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1. Biallelic human SHARPIN loss of function induces autoinflammation and immunodeficiency

Author

Oda, Hirotsugu, Manthiram, Kalpana, Chavan, Pallavi Pimpale, Rieser, Eva, Veli, Önay, Kaya, Öykü, Rauch, Charles, Nakabo, Shuichiro, Kuehn, Hye Sun, Swart, Mariël, Wang, Yanli, Çelik, Nisa Ilgim, Molitor, Anne, Ziaee, Vahid, Movahedi, Nasim, Shahrooei, Mohammad, Parvaneh, Nima, Alipour-olyei, Nasrin, Carapito, Raphael, Xu, Qin, Preite, Silvia, Beck, David B., Chae, Jae Jin, Nehrebecky, Michele, Ombrello, Amanda K., Hoffmann, Patrycja, Romeo, Tina, Deuitch, Natalie T., Matthíasardóttir, Brynja, Mullikin, James, Komarow, Hirsh, Stoddard, Jennifer, Niemela, Julie, Dobbs, Kerry, Sweeney, Colin L., Anderton, Holly, Lawlor, Kate E., Yoshitomi, Hiroyuki, Yang, Dan, Boehm, Manfred, Davis, Jeremy, Mudd, Pamela, Randazzo, Davide, Tsai, Wanxia Li, Gadina, Massimo, Kaplan, Mariana J., Toguchida, Junya, Mayer, Christian T., Rosenzweig, Sergio D., Notarangelo, Luigi D., Iwai, Kazuhiro, Silke, John, Schwartzberg, Pamela L., Boisson, Bertrand, Casanova, Jean-Laurent, Bahram, Seiamak, Rao, Anand Prahalad, Peltzer, Nieves, Walczak, Henning, Lalaoui, Najoua, Aksentijevich, Ivona, and Kastner, Daniel L.

Published
2024
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2. Variant STAT4 and Response to Ruxolitinib in an Autoinflammatory Syndrome

Author

Baghdassarian, Hratch, Blackstone, Sarah A, Clay, Owen S, Philips, Rachael, Matthiasardottir, Brynja, Nehrebecky, Michele, Hua, Vivian K, McVicar, Rachael, Liu, Yang, Tucker, Suzanne M, Randazzo, Davide, Deuitch, Natalie, Rosenzweig, Sofia, Mark, Adam, Sasik, Roman, Fisch, Kathleen M, Pimpale Chavan, Pallavi, Eren, Elif, Watts, Norman R, Ma, Chi A, Gadina, Massimo, Schwartz, Daniella M, Sanyal, Anwesha, Werner, Giffin, Murdock, David R, Horita, Nobuyuki, Chowdhury, Shimul, Dimmock, David, Jepsen, Kristen, Remmers, Elaine F, Goldbach-Mansky, Raphaela, Gahl, William A, O'Shea, John J, Milner, Joshua D, Lewis, Nathan E, Chang, Johanna, Kastner, Daniel L, Torok, Kathryn, Oda, Hirotsugu, Putnam, Christopher D, and Broderick, Lori

Subjects
Biomedical and Clinical Sciences, Clinical Sciences, Genetics, Biotechnology, Clinical Research, Aetiology, 2.1 Biological and endogenous factors, Inflammatory and immune system, Janus Kinases, Nitriles, Pyrazoles, Pyrimidines, Scleroderma, Systemic, Autoimmune Diseases, Mutation, Missense, Gain of Function Mutation, Dermatologic Agents, Anti-Inflammatory Agents, Medical and Health Sciences, General & Internal Medicine, Biomedical and clinical sciences, Health sciences
Abstract

BackgroundDisabling pansclerotic morphea (DPM) is a rare systemic inflammatory disorder, characterized by poor wound healing, fibrosis, cytopenias, hypogammaglobulinemia, and squamous-cell carcinoma. The cause is unknown, and mortality is high.MethodsWe evaluated four patients from three unrelated families with an autosomal dominant pattern of inheritance of DPM. Genomic sequencing independently identified three heterozygous variants in a specific region of the gene that encodes signal transducer and activator of transcription 4 (STAT4). Primary skin fibroblast and cell-line assays were used to define the functional nature of the genetic defect. We also assayed gene expression using single-cell RNA sequencing of peripheral-blood mononuclear cells to identify inflammatory pathways that may be affected in DPM and that may respond to therapy.ResultsGenome sequencing revealed three novel heterozygous missense gain-of-function variants in STAT4. In vitro, primary skin fibroblasts showed enhanced interleukin-6 secretion, with impaired wound healing, contraction of the collagen matrix, and matrix secretion. Inhibition of Janus kinase (JAK)-STAT signaling with ruxolitinib led to improvement in the hyperinflammatory fibroblast phenotype in vitro and resolution of inflammatory markers and clinical symptoms in treated patients, without adverse effects. Single-cell RNA sequencing revealed expression patterns consistent with an immunodysregulatory phenotype that were appropriately modified through JAK inhibition.ConclusionsGain-of-function variants in STAT4 caused DPM in the families that we studied. The JAK inhibitor ruxolitinib attenuated the dermatologic and inflammatory phenotype in vitro and in the affected family members. (Funded by the American Academy of Allergy, Asthma, and Immunology Foundation and others.).

Published
2023

6. Linkage-specific deubiquitylation by OTUD5 defines an embryonic pathway intolerant to genomic variation

Author

Beck, David B, Basar, Mohammed A, Asmar, Anthony J, Thompson, Joyce J, Oda, Hirotsugu, Uehara, Daniela T, Saida, Ken, Pajusalu, Sander, Talvik, Inga, D’Souza, Precilla, Bodurtha, Joann, Mu, Weiyi, Barañano, Kristin W, Miyake, Noriko, Wang, Raymond, Kempers, Marlies, Tamada, Tomoko, Nishimura, Yutaka, Okada, Satoshi, Kosho, Tomoki, Dale, Ryan, Mitra, Apratim, Macnamara, Ellen, Matsumoto, Naomichi, Inazawa, Johji, Walkiewicz, Magdalena, Õunap, Katrin, Tifft, Cynthia J, Aksentijevich, Ivona, Kastner, Daniel L, Rocha, Pedro P, and Werner, Achim

Subjects
Biochemistry and Cell Biology, Biomedical and Clinical Sciences, Genetics, Biological Sciences, Human Genome, Rare Diseases, Pediatric, Generic health relevance, Chromatin, Genomics, Humans, Signal Transduction, Ubiquitin, Ubiquitination, Undiagnosed Diseases Network
Abstract

Reversible modification of proteins with linkage-specific ubiquitin chains is critical for intracellular signaling. Information on physiological roles and underlying mechanisms of particular ubiquitin linkages during human development are limited. Here, relying on genomic constraint scores, we identify 10 patients with multiple congenital anomalies caused by hemizygous variants in OTUD5, encoding a K48/K63 linkage-specific deubiquitylase. By studying these mutations, we find that OTUD5 controls neuroectodermal differentiation through cleaving K48-linked ubiquitin chains to counteract degradation of select chromatin regulators (e.g., ARID1A/B, histone deacetylase 2, and HCF1), mutations of which underlie diseases that exhibit phenotypic overlap with OTUD5 patients. Loss of OTUD5 during differentiation leads to less accessible chromatin at neuroectodermal enhancers and aberrant gene expression. Our study describes a previously unidentified disorder we name LINKED (LINKage-specific deubiquitylation deficiency-induced Embryonic Defects) syndrome and reveals linkage-specific ubiquitin cleavage from chromatin remodelers as an essential signaling mode that coordinates chromatin remodeling during embryogenesis.

Published
2021

7. Mutations that prevent caspase cleavage of RIPK1 cause autoinflammatory disease

Author

Lalaoui, Najoua, Boyden, Steven E, Oda, Hirotsugu, Wood, Geryl M, Stone, Deborah L, Chau, Diep, Liu, Lin, Stoffels, Monique, Kratina, Tobias, Lawlor, Kate E, Zaal, Kristien JM, Hoffmann, Patrycja M, Etemadi, Nima, Shield-Artin, Kristy, Biben, Christine, Tsai, Wanxia Li, Blake, Mary D, Kuehn, Hye Sun, Yang, Dan, Anderton, Holly, Silke, Natasha, Wachsmuth, Laurens, Zheng, Lixin, Moura, Natalia Sampaio, Beck, David B, Gutierrez-Cruz, Gustavo, Ombrello, Amanda K, Pinto-Patarroyo, Gineth P, Kueh, Andrew J, Herold, Marco J, Hall, Cathrine, Wang, Hongying, Chae, Jae Jin, Dmitrieva, Natalia I, McKenzie, Mark, Light, Amanda, Barham, Beverly K, Jones, Anne, Romeo, Tina M, Zhou, Qing, Aksentijevich, Ivona, Mullikin, James C, Gross, Andrew J, Shum, Anthony K, Hawkins, Edwin D, Masters, Seth L, Lenardo, Michael J, Boehm, Manfred, Rosenzweig, Sergio D, Pasparakis, Manolis, Voss, Anne K, Gadina, Massimo, Kastner, Daniel L, and Silke, John

Subjects
Biochemistry and Cell Biology, Biomedical and Clinical Sciences, Biological Sciences, Biodefense, Prevention, Rare Diseases, Vaccine Related, 1.1 Normal biological development and functioning, Underpinning research, Aetiology, 2.1 Biological and endogenous factors, Inflammatory and immune system, Animals, Caspase 3, Caspase 8, Female, Hereditary Autoinflammatory Diseases, Humans, MAP Kinase Kinase Kinases, Male, Mice, Mice, Inbred C57BL, Mice, Knockout, Mutation, Pedigree, Receptor-Interacting Protein Serine-Threonine Kinases, General Science & Technology
Abstract

RIPK1 is a key regulator of innate immune signalling pathways. To ensure an optimal inflammatory response, RIPK1 is regulated post-translationally by well-characterized ubiquitylation and phosphorylation events, as well as by caspase-8-mediated cleavage1-7. The physiological relevance of this cleavage event remains unclear, although it is thought to inhibit activation of RIPK3 and necroptosis8. Here we show that the heterozygous missense mutations D324N, D324H and D324Y prevent caspase cleavage of RIPK1 in humans and result in an early-onset periodic fever syndrome and severe intermittent lymphadenopathy-a condition we term 'cleavage-resistant RIPK1-induced autoinflammatory syndrome'. To define the mechanism for this disease, we generated a cleavage-resistant Ripk1D325A mutant mouse strain. Whereas Ripk1-/- mice died postnatally from systemic inflammation, Ripk1D325A/D325A mice died during embryogenesis. Embryonic lethality was completely prevented by the combined loss of Casp8 and Ripk3, but not by loss of Ripk3 or Mlkl alone. Loss of RIPK1 kinase activity also prevented Ripk1D325A/D325A embryonic lethality, although the mice died before weaning from multi-organ inflammation in a RIPK3-dependent manner. Consistently, Ripk1D325A/D325A and Ripk1D325A/+ cells were hypersensitive to RIPK3-dependent TNF-induced apoptosis and necroptosis. Heterozygous Ripk1D325A/+ mice were viable and grossly normal, but were hyper-responsive to inflammatory stimuli in vivo. Our results demonstrate the importance of caspase-mediated RIPK1 cleavage during embryonic development and show that caspase cleavage of RIPK1 not only inhibits necroptosis but also maintains inflammatory homeostasis throughout life.

Published
2020

9. Antiviral effects of bovine lactoferrin on human norovirus

Author

Oda, Hirotsugu, Kolawole, Abimbola O., Mirabelli, Carmen, Wakabayashi, Hiroyuki, Tanaka, Miyuki, Yamauchi, Koji, Abe, Fumiaki, and Wobus, Christiane E.

Subjects
Norovirus -- Control, Antiviral agents -- Evaluation, Lactoferrins -- Usage -- Health aspects, Biological sciences
Abstract

Human noroviruses cause significant morbidity and mortality worldwide, but lack approved antivirals or vaccines to treat or prevent infections. The recent development of two cell culture systems in human transformed B cells (BJABs) and non-transformed human intestinal enteroid cultures overcomes a main limitation in identifying molecules with anti-norovirus activities. Lactoferrin is an iron-binding glycoprotein found in the milk of most mammals, with broad spectrum antimicrobial activities, including against the related murine norovirus in cell culture. In a Japanese clinical trial, ingestion of lactoferrin reduced the incidence of infectious gastroenteritis in the participants. Because human noroviruses were the most common cause of gastroenteritis in Japan during the clinical trial period, we sought to determine whether lactoferrin could inhibit infection with human norovirus. Our study, using a B cell culture model, demonstrates that lactoferrin reduces human norovirus infection. The mechanism of antiviral action is likely indirect and may involve the induction of innate interferon responses. Therefore, future studies are warranted to test the antiviral efficacy of lactoferrin against human norovirus infection in patients. Key words: human norovirus, lactoferrin, interferon, human B cell. Les norovirus humains sont responsables d'une morbidite et d'une mortalite importantes a travers le monde, mais il n'existe pas d'antiviraux ou de vaccins approuves pour traiter ou prevenir les infections. Le developpement recent de deux systemes de cultures cellulaires consistant en cellules B humaines transformees (BJAB) et en enteroides intestinaux humains non transformes permet de surmonter une des principales limites a l'identification de molecules possedant des activites anti-norovirus. La lactoferrine est une glycoproteine qui lie le fer et que l'on trouve dans le lait de la plupart des mammiferes. Elle possede un large spectre d'activites antimicrobiennes, y compris en culture cellulaire, contre le norovirus murin apparente. Son ingestion reduisait aussi l'incidence de gastroenterites infectieuses chez des participants a une etude clinique au Japon. Puisque les norovirus humains constituaient la cause la plus frequente de gastroenterite au Japon au cours de la periode couverte par l'etude clinique, les auteurs desiraient determiner si la lactoferrine pouvait inhiber l'infection au norovirus humain. Leur etude demontre que la lactoferrine reduit l'infection au norovirus humain dans le modele de cellules B en culture. Le mecanisme d'action antivirale est probablement indirect et peut impliquer l'induction de reponses innees a l'interferon. Consequemment, des etudes futures visant a tester l'efficacite antivirale de la lactoferrine contre l'infection aux norovirus humains chez les patients sont justifiees. [Traduit par la Redaction] Mots-cles : norovirus humain, lactoferrine, interferon, cellule B humaine., Introduction Human noroviruses (HuNoVs) are non-enveloped, positive-sense, single-stranded RNA viruses. They are classified into 10 genogroups and further subdivided into genotypes, of which 5 genotypes encompass over 40 genotypes that [...]

Published
2021
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12. Biallelic human SHARPINloss of function induces autoinflammation and immunodeficiency

Author

Oda, Hirotsugu, Manthiram, Kalpana, Chavan, Pallavi Pimpale, Rieser, Eva, Veli, Önay, Kaya, Öykü, Rauch, Charles, Nakabo, Shuichiro, Kuehn, Hye Sun, Swart, Mariël, Wang, Yanli, Çelik, Nisa Ilgim, Molitor, Anne, Ziaee, Vahid, Movahedi, Nasim, Shahrooei, Mohammad, Parvaneh, Nima, Alipour-olyei, Nasrin, Carapito, Raphael, Xu, Qin, Preite, Silvia, Beck, David B., Chae, Jae Jin, Nehrebecky, Michele, Ombrello, Amanda K., Hoffmann, Patrycja, Romeo, Tina, Deuitch, Natalie T., Matthíasardóttir, Brynja, Mullikin, James, Komarow, Hirsh, Stoddard, Jennifer, Niemela, Julie, Dobbs, Kerry, Sweeney, Colin L., Anderton, Holly, Lawlor, Kate E., Yoshitomi, Hiroyuki, Yang, Dan, Boehm, Manfred, Davis, Jeremy, Mudd, Pamela, Randazzo, Davide, Tsai, Wanxia Li, Gadina, Massimo, Kaplan, Mariana J., Toguchida, Junya, Mayer, Christian T., Rosenzweig, Sergio D., Notarangelo, Luigi D., Iwai, Kazuhiro, Silke, John, Schwartzberg, Pamela L., Boisson, Bertrand, Casanova, Jean-Laurent, Bahram, Seiamak, Rao, Anand Prahalad, Peltzer, Nieves, Walczak, Henning, Lalaoui, Najoua, Aksentijevich, Ivona, and Kastner, Daniel L.

Abstract

The linear ubiquitin assembly complex (LUBAC) consists of HOIP, HOIL-1 and SHARPIN and is essential for proper immune responses. Individuals with HOIP and HOIL-1 deficiencies present with severe immunodeficiency, autoinflammation and glycogen storage disease. In mice, the loss of Sharpinleads to severe dermatitis due to excessive keratinocyte cell death. Here, we report two individuals with SHARPIN deficiency who manifest autoinflammatory symptoms but unexpectedly no dermatological problems. Fibroblasts and B cells from these individuals showed attenuated canonical NF-κB responses and a propensity for cell death mediated by TNF superfamily members. Both SHARPIN-deficient and HOIP-deficient individuals showed a substantial reduction of secondary lymphoid germinal center B cell development. Treatment of one SHARPIN-deficient individual with anti-TNF therapies led to complete clinical and transcriptomic resolution of autoinflammation. These findings underscore the critical function of the LUBAC as a gatekeeper for cell death-mediated immune dysregulation in humans.

Published
2024
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13. Effects of lactoferrin on the production of interferon-[lambda] by the human intestinal epithelial cell line HT-29

Author

Shin, Kouichirou, Oda, Hirotsugu, Wakabayashi, Hiroyuki, Yamauchi, Koji, and Abe, Fumiaki

Subjects
Intestines -- Health aspects, Lactoferrin -- Health aspects, Interferon -- Health aspects, Epithelial cells -- Health aspects, Biological sciences
Abstract

Abstract: We examined the in-vitro effects of bovine lactoferrin (LF) on the production of interferon-[lambda] (IFN-[lambda]), an antiviral cytokine important for the defense of enterocytes, using the human intestinal epithelial [...]

Published
2017
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15. Human LUBAC deficiency leads to autoinflammation and immunodeficiency by dysregulation in TNF-mediated cell death

Author

Oda, Hirotsugu, primary, Manthiram, Kalpana, additional, Chavan, Pallavi Pimpale, additional, Nakabo, Shuichiro, additional, Kuehn, Hye Sun, additional, Beck, David B., additional, Chae, Jae Jin, additional, Nehrebecky, Michele, additional, Ombrello, Amanda K., additional, Romeo, Tina, additional, Deuitch, Natalie, additional, Matthíasardóttir, Brynja, additional, Mullikin, Jim, additional, Stoddard, Jennifer, additional, Niemela, Julie, additional, Anderton, Holly, additional, Lawlor, Kate E., additional, Yoshitomi, Hiroyuki, additional, Yang, Dan, additional, Boehm, Manfred, additional, Davis, Jeremy, additional, Mudd, Pamela, additional, Randazzo, Davide, additional, Tsai, Wanxia Li, additional, Gadina, Massimo, additional, Kaplan, Mariana J., additional, Toguchida, Junya, additional, Mayer, Christian, additional, Rosenzweig, Sergio D., additional, Iwai, Kazuhiro, additional, Silke, John, additional, Boisson, Bertrand, additional, Casanova, Jean-Laurent, additional, Rao, Anand, additional, Lalaoui, Najoua, additional, Aksentijevich, Ivona, additional, and Kastner, Daniel L., additional

Published
2022
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19. Identification of a High‐Frequency Somatic NLRC4 Mutation as a Cause of Autoinflammation by Pluripotent Cell–Based Phenotype Dissection

Author

Kawasaki, Yuri, Oda, Hirotsugu, Ito, Jun, Niwa, Akira, Tanaka, Takayuki, Hijikata, Atsushi, Seki, Ryosuke, Nagahashi, Ayako, Osawa, Mitsujiro, Asaka, Isao, Watanabe, Akira, Nishimata, Shigeo, Shirai, Tsuyoshi, Kawashima, Hisashi, Ohara, Osamu, Nakahata, Tatsutoshi, Nishikomori, Ryuta, Heike, Toshio, and Saito, Megumu K.

Published
2017
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21. Effects of Lactoferrin on Sleep Conditions in Children Aged 12–32 Months: A Preliminary, Randomized, Double-Blind, Placebo-Controlled Trial

Author

Miyakawa, Momoko, Kubo, Shutaro, Oda, Hirotsugu, Motoki, Noriko, Mizuki, Masaru, Tsukahara, Teruomi, Tanaka, Miyuki, Yamauchi, Koji, Abe, Fumiaki, and Nomiyama, Tetsuo

Subjects
children, Clinical Trial Report, sleep, growing-up formula, lactoferrin
Abstract

Purpose To investigate preliminarily the effect of lactoferrin (LF)-fortified formula on sleep conditions in children. Study Design A preliminary, randomized, double-blind, placebo-controlled trial. Methods Healthy children between the ages of 12 and 32 months who attended nursery schools in Japan were divided into two groups and assigned a placebo or LF (48 mg/day)-fortified formula. Children’s sleep conditions were investigated before and after the 13-week intervention using the Japanese Sleep Questionnaire for Preschoolers (JSQ-P). Results Altogether, 109 participants were randomized. Eight participants were eliminated due to lost to follow-up, withdrawal of consent, and ineligibility, with 101 participants (placebo, n = 48; LF, n = 53) included in the full analysis set (FAS) and used for analysis. Wake-up time, bedtime, and nighttime sleep were comparable between the two groups before and after intervention. The change in total JSQ-P T scores tended to improve in the LF group (placebo vs LF: 0.5 ± 6.5 vs −1.9 ± 6.1, p = 0.074), in particular, morning symptoms significantly improved (grumpy in the morning, hard to wake-up, and hard to get out of bed) (placebo vs LF: 0.8 ± 6.2 vs −1.9 ± 6.2, p = 0.028). A better trend was also observed in the LF group regarding restless legs syndrome (RLS)-motor (rubs feet at night and touches feet at night) (placebo vs LF: 2.3 ± 10.7 vs −0.6 ± 13.5, p = 0.083) and insufficient sleep (stays up more than one hour later the day before a holiday and wakes up more than one hour later on a holiday) (placebo vs LF: 0.1 ± 9.8 vs −1.7 ± 8.8, p = 0.095). No adverse drug reactions were found. Conclusion LF intake may improve sleep condition, especially morning symptoms in children above one year of age.

Published
2020

26. Enhanced Chondrogenesis of Induced Pluripotent Stem Cells From Patients With Neonatal-Onset Multisystem Inflammatory Disease Occurs via the Caspase 1–Independent cAMP/Protein Kinase A/CREB Pathway

Author

Yokoyama, Koji, Ikeya, Makoto, Umeda, Katsutsugu, Oda, Hirotsugu, Nodomi, Seishiro, Nasu, Akira, Matsumoto, Yoshihisa, Izawa, Kazushi, Horigome, Kazuhiko, Kusaka, Toshimasa, Tanaka, Takayuki, Saito, Megumu K., Yasumi, Takahiro, Nishikomori, Ryuta, Ohara, Osamu, Nakayama, Naoki, Nakahata, Tatsutoshi, Heike, Toshio, and Toguchida, Junya

Published
2015
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27. Linkage-specific deubiquitylation by OTUD5 defines an embryonic pathway intolerant to genomic variation

Author

Beck, David B., Basar, Mohammed A., Asmar, Anthony J., Thompson, Joyce J., Oda, Hirotsugu, Uehara, Daniela T., Kempers, M.J.E., Rocha, Pedro P., Werner, Achim, Beck, David B., Basar, Mohammed A., Asmar, Anthony J., Thompson, Joyce J., Oda, Hirotsugu, Uehara, Daniela T., Kempers, M.J.E., Rocha, Pedro P., and Werner, Achim

Abstract

Contains fulltext : 230561.pdf (publisher's version ) (Open Access)

Published
2021

29. A Possible Association Between a Nucleotide‐Binding Domain LRR‐Containing Protein Family PYD‐Containing Protein 1 Mutation and an Autoinflammatory Disease Involving Liver Cirrhosis

Author

Yasudo, Hiroki, primary, Ando, Taiki, additional, Maehara, Akie, additional, Ando, Tomoaki, additional, Izawa, Kumi, additional, Tanabe, Atsushi, additional, Kaitani, Ayako, additional, Nomura, Shigeru, additional, Seki, Masafumi, additional, Yoshida, Kenichi, additional, Oda, Hirotsugu, additional, Okamoto, Yoko, additional, Wang, Hexing, additional, Kamei, Anna, additional, Kojima, Mayuki, additional, Kimura, Meiko, additional, Uchida, Koichiro, additional, Nakano, Nobuhiro, additional, Kaneko, Junichi, additional, Ebihara, Nobuyuki, additional, Hasegawa, Kiyoshi, additional, Shimizu, Toshiaki, additional, Takita, Junko, additional, Ogawa, Hideoki, additional, Okumura, Ko, additional, Ogawa, Seishi, additional, Tamura, Naoto, additional, and Kitaura, Jiro, additional

Published
2021
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31. Effects of Lactoferrin on Sleep Conditions in Children Aged 12–32 Months: A Preliminary, Randomized, Double-Blind, Placebo-Controlled Trial

Author

Miyakawa,Momoko, Kubo,Shutaro, Oda,Hirotsugu, Motoki,Noriko, Mizuki,Masaru, Tsukahara,Teruomi, Tanaka,Miyuki, Yamauchi,Koji, Abe,Fumiaki, Nomiyama,Tetsuo, Miyakawa,Momoko, Kubo,Shutaro, Oda,Hirotsugu, Motoki,Noriko, Mizuki,Masaru, Tsukahara,Teruomi, Tanaka,Miyuki, Yamauchi,Koji, Abe,Fumiaki, and Nomiyama,Tetsuo

Abstract

Momoko Miyakawa,1 Shutaro Kubo,1 Hirotsugu Oda,1 Noriko Motoki,2 Masaru Mizuki,2 Teruomi Tsukahara,2 Miyuki Tanaka,1 Koji Yamauchi,1 Fumiaki Abe,1 Tetsuo Nomiyama2 1Food Ingredients and Technology Institute, R&D Division, Morinaga Milk Industry Co., Ltd, Zama, Kanagawa, Japan; 2Department of Preventive Medicine and Public Health, Shinshu University School of Medicine, Matsumoto, Nagano, JapanCorrespondence: Momoko MiyakawaFood Ingredients and Technology Institute, R&D Division, Morinaga Milk Industry Co., Ltd., 1-83, 5-Chome, Higashihara, Zama-City, Kanagawa-Pref. 252-8583, JapanTel +81 46252 3045Fax +81 46252 3017Email mo-miyakawa@morinagamilk.co.jpPurpose: To investigate preliminarily the effect of lactoferrin (LF)-fortified formula on sleep conditions in children.Study Design: A preliminary, randomized, double-blind, placebo-controlled trial.Methods: Healthy children between the ages of 12 and 32 months who attended nursery schools in Japan were divided into two groups and assigned a placebo or LF (48 mg/day)-fortified formula. Children’s sleep conditions were investigated before and after the 13-week intervention using the Japanese Sleep Questionnaire for Preschoolers (JSQ-P).Results: Altogether, 109 participants were randomized. Eight participants were eliminated due to lost to follow-up, withdrawal of consent, and ineligibility, with 101 participants (placebo, n = 48; LF, n = 53) included in the full analysis set (FAS) and used for analysis. Wake-up time, bedtime, and nighttime sleep were comparable between the two groups before and after intervention. The change in total JSQ-P T scores tended to improve in the LF group (placebo vs LF: 0.5 ± 6.5 vs − 1.9 ± 6.1, p = 0.074), in particular, morning symptoms significantly improved (grumpy in the morning, hard to wake-up, and hard to get out of bed) (placebo vs LF: 0.8 ± 6.2 vs − 1.9 ± 6.2, p = 0.028). A better trend was also

Published
2020

32. Effects of Lactoferrin on Subjective Skin Conditions in Winter: A Preliminary, Randomized, Double-Blinded, Placebo-Controlled Trial

Author

Oda, Hirotsugu, Miyakawa, Momoko, Mizuki, Masaru, Misawa, Yuka, Tsukahara, Teruomi, Tanaka, Miyuki, Yamauchi, Koji, Abe, Fumiaki, and Nomiyama, Tetsuo

Subjects
skin, Clinical, Cosmetic and Investigational Dermatology, Clinical Trial Report, moisture, winter, texture, lactoferrin
Abstract

Hirotsugu Oda,1 Momoko Miyakawa,1 Masaru Mizuki,2 Yuka Misawa,3 Teruomi Tsukahara,2 Miyuki Tanaka,1 Koji Yamauchi,1 Fumiaki Abe,1 Tetsuo Nomiyama2 1Food Ingredients and Technology Institute, R&D Division, Morinaga Milk Industry Co., Ltd, Zama, Kanagawa 252-8583, Japan; 2Department of Preventive Medicine and Public Health, Shinshu University School of Medicine, Matsumoto, Nagano 390-8621, Japan; 3Department of Rehabilitation, Nagano Children’s Hospital, Nagano 399-8288, JapanCorrespondence: Hirotsugu OdaFood Ingredients and Technology Institute, R&D Division, Morinaga Milk Industry Co., Ltd., 5-1-83, Zama, Kanagawa 252-8583, JapanTel +81 46 252 3045Fax +81 46 252 3017Email h-oda@morinagamilk.co.jpObjective: To investigate the effects of lactoferrin (LF) on subjective skin conditions in winter.Design: A preliminary, randomized, double-blinded, placebo-controlled trial.Setting and subjects: Healthy adultsin Japan.Interventions: Intake of placebo, 200 mg, or 600 mg of LF for 12 weeks in winter.Endpoints: Changes in the scores of subjective skin conditions.Results: Three hundred and forty-six subjects were randomized. Nine subjects (placebo, n=0; 200 mg, n=5; 600 mg, n=4) withdrew consent, and 7 subjects (placebo, n=4; 200 mg, n=2; 600 mg, n=1) were lost to follow-up, resulting in 330 for a full analysis set.Outcomes: Changes in the scores of moisture were greater in the 600 mg group than in the placebo group. Changes in the scores of moisture were greater in the 200 mg and 600 mg groups, and of texture were greater in the 600 mg group than in the placebo group in female subjects.Conclusion: Intake of LF may improve moisture or texture of skin in winter.Keywords: lactoferrin, winter, skin, moisture, texture &nbsp

Published
2019

33. Somatic Mutations in UBA1 and Severe Adult-Onset Autoinflammatory Disease

Author

Beck, David B., primary, Ferrada, Marcela A., additional, Sikora, Keith A., additional, Ombrello, Amanda K., additional, Collins, Jason C., additional, Pei, Wuhong, additional, Balanda, Nicholas, additional, Ross, Daron L., additional, Ospina Cardona, Daniela, additional, Wu, Zhijie, additional, Patel, Bhavisha, additional, Manthiram, Kalpana, additional, Groarke, Emma M., additional, Gutierrez-Rodrigues, Fernanda, additional, Hoffmann, Patrycja, additional, Rosenzweig, Sofia, additional, Nakabo, Shuichiro, additional, Dillon, Laura W., additional, Hourigan, Christopher S., additional, Tsai, Wanxia L., additional, Gupta, Sarthak, additional, Carmona-Rivera, Carmelo, additional, Asmar, Anthony J., additional, Xu, Lisha, additional, Oda, Hirotsugu, additional, Goodspeed, Wendy, additional, Barron, Karyl S., additional, Nehrebecky, Michele, additional, Jones, Anne, additional, Laird, Ryan S., additional, Deuitch, Natalie, additional, Rowczenio, Dorota, additional, Rominger, Emily, additional, Wells, Kristina V., additional, Lee, Chyi-Chia R., additional, Wang, Weixin, additional, Trick, Megan, additional, Mullikin, James, additional, Wigerblad, Gustaf, additional, Brooks, Stephen, additional, Dell’Orso, Stefania, additional, Deng, Zuoming, additional, Chae, Jae J., additional, Dulau-Florea, Alina, additional, Malicdan, May C.V., additional, Novacic, Danica, additional, Colbert, Robert A., additional, Kaplan, Mariana J., additional, Gadina, Massimo, additional, Savic, Sinisa, additional, Lachmann, Helen J., additional, Abu-Asab, Mones, additional, Solomon, Benjamin D., additional, Retterer, Kyle, additional, Gahl, William A., additional, Burgess, Shawn M., additional, Aksentijevich, Ivona, additional, Young, Neal S., additional, Calvo, Katherine R., additional, Werner, Achim, additional, Kastner, Daniel L., additional, and Grayson, Peter C., additional

Published
2020
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37. Effects of Lactoferrin-Fortified Formula on Acute Gastrointestinal Symptoms in Children Aged 12–32 Months: A Randomized, Double-Blind, Placebo-Controlled Trial

Author

Motoki, Noriko, primary, Mizuki, Masaru, additional, Tsukahara, Teruomi, additional, Miyakawa, Momoko, additional, Kubo, Shutaro, additional, Oda, Hirotsugu, additional, Tanaka, Miyuki, additional, Yamauchi, Koji, additional, Abe, Fumiaki, additional, and Nomiyama, Tetsuo, additional

Published
2020
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39. Mutations that prevent caspase cleavage of RIPK1 cause autoinflammatory disease

Author

Lalaoui, Najoua, primary, Boyden, Steven E., additional, Oda, Hirotsugu, additional, Wood, Geryl M., additional, Stone, Deborah L., additional, Chau, Diep, additional, Liu, Lin, additional, Stoffels, Monique, additional, Kratina, Tobias, additional, Lawlor, Kate E., additional, Zaal, Kristien J. M., additional, Hoffmann, Patrycja M., additional, Etemadi, Nima, additional, Shield-Artin, Kristy, additional, Biben, Christine, additional, Tsai, Wanxia Li, additional, Blake, Mary D., additional, Kuehn, Hye Sun, additional, Yang, Dan, additional, Anderton, Holly, additional, Silke, Natasha, additional, Wachsmuth, Laurens, additional, Zheng, Lixin, additional, Moura, Natalia Sampaio, additional, Beck, David B., additional, Gutierrez-Cruz, Gustavo, additional, Ombrello, Amanda K., additional, Pinto-Patarroyo, Gineth P., additional, Kueh, Andrew J., additional, Herold, Marco J., additional, Hall, Cathrine, additional, Wang, Hongying, additional, Chae, Jae Jin, additional, Dmitrieva, Natalia I., additional, McKenzie, Mark, additional, Light, Amanda, additional, Barham, Beverly K., additional, Jones, Anne, additional, Romeo, Tina M., additional, Zhou, Qing, additional, Aksentijevich, Ivona, additional, Mullikin, James C., additional, Gross, Andrew J., additional, Shum, Anthony K., additional, Hawkins, Edwin D., additional, Masters, Seth L., additional, Lenardo, Michael J., additional, Boehm, Manfred, additional, Rosenzweig, Sergio D., additional, Pasparakis, Manolis, additional, Voss, Anne K., additional, Gadina, Massimo, additional, Kastner, Daniel L., additional, and Silke, John, additional

Published
2019
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40. Effects of Lactoferrin on Subjective Skin Conditions in Winter: A Preliminary, Randomized, Double-Blinded, Placebo-Controlled Trial

Author

Oda,Hirotsugu, Miyakawa,Momoko, Mizuki,Masaru, Misawa,Yuka, Tsukahara,Teruomi, Tanaka,Miyuki, Yamauchi,Koji, Abe,Fumiaki, Nomiyama,Tetsuo, Oda,Hirotsugu, Miyakawa,Momoko, Mizuki,Masaru, Misawa,Yuka, Tsukahara,Teruomi, Tanaka,Miyuki, Yamauchi,Koji, Abe,Fumiaki, and Nomiyama,Tetsuo

Abstract

Hirotsugu Oda,1 Momoko Miyakawa,1 Masaru Mizuki,2 Yuka Misawa,3 Teruomi Tsukahara,2 Miyuki Tanaka,1 Koji Yamauchi,1 Fumiaki Abe,1 Tetsuo Nomiyama2 1Food Ingredients and Technology Institute, R&D Division, Morinaga Milk Industry Co., Ltd, Zama, Kanagawa 252-8583, Japan; 2Department of Preventive Medicine and Public Health, Shinshu University School of Medicine, Matsumoto, Nagano 390-8621, Japan; 3Department of Rehabilitation, Nagano Children’s Hospital, Nagano 399-8288, JapanCorrespondence: Hirotsugu OdaFood Ingredients and Technology Institute, R&D Division, Morinaga Milk Industry Co., Ltd., 5-1-83, Zama, Kanagawa 252-8583, JapanTel +81 46 252 3045Fax +81 46 252 3017Email h-oda@morinagamilk.co.jpObjective: To investigate the effects of lactoferrin (LF) on subjective skin conditions in winter.Design: A preliminary, randomized, double-blinded, placebo-controlled trial.Setting and subjects: Healthy adults in Japan.Interventions: Intake of placebo, 200 mg, or 600 mg of LF for 12 weeks in winter.Endpoints: Changes in the scores of subjective skin conditions.Results: Three hundred and forty-six subjects were randomized. Nine subjects (placebo, n=0; 200 mg, n=5; 600 mg, n=4) withdrew consent, and 7 subjects (placebo, n=4; 200 mg, n=2; 600 mg, n=1) were lost to follow-up, resulting in 330 for a full analysis set.Outcomes: Changes in the scores of moisture were greater in the 600 mg group than in the placebo group. Changes in the scores of moisture were greater in the 200 mg and 600 mg groups, and of texture were greater in the 600 mg group than in the placebo group in female subjects.Conclusion: Intake of LF may improve moisture or texture of skin in winter.Keywords: lactoferrin, winter, skin, moisture, texture &nbsp

Published
2019

42. Second Case of HOIP Deficiency Expands Clinical Features and Defines Inflammatory Transcriptome Regulated by LUBAC

Author

Oda, Hirotsugu, primary, Beck, David B., additional, Kuehn, Hye Sun, additional, Sampaio Moura, Natalia, additional, Hoffmann, Patrycja, additional, Ibarra, Maria, additional, Stoddard, Jennifer, additional, Tsai, Wanxia Li, additional, Gutierrez-Cruz, Gustavo, additional, Gadina, Massimo, additional, Rosenzweig, Sergio D., additional, Kastner, Daniel L., additional, Notarangelo, Luigi D., additional, and Aksentijevich, Ivona, additional

Published
2019
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43. Accurate clinical genetic testing for autoinflammatory diseases using the next-generation sequencing platform MiSeq

Author

00511891, 90634931, Nakayama, Manabu, Oda, Hirotsugu, Nakagawa, Kenji, Yasumi, Takahiro, Kawai, Tomoki, Izawa, Kazushi, Nishikomori, Ryuta, Heike, Toshio, Ohara, Osamu, 00511891, 90634931, Nakayama, Manabu, Oda, Hirotsugu, Nakagawa, Kenji, Yasumi, Takahiro, Kawai, Tomoki, Izawa, Kazushi, Nishikomori, Ryuta, Heike, Toshio, and Ohara, Osamu

Abstract

Autoinflammatory diseases occupy one of a group of primary immunodeficiency diseases that are generally thought to be caused by mutation of genes responsible for innate immunity, rather than by acquired immunity. Mutations related to autoinflammatory diseases occur in 12 genes. For example, low-level somatic mosaic NLRP3 mutations underlie chronic infantile neurologic, cutaneous, articular syndrome (CINCA), also known as neonatal-onset multisystem inflammatory disease (NOMID). In current clinical practice, clinical genetic testing plays an important role in providing patients with quick, definite diagnoses. To increase the availability of such testing, low-cost high-throughput gene-analysis systems are required, ones that not only have the sensitivity to detect even low-level somatic mosaic mutations, but also can operate simply in a clinical setting. To this end, we developed a simple method that employs two-step tailed PCR and an NGS system, MiSeq platform, to detect mutations in all coding exons of the 12 genes responsible for autoinflammatory diseases. Using this amplicon sequencing system, we amplified a total of 234 amplicons derived from the 12 genes with multiplex PCR. This was done simultaneously and in one test tube. Each sample was distinguished by an index sequence of second PCR primers following PCR amplification. With our procedure and tips for reducing PCR amplification bias, we were able to analyze 12 genes from 25 clinical samples in one MiSeq run. Moreover, with the certified primers designed by our short program--which detects and avoids common SNPs in gene-specific PCR primers--we used this system for routine genetic testing. Our optimized procedure uses a simple protocol, which can easily be followed by virtually any office medical staff. Because of the small PCR amplification bias, we can analyze simultaneously several clinical DNA samples with low cost and can obtain sufficient read numbers to detect a low level of somatic mosaic mutations.

Published
2017

49. Reply to Walsh et al

Author

Kawai, Tomoki, primary, Hiejima, Eitaro, additional, Oda, Hirotsugu, additional, Izawa, Kazushi, additional, Yasumi, Takahiro, additional, Nishikomori, Ryuta, additional, Okamoto, Shinya, additional, and Heike, Toshio, additional

Published
2017
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