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 Table of Contents  
CASE REPORT
Year : 2021  |  Volume : 7  |  Issue : 2  |  Page : 139-142

Bilateral focal osteoporotic marrow defect associated with adenomatoid odontogenic tumor of mandible


1 Department of Oral and Maxillofacial Pathology, Dr B.R. Ambedkar Institute of Dental Sciences and Hospital, Patna, Bihar, India
2 Department of Conservative Dentistry and Endodontics, Patna Dental College and Hospital, Patna, Bihar, India
3 Department of Oral and Maxillofacial Pathology, Sree Balaji Dental College and Hospital, Chennai, Tamil Nadu, India
4 Department of Dental, Indira Gandhi Institute of Medical Sciences, Patna, Bihar, India
5 Department of Oral Pathology, Dr B.R. Ambedkar Institute of Dental Sciences and Hospital, Patna, Bihar, India

Date of Submission22-Sep-2020
Date of Acceptance05-Nov-2021
Date of Web Publication17-Aug-2021

Correspondence Address:
Shashi Ranjan
Department of Oral and Maxillofacial Pathology, Dr B.R. Ambedkar Institute of Dental Sciences and Hospital, Patna, Bihar
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jigims.jigims_2_20

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  Abstract 


A case previously treated by surgical enucleation for adenomatoid odontogenic tumor involving from right parasymphysis to left parasymphysis region of mandible in a young female patient 3 years back reported with pain over left posterior region of mandible in a private dental clinic. Clinical examination revealed no dental pathosis in involved area. Panoramic radiograph revealed irregular multilocular radiolucency with foci of radiopacity with sclerotic borders involving periapical region of canine to second molar teeth till lower border of left side mandible. Incidentally, similar radioluceny was seen over the right posterior side of mandible periapically. Previously enucleated midline lesion revealed normal healed area without any sign of recurrence. As the lesion seemed to be bilateral, radiographical differential diagnosis was difficult, and lesions included were florid cementosseous dysplasia, multiple odontogenic keratocyst of basal cell nevus syndrome, ameloblastoma, and metastatic tumor to jaw. Histopathologically, the left posterior lesion was diagnosed as focal osteoporotic marrow defect, and right side lesion was considered as same. We are reporting a case of bilateral focal osteoporotic marrow defect along with previously enucleated adenomatoid odontogenic tumor, and purpose of this case report to correlate a relationship between the two different lesions whether focal osteoporotic marrow defect arise in response to adenomatoid odontogenic tumor or is incidental finding.

Keywords: Adenomatoid odontogenic tumor, bone marrow, focal osteoporotic marrow defect, osteoporosis, osteoporotic bone marrow defect


How to cite this article:
Ranjan S, Issar R, Masthan K M, Babu N A, Singh P, Singh KT. Bilateral focal osteoporotic marrow defect associated with adenomatoid odontogenic tumor of mandible. J Indira Gandhi Inst Med Sci 2021;7:139-42

How to cite this URL:
Ranjan S, Issar R, Masthan K M, Babu N A, Singh P, Singh KT. Bilateral focal osteoporotic marrow defect associated with adenomatoid odontogenic tumor of mandible. J Indira Gandhi Inst Med Sci [serial online] 2021 [cited 2022 Nov 28];7:139-42. Available from: http://www.jigims.co.in/text.asp?2021/7/2/139/331742




  Introduction Top


Hematopoietic marrow may normally be found in the region of the maxillary tuberosity and in the mandibular posterior body (Standish and Shafer 1962).[1] However, these sites may be stimulated by inflammation, repair, and regeneration. Such stimulations produce a radiographically evident lesion. This may present as swelling with pain. Osteoporotic bone marrow defect (OBMD) predominantly occurs in the mandible, particularly in middle-aged women, is asymptomatic and requires no treatment. However, it is seldom considered in a clinical differential diagnosis. These large marrow spaces may appear to be superimposed over the roots of teeth on radiographs simulating the appearance of apical pathosis. The radiographic appearance varied from sharply defined radiolucencies with distinct sclerotic borders to extremely ill-defined areas with a moth-eaten appearance. Crawford et al. reported 17 cases of osteoporotic marrow defects of the jaws and described these as being “poorly defined,” “large radiolucent area,” radiolucent area with abnormal bony configuration,” and “honeycombed radiolucencies” [Table 1].[2]
Table 1: Column reveals different authors' case reports and clinical pictures

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  Case Report Top


A 22-year-old woman reported to a private clinic with pain over the left posterior tooth region. The pain was insidious, dull continuous, and localized over the left mandibular premolar-molar area. Clinical examination did not reveal any tooth pathosis.

Past dental history revealed that the patient was treated for adenomatoid odontogenic tumor involving anterior mandible crossing midline by enucleation about 3 years back in the same clinic.

Coming to the present complaint, a thorough examination of the patient revealed pain over left posterior body of mandible without any clinical enlargement, and the teeth were vital in the involved area. Panoramic radiograph revealed irregular multilocular radiolucency. This radiolucency had foci of radioopacity. The lesion had sclerotic borders involving periapical region. This extended from canine to second molar teeth. Downward it extended up to the lower border of left side mandible. Incidentally, similar radioluceny was seen over the right posterior side of mandible periapically. Previously enucleated midline lesion revealed normal healed area without any sign of recurrence. The right side radiolucent lesion was asymptomatic. The initial differential diagnosis included odontogenic keratocyst, ameloblastoma, and early stage of fibro osseous lesion and metastatic tumor to bone. The last two diagnoses were considered since the inferior margin of the lesion showed sclerotic border that blended into the surrounding bone. Incisional biopsy of the left side lesion histopathologially revealed fibro-fatty marrow tissue with foci of osteoporotic lamellar bone scattered throughout. There was marrow hyperplasia showing myeloid and erythroid components. In addition, megakaryocytes supported in mature fatty tissue were also evident. There was no fatty tissue hyperplasia. Osteoporotic lamellar bone revealed smooth margin with sparse marrow defect osteoblastic rimming and widely dispersed osteocytes. Histopathologial diagnosis of focal osteoporotic was rendered [Figure 1]a, [Figure 1]b, [Figure 1]c, [Figure 1]d.
Figure 1: (a) H and E section shows osteoporotic bone along with fibrofatty marrow tissue (×4 view). (b) H and E section shows osteoporotic bone (×10 view). (c) H and E section shows fibrofatty marrow tissue composed of erythroid and myeloid cell line. Few megakaryocytes seen (arrow) (×10 view). (d) H and E section shows fibrofatty marrow tissue composed of erythroid and myeloid cell line (white arrow). Few megakaryocytes seen (black arrow) (×40 view)

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We thoroughly scrutinized the panoramic radiograph and compared it with the initial radiograph taken before surgical enucleation of adenomatoid odontogenic tumor 3 years back.

Surprisingly, in the earlier radiograph also there was the faint suggestion of the bilateral radiolucent lesion along with central midline radiolucent lesion of mandible before surgical enucleation of midline adenomatoid odontogenic tumor [Figure 2]a. While investigating the recent radiograph, we found normal healed midline lesion. However, when we compared the size and extent of involvement of left radiolucent lesion, it was evident that the lesion was expanding. Earlier radiograph revealed small irregular multilocular radiolucent lesion infiltrating between roots of canine and first premolar tooth of the left side of mandible. Recent radiograph revealed diffuse extension of multilocular radiolucent lesion involving up to the interradicular area of previously uninvolved second premolar and first molar [Figure 2]b.
Figure 2: (a) Previous panormic radiograph, three years back before enucleation of midline lesion of mandible diagnosed as adenomatoid odontogenic tumor (red thin arrow), irregular radiolucent lesion adjacent to midline lesion bilaterally (yellow thick and disintegrated line), left side radiolucent lesion infiltrating between roots of canine and premolar teeth involving lower apex of tooth roots. (b) Recent panormic radiograph demonstrating healed enucleated midline lesion of mandible which was diagnosed as adenomatoid odontogenic tumor (red thin arrow), irregular radiolucent lesion adjacent to midline lesion bilaterally (yellow thick and disintegrated line), left side radiolucent lesion infiltrating between roots of canine, premolars and first molar teeth till mid root level. Recent lesion is clearly showing expansion on comparison with previous radiograph

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Hence, there was definite increase in the size of radiolucency within these 3 years.

[Figure 2]a Previous panormic radiograph, 3 years back before enucleation of midline lesion of mandible diagnosed as adenomatoid odontogenic tumor (red thin arrow), irregular radiolucent lesion adjacent to midline lesion bilaterally (yellow thick and disintegrated line), left side radiolucent lesion infiltrating between roots of canine and premolar teeth involving lower apex of tooth roots.

[Figure 2]b Recent panormic radiograph demonstrating healed enucleated midline lesion of mandible which was diagnosed as adenomatoid odontogenic tumor (red thin arrow), irregular radiolucent lesion adjacent to midline lesion bilaterally (yellow thick and disintegrated line), left side radiolucent lesion infiltrating between roots of canine, and premolars and first molar teeth till mid root level. Recent lesion is clearly showing expansion on comparison with previous radiograph.


  Discussion Top


Active hematopoietic marrow is gradually replaced with fatty marrow as people grow older, but fatty marrow may be quickly replaced with areas of active hematopoiesis as a demand for blood cell formation increases. Occasionally, foci of hematopoietic or fibrofatty marrow will form a localized collection to produce a paucity of trabeculae and to present a radiolucency in the jaws.[2] Sites most frequently mentioned in the jaws are the condylar process, the mandiblular angle, and the maxillary tuberosity.[3]

The pathogenesis is not known, but several suggested possibilities include

  1. Bone resorption secondary to marrow hyperplasia in response to an increased demand for blood cells
  2. Persistent embryologic marrow remnants
  3. Altered regeneration of bony trabeculae in an area of previous trauma, local inflammation, or extraction.[4]


However, the majority of reported lesions in literature are asymptomatic, but Lipani et al.[5] emphasized the OBMD of the jaw bone might behave somewhat aggressively and produce a painful expansion of the cortical margin. The frequent incidence of such lesions in the edentulous segments of the posterior mandible suggests that the proliferation of hematopoietic marrow elements may be stimulated by odontogenic inflammation or be a sequel of repair.[5] Our case might have arisen due to the stimulus of adenomatoid odontogenic tumor of anterior mandible. This might have caused marrow hyperplasia bilaterally to the midline lesion. Another possibility is that it might have aggravated in response to the healing process of enucleation done for the adenomatoid odontogenic tumor.

Makek et al. also reported that five of their twenty patients presented with pain of no dental cause, and one of them had gradual but progressive enlargement of the right mandible, resulting in a lateral open bite deformity. In addition, interestingly, enough in this case, the lesion was bilateral, with no symptoms attributable to the left-sided lesion.[6],[7] Our case was also symptomatic for the left side lesion of mandible and was totally asymptomatic on the right side of mandible. Since bilateral similar lesions have been associated with sickle cell anemia, the patient should be evaluated for same.[3]

The vague or misleading clinical history of pain and swelling as well as the varied radiographic appearance of hematopoietic marrow defect frequently requires consideration in the differential diagnosis of many pathologic entities.[8],[9]

Pain and swelling suggest osteomyelitis, primary neoplasms of bone, metastatic lesions, and occasionally odontogenic cysts and tumors. When teeth are present, X-rays of hematopoietic defects of younger patients may be confused with central giant cell lesion, eosinophilic granuloma, leukemic infiltrate, and certain blood dyscrasias. Most essential for differential diagnosis of osteoporotic defects is a thorough and comprehensive medical history. It is particularly important in the middle-aged female as metastatic lesions of breast, lung, thyroid, and parathyroid are not uncommon. In posthysterectomy patients, there is frequently an osteoporotic appearance of the jaws, and this also must be considered.

Based on clinical and radiographic symptoms, diagnosis of a hematopoietic marrow defect is difficult. In middle-aged females, however, painful lesions in the posterior mandible combined with an ill-defined radiolucency should suggest such a possibility and should be considered in differential diagnosis.

Declaration of patient consent

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

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Standish S, Shafer W. Focal osteoporotic bone marrow defects of the jaws. J Oral Surg 1962;20:123-8.  Back to cited text no. 1
    
2.
Crawford BE, Weathers DR. Osteoporotic marrow defects of the jaws. J Oral Surg 1970;28:600-3.  Back to cited text no. 2
    
3.
Sanner J, Ramin J. Osteoporotic, hematopoietic marrow defect: An osseous manifestation of sickle-cell anemia. J Oral Surg 1977;35:986-8.  Back to cited text no. 3
    
4.
Barker BF, Jensen JL, Howell FV. Focal osteoporotic bone marrow defects of the jaws. An analysis of 197 new cases. Oral Surg Oral Med Oral Pathol 1974;38:404-13.  Back to cited text no. 4
    
5.
Lipani CS, Natiella JR, Greene GW Jr. The hematopoietic defect of the jaws: A report of sixteen cases. J Oral Pathol 1982;11:411-6.  Back to cited text no. 5
    
6.
Correll RW, Wescott WB. Asymptomatic, ill-defined radiolucent area in the posterior body of the mandible. J Am Dent Assoc 1983;107:460-1.  Back to cited text no. 6
    
7.
Makek M, Lello GE. Focal osteoporotic bone marrow defects of the jaws. J Oral Maxillofac Surg 1986;44:268-73.  Back to cited text no. 7
    
8.
Schneider LC, Mesa ML, Fraenkel D. Osteoporotic bone marrow defect: Radiographic features and pathogenic factors. Oral Surg Oral Med Oral Pathol 1988;65:127-9.  Back to cited text no. 8
    
9.
Sa'do B, Ozeki S, Higuchi Y, Nakayama E. Osteoporotic bone marrow defect of the mandible: Report of a case diagnosed by computed tomography scanning. J Oral Maxillofac Surg 1992;50:80-2.  Back to cited text no. 9
    


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