eMedicine - Adamantinoma : Article by Christopher D = Smelser, DO

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Adamantinoma

Last Updated: = July 25, 2002=20

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Synonyms and related = keywords:=20 long bone tumors, classic adamantinomas, differentiated=20 adamantinomas, adamantinomous tumors, primary=20 adamantinomas

AUTHOR INFORMATION=20 Section 1 of = 10   =20 =

Author=20 Information = Introduction=20 Differentials=20 X-ray=20 Cat = Scan MRI=20 Nuclear=20 Medicine = Intervention=20 Pict= ures=20 = Bibliography

Author: Christopher=20 D Smelser, DO, Staff Physician, Department of=20 Radiology, William Beaumont Army Medical Center
Coauthor(s): Robert=20 D Stoffey, DO, Director of Women's Imaging, = Chief of=20 Mammography Section, Department of Radiology, William = Beaumont Army=20 Medical Center; B Wade Mahaney, MD, Staff=20 Physician, Department of Radiology, William Beaumont Army = Medical=20 Center; Sean C Keenan, MD, Staff Physician, = Department of Radiology, William Beaumont Army Medical = Center;=20 Gary E Simmons, MD, Chief, Department of = Radiology,=20 William Beaumont Army Medical Center

Christopher D Smelser, DO, is a member of the following = medical=20 societies: American = Osteopathic=20 Association

Editor(s): Michael A Bruno, MD, Chair,=20 Department of Radiology, Maricopa Medical Center; = Bernard D=20 Coombs, MBChB, PhD, Assistant Professor, Department = of=20 Radiology, University of Colorado Health Sciences Center;=20 Murali Sundaram, MBBS, FRCR, Department of=20 Radiology, Mayo Clinic of Rochester; Robert M = Krasny,=20 MD, Visiting Assistant Professor of Radiology, = University=20 of California at Los Angeles Medical Center; Consulting = Staff, Tower=20 Imaging, Los Angeles, California; and Donald = Resnick,=20 MD, Chief of Osteoradiology Section, Veterans = Affairs=20 Medical Center; Professor, Department of Radiology, Division = of=20 Musculoskeletal Imaging, University of California at San = Diego=20

Background:=20 Adamantinoma is a rare tumor, and its origin remains=20 controversial. Fischer first described the tumor in 1913, and = since then,=20 only approximately 200 cases have been reported. The tumor occurs = almost=20 exclusively in the long bones; tumors in the tibia account for = more than=20 80% of cases. The diaphyseal region is the area most commonly = affected.=20
Adamantinomas are classified into 2 distinct types: classic and = differentiated. Classic adamantinomas usually occur in patients = older than=20 20 years, whereas differentiated adamantinomas occur almost = exclusively in=20 patients younger than 20 years. In addition, the 2 classifications = of=20 adamantinomas have distinct radiographic and histologic = differences.=20
Patients with adamantinomas present with variable signs and = symptoms;=20 most commonly, they report pain and swelling. The tumor is slow = growing,=20 and patients may describe discomfort lasting months to years. = Although=20 ascertaining accurate mortality statistics is difficult because of = the=20 extremely rare nature of this tumor, the 10-year survival rate is = believed=20 to be 10%. Treatment options for adamantinoma are surgical and = include=20 either marginal or en bloc resection. Unfortunately, neither = radiation=20 therapy nor chemotherapy has been proven effective in the = treatment of=20 this insidious tumor.

Pathophysiology: Although adamantinomas can = cause a=20 variety of pathologic and histologic abnormalities, the tumors are = usually=20 lobular and well defined. Generally, adamantinomas remain = intracortical;=20 however, when prior surgery has been performed or when the tumor = is of the=20 classic type, the lesions may extend beyond the periosteum.=20
The histologic features of adamantinomas have a multitude of = variations=20 that are present not only among patients but also among different = areas of=20 the same tumor. The histologic features of differentiated = adamantinomas=20 are predominantly characterized by a pattern of osteofibrous = dysplasia.=20 Conversely, in classic adamantinomas, the cells form a tubular, = basaloid,=20 squamoid, or spindled pattern. As a result of the variety of=20 manifestations of the tumor, multiple biopsies must often be = performed to=20 obtain a representative sample for study.=20
Adamantinomas typically range from 3-15 cm in size, and = metastases=20 occur in approximately 15-20% of patients. Metastases generally = appear in=20 the lungs and local lymph nodes. Adamantinomas can be found in = many other=20 bones in addition to their usual location in the tibia. The = humerus, ulna,=20 femur, fibula, and radius are other possible locations in the long = bones.=20 Occasionally, the tumor occurs in the spine. Adamantinomas are = locally=20 aggressive and extremely slow growing.

Frequency:

In the US: Because only approximately 200=20 well-documented cases of adamantinoma have been identified in = the almost=20 100 years since the tumor has been classified, it can be = described only=20 as extremely rare.

Internationally: Adamantinomas are = extremely rare.=20
Mortality/Morbidity: Despite their = slow-growing=20 nature, adamantinomas can cause significant morbidity and = mortality. The=20 10-year survival rate is estimated to be approximately 10%. In = addition,=20 painful bowing deformity of the tibia is not an infrequent = occurrence in=20 this condition.=20
Race: To the authors' knowledge, no racial = comparative=20 studies have been performed because of the infrequent occurrence = of the=20 tumor and because of the difficult and technologically = sophisticated=20 diagnosis. Knowing how many cases have been missed in the less = developed=20 regions of the world is impossible, just as knowing how many cases = remain=20 undiagnosed in industrialized Western nations is difficult.=20
Sex: Adamantinomas have a male predominance, = with an=20 approximate male-to-female ratio of 1.25:1.=20
Age: The more common classic adamantinoma = usually=20 occurs in individuals older than 20 years, whereas the = differentiated form=20 of the tumor almost exclusively occurs in those younger than 20 = years.=20 However, the tumors have occurred in individuals aged 10-70 years. =
Anatomy: The tibia is by far the most commonly = affected bone; tibial tumors account for 80-85% of primary = adamantinomas.=20 Other long bones are also commonly affected; occasionally, primary = involvement of the spine occurs.
Clinical Details: Patients present with = complaints=20 such as a dull ache or bone pain that has been present for months = to=20 years. Another common complaint is localized swelling. Often, a = history of=20 trauma may be elicited. A palpable mass may be firm or soft; its = diameter=20 is usually in the range of 3-15 cm.
Preferred Examination: Plain radiography, CT, = and MRI=20 may all be used to help assess suspected adamantinomous tumors. = However,=20 CT and MRI are not specific in the differentiation of this tumor = from=20 other conditions; findings often overlapping with those of other = tumors=20 and tumor-like lesions. A variety of tumors and tumor-like lesions = can=20 mimic an adamantinoma. Histologic examination is key to the = identification=20 of an adamantinoma; the histologic features of these tumors have = many=20 variations.
Limitations of Techniques: Limitations of = plain-film=20 radiography include the relatively long list of differential = diagnoses for=20 adamantinoma. Many pathologic conditions that are as rare as or = more=20 common than adamantinoma demonstrate similar characteristics on = plain=20 radiographs, as well as CT scans and MRIs. This fact, coupled with = the=20 limited experience that most radiologists (and physicians in = general) have=20 in dealing with this tumor, makes the diagnosis and treatment of=20 adamantinomas challenging.

INTRODUCTION = Section 2 of = 10

Author=20 Information = Introduction=20 Differentials=20 X-ray=20 Cat = Scan MRI=20 Nuclear=20 Medicine = Intervention=20 Pict= ures=20 = Bibliography

Aneurysmal = Bone Cyst=20
Chondromyxoid = Fibroma=20
Chondrosarcoma=20
Fibrous=20 Dysplasia
Hemangioendothelioma= =20

Other Problems to be Considered:
Osteofibrous dysplasia
Nonossifying or ossifying=20 fibroma
Eosinophilic granuloma
Simple bone cyst

DIFFERENTIALS = Section 3 of = 10   =

Author=20 Information = Introduction=20 Differentials=20 X-ray=20 Cat = Scan MRI=20 Nuclear=20 Medicine = Intervention=20 Pict= ures=20 = Bibliography

Findings: In its early stages, an adamantinoma = appears=20 as an elongated linear lucency on plain radiographs, and no = periosteal=20 reaction is noted in the surrounding bone. In later stages, = cortical=20 sclerosis becomes apparent on plain radiographs.=20
The most common location is in the diaphyseal region of long = bones,=20 especially the tibia. A periosteal reaction and fracture are less = common=20 late-term sequelae of adamantinomas that can also be depicted on = plain=20 radiographs.=20
The differentiation of adamantinoma from fibrous dysplasia and=20 osteofibrous dysplasia may be difficult by using plain radiographs = alone.=20 When questions arise in the diagnosis of the tumor, the histologic = and=20 clinical features must be included to narrow the differential = diagnosis.
Degree of Confidence: Because of the extremely = rare=20 nature of this tumor, the degree of confidence has not been = determined.
False Positives/Negatives: False-positive = findings=20 include fibrous dysplasia, osteofibrous dysplasia, fibroma = (nonossifying=20 or ossifying), bone cyst (aneurysmal or simple), chondrosarcoma,=20 chondromyxoid fibroma, eosinophilic granuloma, and = hemangioendothelioma.=20
Because of the extremely rare nature of this tumor, a list of=20 false-negative findings has not been adequately formulated.

X-RAY = Section 4 of = 10

Author=20 Information = Introduction=20 Differentials=20 X-ray=20 Cat = Scan MRI=20 Nuclear=20 Medicine = Intervention=20 Pict= ures=20 = Bibliography

Quick Find

Author=20 Information
= Introduction
Differentials
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= Intervention
Pict= ures
= Bibliography

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Aneurysmal=20 Bone Cyst

Chondromyxoid=20 Fibroma

Chondrosarcoma=20

Fibrous=20 Dysplasia

Hemangioendothelioma= =20

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Findings: CT scans often are used to study=20 adamantinomas, but the findings are not specific. CT scans of the = lower=20 extremity often reveal a hypoattenuating sclerotic region in the = tibial=20 diaphysis.
Degree of Confidence: Degrees of confidence = have not=20 been accurately determined because of the rarity of this tumor.
False Positives/Negatives: False-positive = findings=20 include fibrous dysplasia, osteofibrous dysplasia, fibroma, bone = cyst, and=20 chondromyxoid fibroma.

CAT SCAN = Section 5 of = 10

Author=20 Information = Introduction=20 Differentials=20 X-ray=20 Cat = Scan MRI=20 Nuclear=20 Medicine = Intervention=20 Pict= ures=20 = Bibliography

Findings: MRI often is used to image = adamantinomas,=20 but the findings are nonspecific. When MRI is used to study = adamantinomas,=20 the tumors demonstrate low signal intensity on T1-weighted = spin-echo=20 images and high signal intensity on T2-weighted images. Because = these=20 appearances are also typical of most tumors, these findings are=20 nonspecific.
Degree of Confidence: Because of the rarity of = this=20 tumor, the degree of confidence cannot be accurately determined at = this=20 time.
False Positives/Negatives: False-positive = findings=20 include fibrous dysplasia, osteofibrous dysplasia, fibroma, bone = cyst, and=20 chondrosarcoma.

MRI Section 6 of = 10

Author=20 Information = Introduction=20 Differentials=20 X-ray=20 Cat = Scan MRI=20 Nuclear=20 Medicine = Intervention=20 Pict= ures=20 = Bibliography

Findings: Use of nuclear medicine to study=20 adamantinomas is a relatively new undertaking; therefore, few data = regarding the tumors have been collected. However, the following = findings=20 are believed to correspond to adamantinomous lesions: increased = blood flow=20 in the region of the tumor, increased blood pooling, and increased = accumulation of technetium-99m methylene diphosphate in the area = of the=20 tumor.

NUCLEAR MEDICINE = Section 7 of = 10    =

Author=20 Information = Introduction=20 Differentials=20 X-ray=20 Cat = Scan MRI=20 Nuclear=20 Medicine = Intervention=20 Pict= ures=20 = Bibliography

Intervention: The only intervention that has = been=20 shown to be effective in these tumors is a surgical approach. = Neither=20 radiation therapy nor chemotherapy has been proven effective.=20
Treatment options for adamantinoma are surgical and include = either=20 marginal or en bloc resection. Marginal resection of the tumor has = been=20 shown to be effective in less than one half of the cases studied; = however,=20 en bloc resection generally prevents local recurrence of the = tumor.

INTERVENTION = Section 8 of = 10

Author=20 Information = Introduction=20 Differentials=20 X-ray=20 Cat = Scan MRI=20 Nuclear=20 Medicine = Intervention=20 Pict= ures=20 = Bibliography

PICTURES = Section 9 of = 10   = =

Author=20 Information = Introduction=20 Differentials=20 X-ray=20 Cat = Scan MRI=20 Nuclear=20 Medicine = Intervention=20 Pict= ures=20 = Bibliography

Caption: = Picture 1.=20 Plain radiograph in a patient with adamantinoma reveals bony = sclerosis, which is typical of a well-established = adamantinoma.=20 Courtesy of UCSD Medical Library.

3D"Click View Full=20 Size Image

eMedicine=20 Zoom View (Interactive!)

Picture = Type:=20 X-RAY

Caption: = Picture 2. CT=20 scan in a patient with adamantinoma reveals an expansile = lesion and=20 superficial erosion on the surface. Courtesy of UCSD Medical = Library.

3D"Click View Full=20 Size Image

eMedicine=20 Zoom View (Interactive!)

Picture = Type:=20 CT

Choi J: Adamantinoma (Angioblastoma). May 3, 1996. Available = at:=20 = http://www.vh.org/Providers/TeachingFiles/RCW/050396/Adamatinoma.html .=20 Accessed January 22, 2002[Full Text].=20
Dorfman HD, Czerniak: Bone Tumors. Mosby-Year Book; 1998: = 949-73.=20
Resnick D: Diagnosis of Bone and Joint Disorders. Vol 6.=20 Philadelphia, PA: W. B. Saunders Co; 1995: 3882-4.=20
Taveras JM: Radiology, Diagnosis, Imaging Intervention. Vol = 5.=20 Philadelphia, PA: Lippincott Williams & Wilkins; 1993.=20
Usui K, Yamamoto W, Shuke N, et al: Scintigraphic findings = of tibial=20 adamantinoma on a three-phase bone scan. Clin Nucl Med 2000 Dec; = 25(12):=20 1057-8[Medline].

BIBLIOGRAPHY = Section 10 of=20 10

Author=20 Information = Introduction=20 Differentials=20 X-ray=20 Cat = Scan MRI=20 Nuclear=20 Medicine = Intervention=20 Pict= ures=20 = Bibliography

NOTE: =
Medicine is a = constantly=20 changing science and not all therapies are clearly = established. New=20 research changes drug and treatment therapies daily. The = authors,=20 editors, and publisher of this journal have used their best = efforts=20 to provide information that is up-to-date and accurate and = is=20 generally accepted within medical standards at the time of=20 publication. However, as medical science is constantly = changing and=20 human error is always possible, the authors, editors, = and=20 publisher or any other party involved with the publication = of this=20 article do not warrant the information in this article is = accurate=20 or complete, nor are they responsible for omissions or = errors in the=20 article or for the results of using this information. The = reader=20 should confirm the information in this article from other = sources=20 prior to use. In particular, all drug doses, indications, = and=20 contraindications should be confirmed in the package = insert. FULL = DISCLAIMER=20

Adamantin= oma=20 excerpt
=A9 Copyright 2003, eMedicine.com, = Inc.

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RADIOLOGY=0A= - Medical Reference

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Table of = Contents
Alphabetical Index of = Topics

Brain/spine
Breast
Cardiac
Chest
Gastrointestinal
Genitourinary
Head And Neck

Multisystem
Musculoskeletal
Obstetrics/gynecology
Pediatrics
Technical/physics
Vascular/interventional

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Chief=0A= Editors
	Felix S = Chew, MD, EdM Kyung J Cho, MD Lawrence M Davis, MD Ziv J = Haskal, MD John Karani, MBBS, FRCR Eugene C Lin, MD Donald = Resnick, MD James G Smirniotopoulos, MD Charles S White, MD =

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Copy=0A= Editors
	Lisa = Davis, MA Deborah M Eng Beth Lehr Kelly Nikkila Tammy = Pomernackas Jennifer Welsh

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CME=0A= Editors
	Robert M = Krasny, MD

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Pharmacy=0A= Editors
	Bernard D = Coombs, MBChB, PhD Walter Huda, PhD

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Medical=0A= Editors

Hussein M = Abdel-Dayem, MD
Zahir Amin, MBBS, MRCP, MD, FRCR
Judith K Amorosa, = MD, FACR
Lori Lee Barr, MD
Mario Bezzi, MD
Orest Boyko, MD, = PhD
Jeffrey C Brandon, MD
James C Brown, MD
Michael A Bruno, = MD
Jocelyn D Chertoff, MD
Harris L Cohen, MD, FACR
Peter S = Conti, MD, PhD
Jeffrey L Creasy, MD
Pamela J DiPiro, = MD
Amilcare Gentili, MD
Charles M Glasier, MD
John L Go, = MD
S Bruce Greenberg, MD
Giuseppe Guglielmi, MD
John L Haddad, = MD
Jacqueline C Hodge, MD
Fredric A Hoffer, MD
J Randy Jinkins, = MD
Robert A Koenigsberg, DO, MSc, FAOCR
D Bradley Koslin, = MD
Glenn Krinsky, MD
Neela Lamki, MD
Henrique M Lederman, MD, = PhD
Leon Lenchik, MD
David S Levey, MD, PhD
Lucien M Levy, MD, = PhD
John M Lewin, MD
Jeffrey A Miller, MD
Hrudaya Nath, MBBS, = MD
Mahesh R Patel, MD
Justin D Pearlman, MD, ME, PhD, = FACC
Steven Perlmutter, MD, FACR
Hugh J Robertson, MD, = FACR
Pamela W Schaefer, MD
Kitt Shaffer, MD, PhD
Lee=0A= Sider, MD, MBA
Satinder P Singh, MD
Gary P Siskin, = MD
Christopher L Sistrom, MD
Robert J Starshak, MD
Moni Stein, = MD
Ludwig G Strauss, MD
Richard Tello, MD
Anthony Watkinson, = MD
Eric P Weinberg, MD
Beverly P Wood, MD, MS
Chi-Shing Zee, = MD

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Managing=0A= Editors
	Edward = Azavedo, MD Joshua A Becker, MD Javier Beltran, MD Lawrence M = Boxt, MD, FACC William G Bradley, Jr, MD, PhD, FACR Kyung J Cho, = MD Douglas M Coldwell, MD, PhD Abraham H Dachman, MD Robert L = DeLaPaz, MD Pamela J DiPiro, MD Bruno D Fornage, MD Arnold C = Friedman, MD, FACR Spencer B Gay, MD John H Harris, Jr, = MD George Hartnell, MD Marta Hernanz-Schulman, MD Theodore E = Keats, MD Marc S Keller, MD Kieran McHugh, MBBCh John D Newell, = Jr, MD, FACR, FCCP, FASER David Andrew Nicholson, BM, BS, = FRCR Wilfred CG Peh, MBBS, MD, FRCPE, FRCPG, FRCR C Douglas = Phillips, MD Philip W Ralls, MD Michael R Rees, BSc, MBChB, FRCP, = DMRD, FRCR William R Reinus, MD, MBA, FACR Karen L Reuter, MD, = FACR Matthew D Rifkin, MD Val Runge, MD Georges M Salamon, = MD Udo P Schmiedl, MD, PhD Paul M Silverman, MD Lynne S = Steinbach, MD Eric J Stern, MD David A Stringer, MBBS, = FRCR Murali Sundaram, MBBS, FRCR W Richard Webb, MD
Article = Excerpts

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