Immunohistochemical analyses to determine pathogenesis of tenosynovitis with psammomatous calcification in the wrist: A case report

  • Authors:
    • Chika Miyasaka
    • Mitsuaki Ishida
    • Yoshitaka Hamada
    • Takeyasu Toyama
    • Koji Tsuta
  • View Affiliations

  • Published online on: May 31, 2019     https://doi.org/10.3892/br.2019.1218
  • Pages: 27-30
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Abstract

Tenosynovitis with psammomatous calcification is an extremely rare clinicopathological condition, which is characterized histopathologically by the presence of numerous psammomatous calcifications surrounded by a granulomatous reaction comprising a mixture of histiocytes and fibroblasts. The pathogenesis of this disease remains unclear, although an association with repetitive tendinous injury has been proposed. The present study describes the details of a case in an elderly Japanese female, and, to the best of our knowledge, the first known immunohistochemical analysis of the mechanism underlying psammomatous calcification formation. A 66‑year‑old Japanese woman presented with pain in the right wrist. The lesion was surgically resected. Histopathological examination revealed a well‑circumscribed lesion composed of psammomatous calcification. The calcification was surrounded by histiocytes, and a few multinucleated giant cells and fibroblastic spindle cells. Immunohistochemical study revealed that these histiocytes were positive for cluster of differentiation 163, and the histiocytes and spindle cells surrounding the psammomatous calcification expressed bone morphogenetic protein‑1 (BMP‑1). Tenosynovitis with psammomatous calcification is hypothesized to be a distinctive subtype of idiopathic calcifying tenosynovitis involving an unusual reactive or degenerative process. BMP‑1 has been demonstrated to be involved in the regulation of hard tissue mineralization, and its expression has been suggested to be associated with psammoma formation in papillary thyroid cancer. To the best of our knowledge, the case report within the present study suggested for the first time that BMP‑1 expression was associated with development of psammomatous calcification in this condition.

Introduction

Tenosynovitis with psammomatous calcification is an extremely rare clinicopathological condition. Since it was first described by Gravanis and Gaffney in 1983(1), only a few additional cases have been examined in the English language literature (2-5). This variant of calcifying tenosynovitis or calcific tendonitis is characterized histopathologically by the presence of numerous psammomatous calcifications surrounded by a granulomatous reaction comprising a mixture of histiocytes and fibroblasts (2-5). Recently, Michal et al (6) investigated a large case series of this disease, and confirmed that the disease exhibited a tendency to affect the fingers or toes of young to middle-aged women, and appeared to be associated with trauma and/or repetitive activity. They concluded that tenosynovitis with psammomatous calcification is a distinctive trauma-associated subtype of idiopathic calcifying tenosynovitis.

However, the pathogenesis of tenosynovitis with psammomatous calcification remains unclear. Although an association of this disease with repetitive tendinous injury has been described previously (2,6), other studies have described cases without a history of trauma (3,4). Bone morphogenetic protein (BMP)-1, also known as procollagen C-peptidase, is a multifunctional protein regulating of hard tissue mineralization (7). BMP-1 expression has been suggested to be associated with ectopic ossification (8) and psammoma formation in papillary thyroid cancer (9). The present study described a case report of tenosynovitis with psammomatous calcification that occurred in the wrist of an elderly female, and the immunohistochemical analysis of the mechanism of psammomatous calcification formation, particularly association with BMP-1 expression.

Case report

A 66-year-old Japanese female presented with pain in the right wrist. She had a history of De Quervain's disease and infliximab use for ulcerative colitis, but no history of trauma to the wrist. Radiographic imaging demonstrated a calcified lesion in the palmar side of the right wrist, around the lunar and capitate bones. The lesion was surgically resected following a clinical diagnosis of ectopic bone formation.

Samples were fixed in 10% formalin at room temperature for 24 h and paraffin-embedded specimens (60˚C, 4 h) of the resected lesion were processed for routine histological examination and immunohistochemical analyses. Immunohistochemical analyses were performed using autostainers (XT System Benchmark; OptiView DAB Universal Kit, Roche Diagnostics) and Autostainer link 48 (Envision FLEX; cat. no. K8000; Dako; Agilent Technologies). The primary antibodies used were a mouse monoclonal antibody against α-smooth muscle actin (α-SMA; clone name, 1A4; 20 min at room temperature; Dako; Agilent Technologies), a rabbit polyclonal antibody against BMP-1 (cat. no. ab205394; 32 min at room temperature; Abcam) and a mouse monoclonal antibody against CD163 (clone name, 10D6; 32 min at room temperature; Leica Microsystems, Ltd). Light microscopic examination of 3-µm H&E-stained (hematoxylin, 3 min and eosin, 5 min at room temperature) sections (magnification, x12.5, x100 and x400) revealed a well-circumscribed lesion with a central cystic cavitation (Fig. 1A), and the notable feature of numerous psammomatous calcifications (Fig. 1B). These calcifications were surrounded by histiocytes, and a few multinucleated giant cells and fibroblastic spindle cells (Fig. 1C). Neither foamy cells nor siderophages were observed within the lesion.

Light microscopic analyses of immunohistochemistry (magnification, x400) revealed that these histiocytes were positive for CD163 (Fig. 1D). A small number of α-SMA-positive spindle cells were also detected, and the histiocytes and spindle cells surrounding the psammomatous calcification expressed BMP-1 (Fig. 1E). Based on these features, a final diagnosis of tenosynovitis with psammomatous calcification was made.

Discussion

In the present report, a case of tenosynovitis with psammomatous calcification was described. In addition, to the best of our knowledge, this was the first time immunohistochemical analysis was used to identify the potential mechanism of psammoma formation. Table I summarizes the clinicopathological features of the case in the present study, and those of previously described cases. As demonstrated, this disease affects patients with a median age of 44 years (14-83 years), with a female predominance (male:female ratio, 4:30), particularly in young to middle-aged women. Of the patients examined previously and in the presents study, 12 of 25 had a history of trauma or repetitive activity. The majority of cases occurred in the hand, in particular in the finger, or the foot, and the most common complaint was a painful mass. A previous study involving the largest case series revealed these aforementioned clinicopathological features of tenosynovitis with psammomatous calcification (6), which is believed to be a distinct clinicopathological condition involving an unusual reactive or degenerative process in the chronically traumatized tendon and peritendinous tissue (2,6). However, the underlying molecular mechanism of development of this disease remains unclear.

Table I

Clinicopathological features of tenosynovitis with psammomatous calcification.

Table I

Clinicopathological features of tenosynovitis with psammomatous calcification.

Author, yearCase no.Age, yearsSexLocationPrimary complaintHistory of trauma or repetitive activity(Refs.)
Gravanis and Gaffney, 1983154MaleRight pectoralis minor tendonNANA(1)
 228FemaleLeft ring finger, PIP jointSwellingNA(1)
Shon and Flope, 2010316FemaleRight foot peritendinous tissuePainful massYes(2)
 419FemaleRight foot peritendinous tissuePoint tendernessYes(2)
 540FemaleRight thumbPainful massYes(2)
 663FemaleRight flexor carpal tendonPersistent painYes(2)
 767FemaleRight ring fingerPainful massYes(2)
 883FemaleRight middle fingerPainful massYes(2)
Robb et al, 2012952FemaleLeft kneePersistent painNo(3)
Kawata et al, 20141035MaleLeft middle finger, PIP jointPainful swellingNo(4)
Fox et al, 20171114MaleRight ring fingerPain, intermittent lockingNo(5)
Michal et al, 20181216FemaleRight footNAYes(6)
 1317FemaleLeft great toePainYes(6)
 1418FemaleLeft IV fingerNerve tinglingYes(6)
 1519FemaleRight foot MTP jointPainNA(6)
 1625FemaleRight toe MTP jointPain and edemaYes(6)
 1733FemaleRight IV MCP jointSwelling with painNo(6)
 1833FemaleRight IV finger PIP jointPainNo(6)
 1938FemaleLeft III finger PIP jointNoneYes(6)
 2040FemaleRight IV finger PIP jointPain and edemaNo(6)
 2141FemaleRight II fingerPainNo(6)
 2244FemaleV finger PIP jointNoneNo(6)
 2344FemaleRight big toeNoneNo(6)
 2444FemaleRight IV fingerNANA(6)
 2547FemaleLeft fingerNANA(6)
 2649FemaleRight handNANA(6)
 2749FemaleLeft big toePain and edemaYes(6)
 2849FemaleRight III fingerPainNo(6)
 2950FemaleLeft thumbPainNA(6)
 3052FemaleLeft kneePainNo(6)
 3159MaleLeft IV fingerNANA(6)
 3263FemaleRight II fingerNANA(6)
 3375FemaleRight elbowPainNo(6)
Present study 66FemaleRight wristPainNo-

[i] MCP, Metacarpophalangeal; MTP, Metatarsophalangeal; NA, not available; PIP, Proximal interphalangeal.

BMP-1, also known as procollagen C-peptidase may convert a variety of precursor proteins, including procollagen and dentin matrix protein, into active forms, resulting in their involvement in cell adhesion and the regulation of hard tissue mineralization (7). Therefore, the present study focused on the association between psammomatous calcification of this lesion and BMP-1 expression. From the data in the present study, the expression of BMP-1 in histiocytes and spindle cells surrounding the psammomatous calcification was clearly observed. This suggests that the expression of BMP-1 may be associated with the development of psammomatous calcification.

In conclusion, the present study described a typical case of tenosynovitis with psammomatous calcification and reviewed its clinicopathological characteristics. The results suggested that the expression of BMP-1 in the histiocytes and spindle cells surrounding the psammomatous calcification may be associated with development of this condition.

Acknowledgements

Not applicable.

Funding

No funding was received.

Availability of data and materials

All data generated or analyzed during this study are included in this published article.

Authors' contributions

CM and MI were responsible for the conception and design of the study. CM, MI, YH, TT and KT were involved in the acquisition and analysis of the data. CM and MI drafted the manuscript. The final version of the manuscript was read and approved by all authors.

Ethics approval and consent to participate

This study was conducted in accordance with the Declaration of Helsinki, and written consent was obtained from the patient.

Patient consent for publication

Written informed consent for publication was obtained from the patient.

Competing interests

The authors declare that they have no competing interests.

References

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Spandidos Publications style
Miyasaka C, Ishida M, Hamada Y, Toyama T and Tsuta K: Immunohistochemical analyses to determine pathogenesis of tenosynovitis with psammomatous calcification in the wrist: A case report. Biomed Rep 11: 27-30, 2019
APA
Miyasaka, C., Ishida, M., Hamada, Y., Toyama, T., & Tsuta, K. (2019). Immunohistochemical analyses to determine pathogenesis of tenosynovitis with psammomatous calcification in the wrist: A case report. Biomedical Reports, 11, 27-30. https://doi.org/10.3892/br.2019.1218
MLA
Miyasaka, C., Ishida, M., Hamada, Y., Toyama, T., Tsuta, K."Immunohistochemical analyses to determine pathogenesis of tenosynovitis with psammomatous calcification in the wrist: A case report". Biomedical Reports 11.1 (2019): 27-30.
Chicago
Miyasaka, C., Ishida, M., Hamada, Y., Toyama, T., Tsuta, K."Immunohistochemical analyses to determine pathogenesis of tenosynovitis with psammomatous calcification in the wrist: A case report". Biomedical Reports 11, no. 1 (2019): 27-30. https://doi.org/10.3892/br.2019.1218