Severe rhabdomyolysis in an infant due to fatty acid oxidation disorder: a case report
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Amelie Krug1, Pascale Perlot2, Aurelie Empain3
1Université Libre de Bruxelles (ULB), Rte de Lennik 808, Anderlecht, 1070, Brussels, Belgium. ameliemskrug@gmail.com.
Insights
Carnitine palmitoyltransferase II deficiency, a rare metabolic myopathy, can cause severe rhabdomyolysis in infants. Genetic analysis is crucial for diagnosis, even with normal initial metabolic tests.
Area of Science:
- Pediatrics
- Genetics
- Biochemistry
Background:
- Rhabdomyolysis, a serious condition with diverse causes, necessitates prompt etiological identification and treatment.
- Early diagnosis and intervention are critical to mitigate severe consequences of rhabdomyolysis.
Purpose of the Study:
- To report a case of severe rhabdomyolysis in an infant.
- To highlight the diagnostic challenges of carnitine palmitoyltransferase II deficiency in early childhood.
- To emphasize the role of genetic analysis in diagnosing metabolic myopathies.
Main Methods:
- Case report of a 14-month-old infant presenting with fever, hypotonia, and elevated creatine phosphokinase.
- Initial metabolic investigations were performed, revealing normal acylcarnitine profiles.
- Genetic analysis identified a homozygous missense variant in the CPT2 gene (NM_000098.3, c.338C>T, p.[Ser113Leu]).
Main Results:
- The infant presented with severe rhabdomyolysis and extremely high creatine phosphokinase levels (>100,000 UI/L).
- Despite normal primary metabolic analyses, genetic testing confirmed Carnitine palmitoyltransferase II deficiency.
- Prophylactic strategies and genetic counseling were implemented for the patient and family.
Conclusions:
- Carnitine palmitoyltransferase II deficiency can manifest as severe rhabdomyolysis in infants.
- Diagnosis can be challenging due to normal initial metabolic screening.
- Genetic analysis is essential for definitive diagnosis of CPT2 deficiency in infants.
Background:
Rhabdomyolysis can develop from numerous etiologies, both acquired and hereditary. Consequences of rhabdomyolysis may be grave, therefore identifying and treating the etiology is crucial.
Case Presentation:
We herein report the occurrence of severe rhabdomyolysis in a previously healthy 14-month-old infant presenting to the emergency department with fever, hypotonia, and generalized discomfort. Analysis revealed extremely high creatine phosphokinase levels (> 100,000 UI/L). Metabolic myopathy was suspected, however, primary metabolic analyses were normal. Carnitine palmitoyltransferase II deficiency was diagnosed following genetic analysis, identifying a homozygous NM_000098.3, c.338C > T, p.[Ser113Leu] missense variant. Prophylactic measures were established to prevent relapse and genetic counseling provided for the sibship.
Conclusion:
This case highlights the difficulty of diagnosing carnitine palmitoyltransferase II deficiency in an infant and the importance of genetic analysis to establish the diagnosis, despite a normal acylcarnitine profile.
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