Literature DB >> 22145491

Autoimmune polyendocrinopathy associated with ring chromosome 18.

Nina Jain1, Pamela J Reitnauer, Kathleen W Rao, Arthur S Aylsworth, Ali S Calikoglu.   

Abstract

Phenotypic and clinical features of individuals with ring chromosome 18 [r(18)] vary with the extent of deletion of the short (18p-) or long arm (18q-). Most patients with r(18), therefore, demonstrate a clinical spectrum of both 18p- and 18q- deletions. Short stature, microcephaly, mental and motor retardation, craniofacial dysmorphism and extremity abnormalities are the most commonly reported features in patients with r(18). Abnormalities of chromosome 18, especially 18p- syndrome, are often reported with autoimmune thyroid disease and growth hormone deficiency, but reports of endocrine abnormalities associated with r(18) are rare. Here, we report a case of an African-American female with hyperthyroidism, type 1 diabetes mellitus, vitiligo and IgA deficiency associated with a r(18) chromosome complement. This patient additionally had mild intellectual disability and dysmorphic features. Karyotype analysis showed a de novo ring chromosome 18 (deletion 18q23-18qter and deletion 18p11.3-18pter). Although this unique association of autoimmune polyglandular endocrinopathy with ring chromosome 18 could be coincidental, we speculate that a gene or genes on chromosome 18 might play a role in the autoimmune process.

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Year:  2011        PMID: 22145491     DOI: 10.1515/jpem.2011.320

Source DB:  PubMed          Journal:  J Pediatr Endocrinol Metab        ISSN: 0334-018X            Impact factor:   1.634


  5 in total

1.  A Case of Ring Chromosome 18 with Single Umbilical Artery Detected During Prenatal Period.

Authors:  Nazan Eras
Journal:  Mol Syndromol       Date:  2020-09-10

2.  A case report of Ring chromosome 18 with systemic Lupus Erythematosus and Crohn's disease.

Authors:  Tina Rezaeizadeh; Encieh Delshad; Nader Mansour Samaei; Naghmeh Gholipour
Journal:  Mol Biol Rep       Date:  2021-11-14       Impact factor: 2.316

3.  Hypothyroidism and levothyroxine-responsive liver dysfunction in a patient with ring chromosome 18 syndrome.

Authors:  Kazuhiro Ohkubo; Kenji Ihara; Shouichi Ohga; Masataka Ishimura; Toshiro Hara
Journal:  Thyroid       Date:  2012-09-04       Impact factor: 6.568

4.  Rheumatoid arthritis in an adult patient with mosaic distal 18q-, 18p- and ring chromosome 18.

Authors:  Alanna Chau; K H Ramesh; Anand D Jagannath; Shitij Arora
Journal:  F1000Res       Date:  2017-11-02

5.  A Rare Association of Monosomy 18p Syndrome and Polyglandular Autoimmune Syndrome Type IIIA.

Authors:  D Dolek-Cetinkaya; Mm Demirpence; A Gorgel; F Salgur; M Bahceci
Journal:  Balkan J Med Genet       Date:  2013-06       Impact factor: 0.519

  5 in total

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