Metastasis of Acinic Cell Carcinoma of the Parotid Gland to The

the number of involved nodes is Bita Esmaeli, MD lymph nodes for detection of metastatic melanoma. Ann Diagn Pathol. 2002;6:257-264. small, a situation more common in Houston, Tex 14. Yu LL, Flotte TJ, Tanabe KK, et al. Detection patients with positive SLNs than in David Reifler, MD of microscopic melanoma metastases in senti- those who develop clinically appar- nel lymph nodes. Cancer. 1999;86:617-627. Grand Rapids, Mich 15. Bostick P, Essner R, Sarantou T, et al. Intraop- ent disease. It is not unreasonable Victor G. Prieto, MD, PhD erative lymphatic mapping for early-stage melaoma of the head and neck. Am J Surg. 1997; to assume that this pattern is also M. Amir Ahmadi, MD true for conjunctival melanomas. 174:536-539. Lillie Hidaji, BA 16. Eicher SA, Clayman GL, Myers JN, et al. A pro- Therefore, the potential for im- spective study of intraoperative lymphatic map- Ebrahim Delpassand, MD pingforheadandneckcutaneousmelanoma.Arch proved regional control and sur- Merrick I. Ross, MD vival offers a rationale for studying Otolaryngol Head Neck Surg. 2002;128:241-246. Houston 17. Jansen L, Koops HS, Nieweg OE, et al. Senti- SLN biopsy in patients with con- nel node biopsy for melanoma in the head and junctival melanoma. neck region. Head Neck. 2000;22:27-33. The authors have no relevant finan- 18. Medina-Franco H, Beenken SW, Heslin MJ, et At present, the role of adju- cial interest in this article. al. Sentinel node biopsy for cutaneous melanoma in the head and neck. Ann Surg Oncol. vant systemic therapy for conjunc- We thank Stephanie Deming for tival melanomas is unclear. Since 2001;8:716-719. her invaluable editorial contribution 19. Gershenwald JE, Colome MI, Thompson W, et nodal disease is a powerful prognos- to this report. al. Patterns of recurrence following a negative tic factor for distant failure, SLN bi- sentinel lymph node biopsy in 243 patients with Corresponding author and re- stage I or II melanoma. J Clin Oncol. 1998;16: opsy may allow for early detection prints: Bita Esmaeli, MD, Ophthal- 2253-2260. of high-risk patients. If this is the mology Section, Department of Plas- 20. Henk JM, Whitelocke RA, Warrington AP, et al. case, then patients with nodal dis- Radiation dose to the lens and cataract formation. tic Surgery, Box 443, M. D. Anderson Int J Radiat Oncol Biol Phys. 1993;25:815-820. ease can be offered protocols for sys- Cancer Center, 1515 Holcombe Blvd, 21. Glat PM, Longaben MT, Jelks EB, et al. Peri- orbital melanocytic lesions: excision and re- temic adjuvant therapy in addition Houston, TX 77030 (e-mail: besmaeli to complete surgical dissection of construction in 70 patients. Plast Reconstr Surg. @mdanderson.org). 1998;102:19-27. the involved basins. Adjuvant 22. Ross M, Reintgen D, Balch C. Selective lymph- therapy may consist of systemic ad- 1. Norregaard JC, Gerner N, Jensen OA, Prause adenectomy: emerging role for lymphatic map- ministration of interferon or chemo- JU. Malignant melanoma of the conjunctiva: oc- ping and sentinel node biopsy in the manage- currence and survival following surgery and ra- ment of early stage melanoma. Semin Surg Oncol. therapy, or a combination of these ap- diotherapy in a Danish population. Graefes Arch 1993;9:219-223. proaches.23-26 Despite the additional Clin Exp Ophthalmol. 1996;234:569-572. 23. O’Brien CJ, Coates AS, Petersen-Schaefer K, et 2. Paridaens ADA, Minassian DC, McCartney al. Experience with 998 cutaneous melano- surgery time required for SLN bi- ACE, Hungerford JL. Prognostic factors in pri- mas of the head and neck over 30 years. Am opsy and the potential risk of facial mary malignant melanoma of the conjunctiva: J Surg. 1991;162:310-314. nerve damage if the nodes are in the a clinicopathological study of 256 cases. Br 24. Kirkwood J, Strawderman M, Ernstoff M, Smith J Ophthalmol. 1994;78:252-259. T, Borden E, Blum R. Interferon-alpha-2b adju- parotid area, the likely improve- 3. Shields CL, Shields JA, Gunduz K, et al. Conjunc- vanttherapyofhigh-riskresectedcutaneousmela- ment in nodal staging and the pos- tival melanoma: risk factors for recurrence, exen- noma: the Eastern Cooperative Oncology Group sible survival benefit offered by this teration, metastasis, and death in 150 consecutive trial EST 1684. J Clin Oncol. 1996;14:7-17. patients. Arch Ophthalmol. 2000;118:1497-1507. 25. Buzaid AC, Colome M, Bedikian A, et al. Phase technique warrant its further study in 4. Jakobiec FA, Rini FJ, Fraunfelder FT, Brownstein II study of neoadjuvant concurrent biochemo- patients with conjunctival mela- S. Cryotherapy for conjunctival primary acquired therapy in melanoma patients with local-regional melanosis and melanoma: experience with 62 metastases. Melanoma Res. 1998;8:549-556. noma and possibly other ocular ad- cases. Ophthalmology. 1988;95:1058-1070. 26. Eton O, East M, Legha SS, et al. Pilot study of intra- nexal tumors with a propensity for 5. Tuomaala S, Eskelin S, Tarkkanen A, et al. arterial cisplatin and intravenous vinblastine and metastasis to the regional nodes. A Population-based assessment of clinical char- dacarbazine in patients with melanoma in-transit acteristics predicting outcome of conjunctival metastases. Melanoma Res. 1999;9:483-489. larger-scale study needs to be under- melanoma in whites. Invest Ophthalmol Vis Sci. taken to evaluate the rate of positiv- 2002;43:3399-3408. ity of SLNs in patients with conjunc- 6. Esmaeli B, Wang X, Youssef A, Gershenwald JE. Patterns of regional and distant metastasis Metastasis of Acinic tival melanoma. Our experience with in patients with conjunctival melanoma: expe- SLN biopsy for conjunctival melano- rience at a cancer center over four decades. Oph- Cell Carcinoma of the thalmology. 2001;108:2101-2105. Parotid Gland to the mas suggests that this technique can 7. Gershenwald JE, Thompson W, Mansfield PF, be done safely and should be consid- et al. Multi-institutional melanoma lymphatic Contralateral Orbit ered for lesions that are thicker than mapping experience: the prognostic value of sentinel lymph node status in 612 stage I or II 1 mm; it should be performed, how- melanoma patients. J Clin Oncol. 1999;17:976- Acinic cell carcinoma is an uncom- ever, in the confines of an institu- 983. mon low-grade malignant tumor of tional review board–approved pro- 8. Esmaeli B. Sentinel lymph node mapping for pa- tientswithcutaneousandconjunctivalmelanoma. the salivary glands, in which some 1 tocol so that observations about the Ophthal Plast Reconstr Surg. 2000;16:170-172. cells resemble normal acinic cells. rate of positivity of SLNs can be re- 9. Esmaeli B, Eicher S, Delpassand E, Prieto VG, Most of these tumors occur in the pa- Popp J, Gershenwald JE. Sentinel lymph node 1,2 liably reported in the future. It is im- biopsy for conjunctival melanoma. Ophthal Plast rotid gland. Women are affected portant to point out that while our Reconstr Surg. 2001;17:436-442. more often than men, and the age at observations in this single patient vali- 10. Esmaeli B. Advances in the management of ma- occurrenceisearlierthaninothersali- lignant eyelid tumors: the role of sentinel node 2 date the concept that SLN biopsy biopsy. Int Ophthalmol Clin. 2002;42:151-162. vary gland cancers. Most cases are should be considered for patients with 11. Wilson MW, Fleming JC, Fleming RM, Haik unilateral, and bilateral involvement high-risk melanomas of the conjunc- BG. Sentinel node biopsy for orbital and ocular adnexal tumors. Ophthal Plast Reconstr Surg. has rarely been reported. Conversely, tiva, they should not be interpreted 2001;17:338-344. anunusualcaseofsynchronousacinic as recommendations for routine SLN 12. Esmaeli B. Commentary on “Sentinel node bi- cell carcinomas of the left parotid and opsy for orbital and ocular adnexal tumors.” biopsy in every patient with conjunc- Ophthal Plast Reconstr Surg. 2001;17:344-345. right submandibular glands has pre- tival melanoma. 13. Prieto VG, Clark SH. Processing of sentinel viously been described.3

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©2003 American Medical Association. All rights reserved. Downloaded From: https://jamanetwork.com/ on 09/25/2021 fore decided to monitor the orbital lesion with repeat magnetic resonance imaging at a later date. A right total parotidectomy with sacrifice of a part of the facial nerve, excision of con- tents of the right submandibular tri- angle, and supramental node sam- pling were performed. Histologic examination demonstrated complete excision of the residual tumor in the parotid bed. There was an is- land of tumor within the excised right submandibular gland that was be- lieved to be either metastatic or a syn- chronous tumor. Postoperative adjuvant radiotherapy was carried out, during which time the patient visited her optometrist with symptoms of blurred vision and diplopia. The lat- Figure 1. Acinic cell tumor arising in the parotid tissue with a small group of normal parotid acini ter was controlled with the addi- (bottom left) (hematoxylin-eosin, original magnification ϫ200). tion of a 10–prism diopter Fresnel prism base out to the distance seg- parotid and submandibular glands, ment of the spectacle. and an enlarged submandibular In June 2002, the patient was lymph node. In January 2002, a right referred to and seen in the Oph- partial parotidectomy with sam- thalmology Department for consid- pling of the right submandibular eration of eyelid surgery to reduce lymph node was performed. Histo- the exposure of the right eye that logic examination showed that the resulted from the surgically in- parotid gland contained acinic cell duced facial nerve palsy. By this carcinoma (Figure 1) extending to time, however, her diplopia had within 0.2 mm of the surgical exci- also increased. sion margin, with a possibility of in- Examination demonstrated 10– complete excision. Four mitoses in and 12–prism diopter esophoria for 40 high-power fields were noted in near and distance, respectively, with the tumor cells. The right subman- slight left proptosis. The previ- dibular lymph node contained a tu- ously arranged follow-up magnetic mor growing in nests of large tu- resonance imaging was done 2 weeks mor cells showing vesicular nuclei later and showed enlargement of the Figure 2. Axial magnetic resonance image of the with prominent nucleoli and granu- orbital lesion with displacement of orbits showing a lesion approximately 1 cm in diameter in the left orbit. lar cytoplasm, suggesting meta- the optic nerve (Figure 3). The dip- static acinic cell carcinoma. lopia was worse, with considerable Although acinic cell carcino- A magnetic resonance image of limitation of elevation, depression, mas rarely metastasize, they have a the head and neck was performed 3 and adduction of the left eye. high tendency to recur locally if they weeks after the surgery and could Excision biopsy of the orbital are incompletely excised. We de- not exclude residual tumor in the pa- mass was performed. At surgery the scribe a patient with acinic cell tu- rotid gland because of postopera- mass was found to lie between the mor arising in the parotid gland, with tive changes in the parotid area. In- medial rectus muscle and the eye. The metastases to the contralateral or- cidentally, the image demonstrated lack of a capsule and its gelatinous bit, submandibular salivary gland, a lesion approximately 1 cm in di- consistency prevented excision in and submandibular lymph node. ameter in the left orbit that was in- toto, with the posterior portion in traconal, lying between the medial particular requiring piecemeal re- Report of a Case. A 54-year-old rectus muscle and the optic nerve, moval. Histologic examination white woman came to the Maxillo- and that did not appear to arise from showed the mass to be acinic cell car- facial Department with a right pa- either structure (Figure 2). cinoma (Figure 4), compatible with rotid mass of 2 months’ duration. Re- Initially, the orbital lesion was the primary tumor previously ex- sults of a fine-needle aspiration not thought to be related to the sali- cised. After exclusion of any other biopsy suggested an epithelial neo- vary gland tumor, as an early metas- distant metastasis with a computed plasm, and ultrasonography of the tasis with contralateral spread from tomographic scan of the abdomen neck showed an unusual appear- the acinic cell carcinoma was be- and chest, radiotherapy to the left or- ance of the superficial portion of the lieved to be very unlikely. It was there- bit was performed.

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©2003 American Medical Association. All rights reserved. Downloaded From: https://jamanetwork.com/ on 09/25/2021 vary lobule. High-grade tumors are poorly differentiated and resemble the early phases of embryonic develop- ment of acini. Controversy persists regard- ing the prognostic value of assign- ing grade by histologic analysis of acinic cell carcinoma.2 In a recent report by Hoffman et al,2 the high- grade cancers were significantly associated with age of more than 30 years, advanced stage, and distant metastasis at initial manifestation. In our case, the acinic cell tumor was found in the parotid gland, the submandibular gland, and a lymph node. The primary tumor seemed to be partially encapsulated, but mitotic figures were seen, Figure 3. Axial magnetic resonance image of the orbits showing considerable enlargement of the tumor, suggesting malignant potential. Me- causing displacement of the optic nerve and the globe. tastasis to the orbit occurred very early in the disease process and was discovered only incidentally. The metastatic tumor in the orbit had grown considerably in 5 months, causing proptosis and diplopia.

Tarek A. Saleh, FRCSEd Kim N. Hakin, FRCOPhth Michael J. Davidson, FRCSEd Taunton, England

We thank David Turner, FRCPath, PhD, Department of Pathology, Taun- ton and Somerset Hospital, Taunton, England, for his advice. Corresponding author: Tarek A. Saleh, FRCSEd, Eye Department, Taunton and Somerset Hospital, Mus- grove Park, Taunton, Somerset TA1 5DA, England.

Figure 4. Metastatic deposit of acinic cell tumor in orbital connective tissue (hematoxylin-eosin, original 1. Ellis GL, Auclair PL. Acinic cell adenocarci- magnification ϫ200). noma. In: Ellis GL, Auclair PL, Gnepp DR, eds. Surgical Pathology of Salivary Glands. Vol 25. Philadelphia, Pa: WB Saunders Co; 1991:299- On follow-up in December, 11 rarely arise from the lacrimal gland.6 337. months after the initial diagnosis of Spiro et al7 reported a distant metas- 2. Hoffman HT, Karnell LH, Robinson RA, Pinks- ton JA, Menck HR. National Cancer Data Base acinic cell carcinoma of the parotid tasis rate of 12%, mainly to lung, bone, report on cancer of the head and neck: acinic cell gland, the patient had no evidence of and brain. On gross pathological ex- carcinoma. Head Neck. 1999;21:297-309. 3. Gustafsson H, Carlso¨o¨ B. Multiple acinic cell car- tumor and had left convergent squint amination, acinic cell carcinoma is cinoma: some histological and ultrastructural fea- causing intermittent diplopia. fairly well circumscribed and may ap- tures of a case. J Laryngol Otol. 1985;99:1183- pear encapsulated. Four histopatho- 1193. 4. Saxena RB, Mathur RN, Sonani SZ. Orbital me- Comment. We found 2 reported logic patterns have been described: tastasis of mixed parotid tumour. Indian J Oph- cases of distant metastasis from pa- solid, microcystic, papillary-cystic, thalmol. 1975;23(2):23-24. rotid gland tumor to the orbit4,5 in and follicular.1 It is not uncommon 5. Thomas KM, Cumberworth VL, McEwan J. Or- bital and skin metastases in a polymorphous low the English-language literature. The for these patterns to coexist in the grade adenocarcinoma of the salivary gland. primary tumors were mixed pa- same tumor. Batsakis et al8 catego- J Laryngol Otol. 1995;109:1222-1225. 4 6. Jang J, Kie JH, Lee SY, et al. Acinic cell carci- rotid tumor and polymorphous low- rized acinic cell carcinoma into low- noma of the lacrimal gland with intracranial ex- 5 grade adenocarcinoma. grade and high-grade neoplasms. tension: a case report. Ophthal Plast Reconstr Surg. Acinic cell carcinoma repre- Low-grade tumors are broadly inter- 2001;17:454-457. 7. Spiro RH, Huvos AG, Strong EW. Acinic cell car- sents approximately 5% to 17% of all preted as those most closely resem- cinoma of salivary origin: a clinicopathologic salivary gland cancers1,2 and can very bling the architecture of a normal sali- study of 67 cases. Cancer. 1978;41:924-935.

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©2003 American Medical Association. All rights reserved. Downloaded From: https://jamanetwork.com/ on 09/25/2021 8. Batsakis JG, Chinn EK, Weimert TA, Work WP, Krause CJ. Acinic cell carcinoma: a clinicopathologic study of thirty-five cases. J Laryngol Otol. 1979;93:325-340.

Retinal Astrocytic Hamartomas: Unexpected Findings in a Giant Panda

We report the unexpected findings of bilateral retinal astrocytic hamartomas in a giant panda. The eyes of Hsing-Hsing, a giant panda that had renal failure, chronic hypertension, degenerative joint disease, and progressive ulcerative keratitis, were examined grossly and histo- logically. Most of the findings were related to Hsing-Hsing’s debilitated condition or to normal anatomic variations between giant pandas and humans. These findings included anatomic features typical of a carnivore and metastatic calci- fication of the cornea and tapetum secondary to renal failure, retinal arteriolar sclerosis and hemorrhage due to hypertension, and bilateral corneal ulcers due to septicemia. Bilateral retinal astrocytic hamartomas were also unexpectedly found. The clinical importance of retinal astrocytic hamartomas is dis- cussed.

Report of a Case. We report a case Figure 1. Hsing-Hsing before he became debilitated. of retinal astrocytic hamartomas of unknown cause in the eyes of a 28- year-old male giant panda (Ailu- pair of musk oxen, as gestures of finding of retinal astrocytic hamar- ropoda melanoleuca). The eyes were goodwill between the United States tomas. There were no other lesions obtained from Hsing-Hsing and the People’s Republic of supportive of tuberous sclerosis. (Figure 1), who was euthanized at China. The pandas were instant There have been only 2 previous the Smithsonian National Zoologi- celebrities seen by millions of reports of ocular pathologic find- cal Park (Washington, DC) because people and taken into the hearts of ings in giant pandas, and neither of advanced renal failure leading to children everywhere. described retinal astrocytic hamar- several degenerative conditions in- Ling-Ling died of heart failure tomas.1,2 cluding chronic renal failure, degen- in 1992. At her death, she and Hsing- Astrocytic hamartomas are erative joint disease, chronic epi- Hsing were aged 23 years and be- benign tumors. In humans, these staxis, decreased mobility, progressive lieved to be the oldest giant pandas tumors are more commonly associ- keratitis, and bilateral corneal ulcers living outside of the People’s Repub- ated with tuberous sclerosis with reduced vision. The Smithso- lic of China. Hsing-Hsing devel- (Bourneville disease) and less fre- nian National Zoological Park (Wash- oped arthritis and was castrated to quently seen in neurofibromatosis ington, DC) submitted both eyes to treat a testicular tumor at the age of 1 (von Recklinghausen disease) the ophthalmic pathology division at 26 years. During the last year, he dis- and neurofibromatosis 2 or as iso- the Armed Forces Institute of Pathol- played remarkable resilience de- lated occurrences.3-5 These hamar- ogy (Washington, DC) for examina- spite kidney failure and associated tomas are usually found in the tion in November 1999. symptoms. retina and optic disc. Because of Hsing-Hsing and his mate Both eyes included findings the strong association between Ling-Ling came to the United related to Hsing-Hsing’s physical tuberous sclerosis and retinal States in 1972 in exchange for a state, in addition to the unexpected astrocytic hamartoma in humans,

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