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BRIEF REPORTS

Fine-Needle Aspiration Cytology


of Desmoplastic Malignant
Melanoma Metastatic
to the Parotid Gland:
Case Report and Review of the Literature
David C. Chhieng, M.D., Joan F. Cangiarella, M.D.,
Jerry Waisman, M.D., and Jean-Marc Cohen, M.D.*

We report a case of desmoplastic malignant melanoma metastatic Case Report


to the parotid gland initially evaluated by fine-needle aspiration.
The cytologic findings consisted of scattered spindle cells in a A 45-year old Caucasian male first presented with a pig-
background of heterogeneous lymphoid cells. The spindle cells mented lesion of the forehead 2 years ago. A wide excision
were scant and displayed mild cytologic atypia. In addition, rare was performed. The histology revealed a malignant mela-
stromal fragments were also present. Cytoplasmic pigment and noma, Clark level IV with a thickness of 3.8 mm. The lesion
intranuclear cytoplasmic inclusions were not seen. The initial
was composed of both epithelioid and spindle-shaped mela-
impression was that of a reactive lymph node with fibrosis. In
retrospect, rare spindle cells displayed moderate atypia. In addi- nocytes. The melanoma recurred in the forehead 1 year later
tion, the stromal fragments were cellular and contained spindle and was again completely excised. Six months later, the
cells with mild atypia. These cytologic findings along with a known patient presented with a 2-week history of a left parotid
history of malignant melanoma should provide clues to the correct mass. Physical examination revealed a 2-cm firm, movable
diagnosis of desmoplastic malignant melanoma. Diagn. Cytopa-
mass of the left parotid gland. The overlying skin was
thol. 2000;22:97–100. r 2000 Wiley-Liss, Inc.
unremarkable. An FNA biopsy was performed and the
Key Words: malignant melanoma; desmoplasia; metastasis; parotid aspiration smears were interpreted as a reactive intraparotid
gland; needle biopsy lymph node. The parotid swelling became smaller after a
course of antibiotic treatment but never completely resolved.
Desmoplastic malignant melanoma (DMM) was first de- An FNA was repeated 6 months later. A cytologic diagnosis
scribed by Conley et al. in 1971.1 It is a rare subtype of of reactive lymphoid hyperplasia with fibrosis was made.
malignant melanoma that is characterized by a proliferation Because of persistence of the parotid swelling despite
of spindle neoplastic cells with a variable stromal re- conservative treatment, a left parotidectomy was performed.
sponse.1–3 The majority of these tumors arise in the sun-
damaged skin of the head and neck region and have a high Materials and Methods
propensity to spread along nerve fascicles.4–7 The cytologic
The FNAs were performed by cytopathologists using 25-
features of malignant melanoma have been well de-
gauge needles. The smears were either air-dried or fixed in
scribed.8–12 However, there is only one case report on the
95% ethanol and then stained with a modified Giemsa or the
cytologic findings of DMM.13 We report a case of DMM
Papanicolaou stain, respectively. The surgical specimen was
involving the parotid gland initially evaluated by fine-needle
fixed in 10% buffered formalin and embedded in semisyn-
aspiration (FNA) biopsy. The cytologic findings are de-
thetic paraffin. Five µm sections were cut and stained with
scribed along with a discussion of the differential diagnosis.
hematoxylin and eosin. Additional sections were obtained
for immunohistochemical study. After deparaffinization, the
Department of Pathology, New York University of Medical Center, New sections were placed in a citrate buffer and treated in a
York, New York microwave oven for 10 min. The immunostaining was
*Correspondence to: Jean-Marc Cohen, M.D., Department of Pathology, performed with antibodies to S-100 protein and HMB-45
Beth Israel Medical Center, First Avenue at 16th Street, New York NY
10003. antigen. Appropriate positive and negative controls were
Received 2 March 1999; Accepted 24 June 1999 performed. Additional material of the surgical specimen was

r 2000 WILEY-LISS, INC. Diagnostic Cytopathology, Vol 22, No 2 97


CHHIENG ET AL.

Fig. 1. The aspiration smear was cellular and consisted of numerous


lymphocytes and rare benign salivary gland acinar cells (open arrow). In Fig. 2. Rare spindle cells with moderate nuclear enlargement and pleomor-
addition, there were scattered spindle cells with elongated and slender phism were noted (arrow). The cytoplasm was delicate and devoid of
nucleus and fine chromatin (solid arrow) (Papanicolaou stain x600). pigment (Diff-Quikt stain x600).

also fixed in glutaldehyde for ultrastructural examination.


Ultrathin sections were cut, stained with uranyl acetate and
lead citrate, and examined under a transmission electron
microscope.

Results
Cytologic Findings
Both aspiration smears were cellular and consisted of a
heterogeneous population of lymphocytes. Rare fragments
of dense fibrous stroma and benign salivary gland tissue
were also noted. In addition, there were scattered spindle
cells in the second aspiration smears (Fig 1). The majority of
the spindle cells appeared as naked nuclei. The cytoplasm,
when present, was scanty, delicate, and devoid of pigment.
The nuclei were elongated and slender with fine chromatin. Fig. 3. Histologic section revealed an infiltrating tumor composed of
spindle cells arranged in fascicles. There was extensive intercellular
The spindle cells were initially interpreted as consistent with collagenous deposition. A prominent lymphoid infiltrate was noted. (Insert)
fibroblasts. In retrospect, rare spindle cells displayed moder- The neoplastic cells displayed mild to moderate atypia. Mitotic figures were
ate atypia with nuclear enlargement, irregular nuclear con- frequent. Cytoplasmic pigmentation was absent (hematoxylin and eosin
x40, insert: x400).
tours, and prominent nucleoli (Fig. 2). Furthermore, the
stromal fragments were cellular and contained atypical
spindle cells. Similar spindle-shaped cells were also noted in
the first aspiration smears, but they were scantier and less in cellularity and composed of spindle cells arranged in
conspicuous than those in the second aspiration smears. fascicles and whorls (Fig. 3). The neoplastic cells displayed
Immunostaining for S-100 protein performed on a destained mild to moderate atypia. Mitotic figures were frequent.
slide was noncontributory. Cytoplasmic pigmentation was absent. There was extensive
intercellular collagenous deposition. A prominent lymphoid
Gross and Histologic Findings infiltrate, particularly in the perivascular areas, was noted.
The surgical specimen consisted of a parotidectomy speci- Perineural invasion was frequent. The histologic findings
men with overlying skin and subcutaneous tissue. The skin were consistent with the diagnosis of DMM. Two intrapa-
appeared unremarkable. On serial sectioning of the parotid rotid lymph nodes were also involved by DMM.
gland, the glandular parenchyma was partly replaced by a The tumor cells were immunoreactive with S-100 protein
poorly circumscribed, firm, white mass, measuring 5 ⫻ but negative for HMB-45 antigen. Ultrastructural examina-
3.5 cm. tion revealed bundles of spindle cells with irregular nuclear
Microscopically, a fibrous-appearing tumor diffusely infil- contours, prominent cytoplasmic processes, and occasional
trated the parotid gland, the adjacent soft tissue, and the deep cell junctions. Basement membrane material was focally
dermis of the overlying skin. The tumor was highly variable present. Rare structures consistent with premelanosomes

98 Diagnostic Cytopathology, Vol 22, No 2


DESMOPLASTIC MALIGNANT MELANOMA

were also seen. These findings supported the diagnosis of myoma, and fibromatosis. It is important to differentiate a
DMM. reparative process with scarring from a locally recurrent
DMM.13 The presence of significant cytologic atypia favors
Discussion
a diagnosis of DMM over a benign process. Malignant
Malignant melanoma can manifest itself in various morpho- lesions that should be distinguished from DMM include
logic patterns and cell types, thus mimicking benign as well sarcomatoid carcinoma, MFH, MPNST, leiomyosarcoma,
as malignant neoplasms. Desmoplastic malignant melanoma and fibrosarcoma. If cytologic evidence of specific differen-
(DMM) is an uncommon subtype and is associated with tiation is not apparent, these lesions may be difficult to
higher frequency of local recurrence.4 The histology has
separate from DMM since the latter often lack cytoplasmic
been well described and is characterized by a spindle cell
pigments and other characteristic features of malignant
proliferation with variable cellularity.1–4,6 The neoplastic
melanoma. The use of ancillary studies such as immunocyto-
cells arrange themselves in interlacing fascicles or whorls
chemical staining may be helpful in the differential diagno-
and exhibit mild to marked polymorphism. Melanin pigment-
containing neoplastic cells as well as melanophages may be sis. The majority of DMMs will be positive for vimentin,
found but are usually absent. A desmoplastic response, in the S-100 protein, and neuron specific enolase.4 HMB-45 anti-
form of collagenization of the intercellular stroma, is almost gen, a marker for malignant melanoma, is usually negative
invariably present. Therefore, some areas, in which there is in DMMs.17 Electron microscopy may also be helpful if
an abundant collagen deposition along with relatively bland premelanosomes and/or melanomas are identified.16
appearing spindle cells, may be mistaken as a benign The differentiation of DMM from MPNST may be
process.14 Neurotropism is found in many but not all DMMs. difficult or even impossible because both tumors have many
Perivascular lymphoid infiltrates are often noted and in some overlapping features. DMM often demonstrates evidence of
tumors can be prominent and misinterpreted as inflamma- schwannian differentiation at both light and electron micro-
tory skin lesions or malignant lymphoma.15 Histologic scopic levels.2,6,18,19 Both tumors are immunoreactive for
patterns resembling a malignant fibrous histiocytoma (MFH), S-100 protein and negative for HMB-45 antigen.6,15 Clinical
a peripheral nerve sheath tumor (PNST), and a smooth information such as a known history of a melanocytic lesion,
muscle tumor have also been described.4,6 There is much the presence of lymph node metastases, or a history of
controversy about the origin of the neoplastic cells. Most neurofibromatosis may be useful in their differentiation.5,7,20
workers believe that the neoplastic cells are melanocytic in Since both entities are of neural crest origin and are
origin and undergo progressive metaplasia towards fibro- aggressive, some authors consider the distinction between
blasts, myofibroblasts, or schwannian cells.1,3–5 DMM and MPNST irrelevant.2,5
The cytologic findings of DMM have rarely been de- DMM often arise in the sun-damaged skin of the head and
scribed in the literature. Egbert et al.16 mentioned the use of neck region of elderly patients.1,2,5–7,13,15,16 There is a slight
FNA in the diagnosis of a metastatic lesion in one of their 25 male predominance.4,6,14 They can arise as a complication of
cases of DMM; they did not describe the cytologic features. many types of malignant melanoma but most frequently
Nance et al.13 described the cytologic findings of DMM from with lentigo maligna.1,5 Although malignant melanoma
a recurrent chest wall lesion and a metastatic lesion to the
frequently involves the parotid gland, only rare examples of
adrenal gland from the same patient. They reported the
DMM involving the parotid gland have been reported. Three
presence of microtissue fragments of spindle cells with
cases of recurrent DMMs involving salivary gland were
hyperchromatic nuclei and prominent nucleoli. On the
reported by Reed and Leonard.5 Auclair et al.21 reported one
contrary, we observed predominantly isolated spindle cells
neurotropic melanoma in their report of 67 cases of sarcoma-
in a background of a heterogeneous population of lymphoid
cells. The latter could represent the prominent lymphoid toid neoplasms of the parotid gland. Labrecque et al.22
infiltrate often seen in DMM or could be secondary to the mentioned a case of parotid gland involvement by recurrent
sampling of the intraparotid lymph node. As the neoplastic DMM. Jennings et al.,23 in their report of five additional
cells could undergo fibroblastic differentiation and appear cases of parotid involvement by DMM, suggested that the
bland, they may be overlooked or be difficult to differentiate mode of spread of DMM to the parotid gland could also be
from reactive fibroblasts. In retrospect, rare spindle cells along nerve fibers in addition to via lymphatic and by direct
with moderate atypia were identified. Intracytoplasmic pig- extension. Superficial parotidectomy and wide local exci-
ment and intranuclear cytoplasmic inclusions were not seen sion with or without radiation therapy have resulted in
in our case as well as others.13 effective local control.5,23
The cytologic differential diagnosis of DMM is broad and In summary, we report the cytologic findings of a case of
includes a variety of benign and malignant spindle cell DMM metastatic to the parotid gland. It is important to
processes. Benign processes that should be considered consider DMM in the differential diagnosis of a spindle cell
include dermatofibroma, schwannoma, neurofibroma, leio- lesion, especially in patients with a history of cutaneous

Diagnostic Cytopathology, Vol 22, No 2 99


CHHIENG ET AL.

malignant melanoma. The use of ancillary studies may be 12. Kline TS, Kannan V. Aspiration biopsy cytology and melanoma.
American J Clin Pathol 1982;77:597–601.
helpful in making the diagnosis.
13. Nance KV, Park HK, Silverman JF. Fine needle aspiration cytology of
desmoplastic malignant melanoma: a case report. Acta Cytol 1991;35:
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100 Diagnostic Cytopathology, Vol 22, No 2

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