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      Multiple Hereditary Osteochondromatosis: A Case Report

      case-report

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          Abstract

          Objectives

          Common carious lesions owing to vomiting are not widespread in children. In this case, we aimed to report an 11-years-old male patient with common carious lesions due to repeated vomitings, chewing and eating difficulty and retarded growth with Multiple Hereditary Osteochondromatosis (MHO).

          Case Report

          An 11-years-old boy was referred to Department of Pediatric Dentistry in Faculty of Dentistry because of eating difficulty owing to common carious lesions. It was seen that the patient growth was generally retarded in extra-oral examination. Some exostoses were also present on the extremities. It was learned that he was previously diagnosed as MHO in Faculty of Medicine. Nausea and vomiting have been commonly occurring after taking of Didronat. Chewing and eating difficulty and inadequate nutrition were present because of bad oral hygiene, carious lesions and remained roots. Growth was negatively affected by malnutrition and MHO.

          Results

          Diet recommendations were given and oral hygiene behaviors were rearranged. Preventive, surgical, restorative and prosthetical dental applications were applied for dental treatments.

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          Narrative [corrected] review: bisphosphonates and osteonecrosis of the jaws.

          Sook-Bin Woo, John Hellstein, John R Kalmar (2006)
          Osteonecrosis of the jaws is a recently described adverse side effect of bisphosphonate therapy. Patients with multiple myeloma and metastatic carcinoma to the skeleton who are receiving intravenous, nitrogen-containing bisphosphonates are at greatest risk for osteonecrosis of the jaws; these patients represent 94% of published cases. The mandible is more commonly affected than the maxilla (2:1 ratio), and 60% of cases are preceded by a dental surgical procedure. Oversuppression of bone turnover is probably the primary mechanism for the development of this condition, although there may be contributing comorbid factors. All sites of potential jaw infection should be eliminated before bisphosphonate therapy is initiated in these patients to reduce the necessity of subsequent dentoalveolar surgery. Conservative débridement of necrotic bone, pain control, infection management, use of antimicrobial oral rinses, and withdrawal of bisphosphonates are preferable to aggressive surgical measures for treating this condition. The degree of risk for osteonecrosis in patients taking oral bisphosphonates, such as alendronate, for osteoporosis is uncertain and warrants careful monitoring.
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            Bisphosphonate-associated osteonecrosis of mandibular and maxillary bone: an emerging oral complication of supportive cancer therapy.

            Stephanie M. Schubert, Cesar Migliorati, Marcelo M Seneda … (2005)
            The current report presented 17 patients with cancer with bone metastases and 1 patient with osteopenia who received treatment with bisphosphonates and who subsequently developed osteonecrosis of the mandible and/or maxilla. The authors reviewed information on 18 patients who were referred to oral medicine or oral surgery specialists for evaluation and treatment of mandibular and/or maxillary bone necrosis from June 2002 to September 2004. To be included in the current review, patients must have been treated with either pamidronate or zoledronic acid to control or prevent metastatic disease, or with alendronate for osteoporosis. All patients with cancer had received chemotherapy while receiving bisphosphonate management. The 17 patients with cancer were receiving active medical care for a malignancy. Cancer treatment included a variety of chemotherapeutic agents. They presented with metastatic disease to bone and were treated intravenously with the bisphosphonates pamidronate or zoledronic acid for a mean time of 25 months (range, 4-41 mos). There were 14 females and 4 males with a mean age of 62 years (range, 37-74 yrs). Malignancies included breast carcinoma (n = 10), multiple myeloma (n = 3), prostate carcinoma (n = 1), ovarian carcinoma (n = 1), prostate carcinoma/lymphoma (n = 1), and breast/ovarian carcinoma (n = 1). One female patient with osteopenia received alendronate. The most common clinical osteonecrosis presentations included infection and necrotic bone in the mandible. Associated events included dental extractions, infection, and trauma. Two patients appeared to develop disease spontaneously, without any clinical or radiographic evidence of local pathology. Despite surgical intervention, antibiotic therapy, hyperbaric oxygen therapy, and topical use of chemotherapeutic mouth rinses, most of the lesions did not respond well to therapy. Discontinuation of bisphosphonate therapy did not assure healing. However, 1 patient with cancer healed after discontinuation of bisphosphonate therapy for 4 months. The findings in the patient population combined with recent literature reports suggested that bisphosphonates may contribute to the pathogenesis of the oral lesions. The risk factors and precise mechanism involved in the formation of the osteonecrosis are not known. This condition represents a new oral complication in patients with cancer and can be termed bisphosphonate-associated osteonecrosis. Lesions in patients with osteoporosis are worrisome and need to be further evaluated.
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              Multiple hereditary osteochondromata.

              Sarah Peterson (1989)
              Multiple hereditary osteochondromata is a disorder consisting of multiple projections of bone (exostoses) capped by cartilage. The lesions are most numerous in the metaphyses of long bones but may appear on diaphyses of long bones and on flat bones and vertebrae. The transmission is autosomal dominant. Sarcomatous transformation is uncommon and probably occurs in fewer than 1% of patients. The more common indications for surgical excision of lesions are pain, growth disturbance, compromised joint motion, cosmesis, and secondary impingement of tendon, nerve, or vessel. Excision of the lesions is effective in relieving pain, improving cosmesis and joint motion, and removing secondary impingement of tendon, nerve, or vessel, and may retard or prevent progressive disturbance of osseous growth. Wrist and ankle deformities are often associated with relative shortening and bowing of the ulna and fibula, respectively; tilt and tapering of the distal radial and tibial epiphyses; and distal radioulnar and tibio-fibular diastasis. These deformities can be effectively treated by ulnar and fibular lengthening combined with hemiphyseal stapling of the distal radius and tibia. Progressive genu valgum is well corrected by placement of staples over the medial side of the physis of the distal femur or proximal tibia or both.
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                Author and article information

                Journal
                Journal ID (nlm-ta): Eur J Dent
                Journal ID (publisher-id): Eur J Dent
                Title: European journal of dentistry
                Publisher: Dental Investigations Society
                ISSN (Print): 1305-7456
                ISSN (Electronic): 1305-7464
                Publication date (Print): July 2007
                Volume: 1
                Issue: 3
                Pages: 183-187
                Affiliations
                [a ] Assistant Professor, Department of Pediatric Dentistry, Faculty of Dentistry, Suleyman Demirel University, Isparta, Turkey
                [b ] Specialist Doctor, Ottoman Oral-Dental Health Department, Ankara, Turkey
                [c ] Associated Professor, Department of Pediatrics, Faculty of Medicine, Suleyman Demirel University, Isparta, Turkey
                Author notes
                Corresponding Author: Dr. Cigdem Kucukesmen Süleyman Demirel University, Department of Pediatric Dentistry, Faculty of Dentistry, 32260 Cunur, Isparta, Turkey. Phone : +90 537 415 11 10 Fax : +90 246 237 06 07 E-mail : kucukesmencigdem@ 123456gmail.com
                Article
                Publisher ID: dent1_p183
                PMC ID: 2638239
                PubMed ID: 19212564
                SO-VID: 047b54c1-8194-4925-88bb-68af9ea2bba1
                Copyright © Copyright 2007 European Journal of Dentistry. All rights reserved.
                History
                Categories
                Subject: Case Reports

                ScienceOpen disciplines: Dentistry
                Keywords: hereditary multiple osteochondromatosis,chondrosarcoma,multiple hereditary exostoses,bone tumor,bisphosphonates
                Data availability:
                ScienceOpen disciplines: Dentistry
                Keywords: hereditary multiple osteochondromatosis, chondrosarcoma, multiple hereditary exostoses, bone tumor, bisphosphonates

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