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      Case Report: Guanfacine and methylphenidate improved chronic lower back pain in autosomal dominant polycystic kidney disease with comorbid attention deficit hyperactivity disorder and autism spectrum disorder

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          Abstract

          Autosomal dominant polycystic kidney disease (ADPKD) is an inherited renal disease characterized by the bilateral development of multiple cysts in the kidneys. Pain management is a clinically important issue, especially because approximately 60% of patients with ADPKD experience chronic pain related to hemorrhage from renal cysts, which significantly reduces their daily life. The cystic fibrosis transmembrane conductance regulator, the molecule responsible for cyst formation in ADPKD, is also the cause of cystic fibrosis. Since attention deficit hyperactivity disorder (ADHD) is known to occur frequently in conjunction with cystic fibrosis, ADPKD may be associated with ADHD. However, to our knowledge, no study has investigated 1) ADHD or autism spectrum disorder (ASD) as comorbidities with ADPKD, 2) the effects of ADHD medications on chronic pain in ADPKD, or 3) cerebral blood flow corresponding to guanfacine (GF) or methylphenidate (MP) treatment for chronic pain. We report the case of a 15-year-old girl with ADPKD, who had chronic back pain associated with ADPKD and had to withdraw from high school because the pain interfered with her daily life. Although she took antihypertensive medications to prevent bleeding, they did not provide adequate blood pressure control. The patient was referred to a child psychiatrist and diagnosed with ASD; however, the pain did not improve. Subsequently, she was referred to our pain center. The diagnosis of ADHD was confirmed and treatment with ADHD medications was initiated. Monotherapy with MP, atomoxetine, and GF resulted in hypertension and hypotension as side effects; however, a combination of MP 18 mg and GF 4 mg provided pain relief and moderate blood pressure control, and the patient was able to go on to college. During the course of treatment, there was an improvement in the distribution of cerebral blood flow in the prefrontal and insular cortices. Confirmation of an ADHD diagnosis comorbid with ASD enabled the use of ADHD medications. The combination of MP and GF improved chronic back pain and high blood pressure due to ADPKD and cerebral blood flow. Screening for ADHD is important in the treatment of ADPKD.

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          A self-assessment scale has been developed and found to be a reliable instrument for detecting states of depression and anxiety in the setting of an hospital medical outpatient clinic. The anxiety and depressive subscales are also valid measures of severity of the emotional disorder. It is suggested that the introduction of the scales into general hospital practice would facilitate the large task of detection and management of emotional disorder in patients under investigation and treatment in medical and surgical departments.
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            The validity of the Hospital Anxiety and Depression Scale. An updated literature review.

            To review the literature of the validity of the Hospital Anxiety and Depression Scale (HADS). A review of the 747 identified papers that used HADS was performed to address the following questions: (I) How are the factor structure, discriminant validity and the internal consistency of HADS? (II) How does HADS perform as a case finder for anxiety disorders and depression? (III) How does HADS agree with other self-rating instruments used to rate anxiety and depression? Most factor analyses demonstrated a two-factor solution in good accordance with the HADS subscales for Anxiety (HADS-A) and Depression (HADS-D), respectively. The correlations between the two subscales varied from.40 to.74 (mean.56). Cronbach's alpha for HADS-A varied from.68 to.93 (mean.83) and for HADS-D from.67 to.90 (mean.82). In most studies an optimal balance between sensitivity and specificity was achieved when caseness was defined by a score of 8 or above on both HADS-A and HADS-D. The sensitivity and specificity for both HADS-A and HADS-D of approximately 0.80 were very similar to the sensitivity and specificity achieved by the General Health Questionnaire (GHQ). Correlations between HADS and other commonly used questionnaires were in the range.49 to.83. HADS was found to perform well in assessing the symptom severity and caseness of anxiety disorders and depression in both somatic, psychiatric and primary care patients and in the general population.
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              Analysis of the human tissue-specific expression by genome-wide integration of transcriptomics and antibody-based proteomics.

              Global classification of the human proteins with regards to spatial expression patterns across organs and tissues is important for studies of human biology and disease. Here, we used a quantitative transcriptomics analysis (RNA-Seq) to classify the tissue-specific expression of genes across a representative set of all major human organs and tissues and combined this analysis with antibody-based profiling of the same tissues. To present the data, we launch a new version of the Human Protein Atlas that integrates RNA and protein expression data corresponding to ∼80% of the human protein-coding genes with access to the primary data for both the RNA and the protein analysis on an individual gene level. We present a classification of all human protein-coding genes with regards to tissue-specificity and spatial expression pattern. The integrative human expression map can be used as a starting point to explore the molecular constituents of the human body.
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                Author and article information

                Contributors
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                URI : https://loop.frontiersin.org/people/2393872/overviewRole: Role:
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                Journal
                Front Pediatr
                Front Pediatr
                Front. Pediatr.
                Frontiers in Pediatrics
                Frontiers Media S.A.
                2296-2360
                01 November 2023
                2023
                : 11
                : 1283823
                Affiliations
                [ 1 ]Department of Anesthesiology and Pain Relief Center, The University of Tokyo Hospital , Tokyo, Japan
                [ 2 ]Department of Pain Medicine, Fukushima Medical University School of Medicine , Fukushima, Japan
                [ 3 ]Department of Pediatrics, Graduate School of Medicine, The University of Tokyo , Tokyo, Japan
                [ 4 ]Department of Pediatric Nephrology, Tokyo Women’s Medical University , Tokyo, Japan
                [ 5 ]Institute for Quantum Medical Science, National Institutes for Quantum Science and Technology , Chiba, Japan
                [ 6 ]Department of Neuropsychiatry, The University of Tokyo Hospital , Tokyo, Japan
                [ 7 ]Nursing Department, The University of Tokyo Hospital , Tokyo, Japan
                [ 8 ]Institute of Engineering Innovation, School of Engineering, The University of Tokyo , Tokyo, Japan
                [ 9 ]Department of Psychiatry, Aizu Medical Center, Fukushima Medical University , Fukushima, Japan
                Author notes

                Edited by: Lovro Lamot, University of Zagreb, Croatia

                Reviewed by: Ghazal Zahed, Shahid Beheshti University, Iran Ivana Trutin, Clinical hospital centre Sestre milosrdnice, Croatia

                [* ] Correspondence: Satoshi Kasahara namahagenator@ 123456gmail.com

                Abbreviations ADPKD, autosomal dominant polycystic kidney disease; ATX, atomoxetine; CFTR, cystic fibrosis transmembrane conductance regulator; GF, guanfacine; MP, methylphenidate.

                Article
                10.3389/fped.2023.1283823
                10646415
                0115a6ec-1426-4438-8615-f3ba1e519f0c
                © 2023 Kasahara, Kanda, Takahashi, Fujioka, Morita, Matsudaira, Sato, Hattori, Momose, Niwa and Uchida.

                This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) and the copyright owner(s) are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.

                History
                : 27 August 2023
                : 17 October 2023
                Page count
                Figures: 4, Tables: 0, Equations: 0, References: 47, Pages: 0, Words: 0
                Funding
                Funded by: Grant-in-Aid for Scientific Research
                Award ID:  
                Funded by: Japan Society for the Promotion of Science
                Award ID: 20K07755
                The author(s) declare financial support was received for the research, authorship, and/or publication of this article.
                This study was supported by a Grant-in-Aid for Scientific Research (C) from the Japan Society for the Promotion of Science (Grant number: 20K07755).
                Categories
                Pediatrics
                Case Report
                Custom metadata
                Pediatric Nephrology

                autosomal dominant polycystic kidney disease,lower back pain,cystic fibrosis transmembrane conductance regulator,attention deficit hyperactivity disorder,autism spectrum disorder,guanfacine,methylphenidate,cerebral blood flow

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