conservative treatment is typically recommended for pregnant patients with pituitary adenoma (PA), surgical treatment is occasionally necessary for those with acute symptoms. Currently, surgical interventions utilized among these patients is poorly studied.
AIM To evaluate the surgical indications, timing, perioperative precautions and postoperative complications of PAs during pregnancy and to provide comprehensive guidance.
METHODS Six patients with PAs who underwent surgical treatment during pregnancy at Peking Union Medical College Hospital between January 1990 and June 2021 were recruited for this study. Another 35 pregnant patients who were profiled in the literature were included in our analysis.
RESULTS The 41 enrolled patients had acute symptoms including visual field defects, severe headaches or vision loss that required emergency pituitary surgeries. PA apoplexies were found in 23 patients. The majority of patients (55.9%) underwent surgery in the second trimester of pregnancy. A multidisciplinary team was involved in patient care from the preoperative period through the postpartum period. With the exception of 1 patient who underwent an induced abortion and 1 fetus that died due to a nuchal cord, 39 patients delivered successfully. Among them, 37 fetuses were healthy until the most recent follow-up.
CONCLUSION PA surgery during pregnancy is effective and safe during the second and third trimesters. Pregnant patients requiring emergency PA surgery require multidisciplinary evaluation and healthcare management.
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Affiliation(s)
- Xin-Yu Jia
- Department of Plastic Surgery, Plastic Surgery Hospital, Chinese Academy of Medical Sciences and Peking Union Medical College, Beijing 100144, China
- Department of Neurosurgery, Key Laboratory of Endocrinology of Ministry of Health, China Pituitary Adenoma Specialist Council, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences and Peking Union Medical College Hospital, Beijing 100730, China
- Department of Plastic Surgery, Chinese Academy of Medical Sciences and Peking Union Medical College, Beijing 100730, China
| | - Xiao-Peng Guo
- Department of Neurosurgery, Key Laboratory of Endocrinology of Ministry of Health, China Pituitary Adenoma Specialist Council, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences and Peking Union Medical College Hospital, Beijing 100730, China
- Department of Neurosurgery, Medical Research Center, State Key Laboratory of Complex Severe and Rare Diseases, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences and Peking Union Medical College Hospital, Beijing 100730, China
| | - Yong Yao
- Department of Neurosurgery, Key Laboratory of Endocrinology of Ministry of Health, China Pituitary Adenoma Specialist Council, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences and Peking Union Medical College Hospital, Beijing 100730, China
- Department of Neurosurgery, Medical Research Center, State Key Laboratory of Complex Severe and Rare Diseases, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences and Peking Union Medical College Hospital, Beijing 100730, China
| | - Kan Deng
- Department of Neurosurgery, Key Laboratory of Endocrinology of Ministry of Health, China Pituitary Adenoma Specialist Council, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences and Peking Union Medical College Hospital, Beijing 100730, China
- Department of Neurosurgery, Medical Research Center, State Key Laboratory of Complex Severe and Rare Diseases, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences and Peking Union Medical College Hospital, Beijing 100730, China
| | - Wei Lian
- Department of Neurosurgery, Key Laboratory of Endocrinology of Ministry of Health, China Pituitary Adenoma Specialist Council, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences and Peking Union Medical College Hospital, Beijing 100730, China
- Department of Neurosurgery, Medical Research Center, State Key Laboratory of Complex Severe and Rare Diseases, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences and Peking Union Medical College Hospital, Beijing 100730, China
| | - Bing Xing
- Department of Neurosurgery, Key Laboratory of Endocrinology of Ministry of Health, China Pituitary Adenoma Specialist Council, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences and Peking Union Medical College Hospital, Beijing 100730, China
- Department of Neurosurgery, Medical Research Center, State Key Laboratory of Complex Severe and Rare Diseases, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences and Peking Union Medical College Hospital, Beijing 100730, China
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Hamblin R, Coulden A, Fountas A, Karavitaki N. The diagnosis and management of Cushing's syndrome in pregnancy. J Neuroendocrinol 2022; 34:e13118. [PMID: 35491087 PMCID: PMC9541401 DOI: 10.1111/jne.13118] [Citation(s) in RCA: 31] [Impact Index Per Article: 10.3] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 02/13/2022] [Revised: 03/01/2022] [Accepted: 03/05/2022] [Indexed: 11/28/2022]
Abstract
Endogenous Cushing's syndrome (CS) is rarely encountered during pregnancy. Clinical and biochemical changes in healthy pregnancy overlap with those seen in pregnancy complicated by CS; the diagnosis is therefore challenging and can be delayed. During normal gestation, adrenocorticotrophic hormone, corticotrophin-releasing hormone, cortisol, and urinary free cortisol levels rise. Dexamethasone administration fails to fully suppress cortisol in pregnant women without CS. Localisation may be hindered by non-suppressed adrenocorticotrophic hormone levels in a large proportion of those with adrenal CS; smaller corticotroph adenomas may go undetected as a result of a lack of contrast administration or the presence of pituitary hyperplasia; and inferior petrosal sinus sampling is not recommended given the risk of radiation and thrombosis. Yet, diagnosis is essential; active disease is associated with multiple insults to both maternal and foetal health, and those cured may normalise the risk of maternal-foetal complications. The published literature consists mostly of case reports or small case series affected by publication bias, heterogeneous definitions of maternal or foetal outcomes or lack of detail on severity of hypercortisolism. Consequently, conclusive recommendations, or a standardised management approach for all, cannot be made. Management is highly individualised: the decision for surgery, medical control of hypercortisolism or adoption of a conservative approach is dependent on the timing of diagnosis (respective to stage of gestation), the ability to localise the tumour, severity of CS, pre-existing maternal comorbidity, and, ultimately, patient choice. Close communication is a necessity with the patient placed at the centre of all decisions, with risks, benefits, and uncertainties around any investigation and management carefully discussed. Care should be delivered by an experienced, multidisciplinary team, with the resources and expertise available to manage such a rare and challenging condition during pregnancy.
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Affiliation(s)
- Ross Hamblin
- Institute of Metabolism and Systems Research, College of Medical and Dental SciencesUniversity of BirminghamBirminghamUK
- Centre for Endocrinology, Diabetes and MetabolismBirmingham Health PartnersBirminghamUK
- Department of Endocrinology, Queen Elizabeth HospitalUniversity Hospitals Birmingham NHS Foundation TrustBirminghamUK
| | - Amy Coulden
- Institute of Metabolism and Systems Research, College of Medical and Dental SciencesUniversity of BirminghamBirminghamUK
- Centre for Endocrinology, Diabetes and MetabolismBirmingham Health PartnersBirminghamUK
- Department of Endocrinology, Queen Elizabeth HospitalUniversity Hospitals Birmingham NHS Foundation TrustBirminghamUK
| | - Athanasios Fountas
- Institute of Metabolism and Systems Research, College of Medical and Dental SciencesUniversity of BirminghamBirminghamUK
- Centre for Endocrinology, Diabetes and MetabolismBirmingham Health PartnersBirminghamUK
- Department of Endocrinology, Queen Elizabeth HospitalUniversity Hospitals Birmingham NHS Foundation TrustBirminghamUK
| | - Niki Karavitaki
- Institute of Metabolism and Systems Research, College of Medical and Dental SciencesUniversity of BirminghamBirminghamUK
- Centre for Endocrinology, Diabetes and MetabolismBirmingham Health PartnersBirminghamUK
- Department of Endocrinology, Queen Elizabeth HospitalUniversity Hospitals Birmingham NHS Foundation TrustBirminghamUK
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Sridharan K, Sahoo J, Palui R, Patil M, Kamalanathan S, Ramesh AS, Kubera NS. Diagnosis and treatment outcomes of Cushing's disease during pregnancy. Pituitary 2021; 24:670-680. [PMID: 33779937 DOI: 10.1007/s11102-021-01142-7] [Citation(s) in RCA: 5] [Impact Index Per Article: 1.3] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Accepted: 03/14/2021] [Indexed: 11/24/2022]
Abstract
PURPOSE We report a case of a pregnant woman with Cushing's disease (CD) and performed a systematic review of literature on diagnosis, treatment, maternal and fetal outcomes of CD in pregnancy. METHODS A PubMed search was performed for manuscripts in English language from inception till June 2020. Cases of CD with hypercortisolism during pregnancy were included and categorized into three groups based on treatment received. Data on diagnostic modalities, CD remission, materno-fetal outcomes were analysed. RESULTS Fifty-five patients of CD with 62 pregnancies were analysed. 24-h urinary free cortisol(UFC) was elevated by a mean of 5.4 ± 4.2 fold upper limit of normal non-pregnant level. 12/19 (63.1%) CD patients had more than threefold elevation of UFC measured during pregnancy. Mean midnight serum cortisol was 753.7 ± 270.5 nmol/l. At a midnight serum cortisol cut off of 440 nmol/l, 15/16 patients were correctly identified as CD. 23.2% underwent trans-sphenoidal surgery (group 1), 16.1% received only medical treatment (group 2) while 60.7% received no treatment (group 3) during pregnancy. Remission rates for CD in groups 1 and 2 were 76.9% and 77.8%, respectively. Adverse maternal and fetal outcomes were seen in 53.9% and 59.3% of the patients, respectively and were not significantly different between groups, although, lesser live births and greater pregnancy losses were seen in group 3. CONCLUSION Midnight serum cortisol had better sensitivity than UFC for diagnosing hypercortisolism due to CD during pregnancy. In general, CD should be treated during pregnancy in order to optimize maternal and fetal outcomes as a trend towards increased live births is seen in treated subjects.
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Affiliation(s)
- Kalyani Sridharan
- Department of Endocrinology and Metabolism, All India Institute of Medical Sciences, Rishikesh, Uttarakhand, India.
| | - Jayaprakash Sahoo
- Department of Endocrinology and Metabolism, Jawaharlal Institute of Post Graduate Medical Education and Research, Dhanvanthri Nagar, Puducherry, India
| | - Rajan Palui
- Consultant Endocrinologist, The Mission Hospital, Durgapur, West Bengal, India
| | - Milind Patil
- Consultant Endocrinologist, Inlaks Budhrani Hospital, Pune, India
| | - Sadishkumar Kamalanathan
- Department of Endocrinology and Metabolism, Jawaharlal Institute of Post Graduate Medical Education and Research, Dhanvanthri Nagar, Puducherry, India
| | - A S Ramesh
- Department of Neurosurgery, Jawaharlal Institute of Post Graduate Medical Education and Research, Dhanvanthri Nagar, Puducherry, India
| | - N S Kubera
- Department of Obstetrics and Gynecology, Jawaharlal Institute of Post Graduate Medical Education and Research, Dhanvanthri Nagar, Puducherry, India
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Jolly K, Darr A, Arlt W, Ahmed S, Karavitaki N. Surgery for Cushing's disease in pregnancy: our experience and a literature review. Ann R Coll Surg Engl 2019; 101:e26-e31. [PMID: 30286651 PMCID: PMC6303815 DOI: 10.1308/rcsann.2018.0175] [Citation(s) in RCA: 11] [Impact Index Per Article: 1.8] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Accepted: 06/13/2018] [Indexed: 12/23/2022] Open
Abstract
Cushing's syndrome in pregnancy is a rare phenomenon and there is limited literature on its management. Cushing's disease in pregnancy is even less common and there is little guidance to help in the treatment for this patient group. Diagnosis of Cushing's syndrome in pregnancy is often delayed due to overlap of symptoms. In addition, there are no validated diagnostic tests or parameters documented. We present a case of a 30-year-old woman presenting to the antenatal clinic at 13 weeks of pregnancy with high suspicion of Cushing's disease. Her 21-week fetal scan showed a congenital diaphragmatic hernia and she underwent pituitary magnetic resonance imaging, which confirm Cushing's disease. She successfully underwent transsphenoidal adenomectomy with histology confirming a corticotroph adenoma. Tests following transsphenoidal surgery confirmed remission of Cushing's disease and she underwent an emergency caesarean section at 38 weeks. Unfortunately, her baby died from complications associated with the congenital abnormality 36 hours after birth. The patient remains in remission following delivery. To date, there have been no reported cases of congenital diaphragmatic hernia associated with Cushing's disease in pregnancy. In addition, we believe that this is only the eighth reported patient to have undergone successful transsphenoidal surgery for Cushing's disease in pregnancy.
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Affiliation(s)
- K Jolly
- Ear, Nose and Throat Department, University Hospital Birmingham, Birmingham, UK
| | - A Darr
- Ear, Nose and Throat Department, University Hospital Birmingham, Birmingham, UK
| | - W Arlt
- Department of Endocrinology, University Hospital Birmingham, Birmingham, UK
| | - S Ahmed
- Ear, Nose and Throat Department, University Hospital Birmingham, Birmingham, UK
| | - N Karavitaki
- Department of Endocrinology, University Hospital Birmingham, Birmingham, UK
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Ciftci Dogansen S, Canbaz B, Canbaz B, Yarman S. UNCOMPLICATED PREGNANCY IN A PATIENT WITH CUSHING'S DISEASE. ACTA ENDOCRINOLOGICA-BUCHAREST 2017; 13:215-219. [PMID: 31149176 DOI: 10.4183/aeb.2017.215] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Subscribe] [Scholar Register] [Indexed: 11/20/2022]
Abstract
The coexistence of Cushing's syndrome (CS) and pregnancy is uncommon due to the suppression of gonadotropin secretion in CS. Adrenocorticotropic hormone (ACTH) dependent CS in pregnancy is less frequent than adrenal causes. The diagnosis of CS during pregnancy is difficult since physiological changes in the hypothalamo-pituitary-adrenal axis may cause dynamic tests to be misinterpreted. Radiological imaging is limited. We report the case of a 27-year old woman with Cushing's disease (CD) diagnosed in the 5th-week of pregnancy. The mild symptoms of hypercortisolism did not lead to serious complications for the mother or the fetus, so insulin was the only treatment used. The pregnancy was completed without any complications, and at 38 weeks of gestation a healthy female infant was delivered vaginally. Complications, such as hypocortisolemia and hypoglycemia, were not observed in the infant. Postpartum tests were consistent with CD. Contrast-enhanced pituitary magnetic resonance imaging revealed a microadenoma which was removed with transsphenoidal surgery. Histopathology revealed a pituitary adenoma with positive immunohistochemical staining for ACTH. Biochemical remission required prednisolone treatment, but the insulin requirement decreased significantly over time. As a conclusion, CD with mild features can be well tolerated during pregnancy, but the mother and the fetus must be monitored closely.
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Affiliation(s)
- S Ciftci Dogansen
- Istanbul University, Istanbul Faculty of Medicine, Department of Internal Medicine, Istanbul, Turkey
| | - B Canbaz
- Istanbul University, Istanbul Faculty of Medicine, Department of Internal Medicine, Istanbul, Turkey
| | - B Canbaz
- Istanbul University, Istanbul Faculty of Medicine, Division of Endocrinology and Metabolism, Istanbul, Turkey
| | - S Yarman
- Istanbul University, Istanbul Faculty of Medicine, Department of Internal Medicine, Istanbul, Turkey
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Abbassy M, Kshettry VR, Hamrahian AH, Johnston PC, Dobri GA, Avitsian R, Woodard TD, Recinos PF. Surgical management of recurrent Cushing's disease in pregnancy: A case report. Surg Neurol Int 2015; 6:S640-5. [PMID: 26682090 PMCID: PMC4672578 DOI: 10.4103/2152-7806.170472] [Citation(s) in RCA: 11] [Impact Index Per Article: 1.1] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/10/2015] [Accepted: 08/21/2015] [Indexed: 12/20/2022] Open
Abstract
Background: Cushing's disease is a condition rarely encountered during pregnancy. It is known that hypercortisolism is associated with increased maternal and fetal morbidity and mortality. When hypercortisolism from Cushing's disease does occur in pregnancy, the impact of achieving biochemical remission on fetal outcomes is unknown. We sought to clarify the impact of successful surgical treatment by presenting such a case report. Case Description: A 38-year-old pregnant woman with recurrent Cushing's disease after 8 years of remission. The patient had endoscopic transsphenoidal of her pituitary adenoma in her 18th week of pregnancy. The patient had postoperative biochemical remission and normal fetal outcome with no maternal complications. Conclusion: Transsphenoidal surgery for Cushing's disease can be performed safely during the second trimester of pregnancy.
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Affiliation(s)
- Mahmoud Abbassy
- Rose Ella Burkhardt Brain Tumor and Neuro-oncology Center, Neurological Institute, Cleveland Clinic, Cleveland, Ohio, USA ; Minimally Invasive Cranial Base and Pituitary Surgery Program, Cleveland Clinic, Cleveland, Ohio, USA
| | - Varun R Kshettry
- Rose Ella Burkhardt Brain Tumor and Neuro-oncology Center, Neurological Institute, Cleveland Clinic, Cleveland, Ohio, USA ; Minimally Invasive Cranial Base and Pituitary Surgery Program, Cleveland Clinic, Cleveland, Ohio, USA
| | - Amir H Hamrahian
- Department of Endocrinology, Diabetes, and Metabolism, Cleveland Clinic, Cleveland, Ohio, USA
| | - Philip C Johnston
- Department of Endocrinology, Diabetes, and Metabolism, Cleveland Clinic, Cleveland, Ohio, USA
| | - Georgianna A Dobri
- Department of Endocrinology, Diabetes, and Metabolism, Cleveland Clinic, Cleveland, Ohio, USA
| | - Rafi Avitsian
- Department of Anesthesiology, Cleveland Clinic, Cleveland, Ohio, USA
| | - Troy D Woodard
- Rose Ella Burkhardt Brain Tumor and Neuro-oncology Center, Neurological Institute, Cleveland Clinic, Cleveland, Ohio, USA ; Minimally Invasive Cranial Base and Pituitary Surgery Program, Cleveland Clinic, Cleveland, Ohio, USA ; Section of Skull Base Rhinology and Sinus Surgery, Head and Neck Institute, Cleveland Clinic, Cleveland, Ohio, USA
| | - Pablo F Recinos
- Rose Ella Burkhardt Brain Tumor and Neuro-oncology Center, Neurological Institute, Cleveland Clinic, Cleveland, Ohio, USA ; Minimally Invasive Cranial Base and Pituitary Surgery Program, Cleveland Clinic, Cleveland, Ohio, USA ; Section of Skull Base Rhinology and Sinus Surgery, Head and Neck Institute, Cleveland Clinic, Cleveland, Ohio, USA
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7
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Abstract
The pituitary gland is significantly affected during gestation in terms of both size and function. Due to this physiologic adaptation, endocrine evaluation and interpretation of imaging is far more complex than in the non-pregnant state. Pituitary disorders are rare in pregnancy, as they are usually associated with gonadal dysfunction, thereby posing difficulties with fertility. This review will focus on pituitary adenomas (prolactinomas, GH-secreting and ACTH-secreting), their diagnostic handicaps and the recommendations for treatment. We will also discuss the two pituitary disorders encountered in pregnancy, Sheehan's syndrome and lymphocytic hypophysitis.
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Affiliation(s)
| | - Maria Boudina
- Unit of Endocrinology, Theagenio Hospital, Aristotle University of Thessaloniki, Greece
| | - Niki Karavitaki
- Department of Endocrinology, Oxford Centre for Diabetes, Endocrinology and Metabolism, Churchill Hospital, Oxford, UK
| | - Eleni Bili
- 1st Department of Obstetrics & Gynaecology, Aristotle University of Thessaloniki, Greece
| | - John Wass
- Department of Endocrinology, Oxford Centre for Diabetes, Endocrinology and Metabolism, Churchill Hospital, Oxford, UK
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8
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Boronat M, Marrero D, López-Plasencia Y, Barber M, Schamann Y, Nóvoa FJ. Successful outcome of pregnancy in a patient with Cushing's disease under treatment with ketoconazole during the first trimester of gestation. Gynecol Endocrinol 2011; 27:675-7. [PMID: 20923280 DOI: 10.3109/09513590.2010.521268] [Citation(s) in RCA: 30] [Impact Index Per Article: 2.1] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 11/13/2022] Open
Abstract
BACKGROUND Ketoconazole is the most widely used medical treatment for Cushing's syndrome, but, because of its potential to cause birth defects, it is not recommended during pregnancy. Specifically, due to its antiandrogenic effects, ketoconazole entails theoretical risks of interfering with the development of external genitalia in male fetuses. CASE A pregnancy was diagnosed at 13 weeks of gestation in a 26-year-old woman with Cushing's disease under treatment with ketoconazole. The drug was withdrawn and the patient underwent transsphenoidal surgery at 16 weeks of pregnancy. She did not develop postsurgical adrenal insufficiency and was treated with metyrapone during the second and third trimesters of gestation. Partum was induced at 34 weeks of pregnancy. The patient delivered a healthy male infant with normal external genitalia. CONCLUSION Treatment with ketoconazole during the critical period of organogenesis did not cause congenital birth defects to the male fetus of a woman with Cushing's disease. This report should be taken into account for future cases of unplanned pregnancies in women with Cushing's syndrome treated with ketoconazole, as well in those cases in which other therapeutic options are not feasible.
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Affiliation(s)
- Mauro Boronat
- Section of Endocrinology and Nutrition, Complejo Hospitalario Universitario Insular-Materno Infantil, Las Palmas de Gran Canaria, Spain.
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9
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Lindsay JR, Nieman LK. The hypothalamic-pituitary-adrenal axis in pregnancy: challenges in disease detection and treatment. Endocr Rev 2005; 26:775-99. [PMID: 15827110 DOI: 10.1210/er.2004-0025] [Citation(s) in RCA: 243] [Impact Index Per Article: 12.2] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 02/07/2023]
Abstract
Pregnancy dramatically affects the hypothalamic-pituitary-adrenal axis leading to increased circulating cortisol and ACTH levels during gestation, reaching values in the range seen in Cushing's syndrome (CS). The cause(s) of increased ACTH may include placental synthesis and release of biologically active CRH and ACTH, pituitary desensitization to cortisol feedback, or enhanced pituitary responses to corticotropin-releasing factors. In this context, challenges in diagnosis and management of disorders of the hypothalamic-pituitary-adrenal axis in pregnancy are discussed. CS in pregnancy is uncommon and is associated with fetal morbidity and mortality. The diagnosis may be missed because of overlapping clinical and biochemical features in pregnancy. The proportion of patients with primary adrenal causes of CS is increased in pregnancy. CRH stimulation testing and inferior petrosal sinus sampling can identify patients with Cushing's disease. Surgery is a safe option for treatment in the second trimester; otherwise medical therapy may be used. Women with known adrenal insufficiency that is appropriately treated can expect to have uneventful pregnancies. Whereas a fetal/placental source of cortisol may mitigate crisis during gestation, unrecognized adrenal insufficiency may lead to maternal or fetal demise either during gestation or in the puerperium. Appropriate treatment and management of labor are reviewed.
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Affiliation(s)
- John R Lindsay
- Reproductive Biology and Medicine Branch, National Institute of Child Health and Human Development, National Institutes of Health, Bethesda, Maryland 20892-1109, USA
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10
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Miyoshi T, Otsuka F, Suzuki J, Inagaki K, Takeda M, Kano Y, Yamashita T, Ogura T, Date I, Tanaka Y, Hashimoto K, Makino H. Periodic secretion of adrenocorticotropin in a patient with Cushing's disease manifested during pregnancy. Endocr J 2005; 52:287-92. [PMID: 16006722 DOI: 10.1507/endocrj.52.287] [Citation(s) in RCA: 6] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 11/23/2022] Open
Abstract
We report the case of 19-year-old woman with cyclical Cushing's disease, in whom plasma adrenocorticotropin (ACTH) was secreted periodically after her first pregnancy. Since the 33rd week of pregnancy, hypertension and proteinuria became clinically remarkable. She gave normal birth at 36th week of pregnancy; however she continued to gain body weight even after delivery and developed typical Cushingoid features. Her ACTH secretion lacked normal daily fluctuation but exhibited periodic change during 1-year observation, showing 119 pg/ml, 34.6 pg/ml and 115 pg/ml at the 4th, 7th and 13th months after delivery. Plasma ACTH levels were increased by corticotropin releasing hormone and metyrapone, while low-dose dexamethasone suppressed cortisol secretion. Gel filtration analysis of the patient's plasma detected big ACTH molecules being eluted with a peak of authentic 1-39 ACTH. Cranial magnetic resonance imaging revealed a 1-cm pituitary mass in right cavernous sinus. The pituitary tumor was removed by transsphenoidal surgery at 13th month after delivery and was pathologically compatible with ACTH-producing pituitary adenoma by immunohistochemistry. This case includes clinically rare subsets of Cushing's syndrome showing periodic ACTH secretion and aberrant ACTH molecules.
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Affiliation(s)
- Tomoko Miyoshi
- Department of Medicine and Clinical Science, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, Japan
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Affiliation(s)
- Nicoletta Polli
- Ospedale San Luca, Istituto Auxologico Italiano IRCCS, University of Milan, Milan, Italy
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12
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Affiliation(s)
- Ron Swensen
- Department of Gynecology and Obstetrics, Loma Linda University School of Medicine, California 92354, USA.
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Antonelli NM, Dotters DJ, Katz VL, Kuller JA. Cancer in pregnancy: a review of the literature. Part II. Obstet Gynecol Surv 1996; 51:135-42. [PMID: 8622837 DOI: 10.1097/00006254-199602000-00023] [Citation(s) in RCA: 28] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 01/31/2023]
Affiliation(s)
- N M Antonelli
- Department of Obstetrics and Gynecology, University of North Carolina School of Medicine, USA
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15
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Antonelli NM, Dotters DJ, Katz VL, Kuller JA. Cancer in pregnancy: a review of the literature. Part I. Obstet Gynecol Surv 1996; 51:125-34. [PMID: 8622836 DOI: 10.1097/00006254-199602000-00022] [Citation(s) in RCA: 125] [Impact Index Per Article: 4.3] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 01/31/2023]
Abstract
Cancer is an important cause of death in the United States in women of childbearing age. Approximately 1 per 1000 pregnant women will develop cancer. This review (Part II follows in this issue) examines the diagnosis, prognosis, and management of cancer during pregnancy; both in terms of the cancer's effect on the pregnancy, and the pregnancy's effect on the cancer. Some diagnostic modalities and some therapies are problematic to the fetus and placenta. However, in most cases and the pregnancy can be managed concurrently with a good outcome for the baby and without compromising the mother's prognosis.
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Affiliation(s)
- N M Antonelli
- Department of Obstetrics and Gynecology, University of North Carolina School of Medicine, Chapel Hill, USA
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