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DOI 10.1002/ajmg.a.63381

Медицинская библиотека · American journal of medical genetics. Part A · 2023

Beckwith-Wiedemann syndrome with multiple hepatic and cutaneous hemangiomas in a female patient of Albanian origin: Diagnostic and therapeutic considerations.

Moutafi Maria, Gkiourtzis Nikolaos, Ververi Athina, Kavga Maria, Morichovitou Anthi, Papadopoulou-Legbelou Kyriaki, Fotoulaki Maria, Panagopoulou Paraskevi

Клинический случайПоддержка исследования: вне правительства СШАСтатьяПеревод в очереди

Аннотация

We describe a 2-month-old female infant with macroglossia, macrosomia, omphalocele, neonatal hypoglycemia, earlobe creases, low nasal bridge, midface retrusion, syndromic facies and multiple cutaneous and hepatic hemangiomas (HH). Genetic evaluation confirmed the diagnosis of Beckwith-Wiedemann Syndrome (BWS) with mosaic uniparental disomy 11 as the underlying genetic mechanism suggested by partial hypermethylation of H19/IGF2:IG-DMR and partial hypomethylation of KCNQ1OT1:TSS-DMR on chromosome 11p15.5. Pediatric endocrinology and cardiology assessments were normal. No malignant liver or renal tumors were detected during the follow-up period. Treatment with propranolol was started for the multiple HH, according to international recommendations. At 3-, 6-, and 9-month follow up, a gradual decrease in the size of the hemangiomas and AFP levels was observed, without side effects. This is the fifth case in the literature combining HH and BWS, and among these, the third case with this specific genetic defect suggesting a possible association between HH and BWS caused by 11 paternal uniparental disomy [upd(11)pat]. The case also highlights that if treatment is warranted, then oral propranolol can be used for the management of infantile HH in BWS patients similarly to non-BWS patients.

Для цитирования: Moutafi Maria, Gkiourtzis Nikolaos, Ververi Athina, Kavga Maria, Morichovitou Anthi, Papadopoulou-Legbelou Kyriaki, Fotoulaki Maria, Panagopoulou Paraskevi. Beckwith-Wiedemann syndrome with multiple hepatic and cutaneous hemangiomas in a female patient of Albanian origin: Diagnostic and therapeutic considerations.. American journal of medical genetics. Part A. 2023. DOI: 10.1002/ajmg.a.63381.

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