(CANCER RESEARCH 52, 6224-6228. November 15. 1992]
Clonal Rearrangement of Chromosome Band 6p21 in the Mesenchymal Component
of Pulmonary Chondroid Hamartoma1
Jonathan A. Fletcher,2 Geraldine S. Pinkus, Kevin Donovan, Rizwan Naeem, David J. Sugarbaker,
Steven Mentzer, Jack L. Pinkus, and Janina Longtine
Department of Pathology [J. A. F., G. S. P.. K. D., R. N., J. L. P., J. L.] and Division of Thoracic Surgery ¡D.J. S., S. M. J, Brigham and Women's Hospital; the
Division of Pediatrie-Oncologi; Dana-Farber Cancer Institute [J. A. F. J; the Division of Hematology-Oncology, Children's Hospital fj. A. F. J; and the Departments
of Pathology, Surgery, and Pediatrics, Harvard Medical School, Boston, Massachusetts 02115
ABSTRACT
Pulmonary chondroid hamartomas (PCH) are biphasic benign tu
mors that contain both mesenchymal and epithelial populations. In this
report we describe two PCH in which clonal translocations at chromo
some band 6p21 were demonstrated in mesenchymal cells. One of these
had a unique translocation, t(6;14Xp21;q24), that was also found in one
of two PCH karyotyped previously. The t(6;14) has not been described
in other varieties of benign or malignant neoplasia. The 6p21 aberra
tions are of particular interest because break points in this chromosomal
region appear to be characteristic of endometrial polyps. Endometrial
polyps, like PCH, are biphasic benign tumors in which mesenchymal
clonality has been demonstrated.
INTRODUCTION
PCH3 are unusual benign tumors that occur in both children
and adults. PCH are generally asymptomatic nodules that arise
in the periphery of the lungs, and most cases are discovered
incidentally during routine radiographie studies or at postmor
tem examination (1,2). Most PCH are composed of epithelial-
lined clefts that are situated within a heterogeneous prolifera
tion of mesenchymal cells. The mesenchymal population in a
given PCH might include fibrous connective tissue, fat, and
mature and/or immature chondroid tissue. Although PCH were
thought originally to represent hyperplastic developmental
remnants, it has been suggested that the epithelial and mesen
chymal components, or the mesenchymal component alone,
might be neoplastic (1-4). Malignant transformation in PCH is
exceptionally rare, however, and cure is accomplished invari
ably by surgical resection (5).
We have described clonal chromosome rearrangements in the
mesenchymal components of two PCH (6). One of those tu
mors contained a balanced translocation (6;14)(p21;q24) and a
chromosome 11 long arm deletion, whereas the other contained
clonal rearrangements of chromosomes 12 and 18. Herein, we
report two additional cases of PCH in which clonal rearrange
ment at chromosome band 6p21 was demonstrated in mesen
chymal cells.
MATERIALS AND METHODS
Case Reports
Case 1. A 37-year-old woman underwent wedge resection of an as
ymptomatic right lower lobe PCH that was 0.8 cm in its greatest di
ameter. 11istologi-ali). the PCH was composed of varied mesenchymal
Received 4/29/92; accepted 9/10/92.
The costs of publication of this article were defrayed in part by the payment of
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ance with 18 U.S.C. Section 1734 solely to indicate this fact.
1This study was supported in part by a Physician-Scientist Award
<lKHCAOI498-02)to J. A. F. from the NIH.
2 To whom requests for reprints should be addressed, at Department of Pathol
ogy. Brigham and Women's Hospital, 75 Francis Street, Boston, MA 02115.
3The abbreviations used are: PCH, pulmonary chondroid hamartomas;
APAAP, alkaline phosphatase anti-alkaline phosphatase.
cell types along with lumina lined by benign columnar or cuboidal
epithelium (Fig. 1). The mesenchymal components included immature
cells within a myxoid stroma (65% of the total mesenchyme), mature
adipose tissue (20% of the total mesenchyme), and mature cartilage
( 15% of the total mesenchyme).
Case 2. A 68-year-old man underwent wedge resection of an asymp
tomatic left lower lobe PCH that was 2. 5 cm in greatest diameter.
Histologically, the PCH was composed predominantly of mesenchymal
cell populations that included immature cells in a loose, myxoid stroma
(80%), scattered foci of adipose tissue (15%), and a minor component
of mature cartilage (5%). The PCH also contained scattered lumina,
lined by cuboidal or columnar epithelium, that in some cases were
associated with chronic inflammatory cells. The epithelial cells were
noted primarily at the periphery of the hamartoma.
Cytogenetic and in Situ Hybridization Analyses
Sterile specimens of each PCH were obtained directly from the fro
zen section room and were disaggregated, cultured, and karyotyped
using methods described previously (6). Metaphase cells were harvested
after 5-6 days in culture. Chromosome abnormalities were described
using the conventions proposed in the International System for Human
Cytogenetic Nomenclature (1991) (7). Fluorescent in situ hybridization
studies were carried out with a spectrum orange-labeled probe cocktail
to chromosome 6 (Imagenetics, Naperville, IL) and a biotinylated probe
cocktail to chromosome 14 (Oncor, Gaithersburg, MD), according to
the manufacturers' protocols.
Immunohistochemical/Cytogenetic Analyses
Combined immunohistochemical/cytogenetic analyses were carried
out on fresh metaphase cell preparations as described previously (6).
The immunohistochemical/cytogenetic analyses used APAAP imnui-
nohistochemical staining to determine tissue lineages of quinacrine-
banded metaphase cells. Mesenchymal and epithelial cell lineages were
established using mouse monoclonal antibodies to vimentin (Dako
Corp., Carpenteria, CA) and keratin proteins (AE1/AE3; Boehringer
Mannheim, Indianapolis. IN), respectively. Incubations with the pri
mary antibodies were followed by incubations with rabbit anti-mouse
immunoglobulin antibodies and APAAP complexes (Dako). APAAP
complexes were detected using naphthol AS-MX phosphate (Sigma
Chemical Co., St. Louis, MO) as substrate and Fast Red TR salt
(Sigma) as chromagen.
RESULTS
Cytogenetic Analyses
Case 1. Phase microscopy at the time of metaphase cell
harvests revealed spindled (mesenchymal) and epithelial mor
phologies in 80 and 20%, respectively, of cultured cells. Twenty
metaphase cells were analyzed after trypsin-Giemsa banding.
Thirteen cells were pseudodiploid: 46,XX,der(6)t(6;14)(p21;q24)-
?ins(6;/-/)(p21 ;q22q23),t(7; 15)(q22;q22),der( 14)t(6; 14)(p21 ;q24)
(Fig. 2), and the remaining seven cells were diploid: 46,XX. The
translocation break points in the t(6;14) were identical to those
in a PCH that we reported previously (Ref. 6, case 2). However,
6224
Research.
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