846 MICROCYSTIC ADNEXAL CARCINOMA

27. Karp NS, McCarthy JG, Schreiber JS, et al: Membranous bone lengthening: A serial histological study. Ann Plast Surg 29:2, 1992

28. Karns P, Nester D, Boyd C, et al: Recovery of neurosensory function following orthognathic surgery. J Oral Maxillofac Surg 48:124, 1990

29. Block MS, Daire J, Stover J, et al: Changes in the inferior

alveolar nerve following mandibular lengthening in the dog using distraction osteogenesis. J Oral Maxillofac Surg 51:652, 1993

30. Staffenberg DA, Wood JR, McCarthy JG: Mandibular lengthen- ing in the canine using an intraoral device. Presented at V International Congress of Craniofacial Surgery, Oaxaca, Mexico, October 1993

J Oral Maxillofac Surg 53:846-849, 1995

Microcystic Adnexal Carcinoma: Report of a Case

MICHAEL L. ROBINSON, MD, DMD,* MARK A. KNIBBE, MD, DDS,* AND JOHN B. ROBERSON, DMD~f

Microcystic adnexal carcinoma (MAC) is a recently

described neoplasm with combined pilar and eccrine differentiation that is characterized by a locally aggres- sive growth pattern. ~ Wallace and Bernstein 2 reported

that MAC occurs primarily on the faces of young and middle-aged women; however Borenstein et al 3 state

that males and females are affected equally. This entity can be clinically and histologically confused with other

malignant and benign cutaneous neoplasms, which

may lead to inadequate initial treatment and extensive recurrence. 4 We present a case of MAC in a middle-

aged man who received radiation therapy as an adoles-

cent for the treatment of acne.

Report of Case

A 53-year-old white man was evaluated in our office after visiting his dermatologist and general dentist. The patient had a cutaneous lesion in the left mental region for approxi- mately 2-years. The lesion was 4 x 4 cm in size and had irregular margins. It was nontender and had an erythematous cutaneous surface (Figs 1, 2). The patient's medical history was essentially negative, although he did receive radiation treatment for acne as an adolescent. The social history was negative for tobacco and alcohol abuse.

An incisional biopsy was performed under local anesthesia in the office on July 31, 1992. Microscopic examination of

* In .private practice, Oral and Maxillofacial Surgery, Edgewood, KY.

"~ Resident, Oral and Maxillofacial Surgery, University of Cincin- nati, Cincinnati, OH.

Address correspondence and reprint requests to Dr Knibbe: 20 Medical Village Dr, Suite 196, Edgewood, KY 41017.

© 1995 American Association of Oral and Maxillofacial Surgeons

0278-2391/95/5307-002053.00/0

the specimen showed stratified squamous epithelium, pile- sebaceous structures, supporting connective tissue, and an adnexal neoplasm (Fig 3). Within the superficial connective tissue were numerous microcysts that were filled with a kera- tinlike material (Fig 4). There were several layers of flattened cuboidal cells lining the microcysts. The individual micro- cysts were not well delineated and extended into the underly- ing connective tissue. The tumor was infiltrating between muscle fibers and approached segments of peripheral nerve (Fig 5). The histologic findings were consistent with a diag- nosis of MAC.

One week later the patient was taken to the operating room for removal of the lesion and immediate reconstruction. The lesion was marked with 1-cm margins from the palpable tumor. The planned excision encompassed an area measuring 6.5 cm anteroposterior and 6 cm superoinferiorly (Fig 6). The excision was carded through subcutaneous tissue and platysma muscle and down to the inferior border of the mandible. The periosteum toward the center of the lesion was excised. The entire thickness of the tumor was elevated with a periosteal elevator. The mental nerve was in the center of the tumor and was sacrificed. The specimen was sent to the pathologist for frozen sections. A frozen section of the remaining stump of the mental nerve was also performed. The integrity of the mandibular bone was maintained, and there was no evidence of bony invasion because the tumor had not penetrated through the periosteum. The frozen sec- tions showed the margins of the surgical specimen as well as the mental nerve stump to be free of tumor.

The digastric muscle then was removed from the inferior border of the mandible and, in combination with the buccina- tor, used to close the periosteal defect and cover the mandib- ular bone. A rhomboid flap was then designed to locally reconstruct the surgical defect. Once the flap was reflected and mobilized, it was transposed into position with minimal tension.

At 1 year postoperatively, the patient remained asymptom- atic and free of detectable recurrence of the lesion. The surgical wound had healed uneventfully, and he had under- gone minor scar revision and dermabrasion of the flap mar- gins (Fig 7). He has now been free of tumor for 30 months.

ROBINSON, KNIBBE, AND ROBERSON 847

FIGURE 1. Clinical photograph showing erythema in the left chin region.

FIGURE 2. Close-up photograph showing a 4 × 4 cm area of erythema and skin changes in left chin area.

Discussion

Goldstein et al, 5 in 1982, described the first cases of MAC. They used the term because they thought the neoplasm did not fit into any previous classification of adnexal tumors) Approximately 30 cases of MAC have been added to the literature since Goldstein's report?

Microcytic adnexal carcinoma shows five character- istic histologic features: 1) strands and islands of fairly benign appearing squamous and basaloid cells, 2) horn cysts in the superficial portion of the tumor, 3) ductal structures lined by two layers of cuboidal cells, 4)

a deeply invasive growth pattern with penetration of perineural spaces, skeletal muscle, and fat, and 5) a desmoplastic stroma, especially in the deeper areas of tumor. 6

The histologic features of MAC are distinct, al- though these lesions can be confused with a number of other similar tumors. Benign adnexal lesions include trichoepithelioma, syringoma, and trichoadenoma. However, the lack of deep dermal or local invasion separate these entities from the MAC. Squamous cell carcinoma, basal cell carcinoma, and sweat duct carci- noma share similar ]histopathologic characteristics with the MAC, but the frequent mitotic activity and cellular atypia common to those malignant lesions usually are not seen in MAC. 2

The term sclerosing sweat duct carcinoma has been used synonymously with MAC. 4 Because of its inva- sive growth and pilar and eccrine differentiation, the term MAC signifies to the clinician an aggressive, in- filtrating lesion. 7 Despite this tumor's aggressive local behavior, there are no reported cases of metastasis. 8

The cellular origin of the MAC is believed to be from a pluripotential adnexal keratocyte of eccrine or

FIGURE 7. Clinical photograph showing 2 years postoperative FIGURE 6. Clinical photograph showing rhomboid flap design, result.

848 MICROCYSTIC ADNEXAL CARCINOMA

FIGURE 5. Photomicrograph showing tumor ceils forming strands in the deeper connective tissue layer. The tumor is infiltrating be- tween muscle fibers and is approaching segments of a peripheral nerve (Hematoxylin-eosin stain, original magnification x25).

FIGURE 3. Photomicrograph showing stratified squamous epithe- lium, pilosebaceous structures, supporting connective tissue, and an adnexal neoplasm, Within the superficial connective tissue there are numerous microcysts filled with a keratinlike material (Hematoxylin- eosin stain, original magnification x5).

FIGURE 4. Photomicrograph detailing a microcyst within the con- nective tissue. Strands and nests of tumor ceils are seen deep in the connective tissue (Hematoxylin-eosin stain, original magnification x25).

follicular derivation. Recent immunocytochemical studies demonstrate antibodies to carcinoembryonic antigen and Leu-M1 and hence favor an origin from glandular tissue. 9 The use of an immunoperoxidase staining technique in identifying carcinoembryonic an- t igen will probably become a valuable tool in evaluat- ing difficult cases. ~° The presence of Langerhans cells in MAC, as reported by Takemiya et a l ," may indicate another characteristic component of this tumor.

Clinicians should be informed about MAC because of its locally aggressive behavior. Careful attention should be paid to a history of radiation therapy as a possible causative factor. Several reported cases of MA C have had a history of such therapy for the treat- ment of acne. Addit ional cases may become more fre- quent given the popularity of this treatment in the past and the 20-year latency period for radiation-induced neoplasia. ~2 Successful treatment is achieved by com- plete surgical resection of the tumor, using frozen sec- tions to check both the peripheral and deep margins.

References

1. Lupton GP, McMarlin SL: Microcytic adnexal carcinoma: Re- port of a case with 30 year follow-up. Arch Dermatol 122:286, 1986

2, Wallace RD, Bernstein PE: Microcystic adnexal carcinoma: Re- port of one case. Ear Nose Throat J 70:789, 1991

3. Borenstein A, Seidman DS, Trau H, et al: Case report: Microcys- tic adnexal carcinoma following radiotherapy in childhood. Am J Med Sci 301:259, 1991

4. Mayer MH, Winton GB, Smith AC, et al: Microcystic adnexal carcinoma (sclerosing sweat duct carcinoma). Plast Reconstr Surg 84:970, 1989

5. Goldstein DJ, Ban" RJ, Santa Cruz DJ: Microcystic adnexal carcinoma: A distinct clinicopathologic entity. Cancer 50:566, 1982

6. Newman L: Microcystic adnexal carcinoma: A case report and review of the literature. Br J Oral Maxillofac Surg 24:448, 1986

7. Nickoloff B J, Fleischmann HE, Carmel J, et al: Microcystic adnexal carcinoma: Immunohistologic observations sug-

ZHENG AND ZHANG 849

gesting dual (pilar and eccrine) differentiation. Arch Derma- tol 122:290, 1986

8. Hamm JC, Argenta LC, Swanson NA: Microcystic adnexal car- cinoma: An unpredictable aggressive neoplasm. Ann Plast Surg 19:173, 1987

9. Birkby CS, Argenyi ZB, Whitaker DC: Microcystic adnexal carcinoma with mandibular invasion and bone marrow re- placement. J Dermatol Surg Oncol 15:308, 1989

10. Cooper PH: Sclerosing carcinomas of sweat ducts (microcystic adnexal carcinoma). Arch Dermatol 122:261, 1986

11. Takemiya M, Shiraishis, Saeki N, et al: Microcystic adnexal carcinoma wilLh Langerhans cells. J Dermatol 15:428, 1988

12. Fleischman HE, Roth RJ, Wood C, et al: Microcystic adnexal carcinoma treated by microscopically controlled excision. J Dermatol Surg Oncol 10:873, 875, 1984

J Oral Maxillofac Surg 53:849-851, 1995

Tuberculosis of the Parotid Gland: A Report of 12 Cases

JIA WEI ZHENG, MD,* AND QUI HONG ZHANG, MDt

Facia l and cervical tuberculosis are most commonly seen in the submandibu la r and cervical lymph nodes. Tuberculosis o f the parot id gland is rare; only a few wel l -def ined cases o f this condi t ion have been repor ted in the literature. 1 F r o m January 1985 to February 1993,

12 patients with parot id gland tuberculosis were treated in our depar tment , none o f which were correct ly diag- nosed preoperat ively . This repor t re t rospect ively ana- lyzes the 12 pat ients ' c l inical data, causes o f misd iag- nosis, cl inical features o f parot id gland tuberculosis , differential diagnosis , and t reatment options.

Materials and Methods

From January 1985 to February 1993, 165 parot idec- tomies were per formed in our department . The postop- erat ive b iopsy reports were examined and 12 patients with tuberculosis o f the parot id gland were identified. The pat ients ' age, sex, past and present histories, and treatment were rev iewed and analyzed.

Report of Cases

There were five males and seven females ranging in age from 2 to 53 years, with a mean age of 33 years. Five patients

Received from the Department of Oral and MaxiUofacial Surgery, the Affiliated Hospital of Shandong Medical University, Jinan, Shan- dong, China.

* Lecturer. ? Professor. Address correspondence and reprint requests to Dr Zheng: Depart-

ment of Oral and Maxillofacial Surgery, The Affiliated Hospital of Shandong Medical University, Jinan, 250012, Shandong Province, People's Republic of China.

© 1995 American Association of Oral and Maxillofaeial Surgeons

0278-2391/95/5307-002153.00/0

were younger than 10 years, five patients were between 11 and 40 years, and two were more than 50 years old. Seven patients had tuberculosis of the left and five patients had tuberculosis of the right parotid gland. The disease presented as a firm swelling in eight patients, with a maximal size of 5.0 x 4.0 X 3.0 cm and a minimal size of 2.5 X 2.0 X 1.5 cm. In the remaining four patients, the tuberculous lesion manifested as periauricular fistulae that frequently dis- charged pus. The shortest history was 1 month; the longest history was 2 years. Four patients had a 1- to 3-month his- tory, six patients had a history of 4 to 12 months, and two patients had a 13- to 24-month history.

No patients with parotid masses had a history of low- grade fever and tuberculosis. Five patients complained of spontaneous pain in the parotid region and two had tender- ness. There was no facial nerve involvement. The mass was generally isolated, mobile, round or oval shaped, and the color and the temperature of the overlying skin was normal. Usually the mass enlarged gradually, but four patients expe- rienced rapid growth before presentation. All the patients had a normal chest radiograph. The white blood cell count was between 5.0 x 109/L and 8.5 x 10g/L, except in two patients in whom it was more than 9.0 x 109/L. The lympho- cyte count was elevated in five patients. Eight patients re- ceived antibiotics for 1 week before surgery, but the masses showed no or minim,'d reduction in size.

Patients with periauricular fistulae were mainly children. There was a history of recurrent swelling around the ear initially; later, the skin over the lesion was perforated and a fistula formed spontaneously or it occurred following surgi- cal drainage of a fluctuant swelling. Bacterial culture of the pus was done in two patients, but no bacterial growth was reported.

Open biopsy and aspiration were not performed before surgery because of concern about spreading tumor cells. Pa- rotid imaging was done in eight patients. Eight patients un- derwent superficial parotidectomy with preservation of the facial nerve based on a clinical diagnosis of a parotid neo- plasm. Four patients were treated with fistula excision only; the fistulae were superficial and adhered to the intraparotid lymph nodes. All the excised specimens were examined by intraoperative frozen sections and postoperative microscopy.