Cancellous bone-like tissue replacement from calcinosis in patients with systemic sclerosis with multiple external root resorption

Takumi Memida; Shinji Matsuda; Takashi Nakamoto; Kazuhisa Ouhara; Mikihito Kajiya; Shintaro Hirata; Eiji Sugiyama; Naoya Kakimoto; Noriyoshi Mizuno

doi:10.1016/j.bonr.2021.101165

Cancellous bone-like tissue replacement from calcinosis in patients with systemic sclerosis with multiple external root resorption

Bone Rep. 2022 Jan 8:16:101165. doi: 10.1016/j.bonr.2021.101165. eCollection 2022 Jun.

Authors

Takumi Memida¹, Shinji Matsuda¹, Takashi Nakamoto², Kazuhisa Ouhara¹, Mikihito Kajiya¹, Shintaro Hirata³, Eiji Sugiyama^{3

4}, Naoya Kakimoto², Noriyoshi Mizuno¹

Affiliations

¹ Department of Periodontal Medicine, Applied Life Sciences, Institute of Biomedical & Health Sciences, Hiroshima University, 1-2-3 Kasumi, Minami-ku, Hiroshima 734-8553, Japan.
² Department of Oral and Maxillofacial Radiology, Institute of Biomedical and Health Sciences, Hiroshima University, 1-2-3 Kasumi, Minami-ku, Hiroshima 734-8553, Japan.
³ Department of Clinical Immunology and Rheumatology, Hiroshima University Hospital, 1-2-3 Kasumi, Minami-ku, Hiroshima 734-8553, Japan.
⁴ Department of Internal Medicine, JA Yoshida General Hospital, 3666 Yoshida, Yoshida-cho, Akitakata, Hiroshima 731-0501, Japan.

Abstract

Calcinosis is frequently observed in patients with systemic sclerosis (SSc). The fundamental treatment of calcinosis has not yet been established. During follow-up, calcinosis in the subcutaneous surface is often spontaneously extracted or remains confined by fibrous tissues. We previously identified a new symptom in SSc patients, multiple external root resorption (MERR), and these patients had calcifications in the nasal spine. Here, we report for the first time that calcinosis at the nasal spine in patients with MERR can be replaced by cancellous bone-like tissue. Patients 1 and 2 were a 62-year-old Japanese female and a 45-year-old Japanese female (respectively) with MERR who had been previously treated for SSc (Patient 1: limited type, positive for anti-centromere antibody; Patient 2: diffuse type, positive for anti-Scl70 and anti-SS-A antibodies). Patient 3 was a 57-year-old female with MERR who had been previously treated for SSc (diffuse type, positive anti-Scl-70 antibody) and underwent denosumab injection for osteoporosis. Cone-beam computed tomography (CBCT) and CT images in the calcifications at the nasal spine in Patient 1 and 2 were replaced with cancellous bone-like tissue, but not in Patient 3. Serum laboratory examination was performed to assess the systemic bone disease. All three patients had normal clinical data within the references, apart from slightly higher 1,25-dihydroxyvitamin D levels in Patient 1. SSc patients with calcinosis in the maxillofacial area need to be examined carefully for bone replacement using CBCT or CT.

Keywords: BAP, bone alkaline phosphatase; CBCT, cone-beam computed tomography; CREST, calcinosis, Raynaud's phenomenon, esophageal dysmotility, sclerodactyly, and telangiectasia; Calcinosis; FOP, fibrodysplasia ossificans progressive; HO, heterotopic ossification; Heterotopic ossification; MERR, multiple external root resorption; Multiple external root resorption; NSAID, non-steroidal anti-inflammatory drugs; POH, progressive osseous heteroplasia; PTH, parathyroid hormone; RANK, receptor activator of nuclear factor-kappa B; SSc, systemic sclerosis; Systemic sclerosis.

Publication types

Case Reports