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Fatal childhood calciphylaxis in a 10-year-old and literature review
Jing Feng1, Mona Gohara, Rossitza Lazova
1Department of Dermatology, Yale University School of Medicine, New Haven, Connecticut 06520-8059, USA.
Insights
Calciphylaxis, a rare vascular disorder, is detailed in a 10-year-old boy with end-stage renal disease. Pediatric cases suggest higher risks in boys and potential resistance to treatments, necessitating further research.
Area of Science:
- Nephrology
- Dermatology
- Vascular Biology
Background:
- Calciphylaxis is a rare, severe condition causing vascular calcification and skin necrosis.
- It primarily affects adults with end-stage renal disease (ESRD), with few pediatric cases documented.
Observation:
- This report details a fatal calciphylaxis case in a 10-year-old boy with sarcoidosis-induced ESRD.
- Review of pediatric calciphylaxis indicates increased risk in boys with ESRD and secondary hyperparathyroidism.
- Common features include distal extremity/visceral organ involvement, acral necrosis, and potential treatment resistance.
Findings:
- Hyperesthetic pain is a frequent early symptom in pediatric calciphylaxis.
- Diagnostic tools like soft tissue x-rays or xeroradiography can aid in diagnosis.
- Parathyroidectomy combined with medical management may offer the best treatment outcome.
Implications:
- This case highlights the critical need for recognizing and managing calciphylaxis in pediatric ESRD patients.
- Findings suggest unique risk factors and disease progression patterns in children compared to adults.
- Further research is warranted to optimize treatment strategies for pediatric calciphylaxis.
Abstract:
Calciphylaxis is a rare but highly morbid disorder of vascular calcification and skin necrosis, affecting 1% to 4% of adults with end-stage renal disease. Only three affected children have previously been described. We report an unusual instance of fatal calciphylaxis involving a 10-year-old boy, in the setting of sarcoidosis-induced end-stage renal disease. A review of pediatric calciphylaxis cases suggests: (a) increased risk in boys with end-stage renal disease and secondary hyperparathyroidism; (b) frequent distal extremity and visceral organ involvement, (c) worse prognosis with acral necrosis, and (d) possible increased resistance to medical treatment compared to adult patients. Hyperesthetic pain is a common sentinel symptom, and soft tissue x-rays or xeroradiography or both may aid in the diagnosis of pediatric calciphylaxis. Parathyroidectomy in combination with supportive medical management may be the best treatment option in stopping the progression of disease in the pediatric population.
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