Claudia Mandato1, Alessandro Rossi2, Mariano Caldore3, Marta Lamba3, Michele Rocco4, Renata Auricchio2, Pietro Vajro5, Paolo Siani3. 1. Department of Pediatrics, AORN Santobono-Pausilipon, via M. Fiore, 80129 Naples, Italy. Electronic address: cla.mandato@gmail.com. 2. Department of Traslational Medicine, University of Naples Federico II, 80131 Naples, Italy. 3. Department of Pediatrics, AORN Santobono-Pausilipon, via M. Fiore, 80129 Naples, Italy. 4. Department of Services, Pathology, AORN Santobono-Pausilipon, via M. Fiore, 80129 Naples, Italy. 5. Department of Medicine, Surgery, and Dentistry "Scuola Medica Salernitana", Pediatrics Section, University of Salerno, 84081, Baronissi (Salerno), Italy.
Abstract
BACKGROUND: Rhabdomyolysis is a rare, potentially life-threatening condition, caused by multiple disorders. The association with Coeliac Disease (CD) has been rarely reported and in these cases muscular damage was imputed to hypokalemia. Herein we describe a new case of severe rhabdomyolysis in a child subsequently diagnosed as affected by CD, and review previous reports. CASE PRESENTATION: A 3-year-old boy was referred for diarrhea, brown urine, muscular pain/weakness, and no history of muscular trauma. At entry, laboratory tests showed elevated levels of creatine kinase (CK) (x100 unv) and aspartate aminotransferase (AST) (x10 unv), alanine aminotrasferase (ALT) (x5 unv); electrolytes were within the reference range. Twenty-four hours after admission serum CK peaked 115,000 U/L and transaminases increased up to 30 times unv. Hyperhydration treatment was started with renal function monitoring. Urine output decreased little, while serum creatinine and urea nitrogen stayed within the reference range. Serum potassium levels went down to 2.8 mEq/L at day 3, in spite of supplementation. The patient completely recovered at day 16. Main metabolic causes of rhabdomyolysis were ruled out by appropriate tests. Because of rarely reported cases of CD/rhabdomyolysis, anti-tissue transglutaminase (tTG) antibodies were measured and found positive (IgA 34 U/mL, unv <9). HLA typing was DQA1 05:02, DQB1 03:02. As jejunal biopsy showed patchy villous atrophy, gluten free diet (GFD) was prescribed. One year after starting GFD, histology was normal. REVIEW OF LITERATURE: Literature (search engines: PUB MED and GOOGLE SCHOLAR) from 1980 to 2016 retrieved 8 cases (age range: 12 to 75 years old) previously described. CONCLUSION: The present case suggests to check for CD in children with severe rhabdomyolysis. Because severe rhabdomyolysis itself may elevate the serum potassium levels, hypokalemia might go unrecognized as the cause of muscular damage.
BACKGROUND:Rhabdomyolysis is a rare, potentially life-threatening condition, caused by multiple disorders. The association with Coeliac Disease (CD) has been rarely reported and in these cases muscular damage was imputed to hypokalemia. Herein we describe a new case of severe rhabdomyolysis in a child subsequently diagnosed as affected by CD, and review previous reports. CASE PRESENTATION: A 3-year-old boy was referred for diarrhea, brown urine, muscular pain/weakness, and no history of muscular trauma. At entry, laboratory tests showed elevated levels of creatine kinase (CK) (x100 unv) and aspartate aminotransferase (AST) (x10 unv), alanine aminotrasferase (ALT) (x5 unv); electrolytes were within the reference range. Twenty-four hours after admission serum CK peaked 115,000 U/L and transaminases increased up to 30 times unv. Hyperhydration treatment was started with renal function monitoring. Urine output decreased little, while serum creatinine and ureanitrogen stayed within the reference range. Serum potassium levels went down to 2.8 mEq/L at day 3, in spite of supplementation. The patient completely recovered at day 16. Main metabolic causes of rhabdomyolysis were ruled out by appropriate tests. Because of rarely reported cases of CD/rhabdomyolysis, anti-tissue transglutaminase (tTG) antibodies were measured and found positive (IgA 34 U/mL, unv <9). HLA typing was DQA1 05:02, DQB1 03:02. As jejunal biopsy showed patchy villous atrophy, gluten free diet (GFD) was prescribed. One year after starting GFD, histology was normal. REVIEW OF LITERATURE: Literature (search engines: PUB MED and GOOGLE SCHOLAR) from 1980 to 2016 retrieved 8 cases (age range: 12 to 75 years old) previously described. CONCLUSION: The present case suggests to check for CD in children with severe rhabdomyolysis. Because severe rhabdomyolysis itself may elevate the serum potassium levels, hypokalemia might go unrecognized as the cause of muscular damage.