Spinal Canal Involvement in Solitary Infantile Myofibromatosis: Case Report

Abstract

Infantile myofibromatosis involving the spinal canal is very rare; only 11 cases have been reported so far in the literature. The authors present a case of an 18-month-old male child who presented with the history of dribbling of urine and weakness in bilateral lower limbs since 2 months. MRI of spine revealed single intramedullary intradural space-occupying lesion (SOL) at D1 to D2.

The patient underwent laminectomy with excision of SOL with biopsy report suggestive of benign nerve sheath tumor, and immunohistochemistry report revealed desmin negative, smooth muscle actin positive, and S-100 focally positive infantile myofibromatosis. The patient gradually recovered and had a clear stream of urine with improved movements and tone of bilateral lower limbs at the time of discharge.

Keywords

solitary infantile myofibromatosis - spinal canal - intramedullary

• References

• 1 Stout AP. Juvenile fibromatoses. Cancer 1954; 7 (05) 953-978
• 2 Chung EB, Enzinger FM. Infantile myofibromatosis. Cancer 1981; 48 (08) 1807-1818
• 3 Beyer WF, Kraus J, Glückert K, Goldmann AR. [Solitary infantile myofibromatosis of the sacrum]. Z Orthop Ihre Grenzgeb 1990; 128 (05) 473-476 (Ger)
• 4 Asirvatham R, Moreau PG, Antonius JI. Solitary infantile myofibromatosis of axis. A case report. Spine 1994; 19 (01) 80-82
• 5 Tamburrini G, Gessi M, Colosimo Jr C, Lauriola L, Giangaspero F, Di Rocco C. Infantile myofibromatosis of the central nervous system. Childs Nerv Syst 2003; 19 (09) 650-654
• 6 Kim EJ, Wang KC, Lee JY. , et al. Congenital solitary infantile myofibromatosis involving the spinal cord. J Neurosurg Pediatr 2013; 11 (01) 82-86
• 7 Cardia E, Molina D, Zaccone C, la Rosa G, Napoli P. Intracranial solitary-type infantile myofibromatosis. Childs Nerv Syst 1993; 9 (04) 246-249
• 8 Kaplan SS, Ojemann JGM, Grange DK, Fuller C, Park TS. Intracranial infantile myofibromatosis with intraparenchymal involvement. Pediatr Neurosurg 2002; 36 (04) 214-217
• 9 Rutigliano JM, Pollack IF. Ahdab- Barmada M, Pang D, Leland-Albright A. Intracranial infantile myofibromatosis. J Neurosurg 1994; 81: 539-543
• 10 Roggli VL, Kim HS, Hawkins E. Congenital generalized fibromatosis with visceral involvement. A case report. Cancer 1980; 45 (05) 954-960
• 11 Holzer-Fruehwald L, Blaser S, Rossi A, Fruehwald-Pallamar J, Thurnher MM. Imaging findings in seven cases of congenital infantile myofibromatosis with cerebral, spinal, or head and neck involvement. Neuroradiology 2012; 54 (12) 1389-1398
• 12 Sinha AK, Jesudason EC, Mallucci CL, Losty PD, Kokai G, Pizer BL. Infantile myofibromatosis of the craniovertebral junction. Br J Neurosurg 2008; 22 (04) 575-577