Coats Plus syndrome: a diagnostic and therapeutic challenge in pediatric gastrointestinal hemorrhage
Cerebroretinal microangiopathy with calcifications and cysts formerly known as Coats plus syndrome is a rare multisystemic autosomal recessive disease that affects the eyes, brain, bone, and gastrointestinal system. Intestinal telangiectasia are components of vascular malformations characterized by...
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| Veröffentlicht in: | The Turkish journal of pediatrics 2022-01, Vol.64 (1), p.166-170 |
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| container_title | The Turkish journal of pediatrics |
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| creator | Hosnut, Ferda Ozbay Sahin, Gulseren Akcaboy, Meltem |
| description | Cerebroretinal microangiopathy with calcifications and cysts formerly known as Coats plus syndrome is a rare multisystemic autosomal recessive disease that affects the eyes, brain, bone, and gastrointestinal system. Intestinal telangiectasia are components of vascular malformations characterized by gastrointestinal system bleedings. Recurrent gastrointestinal system bleedings have been reported as being due to hepatic failure or vascular malformations of the gastrointestinal system tract.
Here we report a patient who presented with recurrent gastrointestinal system bleeding episodes, bilateral exudative retinopathy, intracranial calcification and was diagnosed with Coats plus syndrome. Recurrent gastrointestinal system bleeding was controlled by monthly octreotide treatment.
Coats plus syndrome presenting with vascular malformations should always be kept in mind in a patient with recurrent gastrointestinal bleeding and accompanying systemic physical findings. Octreotide treatment is an important option for patients with life threatening gastrointestinal system bleeding. Long term use of octreotide treatment can be used successfully in selected pediatric cases. |
| doi_str_mv | 10.24953/turkjped.2020.3315 |
| format | Article |
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Here we report a patient who presented with recurrent gastrointestinal system bleeding episodes, bilateral exudative retinopathy, intracranial calcification and was diagnosed with Coats plus syndrome. Recurrent gastrointestinal system bleeding was controlled by monthly octreotide treatment.
Coats plus syndrome presenting with vascular malformations should always be kept in mind in a patient with recurrent gastrointestinal bleeding and accompanying systemic physical findings. Octreotide treatment is an important option for patients with life threatening gastrointestinal system bleeding. Long term use of octreotide treatment can be used successfully in selected pediatric cases.</description><identifier>ISSN: 0041-4301</identifier><identifier>EISSN: 2791-6421</identifier><identifier>DOI: 10.24953/turkjped.2020.3315</identifier><identifier>PMID: 35286046</identifier><language>eng</language><publisher>Turkey: Akdema Informatics and Publishing</publisher><subject>Anemia ; Ataxia ; Biopsy ; Birth weight ; Blood ; Brain Neoplasms ; Calcification ; Calcinosis ; Central Nervous System Cysts ; Child ; Colonoscopy ; Cysts ; Gastrointestinal bleeding ; Gastrointestinal Hemorrhage - diagnosis ; Gastrointestinal Hemorrhage - etiology ; Gastrointestinal system ; Hair ; Health aspects ; Hemoglobin ; Humans ; Hypertension ; Laboratories ; Leukoencephalopathies ; Muscle Spasticity ; Mutation ; Octreotide - therapeutic use ; Patients ; Pediatrics ; Retinal Diseases ; Seizures ; Syndrome ; Vascular Malformations</subject><ispartof>The Turkish journal of pediatrics, 2022-01, Vol.64 (1), p.166-170</ispartof><rights>COPYRIGHT 2022 Akdema Informatics and Publishing</rights><rights>Copyright Hacettepe University Faculty of Medicine Jan/Feb 2022</rights><lds50>peer_reviewed</lds50><oa>free_for_read</oa><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c373t-1c178fa49654040558077d648350bf5f0e65bdc77c869124d211397a7132e5943</citedby></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><link.rule.ids>314,776,780,27901,27902</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/35286046$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Hosnut, Ferda Ozbay</creatorcontrib><creatorcontrib>Sahin, Gulseren</creatorcontrib><creatorcontrib>Akcaboy, Meltem</creatorcontrib><title>Coats Plus syndrome: a diagnostic and therapeutic challenge in pediatric gastrointestinal hemorrhage</title><title>The Turkish journal of pediatrics</title><addtitle>Turk J Pediatr</addtitle><description>Cerebroretinal microangiopathy with calcifications and cysts formerly known as Coats plus syndrome is a rare multisystemic autosomal recessive disease that affects the eyes, brain, bone, and gastrointestinal system. Intestinal telangiectasia are components of vascular malformations characterized by gastrointestinal system bleedings. Recurrent gastrointestinal system bleedings have been reported as being due to hepatic failure or vascular malformations of the gastrointestinal system tract.
Here we report a patient who presented with recurrent gastrointestinal system bleeding episodes, bilateral exudative retinopathy, intracranial calcification and was diagnosed with Coats plus syndrome. Recurrent gastrointestinal system bleeding was controlled by monthly octreotide treatment.
Coats plus syndrome presenting with vascular malformations should always be kept in mind in a patient with recurrent gastrointestinal bleeding and accompanying systemic physical findings. Octreotide treatment is an important option for patients with life threatening gastrointestinal system bleeding. Long term use of octreotide treatment can be used successfully in selected pediatric cases.</description><subject>Anemia</subject><subject>Ataxia</subject><subject>Biopsy</subject><subject>Birth weight</subject><subject>Blood</subject><subject>Brain Neoplasms</subject><subject>Calcification</subject><subject>Calcinosis</subject><subject>Central Nervous System Cysts</subject><subject>Child</subject><subject>Colonoscopy</subject><subject>Cysts</subject><subject>Gastrointestinal bleeding</subject><subject>Gastrointestinal Hemorrhage - diagnosis</subject><subject>Gastrointestinal Hemorrhage - etiology</subject><subject>Gastrointestinal system</subject><subject>Hair</subject><subject>Health aspects</subject><subject>Hemoglobin</subject><subject>Humans</subject><subject>Hypertension</subject><subject>Laboratories</subject><subject>Leukoencephalopathies</subject><subject>Muscle Spasticity</subject><subject>Mutation</subject><subject>Octreotide - therapeutic use</subject><subject>Patients</subject><subject>Pediatrics</subject><subject>Retinal Diseases</subject><subject>Seizures</subject><subject>Syndrome</subject><subject>Vascular Malformations</subject><issn>0041-4301</issn><issn>2791-6421</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2022</creationdate><recordtype>article</recordtype><sourceid>EIF</sourceid><sourceid>8G5</sourceid><sourceid>BENPR</sourceid><sourceid>GUQSH</sourceid><sourceid>M2O</sourceid><recordid>eNpFkMtOwzAQRS0EgvL4AiRkiXXK-J2wQxUvCQkWsLZcx0lTkrjYzoK_x4VWrEYzc-_M1UHoksCc8kqwmzSFz_XG1XMKFOaMEXGAZlRVpJCckkM0A-Ck4AzICTqNcQ1AFVTqGJ0wQUsJXM5QvfAmRfzWTxHH77EOfnC32OC6M-3oY-osNmON08oFs3HTtrcr0_dubB3uRpzfdyaFPG5NTMF3Y3LZNZoer9zgQ1iZ1p2jo8b00V3s6hn6eLh_XzwVL6-Pz4u7l8IyxVJBLFFlY3glBQcOQpSgVC15yQQsG9GAk2JZW6VsKStCeU0JYZUyijDqRMXZGbr-u7sJ_mvKOfTaTyFniZpKDqoqJZX_qtb0Tndj41Mwduii1XdZopiUILKK_als8DEG1-hN6AYTvjUB_ctf7_nrLX-95Z9dV7sE03LIm71nD5z9AGvsgfo</recordid><startdate>20220101</startdate><enddate>20220101</enddate><creator>Hosnut, Ferda Ozbay</creator><creator>Sahin, Gulseren</creator><creator>Akcaboy, Meltem</creator><general>Akdema Informatics and Publishing</general><general>Hacettepe University Faculty of Medicine</general><scope>CGR</scope><scope>CUY</scope><scope>CVF</scope><scope>ECM</scope><scope>EIF</scope><scope>NPM</scope><scope>AAYXX</scope><scope>CITATION</scope><scope>3V.</scope><scope>4T-</scope><scope>4U-</scope><scope>7X7</scope><scope>7XB</scope><scope>88E</scope><scope>8AO</scope><scope>8FI</scope><scope>8FJ</scope><scope>8FK</scope><scope>8G5</scope><scope>ABUWG</scope><scope>AFKRA</scope><scope>AZQEC</scope><scope>BENPR</scope><scope>CCPQU</scope><scope>DWQXO</scope><scope>EDSIH</scope><scope>FYUFA</scope><scope>GHDGH</scope><scope>GNUQQ</scope><scope>GUQSH</scope><scope>K9.</scope><scope>M0S</scope><scope>M1P</scope><scope>M2O</scope><scope>MBDVC</scope><scope>PQEST</scope><scope>PQQKQ</scope><scope>PQUKI</scope><scope>PRINS</scope><scope>Q9U</scope></search><sort><creationdate>20220101</creationdate><title>Coats Plus syndrome: a diagnostic and therapeutic challenge in pediatric gastrointestinal hemorrhage</title><author>Hosnut, Ferda Ozbay ; Sahin, Gulseren ; Akcaboy, Meltem</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c373t-1c178fa49654040558077d648350bf5f0e65bdc77c869124d211397a7132e5943</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2022</creationdate><topic>Anemia</topic><topic>Ataxia</topic><topic>Biopsy</topic><topic>Birth weight</topic><topic>Blood</topic><topic>Brain Neoplasms</topic><topic>Calcification</topic><topic>Calcinosis</topic><topic>Central Nervous System Cysts</topic><topic>Child</topic><topic>Colonoscopy</topic><topic>Cysts</topic><topic>Gastrointestinal bleeding</topic><topic>Gastrointestinal Hemorrhage - diagnosis</topic><topic>Gastrointestinal Hemorrhage - etiology</topic><topic>Gastrointestinal system</topic><topic>Hair</topic><topic>Health aspects</topic><topic>Hemoglobin</topic><topic>Humans</topic><topic>Hypertension</topic><topic>Laboratories</topic><topic>Leukoencephalopathies</topic><topic>Muscle Spasticity</topic><topic>Mutation</topic><topic>Octreotide - therapeutic use</topic><topic>Patients</topic><topic>Pediatrics</topic><topic>Retinal Diseases</topic><topic>Seizures</topic><topic>Syndrome</topic><topic>Vascular Malformations</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Hosnut, Ferda Ozbay</creatorcontrib><creatorcontrib>Sahin, Gulseren</creatorcontrib><creatorcontrib>Akcaboy, Meltem</creatorcontrib><collection>Medline</collection><collection>MEDLINE</collection><collection>MEDLINE (Ovid)</collection><collection>MEDLINE</collection><collection>MEDLINE</collection><collection>PubMed</collection><collection>CrossRef</collection><collection>ProQuest Central (Corporate)</collection><collection>Docstoc</collection><collection>University Readers</collection><collection>Health & Medical Collection</collection><collection>ProQuest Central (purchase pre-March 2016)</collection><collection>Medical Database (Alumni Edition)</collection><collection>ProQuest Pharma Collection</collection><collection>Hospital Premium Collection</collection><collection>Hospital Premium Collection (Alumni Edition)</collection><collection>ProQuest Central (Alumni) (purchase pre-March 2016)</collection><collection>Research Library (Alumni Edition)</collection><collection>ProQuest Central (Alumni Edition)</collection><collection>ProQuest Central UK/Ireland</collection><collection>ProQuest Central Essentials</collection><collection>ProQuest Central</collection><collection>ProQuest One Community College</collection><collection>ProQuest Central Korea</collection><collection>Turkey Database</collection><collection>Health Research Premium Collection</collection><collection>Health Research Premium Collection (Alumni)</collection><collection>ProQuest Central Student</collection><collection>Research Library Prep</collection><collection>ProQuest Health & Medical Complete (Alumni)</collection><collection>Health & Medical Collection (Alumni Edition)</collection><collection>Medical Database</collection><collection>Research Library</collection><collection>Research Library (Corporate)</collection><collection>ProQuest One Academic Eastern Edition (DO NOT USE)</collection><collection>ProQuest One Academic</collection><collection>ProQuest One Academic UKI Edition</collection><collection>ProQuest Central China</collection><collection>ProQuest Central Basic</collection><jtitle>The Turkish journal of pediatrics</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Hosnut, Ferda Ozbay</au><au>Sahin, Gulseren</au><au>Akcaboy, Meltem</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Coats Plus syndrome: a diagnostic and therapeutic challenge in pediatric gastrointestinal hemorrhage</atitle><jtitle>The Turkish journal of pediatrics</jtitle><addtitle>Turk J Pediatr</addtitle><date>2022-01-01</date><risdate>2022</risdate><volume>64</volume><issue>1</issue><spage>166</spage><epage>170</epage><pages>166-170</pages><issn>0041-4301</issn><eissn>2791-6421</eissn><abstract>Cerebroretinal microangiopathy with calcifications and cysts formerly known as Coats plus syndrome is a rare multisystemic autosomal recessive disease that affects the eyes, brain, bone, and gastrointestinal system. Intestinal telangiectasia are components of vascular malformations characterized by gastrointestinal system bleedings. Recurrent gastrointestinal system bleedings have been reported as being due to hepatic failure or vascular malformations of the gastrointestinal system tract.
Here we report a patient who presented with recurrent gastrointestinal system bleeding episodes, bilateral exudative retinopathy, intracranial calcification and was diagnosed with Coats plus syndrome. Recurrent gastrointestinal system bleeding was controlled by monthly octreotide treatment.
Coats plus syndrome presenting with vascular malformations should always be kept in mind in a patient with recurrent gastrointestinal bleeding and accompanying systemic physical findings. Octreotide treatment is an important option for patients with life threatening gastrointestinal system bleeding. Long term use of octreotide treatment can be used successfully in selected pediatric cases.</abstract><cop>Turkey</cop><pub>Akdema Informatics and Publishing</pub><pmid>35286046</pmid><doi>10.24953/turkjped.2020.3315</doi><tpages>5</tpages><oa>free_for_read</oa></addata></record> |
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| subjects | Anemia Ataxia Biopsy Birth weight Blood Brain Neoplasms Calcification Calcinosis Central Nervous System Cysts Child Colonoscopy Cysts Gastrointestinal bleeding Gastrointestinal Hemorrhage - diagnosis Gastrointestinal Hemorrhage - etiology Gastrointestinal system Hair Health aspects Hemoglobin Humans Hypertension Laboratories Leukoencephalopathies Muscle Spasticity Mutation Octreotide - therapeutic use Patients Pediatrics Retinal Diseases Seizures Syndrome Vascular Malformations |
| title | Coats Plus syndrome: a diagnostic and therapeutic challenge in pediatric gastrointestinal hemorrhage |
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