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CASE REPORT
Adv Biomed Res 2018,  7:78

Osseous Metaplasia in Rectal Polyp: A Case Report with Review of Probable Pathogenesis


1 Department of Pathology, School of Medicine, Isfahan University of Medical Sciences, Isfahan, Iran
2 Department of Surgery, School of Medicine, Isfahan University of Medical Sciences, Isfahan, Iran

Date of Web Publication23-May-2018

Correspondence Address:
Dr. Azar Naimi
Department of Pathology, School of Medicine, Isfahan University of Medical Sciences, Isfahan
Iran
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/abr.abr_169_16

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  Abstract 


Heterotopic bone formation is a rare event in gastrointestinal lesions, especially in the pediatric population. Osseous metaplasia is exceedingly rare in colonic polyps. We present a case of rectal juvenile polyp with stromal osseous metaplasia in a 10-year-old male child. This histologic finding is very rare in children with colorectal polyps.

Keywords: Juvenile, osseous metaplasia, pediatric, rectal polyp


How to cite this article:
Naimi A, Hosseinpour M. Osseous Metaplasia in Rectal Polyp: A Case Report with Review of Probable Pathogenesis. Adv Biomed Res 2018;7:78

How to cite this URL:
Naimi A, Hosseinpour M. Osseous Metaplasia in Rectal Polyp: A Case Report with Review of Probable Pathogenesis. Adv Biomed Res [serial online] 2018 [cited 2018 Dec 11];7:78. Available from: http://www.advbiores.net/text.asp?2018/7/1/78/233025




  Introduction Top


Osseous metaplasia is rarely detected in the gastrointestinal tract. It is a phenomenon that has been described in a wide variety of tissue types with respect to mainly neoplastic and rarely nonneoplastic conditions. In colonic polyps, its occurrence is extremely rare.[1]

Heterotopic bone formation in the digestive tract usually occurs in the colorectal area in association with benign or malignant epithelial tumors.[2]

Here, we report a rare case of heterotopic bone formation in a juvenile polyp, with discussing potential etiologies.


  Case Report Top


A 10-year-old male child with a history of rectorrhagia since the past 2 weeks visited to the gastrointestinal clinic of our hospital.

The local examination of the rectum revealed no abnormal finding.

A colonoscopic examination revealed a single pedunculated polyp at 5 cm of anal verge measuring 3 cm × 2 cm × 1 cm.

The rest of the colonoscopic examination was unremarkable. Thereafter, according to rather large size of polyp, the patient referred to a surgeon and a polypectomy was performed under general anesthesia and the polypoid mass was sent to the laboratory.

On gross examination, one polypoid tissue fragment of gray-white color with rather soft consistency was seen. The tissue was embedded totally. On microscopic examination, sections showed a polypoid colonic mucosa which was ulcerated, with granulation tissue, edema, and a mixed inflammatory cell infiltrate, bony trabeculae in lamina propria, which were surrounded by osteoblastic cells, was encountered also [Figure 1]a and [Figure 1]b.
Figure 1: (a) Juvenile polyp histology predominantly composed of granulation tissue (H and E, ×40). (b) Osseous metaplasia (arrow) in polyp stroma

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  Discussion Top


The osseous metaplasia has been reported in a wide variety of tissue types such as prostate, uterus, breasts, skin appendages, pulmonary system, and gastrointestinal organs with respect to both neoplastic and nonneoplastic conditions.[1],[3]

Osseous metaplasia has been described in various types of gastrointestinal polyps as well as in mucin-producing tumors. These include adenomatous, hyperplastic, and hamartomatous polyps.[4]

Although it mostly occurs in adenomatous polyps, only rare cases of osseous metaplasia have been reported in benign colonic polyps.[5]

In 1981, Sperling described bone formation within a benign lesion, a rectal polyp. Since then, it has been described a few cases of gastrointestinal polyps with osseous metaplasia. The majority of the occurrences have been rectal or left-sided lesions.[6] Other case reports comment on lesions located in the ileum,[7] stomach,[8] and Barrett's esophagus.[2] In one case report of a patient with Peutz–Jeghers syndrome, osseous metaplasia was found in 3 of 15 polyps in the jejunum.[9]

In a review of case studies among the cases with juvenile polyps, a majority of the patients were males, with a mean age of 7.4 years (range 3–15 years). The polyp size varied from 5 mm to 20 mm, with a mean size of 9 mm.

Histologically, necrosis, inflammation, preexisting calcification, increased vascularity, active chronic inflammation, ulceration, and extracellular mucin deposition were reported to be associated with heterotopic bone formation in tumors.[2],[10]

Various mechanisms have been proposed for the mechanism of bone formation.[4]

van Patter and Whittick [11] and Sanerkin [12] thought that mucinous stromal infiltration was associated with osseous metaplasia and, similar to dystrophic calcification, frequently occurred close to tumor necrosis or squamous metaplasia. Groisman et al.[13] first described bone formation within a benign tubulovillous adenoma without signs of necrosis or mucinous accumulation.[6]

In 1989, Randall et al. suggested alkaline phosphatase expression, a marker of osteoblasts from proliferating mesenchymal cells. Recently, most of the studies have suggested expression of bone morphogenic proteins (BMPs) in the setting of osseous metaplasia.[5] BMP2 and BMP4 were noted in the stromal fibroblasts. Kypson suggested BMP2 expression in rectal adenocarcinoma with osseous metaplasia.[14]

In in vitro studies, under the influence of four specific transcription factors (Oct3/4, Sox2, c-Myc, and Klf4), they have been found to generate pluripotent stem cells. Subsequently, these stem cells have demonstrated the capability to differentiate into different cell types.[15],[16] Hence, mature fibroblasts which present within the stromal component are under the influence of these similar transcription factors, leading to differentiation of stem cells to osteoblasts.[5]

Other factors of osteogenesis may include growth factors such as transforming growth factor β1 and β2 or other paracrine factors;[17] however, this warrants further investigation.

An osseous metaplasia is clinically and prognostically insignificant, and most of the times, it is an incidental finding.[6]


  Conclusion Top


Although detecting bone tissue in intestinal polyp stroma is a rare event, it cannot exclude the diagnosis of guvenile polyp.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no confl icts of interest.



 
  References Top

1.
Garg M, Kaur J, Bindroo S, Sharma A, Mahajan NC. Metaplastic ossification in a juvenile rectal polyp: A rare histological finding. J Clin Diagn Res 2013;7:908-10.  Back to cited text no. 1
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2.
Haque S, Eisen RN, West AB. Heterotopic bone formation in the gastrointestinal tract. Arch Pathol Lab Med 1996;120:666-70.  Back to cited text no. 2
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3.
Nakajima H, Iwane S, Mikami T, Nara H, Yamagata K, Morita T, et al. Osseous metaplasia in benign rectal polyps. J Clin Gastroenterol 1997;25:558-9.  Back to cited text no. 3
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4.
Bhat V, Roopa A, Shariff S. Juvenile rectal polyp with osseous metaplasia – A rare case with review of literature. Int J Health Sci Res 2012;2:114-6.  Back to cited text no. 4
    
5.
Bhattacharya N, Sarkar T. Osseous metaplasia in a juvenile polyp: A rare case report in India. J Clin Diagn Res 2013;7:2004-5.  Back to cited text no. 5
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Hjelkrem M, Fair K, Pabby A. Osseous metaplasia in a colon polyp. Pract Gastroenterol 2012;36:58-62.  Back to cited text no. 6
    
7.
Dukes CE. Ossification in rectal cancer: (Section of surgery: Sub-section of proctology). Proc R Soc Med 1939;32:1489-94.  Back to cited text no. 7
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8.
Ohtsuki Y, Danbara Y, Takeda I, Takahashi K, Hayashi K, Sonobe H, et al. Metaplastic bone formation in a hyperplastic polyp of the stomach: A case report. Acta Med Okayama 1987;41:43-6.  Back to cited text no. 8
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9.
Oono Y, Fu KL, Nakamura H, Iriguchi Y, Oda J, Mizutani M, et al. Bone formation in a rectal inflammatory polyp. World J Gastrointest Endosc 2010;2:104-6.  Back to cited text no. 9
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10.
Narita T, Ohnuma H, Yokoyama S. Peutz-Jeghers syndrome with osseous metaplasia of the intestinal polyps. Pathol Int 1995;45:388-92.  Back to cited text no. 10
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11.
van Patter HT, Whittick JW. Heterotopic ossification in intestinal neoplasms. Am J Pathol 1955;31:73.  Back to cited text no. 11
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12.
Sanerkin NG. Squamous metaplasia and ossification in colonic adenocarcinoma. J Pathol Bacteriol 1968;95:547-9.  Back to cited text no. 12
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13.
Groisman GM, Benkov KJ, Adsay V, Dische MR. Osseous metaplasia in benign colorectal polyps. Arch Pathol Lab Med 1994;118:64-5.  Back to cited text no. 13
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14.
Kypson AP, Morphew E, Jones R, Gottfried MR, Seigler HF. Heterotopic ossification in rectal cancer: Rare finding with a novel proposed mechanism. J Surg Oncol 2003;82:132-6.  Back to cited text no. 14
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15.
Wernig M, Meissner A, Foreman R, Brambrink T, Ku M, Hochedlinger K, et al. In vitro reprogramming of fibroblasts into a pluripotent ES-cell-like state. Nature 2007;448:318-24.  Back to cited text no. 15
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16.
Takahashi K, Tanabe K, Ohnuki M, Narita M, Ichisaka T, Tomoda K, et al. Induction of pluripotent stem cells from adult human fibroblasts by defined factors. Cell 2007;131:861-72.  Back to cited text no. 16
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17.
Teot LA, O'Keefe RJ, Rosier RN, O'Connell JX, Fox EJ, Hicks DG. Extraosseous primary and recurrent giant cell tumors: Transforming growth factor-beta1 and -beta2 expression may explain metaplastic bone formation. Hum Pathol 1996;27:625-32.  Back to cited text no. 17
    


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