Case Reports / Journal of Clinical Neuroscience 16 (2009) 1658–1660
Skull osteohypertrophy as a complication of bone wax Tomoya Kamide, Mitsutoshi Nakada *, Yuichi Hirota, Yutaka Hayashi, Yasuhiko Hayashi, Naoyuki Uchiyama, Jun-ichiro Hamada Department of Neurosurgery, Division of Neuroscience, Graduate School of Medical Science, Kanazawa University, 13-1 Takaramachi, Kanazawa, Ishikawa 920-8641, Japan
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Article history: Received 15 February 2009 Accepted 4 March 2009
Keywords: Bone wax Osteohypertrophy Exophthalmos Foreign body
a b s t r a c t A 51-year-old woman with bone wax–induced osteohypertrophy presented with exophthalmos 9 years after a craniotomy for a right internal carotid bifurcation aneurysm. CT scans revealed thickening of the frontotemporal bone ﬂap and surrounding bone, thickening of the upper and lateral orbital walls, and a limited intraorbital cavity. Intraoperative ﬁndings revealed residual bone wax under the bone ﬂap, grayish-white discoloration of the ﬂap, and degeneration of the temporal muscle. Pathological examination revealed granulation and osteogenesis due to a foreign body. To our knowledge, this is the ﬁrst report of bone wax–induced hyperostosis leading to exophthalmos. Ó 2009 Elsevier Ltd. All rights reserved.
1. Introduction Bone wax is a safe agent widely used to control bleeding from bone surfaces and cut edges. Bone wax–induced osteohypertrophy has not been reported to our knowledge, although other adverse effects are known.1–8 2. Case report A 51-year-old woman noticed a gradual protrusion of her right eyeball 9 years after a right frontotemporal craniotomy for an internal carotid bifurcation aneurysm. CT scans revealed thickening of the bone ﬂap, frontal base, and upper and lateral orbital walls (Fig. 1A and B). Osteohypertrophy was noted around one hydroxyapatite burr hole button (Pentax, Tokyo, Japan) but was not seen around other buttons at different sites (Fig. 1C). Bone scintigraphy revealed hyperaccumulation of radioisotope in the osteohypertrophic regions (Fig. 1D). There were no other physical or neurological defects, abnormal laboratory data, or history of allergic reactions. The surgical ﬁndings were: (i) degeneration of the muscle covering the bone ﬂap; and (ii) grayish-white discoloration and considerable thickening of the bone ﬂap, especially the frontal base. The removal of the bone ﬂap bearing the burr hole button revealed residual bone wax (equivalent to the size of the tip of a little ﬁnger) under the button at the center of the thickened region (Fig. 2A). This location of bone wax corresponded to the CT scan ﬁndings (Fig. 1A and B; black arrows). Changes were not observed around the other 3 buttons. The bone wax was removed, and cranioplasty was performed using an artiﬁcial bone (Codman Cranioplastic ; Johnson & Johnson Medical, Bracknell, England). Histological examination of the tissue surrounding the bone wax, including the hypertrophic bone, conﬁrmed granulation caused by a foreign body and osteogenesis proximal to the granulation (Fig. 2B–D). The postoperative course was uneventful. Although the exophthalmos did not dramatically change, progression of the osteohypertrophy was stalled after 6 months (as observed on a CT scan). TM
3. Discussion Adverse effects of bone wax have been reported including: (i) induction of a foreign body granulomatous reaction that prevents * Corresponding author. Tel.: +81 76 265 2384; fax: +81 76 234 4262. E-mail address: [email protected]
bone healing; (ii) osteogenesis; and (iii) bacterial clearance.1–8 Localized pain, warmth, swelling, edema, and tenderness are the main clinical symptoms.9–11 To our knowledge, this is the ﬁrst report of bone wax–induced osteohypertrophy that caused exophthalmos 9 years after the ﬁrst neurosurgery. In our patient, the bone wax comprised bees wax (77%), almond oil (21%), and salicylic acid (2%). This is not remarkably different from the bone wax composition described in other reports; therefore, the composition of the bone wax used in our patient was not unique. The mechanism of osteohypertrophy is unknown. Histological examination revealed severe granulation with multinucleate giant cells in the soft tissue surrounding the bone wax. Inﬂammatory cells in the bone marrow and osteoblasts on the bone surface were noted. Allison et al. also reported osteoblast accumulation on the bone surface after bone wax was used for a mandibular bone fracture.2 We speculate that chronic inﬂammation eventually extended to the bone marrow, resulting in osteoblast activation and osteogenesis. Activation may also occur as a result of the interaction between the burr hole button and bone wax. Burr hole buttons, which consist of hydroxyapatite, are bone substitutes to implant the bone defects. Although the chemical reaction of bone wax with hydroxyapatite has never been reported, simultaneous use might induce osteoblast activation. Verborgt et al. reported the occurrence of a retroperitoneal tumor associated with bone wax 19 years after orthopedic surgery.12 Previous reports have described bone wax–associated complications that occur within 2 years. The onset of complications 9 years after surgery, as observed in our patient, is uncommon. We speculate that the bone wax was deposited under the burr hole button (not under the skin), causing hyperostosis without severe inﬂammation of the surrounding soft tissues; therefore, no symptoms were observed until the manifestation of exophthalmos. Osteohypertrophy mostly manifests in ﬁbrous dysplasia, Paget’s disease, or bone tumors. Diagnosis is based on clinical history, CT scans, bone scintigraphy, and other diagnostic methods. Tehranzadeh et al. reported that CT scans can successfully distinguish Paget’s disease from ﬁbrous dysplasia.13 Bone scintigraphy is also useful for detecting other hypertrophic regions and determining focal or systemic disorders. In our patient, CT scans revealed osteohypertrophy localized around the burr hole button, and a groundglass appearance, a characteristic of Paget’s disease, was not observed. Bone scintigraphy revealed local hyperaccumulation corresponding to the lesion, without other lesions, indicating a focal
Case Reports / Journal of Clinical Neuroscience 16 (2009) 1658–1660
Fig. 1. (A) Axial CT scan with bone window showing the thickened frontotemporal bone, including the bone ﬂap. A bone button is present in the center of the surrounding thickened bone (white arrow) and a free space is observed under the bone button (black arrow). (B) Coronal reconstructed CT scan showing the thickened walls of the orbit; the free space (black arrow) and the bone button (white arrow) can be seen. (C) Axial CT scan conﬁrming that osteohypertrophy was not seen around other buttons at a different site (white arrow). (D) Bone scintigraph showing hyperaccumulation corresponding to the thickened bone observed in the CT scan.
Fig. 2. (A) Intraoperative photograph showing the bone wax (white arrow) under the bone button (asterisk). (B) Photomicrograph showing the section of the bone adjacent to the bone wax. Chronic inﬂammation is observed in the soft tissue overlying the bone wax (hematoxylin and eosin [H&E], 100). (C) Photomicrograph showing the accumulation of osteoblasts on the surface of the bone (arrow) (H&E, 200). (D) Photomicrograph of a soft tissue section surrounding the bone wax showing the granulomatous reaction to the foreign body by multinucleated giant cells (H&E, 400).
Case Reports / Journal of Clinical Neuroscience 16 (2009) 1660–1662
disorder. A bone tumor was the preoperative differential diagnosis. The ﬁnal diagnosis was made after pathological analysis.14 The course of hyperosteotrophy and symptoms varies between patients,9,10,15 and manifestation of a complication should indicate surgery for the removal of the foreign agent. In our patient, we removed the bone wax and a part of the thickened bone, but some parts, such as the surrounding orbit, were not removed. We are monitoring the patient for symptoms and residual hyperostosis. We believe that the use of bone wax should be restricted to a minimum, and all excess fragments and pieces should be removed from the soft tissue. Bone wax is a foreign agent and surgeons should be aware of the potential complications that could arise from its use. References 1. Alberius P, Klinge B, Sjogren S. Effects of bone wax on rabbit cranial bone lesions. J Craniomaxillofac Surg 1987;15:63–7. 2. Allison RT. Foreign body reactions and an associated histological artifact due to bone wax. Br J Biomed Sci 1994;51:14–7. 3. Aurelio J, Chenail B, Gerstein H. Foreign-body reaction to bone wax. Report of a case. Oral Surg Oral Med Oral Pathol 1984;58:98–100.
4. Gibbs L, Kakis A, Weinstein P, et al. Bone wax as a risk factor for surgical site infection following neurospinal surgery. Infect Control Hosp Epidemiol 2004;25:346–8. 5. Johnson P, Fromm D. Effects of bone wax on bacterial clearance. Surgery 1981;89:206–9. 6. Lavigne M, Boddu Siva Rama KR, Doyon J, et al. Bone-wax granuloma after femoral neck osteoplasty. Can J Surg 2008;51:58–60. 7. Patel RB, Kwartler JA, Hodosh RM. Bone wax as a cause of foreign body granuloma in the cerebellopontine angle. Case illustration. J Neurosurg 2000;92:362. 8. Sorrenti SJ, Cumming WJ, Miller D. Reaction of the human tibia to bone wax. Clin Orthop 1984;182:293–6. 9. Julsrud ME. A surgical complication: allergic reaction to bone wax. J Foot Surg 1980;19:152–4. 10. Katz SE, Rootmann J. Adverse effects of bone wax in surgery of the orbit. Ophthal Plast Reconstr Surg 1996;12:121–6. 11. Tay HL, Tan LKS. Surgical bone wax causing epistaxis. J Laryngol Otol 1996;110:267–8. 12. Verborgt O, Verellen K, Thielen FV, et al. A retroperitoneal tumor as a late complication of the use of bone wax. Acta Orthop Belg 2000;66:389–91. 13. Tehranzadeh J, Fung Y, Donohue M, et al. Computed tomography of Paget disease of the skull versus ﬁbrous dysplasia. Skeletal Radiol 1998;27:664–72. 14. Wagner PH, Heilmann P, Schulz A, et al. Fibrous dysplasia: differential diagnosis from Paget’s disease. Dtsch Med Wochenschr 2002;127:2264–8. 15. Anﬁnsen OG, Sudmann B, Rait M, et al. Complications secondary to the use of standard bone wax in seven patients. J Foot Ankle Surg 1993;32:505–8.
Immunohistochemical ﬁndings of multiple ossiﬁed en plaque meningiomas in the thoracic spine Kenzo Uchida a,*, Hideaki Nakajima a, Takafumi Yayama a, Ryuichiro Sato a, Shigeru Kobayashi a, Erisa S. Mwaka a,b, Yoshiaki Imamura c, Hisatoshi Baba a a
Department of Orthopaedics and Rehabilitation Medicine, Fukui University, Faculty of Medical Sciences, Matsuoka Shimoaizuki 23, Eiheiji, Fukui 910-1193, Japan Department of Orthopaedic Surgery, Makerere University Medical School, Kampala, Republic of Uganda c Department of Clinical Pathology, Fukui University, Faculty of Medical Sciences, Fukui, Japan b
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Article history: Received 17 December 2008 Accepted 19 March 2009
Keywords: Calciﬁcation Epiconus syndrome Immunohistochemistry Meningioma Ossiﬁcation Spinal cord
a b s t r a c t We describe a 76-year-old woman who presented with painful muscle weakness and numbness in both legs secondary to grossly ossiﬁed meningiomas at the T8 and T11–T12 vertebral levels. Midline durotomy followed by en bloc excision of the tumours, together with partial resection of the dura mater and the arachnoid, was performed. Histopathological examination showed both tumours consisted of three different histological patterns: psammomatous, ﬁbrous, and metaplastic subtypes. Immunostaining for bone morphogenetic protein-2 and vascular endothelial growth factor was signiﬁcantly positive in the areas surrounding mature bone tissues in the metaplastic and psammomatous subtypes.
1. Introduction Meningioma of the spinal cord is a common intradural extramedullary neoplasm, comprising 25–43% of all spinal cord tumours that emanate from the meninges, with the highest frequency from the anterolateral aspect of the arachnoid.1,2 Psammomatous meningothelial meningioma is an infrequent lesion that appears as a calciﬁed mass on plain radiograph or CT scan.3–5 The ossiﬁed spinal meningioma en plaque3,6 is extremely rare, with a reported incidence of 1–5% of all spinal meningiomas.3,7 However, the mecha* Corresponding author. Tel.: +81 776 61 8383; fax: +81 776 61 8125. E-mail address: [email protected]
Ó 2009 Elsevier Ltd. All rights reserved.
nism responsible for the ossiﬁcation process remains poorly understood. We describe a patient with multiple ossiﬁed spinal en plaque meningiomas, and report the immunohistochemical ﬁndings. 2. Case report A 76-year-old woman presented with a 2-year history of painful muscle weakness and numbness in both legs, with right-side predominance. She also complained of fatigue, a sensation of heaviness, weakness of both lower extremities and urinary incontinence. Walking increased radicular-type leg pain, which resembled spinal cord intermittent claudication.