639
Short report letters Acknowledgements The authors would like to thank Norimasa Iwasaki, MD, PhD; Noriaki Suematsu, MD; Shigeharu Uchiyama, MD, PhD; Hiroshi Yamazaki, MD; Toshimitsu Momose, MD, PhD; Hiroyuki Matsuki, MD; Yoshihiro Sugimoto, MD; Isao Sasaki, MD; Koichi Nakamura, MD; and Toshiro Itsubo, MD, for their assistance in collecting patient data, and Mr. Trevor Ralph for his English-language editorial assistance.
Conflict of interests None declared.
Ethical approval All protocols for this retrospective study were approved by the institutional review board of each participating hospital.
References Cooney WP, Bussey R, Dobyns JH, Linsheid RL. Difficult wrist fractures. Clin Orthop Relat Res. 1987, 214: 136–47. Itsubo T, Hayashi M, Uchiyama S, Hirachi K, Minami A, Kato H. Differential onset patterns and causes of carpal tunnel syndrome after distal radius fracture: a retrospective study of 105 wrists. J Orthop Sci. 2010, 15: 518–23. Lichtman DM, Alexander AH, Mack GR, Gunther SF. Kienbock’s disease—update on silicone replacement arthroplasty. J Hand Surg. 1982, 7: 343–7. Schuind F, Eslami S, Ledoux P. Kienböck’s disease. J Bone Joint Surg Br. 2008, 90: 133–9.
patient did not complain about any loss of sensation to the tip of the fingers. On clinical examination, a non-pulsating, tender, firm but mobile 3 × 3 cm subcutaneous lesion was identified ulnar to the midline at the distal wrist crease. Light touch of the overlying skin elicited uncomfortable hypersensitivity. Tinel’s sign over the carpal tunnel and the Guyon’s canal was negative. Phalen’s test and Durkan’s test were also negative. Compression test for the Guyon’s canal was negative. A magnetic resonance imaging scan confirmed a lesion arising from the median nerve, suggestive of a nerve sheath tumour (Figure 1). Surgical exploration under regional anaesthesia revealed a very vascular encapsulated mass within the median nerve, extending into the carpal tunnel (Figure 2(a)) and causing separation of the fascicles (Figure 2(b)). A prominent branch of the median artery was feeding into the mass and the artery required ligation. The mass was then shelled out under the microscope preserving all fascicles, which were then realigned into their normal anatomical position l. The sheath was left unsutured to allow egress of any bleeding. The carpal tunnel was released. Histology reported a completely excised angioleiomyoma with numerous ectatic vascular channels. Postoperatively, there was no neurovascular deficit and there was no recurrence at one year. Angioleiomyoma (or angiomyoma) is an uncommon benign vascular tumour in the extremities
M. Hayashi, M. Makoto and H. Kato Department of Orthopaedic Surgery, Shinshu University School of Medicine, Matsumoto, Nagano, and Department of Orthopaedic Surgery, Hokkaido University School of Medicine, Sapporo, Hokkaido, Japan. Corresponding author: mhayashi@ shinshu-u.ac.jp © The Author(s) 2013 Reprints and permissions: sagepub.co.uk/journalsPermissions.nav doi: 10.1177/1753193413481937 available online at http://jhs.sagepub.com
Intraneural angioleiomyoma of the median nerve at the wrist Dear Sir, A 63-year-old right-hand dominant woman with mild Parkinson’s disease presented with a 2-year history of a slowly growing mass and worsening discomfort on the palmar aspect of her left wrist. There were no symptoms of nerve compression and the
Figure 1. Sagittal magnetic resonance imaging of the left wrist illustrating the mass.
Downloaded from jhs.sagepub.com at University of Victoria on November 14, 2015
640
The Journal of Hand Surgery (Eur) 40(6)
Segmental neurofibromatosis of the upper extremity: a case report Dear Sir,
Figure 2. Intra-neural angioleiomyoma volar aspect of the left wrist (a) anterior surface; (b) posterior surface of the median nerve; illustrating the dispersed fascicles of median nerve.
(Houdek et al. 2013). Only one case of intra-neural angioleiomyoma of the median nerve at the wrist level has previously been reported (Piers et al., 1996). This lesion can be completely enucleated without resulting in neurological deficits as in radical excision of infiltrating tumours like neurofibromas, hemangiomas or lipofibromatous hamartomas. Acknowledgement We thank Mr David Elliot for his help in editing the letter.
Conflict of interests None declared.
References Houdek M, Rose PS, Shon W, Kakar S. Angioleiomyoma of the upper extremity. J Hand Surg Am. 2013, 38: 1579–83. Piers W, Terrono AL, Hayek J, Millender LH. Angiomyoma (vascular leiomyoma) of the median nerve. J Hand Surg Am. 1996, 21: 285–6.
SS Jing and T Giesen Orthopedic and Trauma Department, Southend University Hospital, Westcliff-on-Sea, UK. Corresponding author: [email protected]
© The Author(s) 2013 Reprints and permissions: sagepub.co.uk/journalsPermissions.nav doi: 10.1177/1753193413510441 available online at http://jhs.sagepub.com
A 71-year-old woman presented with multiple nodules on her left hand, localized to the ulnar side of both the palm and dorsum, with associated numbness in the ulnar nerve distribution (Figure 1A, B). She did not experience any pain or hand dysfunction. The nodules had been growing progressively for over 20 years. The nodules were relatively hard and were mobile with respect to the surrounding deep tissues. There were no nodules or areas of abnormal skin pigmentation elsewhere on her body. We diagnosed them as multiple tumours of the hand and planned excision biopsy. The nodules were excised via incisions on the palmar and dorsal surfaces of the hand. Small areas of adherent skin were resected together with the superficial nodules. The deeper nodules were encapsulated without adherence to the adjacent tissues, enabling complete excision (Figure 1C). Histopathological examination confirmed neurofibromatosis. Postoperatively, grip strength and movements were normal. She had follow up after 1 year, and no tumour recurrence was found. The first case of segmental neurofibromatosis was reported in 1931. Many cases of segmental neurofibromatosis have been reported since then, but among them only two involved the upper limb (Gonzalez et al., 2007). Our patient’s lesions were segmental, unilateral, not associated with extracutaneous lesions, and nonfamilial, which is consistent with type V (i.e., segmental form) according to the Riccardi classification (Gonzalez et al., 2007). Roth et al. (1987) proposed a further classification of segmental neurofibromatosis into four subtypes: I, true segmental; II, localized with deep involvement (nonfamilial); III, hereditary segmental (no deep involvement, familial); and IV, bilateral segmental (no deep involvement, nonfamilial). The neurofibromatosis type I gene has been mapped on chromosome 17q11.2. Many mutations of this gene have been identified, and less than 40% of these can be detected by current methods. Recently, Tinschert et al. (2000) reported that segmental neurofibromatosis is caused by somatic mutation of neurofibromatosis type I. Segmental neurofibromatosis should therefore not be regarded as a distinct entity from neurofibromatosis type I, and all patients with suspected segmental neurofibromatosis should undergo a thorough physical examination and be offered genetic counselling, if necessary. Magnetic resonance imaging is considered an important modality for diagnosing segmental neurofibromatosis. T1-weighted images show round
Downloaded from jhs.sagepub.com at University of Victoria on November 14, 2015
Short report letters Acknowledgements The authors would like to thank Norimasa Iwasaki, MD, PhD; Noriaki Suematsu, MD; Shigeharu Uchiyama, MD, PhD; Hiroshi Yamazaki, MD; Toshimitsu Momose, MD, PhD; Hiroyuki Matsuki, MD; Yoshihiro Sugimoto, MD; Isao Sasaki, MD; Koichi Nakamura, MD; and Toshiro Itsubo, MD, for their assistance in collecting patient data, and Mr. Trevor Ralph for his English-language editorial assistance.
Conflict of interests None declared.
Ethical approval All protocols for this retrospective study were approved by the institutional review board of each participating hospital.
References Cooney WP, Bussey R, Dobyns JH, Linsheid RL. Difficult wrist fractures. Clin Orthop Relat Res. 1987, 214: 136–47. Itsubo T, Hayashi M, Uchiyama S, Hirachi K, Minami A, Kato H. Differential onset patterns and causes of carpal tunnel syndrome after distal radius fracture: a retrospective study of 105 wrists. J Orthop Sci. 2010, 15: 518–23. Lichtman DM, Alexander AH, Mack GR, Gunther SF. Kienbock’s disease—update on silicone replacement arthroplasty. J Hand Surg. 1982, 7: 343–7. Schuind F, Eslami S, Ledoux P. Kienböck’s disease. J Bone Joint Surg Br. 2008, 90: 133–9.
patient did not complain about any loss of sensation to the tip of the fingers. On clinical examination, a non-pulsating, tender, firm but mobile 3 × 3 cm subcutaneous lesion was identified ulnar to the midline at the distal wrist crease. Light touch of the overlying skin elicited uncomfortable hypersensitivity. Tinel’s sign over the carpal tunnel and the Guyon’s canal was negative. Phalen’s test and Durkan’s test were also negative. Compression test for the Guyon’s canal was negative. A magnetic resonance imaging scan confirmed a lesion arising from the median nerve, suggestive of a nerve sheath tumour (Figure 1). Surgical exploration under regional anaesthesia revealed a very vascular encapsulated mass within the median nerve, extending into the carpal tunnel (Figure 2(a)) and causing separation of the fascicles (Figure 2(b)). A prominent branch of the median artery was feeding into the mass and the artery required ligation. The mass was then shelled out under the microscope preserving all fascicles, which were then realigned into their normal anatomical position l. The sheath was left unsutured to allow egress of any bleeding. The carpal tunnel was released. Histology reported a completely excised angioleiomyoma with numerous ectatic vascular channels. Postoperatively, there was no neurovascular deficit and there was no recurrence at one year. Angioleiomyoma (or angiomyoma) is an uncommon benign vascular tumour in the extremities
M. Hayashi, M. Makoto and H. Kato Department of Orthopaedic Surgery, Shinshu University School of Medicine, Matsumoto, Nagano, and Department of Orthopaedic Surgery, Hokkaido University School of Medicine, Sapporo, Hokkaido, Japan. Corresponding author: mhayashi@ shinshu-u.ac.jp © The Author(s) 2013 Reprints and permissions: sagepub.co.uk/journalsPermissions.nav doi: 10.1177/1753193413481937 available online at http://jhs.sagepub.com
Intraneural angioleiomyoma of the median nerve at the wrist Dear Sir, A 63-year-old right-hand dominant woman with mild Parkinson’s disease presented with a 2-year history of a slowly growing mass and worsening discomfort on the palmar aspect of her left wrist. There were no symptoms of nerve compression and the
Figure 1. Sagittal magnetic resonance imaging of the left wrist illustrating the mass.
Downloaded from jhs.sagepub.com at University of Victoria on November 14, 2015
640
The Journal of Hand Surgery (Eur) 40(6)
Segmental neurofibromatosis of the upper extremity: a case report Dear Sir,
Figure 2. Intra-neural angioleiomyoma volar aspect of the left wrist (a) anterior surface; (b) posterior surface of the median nerve; illustrating the dispersed fascicles of median nerve.
(Houdek et al. 2013). Only one case of intra-neural angioleiomyoma of the median nerve at the wrist level has previously been reported (Piers et al., 1996). This lesion can be completely enucleated without resulting in neurological deficits as in radical excision of infiltrating tumours like neurofibromas, hemangiomas or lipofibromatous hamartomas. Acknowledgement We thank Mr David Elliot for his help in editing the letter.
Conflict of interests None declared.
References Houdek M, Rose PS, Shon W, Kakar S. Angioleiomyoma of the upper extremity. J Hand Surg Am. 2013, 38: 1579–83. Piers W, Terrono AL, Hayek J, Millender LH. Angiomyoma (vascular leiomyoma) of the median nerve. J Hand Surg Am. 1996, 21: 285–6.
SS Jing and T Giesen Orthopedic and Trauma Department, Southend University Hospital, Westcliff-on-Sea, UK. Corresponding author: [email protected]
© The Author(s) 2013 Reprints and permissions: sagepub.co.uk/journalsPermissions.nav doi: 10.1177/1753193413510441 available online at http://jhs.sagepub.com
A 71-year-old woman presented with multiple nodules on her left hand, localized to the ulnar side of both the palm and dorsum, with associated numbness in the ulnar nerve distribution (Figure 1A, B). She did not experience any pain or hand dysfunction. The nodules had been growing progressively for over 20 years. The nodules were relatively hard and were mobile with respect to the surrounding deep tissues. There were no nodules or areas of abnormal skin pigmentation elsewhere on her body. We diagnosed them as multiple tumours of the hand and planned excision biopsy. The nodules were excised via incisions on the palmar and dorsal surfaces of the hand. Small areas of adherent skin were resected together with the superficial nodules. The deeper nodules were encapsulated without adherence to the adjacent tissues, enabling complete excision (Figure 1C). Histopathological examination confirmed neurofibromatosis. Postoperatively, grip strength and movements were normal. She had follow up after 1 year, and no tumour recurrence was found. The first case of segmental neurofibromatosis was reported in 1931. Many cases of segmental neurofibromatosis have been reported since then, but among them only two involved the upper limb (Gonzalez et al., 2007). Our patient’s lesions were segmental, unilateral, not associated with extracutaneous lesions, and nonfamilial, which is consistent with type V (i.e., segmental form) according to the Riccardi classification (Gonzalez et al., 2007). Roth et al. (1987) proposed a further classification of segmental neurofibromatosis into four subtypes: I, true segmental; II, localized with deep involvement (nonfamilial); III, hereditary segmental (no deep involvement, familial); and IV, bilateral segmental (no deep involvement, nonfamilial). The neurofibromatosis type I gene has been mapped on chromosome 17q11.2. Many mutations of this gene have been identified, and less than 40% of these can be detected by current methods. Recently, Tinschert et al. (2000) reported that segmental neurofibromatosis is caused by somatic mutation of neurofibromatosis type I. Segmental neurofibromatosis should therefore not be regarded as a distinct entity from neurofibromatosis type I, and all patients with suspected segmental neurofibromatosis should undergo a thorough physical examination and be offered genetic counselling, if necessary. Magnetic resonance imaging is considered an important modality for diagnosing segmental neurofibromatosis. T1-weighted images show round
Downloaded from jhs.sagepub.com at University of Victoria on November 14, 2015
