A rare cause of carpal tunnel syndrome in childhood: Benign recurrent intrahepatic cholestasis

• Autorzy: Necati Balamtekin , Nuray Uslu , Cagri Temucin , Hulya Demir , Inci Saltik Temizel
• Języki publikacji: EN

Abstrakty

EN

Carpal tunnel syndrome and benign recurrent intrahepatic cholestasis are rare conditions in childhood. Benign intrahepatic cholestasis is characterized by repeated self-limited attacks of cholestasis that can start at any age and last from weeks to months. The patients are asymptomatic between these attacks. We report a 16 year-old male patient with benign recurrent intrahepatic cholestasis who developed carpal tunnel syndrome during a cholestatic attack. He was admitted with complaints of jaundice, pruritus and pain, tingling and muscle weakness in both hands for 15 days. Nerve conduction studies revealed findings compatible with carpal tunnel syndrome. He was started on ursodeoxycholic acid, fat soluble vitamins and cholestyramine and cholestasis regressed after four weeks of therapy. With the improvement of cholestasis, the symptoms of carpal tunnel syndrome also disappeared. In conclusion, benign recurrent intrahepatic cholestasis can be a rare cause of carpal tunnel syndrome in childhood. We also advocate treating the underlying disease as an appropriate conservative treatment before surgery.

Słowa kluczowe

EN

Child; Carpal tunnel syndrome; Benign recurrent intrahepatic cholestasis

• Czasopismo: Open Medicine
• Rocznik: 2010
• Tom: 5
• Numer: 6
• Strony: 742-744

Daty

wydano

2010-12-01

online

2010-10-07

Twórcy

autor

Necati Balamtekin

• Faculty of Medicine, Department of Pediatrics, Section of Gastroenterology, Hepatology and Nutrition, Hacettepe University, 06100, Ankara, Turkey,

autor

Nuray Uslu

• Faculty of Medicine, Department of Pediatrics, Section of Gastroenterology, Hepatology and Nutrition, Hacettepe University, 06100, Ankara, Turkey

autor

Cagri Temucin

• Faculty of Medicine, Department of Neurology, Hacettepe University, 06100, Ankara, Turkey

autor

Hulya Demir

• Faculty of Medicine, Department of Pediatrics, Section of Gastroenterology, Hepatology and Nutrition, Hacettepe University, 06100, Ankara, Turkey

autor

Inci Saltik Temizel

• Faculty of Medicine, Department of Pediatrics, Section of Gastroenterology, Hepatology and Nutrition, Hacettepe University, 06100, Ankara, Turkey

Bibliografia

• [1] Van Meir N, De Smet L. Carpal tunnel syndrome in children. J Pediatr Orthop B 2005; 14: 42–45 [Crossref][PubMed]
• [2] Luketic VA, Shiffman ML. Benign recurrent intrahepatic cholestasis. Clin Liver Dis 2004; 8: 133–149 http://dx.doi.org/10.1016/S1089-3261(03)00133-8[Crossref]
• [3] Gupta V, Kumar M, Bhatia BD. Benign recurrent intrahepatic cholestasis. Indian J Pediatr 2005; 72: 793–794 http://dx.doi.org/10.1007/BF02734154[Crossref]
• [4] Cançado EL, Leitao RM, Carrilho FJ, Laudanna AA. Unexpected clinical remission of cholestasis after rifampicin therapy in patients with normal or slightly increased levels of gamma-glutamyl transpeptidase. Am J Gastroenterol 1998; 93: 1510–1517 [PubMed]
• [5] Nicolas I, Pons JA, Vargas A, Gallardo F, Albaladejo A, Nicolas JM.. Ursodeoxycholic acid treatment shortens the course of cholestasis in two patients with benign recurrent intrahepatic cholestasis. Gastroenterol Hepatol 2003; 26: 421–423 http://dx.doi.org/10.1157/13050355[Crossref]
• [6] Voermans NC, van Alfen N, Tolboom JJ, Koetsveld AC, Sie LT. Pediatric median neuropathy due to pruritus in Alagille syndrome. Pediatric Neurology 2006; 35: 216–219 http://dx.doi.org/10.1016/j.pediatrneurol.2006.02.011[Crossref]
• [7] Figus A, Iwuagwu FC, Elliot D. Subacute nerve compressions after trauma and surgery on the hand. Plast Reconstr Surg 2007; 120: 705–712 http://dx.doi.org/10.1097/01.prs.0000270304.26018.1f[Crossref][WoS]
• [8] Dahlin LB. Aspects on pathophysiology of nerve entrapments and nerve compression injuries. Neurosurg Clin N Am 1991; 2: 21–29 [PubMed]

Identyfikatory

DOI

10.2478/s11536-009-0142-2