AASE SYNDROME

 

AASE SYNDROME

 

DEFINITION:

A disorder characterized by congenital hypoplasic anemia and triphalangeal thumbs.

EPIDEMIOLOGY:

  • incidence: rare
  • age of onset:
    • newborn (anemia)
  • risk factors:
    • familial - autosomal recessive
      • chrom.#: ?
      • gene: ?

PATHOGENESIS:

1. Background

  • one of at least 3 autosomal recessive syndromes associated with hematopoietic and skeletal anomalies
    • other syndromes:
      • Fanconi Anemia
      • Thrombocytopenia-Absent Radius (TAR) Syndrome
  • etiology is unknown but is considered to be one of at least 3 disorders where there is a congenital deficiency in erythroid precursors
    • other congenital single cytopenias:
      • Diamond-Blackfan Syndrome
      • Congenital Dyserythropoietic Anemia

CLINICAL FEATURES:

1. Anemia

  • onset in newborn period but tends to improve with age
  • pallor and lethargy

2. Musculoskeletal Manifestations

  • short stature
  • triphalangeal thumbs with mild radial hypoplasia
  • narrow shoulders
  • late closure of fontanelles

3. Others

  • cardiac anomalies - VSD

INVESTIGATIONS:

1. Serum

  • Hb: normocytic or macrocytic, increased HbF, low reticulocytes
  • variable leukopenia

2. Imaging Studies

1. Skeletal X-rays

  • upper limbs and thumbs

3. Chromosomal Breakage Analysis

  • normal

MANAGEMENT:

1. Supportive

  • steroids (prednisone) and/or PRBC transfusions
  • anemia tends to improve with time

 

 

Pediatric Database - AASE SYNDROME

Pediatric Organization - Pedbase [at] Gmail.com