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Journal of Nippon Medical School

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-Case Reports-

A Case of Fibrolipomatous Hamartoma of the Digital Nerve without Macrodactyly

Mitsuhiko Nanno1,2, Takuya Sawaizumi1,3 and Shinro Takai1,3

1Department of Restorative Medicine of Neuro-musculoskeletal System, Orthopaedic Surgery, Graduate School of Medicine, Nippon Medical School
2Department of Orthopaedic Surgery, Nippon Medical School, Musashi Kosugi Hospital
3Department of Orthopaedic Surgery, Nippon Medical School


Fibrolipomatous hamartoma of nerves without macrodactyly is a rare lesion characterized by fibrofatty proliferation causing epineural and perineural fibrosis with fatty infiltration around the nerve bundles. We report an unusual case of fibromatous hamartoma of the ulnar digital nerve of the thumb in a 43-year-old woman. Magnetic resonance imaging revealed a large fusiform mass along the nerve. The findings were unusual and pathognomonic and included a coaxial cable-like appearance on axial sections and a spaghettilike appearance on coronal sections on both T1- and T2-weighted images; these findings were useful for the diagnosis and preoperative evaluation of this lesion. Surgical exploration revealed a yellow, cordlike mass of the digital nerve enlarged by fat. Gross excision could not be done without extensive damage to the nerve. Therefore, a limited excision with biopsy of the fibrolipomatous tissue around the nerve bundles was performed. The histological appearance was consistent with fibrolipomatous hamartoma. There was no recurrence of the mass and no neurological deficit 3 years after surgery. Some authors have suggested that invasive excision can cause catastrophic sensory or motor deficits because of the extensive fatty infiltration of the nerve fascicles. In conclusion, the recommended treatment for this lesion is limited excision with only biopsy to confirm the diagnosis.

J Nippon Med Sch 2011; 78: 388-392

Keywords
fibrolipomatous hamartoma, digital nerve, magnetic resonance imaging, treatment, surgery

Correspondence to
Mitsuhiko Nanno, Department of Orthopaedic Surgery, Nippon Medical School, Musashi Kosugi Hospital, 1-396 Kosugi-cho, Nakahara-ku, Kawasaki, Kanagawa 211-8533, Japan
nanno-mi@ga2.so-net.ne.jp

Received, June 7, 2011
Accepted, June 22, 2011