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  1. Article ; Online: M-type phospholipase A2 receptor-associated membranous nephropathy in a patient with Noonan-like syndrome with loose anagen hair.

    Fujishige, Shuta / Ogawa, Yayoi / Aoyagi, Hayato / Okamoto, Takayuki

    Pediatrics international : official journal of the Japan Pediatric Society

    2023  Volume 65, Issue 1, Page(s) e15643

    MeSH term(s) Humans ; Glomerulonephritis, Membranous/diagnosis ; Receptors, Phospholipase A2 ; Loose Anagen Hair Syndrome ; Noonan Syndrome ; Autoantibodies
    Chemical Substances Receptors, Phospholipase A2 ; Autoantibodies
    Language English
    Publishing date 2023-12-26
    Publishing country Australia
    Document type Journal Article
    ZDB-ID 1470376-2
    ISSN 1442-200X ; 1328-8067
    ISSN (online) 1442-200X
    ISSN 1328-8067
    DOI 10.1111/ped.15643
    Database MEDical Literature Analysis and Retrieval System OnLINE

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  2. Article ; Online: Adrenal function during long-term ACTH therapy for patients with developmental and epileptic encephalopathy.

    Ueda, Yuki / Fujishige, Shuta / Goto, Takeru / Kimura, Shuhei / Namatame, Noriko / Narugami, Masashi / Nakakubo, Sachiko / Nakajima, Midori / Egawa, Kiyoshi / Kaneko, Naoya / Nakayama, Kanako / Hishimura, Nozomi / Yamaguchi, Takeshi / Nakamura, Akie / Shiraishi, Hideaki

    Epilepsia open

    2021  Volume 7, Issue 1, Page(s) 194–200

    Abstract: Some patients with developmental and epileptic encephalopathy (DEE) respond to adrenocorticotropic hormone (ACTH) therapy but relapse soon after. While long-term ACTH therapy (LT-ACTH) has been attempted for these patients, no previous studies have ... ...

    Abstract Some patients with developmental and epileptic encephalopathy (DEE) respond to adrenocorticotropic hormone (ACTH) therapy but relapse soon after. While long-term ACTH therapy (LT-ACTH) has been attempted for these patients, no previous studies have carefully assessed adrenal function during LT-ACTH. We evaluated the effectiveness of LT-ACTH, as well as adverse effects (AE), including their adrenal function in three DEE patients. Patients underwent a corticotropin-releasing hormone (CRH) stimulation test during LT-ACTH, and those with peak serum cortisol below 15 μg/dL were considered to be at high risk of adrenal insufficiency (AI). Two of three responded, and their life-threatening seizures with postgeneralized electroencephalogram (EEG) suppression decreased. Although no individuals had serious AE, CRH stimulation test revealed relatively weak responses, without reaching normal cortisol peak level (18 μg/dL). Hydrocortisone replacement during stress was prepared in a case with lower cortisol peak than our cutoff level. LT-ACTH could be a promising treatment option for cases of DEE that relapse soon after effective ACTH treatment. The longer duration and larger cumulative dosage in LT-ACTH than in conventional ACTH could increase the relative risk of AI. Careful evaluation with pediatric endocrinologists, including hormonal stimulation tests, might be useful for continuing this treatment safely.
    MeSH term(s) Adrenal Insufficiency/chemically induced ; Adrenal Insufficiency/drug therapy ; Adrenocorticotropic Hormone/therapeutic use ; Brain Diseases ; Child ; Corticotropin-Releasing Hormone ; Humans ; Hydrocortisone/therapeutic use
    Chemical Substances Adrenocorticotropic Hormone (9002-60-2) ; Corticotropin-Releasing Hormone (9015-71-8) ; Hydrocortisone (WI4X0X7BPJ)
    Language English
    Publishing date 2021-12-14
    Publishing country United States
    Document type Journal Article
    ISSN 2470-9239
    ISSN (online) 2470-9239
    DOI 10.1002/epi4.12566
    Database MEDical Literature Analysis and Retrieval System OnLINE

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