Acute Lymphoblastic Leukemia with Isolated Adrenocorticotropic Hormone Deficiency.

Yamaguchi, Hiroki; Nakamura, Hiroyuki; Mamiya, Yasutaka; Yamämoto, Yasushi; Takagi, Kenji; Sugihara, Hitoshi; Gomi, Seiji; Inokuchi, Koiti; Hasegawa, S; Shibazaki, Tamotsu; Dan, Kazuo; Wakabayashi, Ichiji

doi:10.2169/internalmedicine.36.819

Cited by 14 publications

(5 citation statements)

References 7 publications

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“…In addition, an epitope of endogenous proteins has been presented by MHC class I molecules in the anterior pituitary cells [ 27 ], which enables the recognition of specific T-cell receptors on cytotoxic T cells. Interestingly, a case of acquired IAD as a form of paraneoplastic syndrome was caused by autoimmunity against corticotrophs with the ectopic ACTH expression in the complicated tumor [ 15 ], and several cases of IAD were complicated with malignant tumors have been reported [ 28 , 29 ]. Interestingly, ectopic ACTH, expression is not rare in various cancers, regardless of the type and ectopic expression of the other pituitary hormones is extremely rare [ 15 ].…”

Section: Discussionmentioning

confidence: 99%

Mechanistic insights into immune checkpoint inhibitor-related hypophysitis: a form of paraneoplastic syndrome

Kanie

Iguchi

Bando

et al. 2021

Cancer Immunol Immunother

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Background Immune checkpoint inhibitors (ICIs) as a cancer immunotherapy have emerged as a treatment for multiple advanced cancer types. Because of enhanced immune responses, immune-related adverse events (irAEs), including endocrinopathies such as hypophysitis, have been associated with the use of ICIs. Most underlying mechanisms of ICI-related hypophysitis remain unclear, especially for programmed cell death-1 (PD-1)/PD-1 ligand 1 (PD-L1) inhibitors. We hypothesized that ICI-related hypophysitis is associated with paraneoplastic syndrome caused by ectopic expression of pituitary-specific antigens. Methods Twenty consecutive patients with ICI-related hypophysitis between 2017 and 2019 at Kobe University Hospital were retrospectively analyzed. Circulating anti-pituitary antibodies were detected using immunofluorescence staining and immunoblotting. Ectopic expression of pituitary autoantigens in tumor specimens was also examined. Results Eighteen patients were treated with PD-1/PD-L1 inhibitors, and two were treated with a combination of cytotoxic T-lymphocyte antigen-4 (CTLA-4) and PD-1 inhibitors. All patients showed adrenocorticotropic hormone (ACTH) deficiency and additionally, three showed thyroid-stimulating hormone (TSH) deficiency, and one showed gonadotropin-releasing hormone (GnRH) deficiency. Among these patients, three exhibited anti-pituitary antibodies, two with anti-corticotroph antibody and one with anti-somatotroph antibody. Interestingly, the anti-corticotroph antibody recognized proopiomelanocortin (POMC) and those two patients exhibited ectopic ACTH expression in the tumor, while the patients without anti-corticotroph antibody did not. Conclusions We demonstrated 10% of PD-1/PD-L1 inhibitors-related hypophysitis were associated with the autoimmunity against corticotrophs and maybe caused as a form of paraneoplastic syndrome, in which ectopic expression of ACTH in the tumor was observed. It is also suggested that the pathophysiology is heterogenous in ICI-related hypophysitis.

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Section: Discussionmentioning

confidence: 99%

Mechanistic insights into immune checkpoint inhibitor-related hypophysitis: a form of paraneoplastic syndrome

Kanie

Iguchi

Bando

et al. 2021

Cancer Immunol Immunother

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“…Only one patient with acute lymphoblastic leukemia and one with gastric cancer have been previously reported [3,4]. Kamiya and Murakami reported a type 2 diabetic patient with concurrent IAD and gastric cancer who was positive for antipituitary antibody and had empty sella [4].…”

Section: Discussionmentioning

confidence: 99%

“…Although the number of diagnosed cases of IAD is increasing because knowledge of the disease and performance of detailed endocrinology testing have become more widespread [2], IAD in combination with malignant neoplasia is very rare [3,4]. Here we describe a patient with gastric cancer who developed unexpected IAD during chemotherapy.…”

Section: Introductionmentioning

confidence: 99%

Isolated adrenocorticotropic hormone deficiency development during chemotherapy for gastric cancer: a case report

et al. 2014

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IntroductionIsolated adrenocorticotropic hormone deficiency is an endocrinological disorder characterized by loss of adrenocorticotropic hormone and resultant adrenal insufficiency. Affected patients often present with fatigue, anorexia, and hyponatremia. Although the number of reported cases has been recently increasing, isolated adrenocorticotropic hormone deficiency combined with malignant neoplasia is very rare. Here we describe a patient with gastric cancer who developed unexpected isolated adrenocorticotropic hormone deficiency during chemotherapy.Case presentationA 72-year-old Japanese man was admitted to our hospital because of febrile neutropenia due to chemotherapy for gastric cancer recurrence. Although the neutropenia and fever immediately improved, he became unable to take any oral medications and was bedridden 1 week after admission. His serum sodium level abruptly decreased to 122mEq/L on the fifth day of hospitalization. We performed endocrinological studies to investigate the cause of his hyponatremia and plasma hyposmolality. His plasma adrenocorticotropic hormone and cortisol levels were very low. However, his serum levels of all other anterior pituitary hormones were slightly elevated. We then performed a corticotropin-releasing hormone test, which showed that neither his plasma adrenocorticotropic hormone nor cortisol level responded to corticotropin-releasing hormone stimulation. We definitively diagnosed isolated adrenocorticotropic hormone deficiency based on these findings. Hydrocortisone replacement therapy was begun at 20mg/day, resulting in a marked improvement in his anorexia and general fatigue within a few days. His serum sodium level was also normalized immediately after the administration of hydrocortisone. He was discharged from our hospital on the 50th day of hospitalization.ConclusionsThe present case is the second report of a patient with concurrent isolated adrenocorticotropic hormone deficiency and gastric cancer and the first report of a patient diagnosed with isolated adrenocorticotropic hormone deficiency during the course of chemotherapy for a solid malignant neoplasm. Although the symptoms and signs described in the present report are common observations during chemotherapy, it is important to consider not only the adverse effects of antineoplastic agents, but also isolated adrenocorticotropic hormone deficiency as a differential diagnosis. Hydrocortisone replacement therapy for isolated adrenocorticotropic hormone deficiency effectively avoids the unnecessary cessation of chemotherapy.

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“…A case of bilateral adrenal hemorrhage in a patient with acute myeloid leukemia (AML) has also been reported [98]. Furthermore, adrenal insufficiency may be the result of isolated adrenocorticotropic hormone (ACTH) deficiency in ALL patients [99]. Patients with hemoglobinopathies, including thalassemias and sickle cell disease, may present with several endocrine disorders, including hypogonadotropic hypogonadism, DM, hypothyroidism, hypoparathyroidism, and adrenal insufficiency, mainly due to iron overload [100,101].…”

Section: Hyponatremia Due To Disorders Of Endocrine System and Metabomentioning

confidence: 99%

Hyponatremia in Patients with Hematologic Diseases

Koumpis

Florentin

Hatzimichael

et al. 2020

JCM

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Hyponatremia is the most common electrolyte disorder in clinical practice and is associated with increased morbidity and mortality. It is frequently encountered in hematologic patients with either benign or malignant diseases. Several underlying mechanisms, such as hypovolemia, infections, toxins, renal, endocrine, cardiac, and liver disorders, as well as the use of certain drugs appear to be involved in the development or the persistence of hyponatremia. This review describes the pathophysiology of hyponatremia and discusses thoroughly the contributing factors and mechanisms that may be encountered specifically in patients with hematologic disorders. The involvement of the syndrome of inappropriate antidiuretic hormone (SIADH) secretion and renal salt wasting syndrome (RSWS) in the development of hyponatremia in such patients, as well as their differential diagnosis and management, are also presented. Furthermore, the distinction between true hyponatremia and pseudohyponatremia is explained. Finally, a practical algorithm for the evaluation of hyponatremia in hematologic patients, as well as the principles of hyponatremia management, are included in this review.

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Acute Lymphoblastic Leukemia with Isolated Adrenocorticotropic Hormone Deficiency.

Cited by 14 publications

References 7 publications

Mechanistic insights into immune checkpoint inhibitor-related hypophysitis: a form of paraneoplastic syndrome

Mechanistic insights into immune checkpoint inhibitor-related hypophysitis: a form of paraneoplastic syndrome

Isolated adrenocorticotropic hormone deficiency development during chemotherapy for gastric cancer: a case report

Hyponatremia in Patients with Hematologic Diseases

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