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Combined Occurrence of Alstrom Syndrome and Bronchiectasis

Avni Kaya, Zerrin Orbak, Atilla Çayır, Hakan Döneray, Sener Tasdemir, Aysegul Ozanturk, Fatih Bingel

PEDIATRICS Cilt 133 · Sayı 3 · 01.03.2014
DOI ile aç

Özet

Alstrom syndrome (Online Mendelian Inheritance in Man ALMS #203800) is a rare hereditary disorder caused by mutations in the gene ALMS1. This rare disorder's characteristics are cone-rod dystrophy resulting in blindness in childhood, insulin-resistant type 2 diabetes mellitus, truncal obesity, progressive sensorineural hearing loss, dilated cardiomyopathy, craniofacial features, hypothyroidism, elevation in liver transaminases, renal insufficiency, gonadal dysfunction, and menstrual irregularities. A 13.5-year-old girl was admitted to the hospital for complaints of excessive water consumption and urination over the previous 2 years. The patient's parents were third-degree relatives. At physical examination, hyperpigmentation was present over the areola and acanthosis nigricans under the arms and on the neck. Audio-logic examination revealed bilateral sensorineural hearing loss, and bilateral cataract was determined at ocular examination. The patient was monitored by the chest diseases department due to bronchiectasis. HbA1c was 13.1%. In mutation screening study, 2 novel mutations c.5586T > G; p.Tyr1862* and c.2905insT; p.L968fs*4 were detected in the ALMS1 gene. Saccharin test was positive. We emphasize that Alstrom syndrome may be complicated by bronchiectasis.

Anahtar kelimeler

DIABETES-MELLITUS Bronchiectasis Acanthosis nigricans

Sürdürülebilir Kalkınma Amaçları

  • SKA 3: Sağlıklı Bireyler

Atıf için künye

Kaya, A., Orbak, Z., Çayır, A., Döneray, H., Tasdemir, S., Ozanturk, A., & Bingel, F. (2014). Combined Occurrence of Alstrom Syndrome and Bronchiectasis. PEDIATRICS, 133(3). https://doi.org/10.1542/peds.2013-0284

BibTeX
@article{kaya2014-222624,
  author = {Kaya, Avni and Orbak, Zerrin and Çayır, Atilla and Döneray, Hakan and Tasdemir, Sener and Ozanturk, Aysegul and Bingel, Fatih},
  title = {Combined Occurrence of Alstrom Syndrome and Bronchiectasis},
  journal = {PEDIATRICS},
  year = {2014},
  volume = {133},
  number = {3},
  issn = {0031-4005},
  doi = {10.1542/peds.2013-0284},
  url = {https://akademik.atauni.edu.tr/yayinlar/222624-combined-occurrence-of-alstrom-syndrome-and-bronchiectasis}
}