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Turner Syndrome Associated with Ulcerative Colitis

Identifieur interne : 003C11 ( Pmc/Checkpoint ); précédent : 003C10; suivant : 003C12

Turner Syndrome Associated with Ulcerative Colitis

Auteurs : Junji Takaya ; Masayuki Teraguchi ; Yumiko Ikemoto ; Ken Yoshimura ; Fumiko Yamato ; Hirohiko Higashino ; Yohnosuke Kobayashi [Japon] ; Kazunari Kaneko

Source :

RBID : PMC:4004840

Abstract

We report the case of a 7-yr-old girl with Turner syndrome, ulcerative colitis (UC) and coarctation of the aorta. The diagnosis of Turner syndrome was made in early infancy (karyotype analysis 45, X). Growth hormone treatment was started at 3 yr and 2 mo of age. From the age of 4 yr and 5 mo, the patient suffered from persistent diarrhea with traces of blood and intermittent abdominal discomfort. As these symptoms gradually deteriorated, she was referred to our clinic at the age of 7 yr for further evaluation. Barium enema showed aphtha and loss of the fine network pattern in the descending colon and rectum. An endoscopic examination showed ulceration, edema, friability, and erythema beginning in the rectum and extending up to the splenic flexure of the descending colon. The histology of the descending colon area showed severe stromal infiltration of inflammatory cells. These endoscopic findings and the histological findings were consistent with UC. Thus, based on these findings, the patient was diagnosed as having UC. Mesalazine therapy was initiated at this time. The patient is currently being treated with mesalazine (1,000 mg/day) and abdominal symptoms and bloody diarrhea have disappeared. GH therapy was not interrupted during the therapy for UC. Retrospectively, growth hormone improved growth velocity (9 cm/year) during the first year of treatment, however from the age of 4 yr, growth velocity decreased (4–5 cm/yr) in spite of the GH treatment. Conclusion: Patients with Turner syndrome and gastrointestinal symptoms should be investigated for inflammatory bowel diseases. Growth velocity is useful for evaluating the presence of inflammatory bowel diseases and other systemic diseases.


Url:
DOI: 10.1297/cpe.15.97
PubMed: 24790328
PubMed Central: 4004840


Affiliations:


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PMC:4004840

Le document en format XML

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<name sortKey="Teraguchi, Masayuki" sort="Teraguchi, Masayuki" uniqKey="Teraguchi M" first="Masayuki" last="Teraguchi">Masayuki Teraguchi</name>
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<name sortKey="Ikemoto, Yumiko" sort="Ikemoto, Yumiko" uniqKey="Ikemoto Y" first="Yumiko" last="Ikemoto">Yumiko Ikemoto</name>
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<name sortKey="Yoshimura, Ken" sort="Yoshimura, Ken" uniqKey="Yoshimura K" first="Ken" last="Yoshimura">Ken Yoshimura</name>
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<name sortKey="Yamato, Fumiko" sort="Yamato, Fumiko" uniqKey="Yamato F" first="Fumiko" last="Yamato">Fumiko Yamato</name>
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<p> We report the case of a 7-yr-old girl with Turner syndrome, ulcerative colitis (UC) and coarctation of the aorta. The diagnosis of Turner syndrome was made in early infancy (karyotype analysis 45, X). Growth hormone treatment was started at 3 yr and 2 mo of age. From the age of 4 yr and 5 mo, the patient suffered from persistent diarrhea with traces of blood and intermittent abdominal discomfort. As these symptoms gradually deteriorated, she was referred to our clinic at the age of 7 yr for further evaluation. Barium enema showed aphtha and loss of the fine network pattern in the descending colon and rectum. An endoscopic examination showed ulceration, edema, friability, and erythema beginning in the rectum and extending up to the splenic flexure of the descending colon. The histology of the descending colon area showed severe stromal infiltration of inflammatory cells. These endoscopic findings and the histological findings were consistent with UC. Thus, based on these findings, the patient was diagnosed as having UC. Mesalazine therapy was initiated at this time. The patient is currently being treated with mesalazine (1,000 mg/day) and abdominal symptoms and bloody diarrhea have disappeared. GH therapy was not interrupted during the therapy for UC. Retrospectively, growth hormone improved growth velocity (9 cm/year) during the first year of treatment, however from the age of 4 yr, growth velocity decreased (4–5 cm/yr) in spite of the GH treatment. Conclusion: Patients with Turner syndrome and gastrointestinal symptoms should be investigated for inflammatory bowel diseases. Growth velocity is useful for evaluating the presence of inflammatory bowel diseases and other systemic diseases.</p>
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<journal-id journal-id-type="nlm-ta">Clin Pediatr Endocrinol</journal-id>
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<article-title>Turner Syndrome Associated with Ulcerative Colitis</article-title>
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<contrib contrib-type="author">
<name>
<surname>Takaya</surname>
<given-names>Junji</given-names>
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<name>
<surname>Teraguchi</surname>
<given-names>Masayuki</given-names>
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<name>
<surname>Ikemoto</surname>
<given-names>Yumiko</given-names>
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<surname>Yoshimura</surname>
<given-names>Ken</given-names>
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<surname>Yamato</surname>
<given-names>Fumiko</given-names>
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<name>
<surname>Higashino</surname>
<given-names>Hirohiko</given-names>
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<name>
<surname>Kobayashi</surname>
<given-names>Yohnosuke</given-names>
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<sup>1</sup>
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<name>
<surname>Kaneko</surname>
<given-names>Kazunari</given-names>
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<aff id="aff1">Department of Pediatrics, Kansai Medical University, Moriguchi, Osaka 570-8506, Japan</aff>
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<corresp id="cor">Correspondence: Dr. Junji Takaya, Department of Pediatrics, Kansai Medical University, 10-15 Fumizonocho, Moriguchi, Osaka 570-8506, Japan. E-mail:
<email xlink:href="takaya@takii.kmu.ac.jp">takaya@takii.kmu.ac.jp</email>
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<pub-date pub-type="epub">
<day>02</day>
<month>8</month>
<year>2006</year>
</pub-date>
<pub-date pub-type="ppub">
<year>2006</year>
</pub-date>
<volume>15</volume>
<issue>3</issue>
<fpage>97</fpage>
<lpage>100</lpage>
<history>
<date date-type="received">
<day>10</day>
<month>1</month>
<year>2006</year>
</date>
<date date-type="accepted">
<day>18</day>
<month>4</month>
<year>2006</year>
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<copyright-statement>2006©The Japanese Society for Pediatric Endocrinology</copyright-statement>
<copyright-year>2006</copyright-year>
<license license-type="open-access" xlink:href="http://creativecommons.org/licenses/by-nc-nd/3.0/">
<license-p>This is an open-access article distributed under the terms of the Creative Commons Attribution Non-Commercial No Derivatives (by-nc-nd) License. </license-p>
</license>
</permissions>
<abstract>
<p> We report the case of a 7-yr-old girl with Turner syndrome, ulcerative colitis (UC) and coarctation of the aorta. The diagnosis of Turner syndrome was made in early infancy (karyotype analysis 45, X). Growth hormone treatment was started at 3 yr and 2 mo of age. From the age of 4 yr and 5 mo, the patient suffered from persistent diarrhea with traces of blood and intermittent abdominal discomfort. As these symptoms gradually deteriorated, she was referred to our clinic at the age of 7 yr for further evaluation. Barium enema showed aphtha and loss of the fine network pattern in the descending colon and rectum. An endoscopic examination showed ulceration, edema, friability, and erythema beginning in the rectum and extending up to the splenic flexure of the descending colon. The histology of the descending colon area showed severe stromal infiltration of inflammatory cells. These endoscopic findings and the histological findings were consistent with UC. Thus, based on these findings, the patient was diagnosed as having UC. Mesalazine therapy was initiated at this time. The patient is currently being treated with mesalazine (1,000 mg/day) and abdominal symptoms and bloody diarrhea have disappeared. GH therapy was not interrupted during the therapy for UC. Retrospectively, growth hormone improved growth velocity (9 cm/year) during the first year of treatment, however from the age of 4 yr, growth velocity decreased (4–5 cm/yr) in spite of the GH treatment. Conclusion: Patients with Turner syndrome and gastrointestinal symptoms should be investigated for inflammatory bowel diseases. Growth velocity is useful for evaluating the presence of inflammatory bowel diseases and other systemic diseases.</p>
</abstract>
<kwd-group>
<kwd>Turner syndrome</kwd>
<kwd>ulcerative colitis</kwd>
<kwd>growth velocity</kwd>
<kwd>GH therapy</kwd>
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<name sortKey="Ikemoto, Yumiko" sort="Ikemoto, Yumiko" uniqKey="Ikemoto Y" first="Yumiko" last="Ikemoto">Yumiko Ikemoto</name>
<name sortKey="Kaneko, Kazunari" sort="Kaneko, Kazunari" uniqKey="Kaneko K" first="Kazunari" last="Kaneko">Kazunari Kaneko</name>
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<name sortKey="Teraguchi, Masayuki" sort="Teraguchi, Masayuki" uniqKey="Teraguchi M" first="Masayuki" last="Teraguchi">Masayuki Teraguchi</name>
<name sortKey="Yamato, Fumiko" sort="Yamato, Fumiko" uniqKey="Yamato F" first="Fumiko" last="Yamato">Fumiko Yamato</name>
<name sortKey="Yoshimura, Ken" sort="Yoshimura, Ken" uniqKey="Yoshimura K" first="Ken" last="Yoshimura">Ken Yoshimura</name>
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<name sortKey="Kobayashi, Yohnosuke" sort="Kobayashi, Yohnosuke" uniqKey="Kobayashi Y" first="Yohnosuke" last="Kobayashi">Yohnosuke Kobayashi</name>
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