scholarly journals Diagnosis and management of adrenocorticotropic hormone-secreting pituitary carcinoma: a case report and review of the literature

2021 ◽  
pp. FRD
Author(s):  
Angeline JJ Shen ◽  
James King ◽  
Peter G Colman ◽  
Christopher J Yates

Adrenocorticotropic hormone (ACTH)-secreting pituitary carcinomas (PC) are rare. The natural history and management of these carcinomas are poorly understood. We conducted a literature review using The MEDLINE database, including the search terms; ‘ACTH’ and ‘pituitary carcinoma’. We also describe in detail a case of ACTH-secreting PC. A total of 61 case reports were reviewed. Median age of diagnosis was 45 years (IQR: 34–54). Metastases to multiple organs were common (61%). Adjuvant therapy especially radiotherapy (78%), temozolomide (34%) and other medical therapy (29%) were frequently employed. The mortality was 53% with a median time to death from diagnosis of 1 year (IQR: 1–3). In conclusion, ACTH-secreting PC are associated with high mortality and a multidisciplinary team approach is recommended for optimal care due to the emerging modalities with possible efficacy.

2021 ◽  
Vol 184 (1) ◽  
pp. K1-K5 ◽  
Author(s):  
Bastiaan Sol ◽  
Jeroen M K de Filette ◽  
Gil Awada ◽  
Steven Raeymaeckers ◽  
Sandrine Aspeslagh ◽  
...  

Background Pituitary carcinomas are rare but aggressive and require maximally coordinated multimodal therapies. For refractory tumors, unresponsive to temozolomide (TMZ), therapeutic options are limited. Immune checkpoint inhibitors (ICI) may be considered for treatment as illustrated in the present case report. Case We report a patient with ACTH-secreting pituitary carcinoma, progressive after multiple lines of therapy including chemotherapy with TMZ, who demonstrated disease stabilization by a combination of ipilimumab (anti-CTLA-4) and nivolumab (anti-PD-1) ICI therapy. Discussion Management of pituitary carcinoma beyond TMZ remains ill-defined and relies on case reports. TMZ creates, due to hypermutation, more immunogenic tumors and subsequently potential candidates for ICI therapy. This case report adds support to the possible role of ICI in the treatment of pituitary carcinoma. Conclusion ICI therapy could be a promising treatment option for pituitary carcinoma, considering the mechanisms of TMZ-induced hypermutation with increased immunogenicity, pituitary expression of CTLA-4 and PD-L1, and the frequent occurrence of hypophysitis as a side effect of ICI therapy.


2015 ◽  
Vol 2015 ◽  
pp. 1-4 ◽  
Author(s):  
Clarissa Groberio Borba ◽  
Rafael Loch Batista ◽  
Nina Rosa de Castro Musolino ◽  
Vanielle Carvalho Machado ◽  
Ana Elisa Evangelista Alcantara ◽  
...  

Pituitary carcinomas are very rare tumors that in most cases produce prolactin and adrenocorticotropic hormone (ACTH). It is a challenge to diagnosis of a pituitary carcinoma before disclosed symptomatic metastasis. We report the case of a female patient with Cushing’s disease who underwent three transsphenoidal surgeries, with pathological findings of common ACTH pituitary adenoma including Ki-67 expression <3%. She achieved hypocortisolism after the 3rd surgery although ACTH levels remained slightly elevated. The patient returned some time later with fast worsening of hypercortisolism. Magnetic resonance imaging showed clivus invasion, which led to a fourth surgery and radiation. This time, immunohistochemistry revealed strong Ki-67 (10% to 15%) and p53 expression. Liver and lumbar spine metastases were found on workup. The patient died after few months due to lung infection. Pituitary carcinomas are rare, and the transformation of an ACTH-secreting pituitary adenoma into a carcinoma is exceptional. The difficulty of defining markers for the diagnosis of carcinoma, before metastasis diagnosis, in order to change the management of the disease, is a challenge.


2021 ◽  
Vol 21 (1) ◽  
Author(s):  
Hideto Ikarashi ◽  
Naohiko Aketa ◽  
Eisuke Shimizu ◽  
Yoji Takano ◽  
Tetsuya Kawakita ◽  
...  

Abstract Background Chronic ocular graft-versus-host disease (oGVHD) is an ocular comorbidity of graft-versus-host disease (GVHD) that usually occurs concurrently with systemic manifestations. Failure to detect and treat oGVHD in its early stages may lead to progression of ocular signs and symptoms leading to oGVHD that is refractory to conventional treatment. Case presentation We report the clinical course of a 19-year-old male and a 59-year-old female with severe and progressive chronic oGVHD without concurrent systemic signs of chronic graft-versus-host disease (cGVHD). Although their systemic conditions had been stable, both suffered from severe oGVHD and were referred to our clinic. Both cases exhibited marked improvement in conjunctival inflammation and fibrotic changes after amniotic membrane transplantation (AMT). Both cases underwent keratoplasty eventually to stabilize ocular surface conditions and to improve visual function. Conclusions We reported the clinical outcomes of 2 cases of chronic oGVHD without concurrent systemic comorbidities that were treated with AMT. The clinician should be aware that cGVHD may persist in target organs even in the absence of concurrent systemic comorbidities following seemingly successful systemic treatment. A multidisciplinary team approach is essential in the early detection and therapeutic intervention for chronic oGVHD.


2015 ◽  
Vol 129 (5) ◽  
pp. 410-415 ◽  
Author(s):  
I Syed ◽  
V S Sunkaraneni

AbstractBackground:There are currently no guidelines in the UK for the specific management of hereditary haemorrhagic telangiectasia related epistaxis. The authors aimed to review the literature and provide an algorithm for the management of hereditary haemorrhagic telangiectasia related epistaxis.Method:The Medline and Embase databases were interrogated on 15 November 2013 using the search items ‘hereditary haemorrhagic telangiectasia’ (title), ‘epistaxis’ (title) and ‘treatment’ (title and abstract), and limiting the search to articles published in English.Results:A total of 46 publications were identified, comprising 1 systematic review, 2 randomised, controlled trials, 27 case series, 9 case reports, 4 questionnaire studies and 3in vitrostudies.Conclusion:There is a lack of high-level evidence for the use of many of the available treatments for the specific management of epistaxis in hereditary haemorrhagic telangiectasia. Current management should be based on a multidisciplinary team approach involving both a hereditary haemorrhagic telangiectasia physician and an ENT surgeon, especially when systemic therapy is being considered. The suggested treatment algorithm considers that the severity of epistaxis merits intervention at different levels of the treatment ladder. The patient should be assessed using a reproducible validated assessment tool, for example an epistaxis severity score, to guide treatment. More research is required, particularly in the investigation of topical agents targeting the development and fragility of telangiectasiae in hereditary haemorrhagic telangiectasia.


2021 ◽  
pp. bmjspcare-2020-002732
Author(s):  
R Andrew Seaton ◽  
Lesley Cooper ◽  
Jack Fairweather ◽  
Stephen Fenning ◽  
Libby Ferguson ◽  
...  

ObjectivesDevelopment of evidence-based good practice recommendations for clinicians considering the use of antibiotics in patients towards the end of life.DesignA multiprofessional group of experts in end-of-life care and antimicrobial stewardship was convened. Findings from a scoping review of the literature and a consultation of clinicians were triangulated. Expert discussion was used to generate consensus on how to approach decision-making.SettingRepresentatives from hospital and a range of community health and care settings.ParticipantsMedical, pharmacy and nursing professionals.Main outcome measuresGood practice recommendations based on published evidence and the experience of prescribers in Scotland.ResultsThe findings of 88 uncontrolled, observational studies of variable quality were considered alongside a survey of over 200 prescribers. No national or international guidelines were identified. Antibiotic use towards the end of life was common but practice was highly variable. The potential harms associated with giving antibiotics tended to be less well considered than the potential benefits. Antibiotics often extended the length of time to death but this was sometimes at the cost of higher symptom burden. There was strong consensus around the importance of effective communication with patients and their families and making treatment decisions aligned to a patient’s goals and priorities.ConclusionsGood practice recommendations were agreed with focus on three areas: making shared decisions about future care; agreeing clear goals and limits of therapy; reviewing all antibiotic prescribing decisions regularly. These will be disseminated widely to support optimal care for patients towards the end of life. A patient version of the recommendations has also been produced to support implementation.


2013 ◽  
Vol 29 (6) ◽  
pp. 327-333 ◽  
Author(s):  
Tracey G. Simon ◽  
Joanna Bradley ◽  
Adisa Jones ◽  
Gerardo Carino

We describe the case of a patient with hemolysis-associated Clostridium perfringens septicemia and review all similar cases published in the literature since 1990, with specific focus on the relationship between treatment strategy and survival. We searched PubMed for all published cases of C. perfringens-associated hemolysis, using the medical subject terms “clostridia,” “clostridial sepsis,” and/or “hemolysis.” All case reports, case series, review articles, and other relevant references published in the English literature since 1990 were included in this study. There were no exclusion criteria. Each case was examined with respect to presenting features of illness, antibiotic regimen, time-to-antibiotic therapy, additional interventions, complications, and patient survival. These variables were entered into a data set and then systematically analyzed with the aid of a statistician, using serial t tests and chi-square analyses. Since 1990, 50 patients of C. perfringens septicemia with hemolysis have been reported. Median age was 61 years (range 31-84), and 58% were male. Mortality was 74%, with a median time to death of 9.7 hours (range 0-96 hours). Of the patients, 35 (70%) were treated medically, while 15 (30%) received antibiotics and surgery. Surgical intervention was associated with significantly improved survival (risk ratio [RR] 0.23, 95% confidence interval [CI] 0.10, 0.53) as was the use of a combination of penicillin and clindamycin (RR of death 0.46, 95% CI 0.25, 0.83). Four patients utilizing hyperbaric oxygen therapy (HBOT) have been reported, and all patients survived. In cases of clostridial sepsis with hemolysis, strong predictors of survival include early initiation of appropriate antibiotics as well as surgical removal of infected foci. The HBOT may also be associated with survival. The disease often progresses rapidly to death, so rapid recognition is critical for the patient survival.


2005 ◽  
Vol 39 (11) ◽  
pp. 1897-1902 ◽  
Author(s):  
Masha SH Lam

OBJECTIVE To review the clinical evidence for routine use of bleomycin test dosing. DATA SOURCES English-language review articles, references from retrieved articles, case reports, and clinical trials were identified from a MEDLINE literature search (1966–July 2005). Key search terms included bleomycin, test dose, anaphylactic reactions, and hypersensitivity. Information from an unpublished E-mail survey, the manufacturer, and the Internet was also used. DATA SYNTHESIS Early clinical trials and isolated case reports suggest that bleomycin-induced acute hypersensitivity reactions occur in 1% of patients with lymphoma and <0.5% of those with solid tumors. The reactions are mainly characterized by high-grade fever, chills, hypotension, and in a few cases, cardiovascular collapse, which can lead to death. The exact mechanism of these reactions is unclear, but is thought to be related to the release of endogenous pyrogens from the host cells. Evidence does not suggest any correlation between doses and the onset or severity of the reactions. Supportive care, including hydration, steroids, antipyretics, and antihistamines, may resolve the symptoms. However, it may not completely prevent recurrences. CONCLUSIONS The incidence of acute hypersensitivity or hyperpyrexic reactions associated with bleomycin is very low, but the reaction is potentially fatal. Clinicians should monitor their patients for any signs and symptoms of acute hyperpyrexic reactions during bleomycin administration. Since the onset of the reactions can occur with any dose of bleomycin and at any time, routine test dosing does not seem to predict when drug reactions may occur.


2018 ◽  
Vol 103 (10) ◽  
pp. 3925-3930 ◽  
Author(s):  
Andrew L Lin ◽  
Philip Jonsson ◽  
Viviane Tabar ◽  
T Jonathan Yang ◽  
John Cuaron ◽  
...  

Abstract Context Pituitary carcinoma is a rare and aggressive malignancy with a poor prognosis and few effective treatment options. Case A 35-year-old woman presented with an aggressive ACTH-secreting pituitary adenoma that initially responded to concurrent temozolomide and capecitabine prior to metastasizing to the liver. Following treatment with ipilimumab and nivolumab, the tumor volume of the dominant liver metastasis reduced by 92%, and the recurrent intracranial disease regressed by 59%. Simultaneously, her plasma ACTH level decreased from 45,550 pg/mL to 66 pg/mL. Molecular Evaluation Both prospective clinical sequencing with Memorial Sloan Kettering–Integrated Mutation Profiling of Actionable Cancer Targets and retrospective whole-exome sequencing were performed to characterize the molecular alterations in the chemotherapy-naive pituitary adenoma and the temozolomide-resistant liver metastasis. The liver metastasis harbored a somatic mutational burden consistent with alkylator-induced hypermutation that was absent from the treatment-naive tumor. Resistance to temozolomide treatment, acquisition of new oncogenic drivers, and subsequent sensitivity to immunotherapy may be attributed to hypermutation. Conclusion Combination treatment with ipilimumab and nivolumab may be an effective treatment in pituitary carcinoma. Clinical sequencing of pituitary tumors that have relapsed following treatment with conventional chemotherapy may identify the development of therapy-induced somatic hypermutation, which may be associated with treatment response to immunotherapy.


2018 ◽  
Vol 99 (4) ◽  
pp. 211-215
Author(s):  
I. Z. Korobkova ◽  
D. A. Dremin ◽  
S. M. Kacalov ◽  
I. V. Kirsan ◽  
A. A. Ugrimov ◽  
...  

The paper describes a clinical example of the topical diagnosis of adrenocorticotropic hormone (ACTH)-producing typical peripheral pulmonary carcinoid. The first stage in its diagnosis was to rule out the production of ACTH by the pituitary gland. The paper presents information on the most common localization of functioning neuroendocrine tumors, as well as a diagnostic algorithm to search for an ectopic focus of the ACTH-secreting tumor that causes hypercorticism. Taking into account that bronchopulmonary neuroendocrine tumors with ectopic hormone production occur rarely (5%), a clinical example is given to demonstrate the capabilities of imaging techniques and standards for their implementation using an integrated approach.


Pharmacy ◽  
2019 ◽  
Vol 7 (3) ◽  
pp. 110 ◽  
Author(s):  
Yuman Lee ◽  
Nicole Bradley

Understanding antibiotic allergies and the risk of cross-sensitivity between and within antibiotic classes can have a substantial impact on patient care. The purpose of this review article is to provide insight into carbapenem allergies, describing the overall incidence, risk factors, and in-class cross-sensitivity. A PubMed search was conducted using the following search terms: carbapenem, allergy, cross-sensitivity, incidence, imipenem/cilastatin, meropenem, ertapenem, and doripenem. Article bibliographies and relevant drug monographs were also reviewed. The overall reported incidence of carbapenem allergy is 0.3%–3.7%. Risk of cross-sensitivity between penicillins and carbapenems is less than 1% in patients with a positive penicillin skin test. Data on cross-sensitivity between cephalosporins and carbapenems are limited; however, the risk appears to also be low. No clinical studies have described cross-sensitivity between the carbapenem agents thus far. The limited data available from case reports demonstrates a lack of cross-sensitivity between the individual carbapenems, suggesting that an alternative carbapenem may cautiously be used in patients with a reported carbapenem allergy.


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