The challenges of managing coexistent disorders with phenylketonuria: 30 cases

Mol Genet Metab. 2015 Dec;116(4):242-51. doi: 10.1016/j.ymgme.2015.10.001. Epub 2015 Oct 9.

Abstract

Introduction: The few published case reports of co-existent disease with phenylketonuria (PKU) are mainly genetic and familial conditions from consanguineous marriages. The clinical and demographic features of 30 subjects with PKU and co-existent conditions were described in this multi-centre, retrospective cohort study.

Methods: Diagnostic age of PKU and co-existent condition, treatment regimen, and impact of co-existent condition on blood phenylalanine (Phe) control and PKU management were reported.

Results: 30 patients (11 males and 19 females), with PKU and a co-existent condition, current median age of 14 years (range 0.4 to 40 years) from 13 treatment centres from Europe and Turkey were described. There were 21 co-existent conditions with PKU; 9 were autoimmune; 6 gastrointestinal, 3 chromosomal abnormalities, and 3 inherited conditions. There were only 5 cases of parental consanguinity. Some patients required conflicting diet therapy (n=5), nutritional support (n=7) and 5 children had feeding problems. There was delayed diagnosis of co-existent conditions (n=3); delayed treatment of PKU (n=1) and amenorrhea associated with Grave's disease that masked a PKU pregnancy for 12 weeks. Co-existent conditions adversely affected blood Phe control in 47% (n=14) of patients. Some co-existent conditions increased the complexity of disease management and increased management burden for patients and caregivers.

Conclusions: Occurrence of co-existent disease is not uncommon in patients with PKU and so investigation for co-existent disorders when the clinical history is not completely consistent with PKU is essential. Integrating care of a second condition with PKU management is challenging.

Keywords: Co-existent; Diet; Phenylalanine; Phenylketonuria; Sapropterin.

Publication types

  • Case Reports
  • Multicenter Study

MeSH terms

  • Adolescent
  • Adult
  • Autoimmune Diseases / blood
  • Autoimmune Diseases / complications
  • Autoimmune Diseases / diagnosis
  • Autoimmune Diseases / therapy*
  • Biopterins / analogs & derivatives
  • Biopterins / therapeutic use
  • Child
  • Child, Preschool
  • Chromosome Aberrations*
  • Consanguinity
  • Diet
  • Disease Management*
  • Europe
  • Female
  • Gastrointestinal Diseases / blood
  • Gastrointestinal Diseases / complications
  • Gastrointestinal Diseases / diagnosis
  • Gastrointestinal Diseases / therapy*
  • Humans
  • Infant
  • Male
  • Phenylalanine / blood*
  • Phenylketonurias / blood
  • Phenylketonurias / complications
  • Phenylketonurias / diagnosis
  • Phenylketonurias / therapy*
  • Pregnancy
  • Retrospective Studies
  • Turkey

Substances

  • Biopterins
  • Phenylalanine
  • sapropterin