Syringomatous tumors of minor salivary gland origin

HUMAN

PATtlOI.OGY--VOI.UME

T A B L E 1. Case I 2 3 4 5 6 7 8

13, N U M B E R 2

Februmy 1982

H E T E R O T O P I C C E R E B R A L TISSUE IN T H E L U N G S : L I T E R A T U R E REVIEW* Age Newborn 4 )ears 5 i )'ears Newborn Stillborn 4 days 5 hours . I/2 hour

CNS Findings

Author (Year)

Microcephaly with encephalocele Frontal meningocele Normal Anencephaly with meningocele Anencephal)" Anencephaly Anencephal)' Anencephaly with gastroschisis

Askanazy (1908) Hiickel (1929) King (1938) Gruenwald (19-tl) Potter and Young, case I (1942) Potter anti Young, case 2 (19-t2) Kanlxmr et al. (1979) Chen et al. (1982)

* Adapted from Valdes-l)apena. 7

p a r e n c h y m a , b u t r a t h e r e x t e n d e d into tile a d j a c e n t l u n g parenchyma through the alveolar spaces, leaving the b r a n c h e s o f b r o n c h i a l t r e e s a n d b l o o d vessels intact. I n tile r e p o r t by K a n b o u r et al., tz t h e g l a n d u l a r s t r u c t u r e l i n e d by tall c o l u n m a r e p i t h e l i t t m xvas c o n s i d e r e d e p e n d y m a . O u r review favors the notion that the glandular structures were branches of the bronchial trees trapped within proliferating n e u r a l tissue.

ACKNOWLEDGMENT T h e a u t h o r s a r e g r a t e f u l to Ms. D a w n T h o r n h i l l a n d Ms. V i r g i n i a W e j a k f o r t h e p r e l ) a r a t i o n o f t h e m a n u s c r i p t .

REFERENCES 1. Potter EL, CraigJM: l'atholog3' of the Fetus and the Infant, 3rd Ed. New York, Yearlxmk Medical Publishers, 1975, p 310.

2. Lemire RJ, Beckwith JB, Warkany J: Anencephaly. New York, Raven Press, 1978, p 74. 3. Gruenwald P: Emboli of brain tissue in fetal lungs. AmJ Pathol 17:879, 19ti. 4. McMillan JB: Emboli of cercbral tissue ira the lungs following severe head injury. Am J Pathol 32:405, 1956. 5. Krakower C: Pulmonary embolus containing cerebral tissue. Arch Pathol 22:113, 1936. 6. Oppenheimer EH: Massive pulmonary embolization by cerebral cortical tissue. BullJotms tlopkins ttosp 94:86, 1954. 7. Valdes-Dapena MA, Are)' JB: Puhnonary emlxfli of cerebral origin in the newborn. A report of two cases. Arch Pathol 8.t:643, 1967. 8. King WI: Tumor (embrynma) of lung containing brain tissue. Med Bull VA 15:181, 1938. 9. Potter EL, Young RL: Heterntopic brain tissue in the hmg of two anencephalic monsters..Arch Pathol 34:1009, 19-t2. 1O. Askanaz)' M: C,ontributinn to the relationship between malformation and tumor based on an observation of a unique encephalocele with gliomas in the lung. Arch Path Anat Inst Tfibingen 6:433, 1908. ! I. Hfickel R: On ectopic glial tissue in the lung of a newborn with frontal encephalocele. Verhandl Deutsch Path Gesellsch 24:272, 1929. 12. Kanbour AI, Barmada MA, Klionsky B, et al: Anencephaly and heterotopic central nervous tissue in hmgs. Arch Pathol Lab Med 103:116, 1979.

SYRINGOMATOUS T U M O R S OF MINOR SALIVARY GLAND ORIGIN CURTIS A. JOIlNSTON, MD,* AND CYRIL TOKER, MDI"

Two intraoral tumors that appear histologically imlistinguishable from syringoma of the skin are discussed. The two patients with these tumors have shown no evidence of recurrence during 36 and 16 months of observation, respectively. We propose that syringomatous tumors of minor saliva D' gland origin may constitute a pathologically and clinically distinct categoD" of intraoral tumors. thtm Pathol 13:182-184, 1982. S y r i n g o m a is a t u m o r o f e p i d e r m a l a p p e n d a g e s t h a t is b e l i e v e d to r e p r e s e n t a n a d e n o m a o f i n t r a e p i d e r t n a l e c c r i n e d u c t s , t T h e lesion is r e p o r t e d to arise m o s t o f t e n in p u b e r t a l w o m e n at m u l t i p l e sites o n t h e face, t r u n k , a n d tlfighs. Characteristic microscopic morphologic features are seen: t h e lesion is c o m p o s e d o f n u m e r o u s isolated d u c t s s c a t t e r e d in a f i b r o u s m a t r i x . T h e s e d u c t s a r e l i n e d w i t h o n e o r two l a y e r s o f r a t h e r u t f i f o r m e p i t h e l i a l cells a n d o f t e n c o n t a i n a n l o r p h o t t s debris. W e r e p o r t two cases o f i n t r a o r a l t u m o r s d i s p l a y i n g histopathologic features characteristic of syringoma. * Resideqt in l'athology. t Professor and llead, Division o f Surgical Pathology. Received from the Department of PathologT, University of Mar)land Hospital, Bahinmre, Md. Address eorrcspondence to Curtis A. Johnston, MD, Department o f Pathology, University o f Maryland llosi)itai , 22 South Greene St, Baltimore, MD 21201.

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CASE REPORTS Case I

T h i s 6 7 - y e a r - o l d w h i t e w o m a n was first s e e n in M a r c h 1977; s h e h a d a two- to t h r e e - m o n t h h i s t o r y o f a m i d lower-lip n o d u l e . Local excision o f tile n o d u l e was p e r f o r m e d . Itistologically, b e n e a t h t h e s q t t a m o u s m u c o s a l e p i t h e l i u m (fig. 1), t u b u l a r o r duct-like s t r u c t u r e s w e r e s e e n s c a t t e r e d in loose f i b r o u s tissue p e r i p h e r a l l y a n d in m o r e c o m p a c t f i b r o u s s t r o m a c e n t r a l l y . T h e m a r g i n o f t h e lesion was i l l - d e f i n e d , w i t h a n " i n f i l t r a t i v e " n l a r g i n focally e n c r o a c h i n g u p o n a m i n o r salivary g l a n d . T h e d u c t s w e r e l i n e d p r e d o m i n a n t l y by a single layer o f tall c u b o i d cells, v a r y i n g f r o t h r e c t a n g t , l a r to s q u a r e in c o n f i g u r a t i o n . O c c a sional d u c t s c o n t a i n e d a n a m o r p h o u s e o s i n o p h i l i c Inaterial. Nuclei w e r e r e g u l a r in size, a l t h o u g h a small p r o p o r t i o n o f h y p e r c h r o m a t i c n n c l e i w e r e seen. No m i t o s e s w e r e f o u n d . T h e lesion was r e p o r t e d as a s y r i n g o m a t o u s t u m o r o f m i n o r salivary g l a n d o r i g i n . T h e r e has b e e n n o e v i d e n c e o f r e c u r r e n c e a f t e r 36 m o n t h s o f follow-up.

Case 2 This 57-year-ohl white woman underwent biopsy of a n o d n l a r lesion w i t h i n t h e u p p e r lip in S e p t e m b e r 1979. I listologically, b e n e a t h t h e buccal e p i t h e l i u n a o f tile lip (fig.

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CASE S T U D I E S

FiKure 1. Case l. (See text f o r discussion.) (Hematoxylin-eosin stain. • 2), regular tubular or duct-like structures were seen scattered within fibrous c o n n e c t i v e tissue. T h e s e t u b u l a r structures were lined predominantly by a single row of somewhat flattened cuboid cells, occasionally reachiug two

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layers in thickness. An a m o r p h o u s eosinophilic material was noted in some ducts, identical in appearance to that seen in case 1. Also as in case 1, the ducts appeared "infiltrative" in distribution, without circumscription, and they s u r r o u n d e d

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t l U M A N P A T I I O L O G Y ~ V O L U M E 13, NUMBER 2 Febntary 1982 portions of three lninor salivary glands. No definite conncction with these salivary glands was identificd. Nuclei were regular in size and chrotnatin distribution. No lnitoses were found. T h e h i s t o p a t h o l o g i c f i n d i n g s were i n t e r p r e t e d elsewhere as invasive adenocarcinoma. As a result, one m o n t h later approximately one third of tile u p p e r lip was resected, with retation o f a skin tlap for plastic reconstruction. In December 1979, revision of the skin flap was performed. T h e r e llas been no evidence of recurrence or metastasis after 16 months o f follow-up. DISCUSSION T h e histologic features of these two cases are identical to those of syringoma of the skin. These lesions were both located, however, beneath tile buccal surface o f the lip, and both lesions s u r r o u n d e d - m i n o r salivary glands. In con-

tradistinctionto the mixed tulnor (pleomorl~llic adenolna), myxoid a n d chondroid lnetaplasia were coinpletel)- absent, and these lesions were trot well circumscribed.2 T h e histogenetic kinship between eccrine sweat glands and salivary glands is well known. It is not unreasolmble to expect the development of similar tumors from these organs. T h e mixed t u m o r of the skin, which is thought to be of eccrine origin, 3 has bccn well described. We propose that tulnors lnorphologicall)' identical to syringoma of the skin may arise within oral mucosa from minor salivary glands. REFERENCES 1. Lever WF. Ilistopatholog3" of the Skin, 5th Ed. Philadelphia.JB Lippincott Co. 1975. 2. ThachrayAC, LucasRB. Tumors of the MajorSalivatT Glands.Atlasof Tumor Patholog)', Second Series, Fascicle 10. Washington, DC, Armed Forces Instituteof Pathology, 1974. 3. Ilernandez l']J. Mixed tumors of the skin of the salivarygland t)pe: a light and electron microscopicstudy,J InvestDermotol66:.t9, 1976.

Correspondence T H E F U T U R E OF C L I N I C A L P A T H O L O G Y

To the Editor:--I would like to respond to the Open F o r u m discussion of the future o f clinical pathology and the role of the clinical pathologist, t I have encountered lnore than one medical and hospital adnfinistrative staff with very real questions about the role of a physician director of clinical laboratories. T h e previous experience o f most staff members had bccn largely with pyre anatomic pathologists who expressed variations of the philosophy that "the clinical labs will r u n themseh'es" and that the pathologist's place is within tile laboratory. Without exception, as soon as the hospital adlninistrators and clinical physicians are exposed to the results of active and intensive direction o f clinical laboratories by a clinical pathologist and active clinical consultation as a rotttine part of tile laboratory practice, these people were absolved of an)" and all questions as to tile "true role" of the clinical pathologist. (The term "clinical" pathologist" is probably not ideal if it can imply a "noncliltical" pathologist. Perhaps the terln "laboratory medicine" or "laboratory ph)'sicialf' would be better since almtomic patholog)' certainly should be practiced as a clinical area of medicine.) I feel that there is no problem at all with survival of laboratory medicine as a medical specialty--in fact, once clinical physicians become aware of the availabilit)' of a laboratory ph)sician as an active consultant in the operating rooln, the emergenc)' room, the intensive care unit, or the routine wards, the main probleln is overutilization of this service, and, as Dr. Senhauser states, a lack of tilne swiftly becomes tile laboratory physician's main probleln. All respondents to Dr. Stefallilfi's essay referred to the paralnount importance of a pathologist practicing as a clinical physician consultant. Ttfis is truly tile key to e n h a n c e m e n t and preservation of tile pathologist's image as a physician. I f tiffs is accomplished, there will be little need for active or deliberate media enhancenaent of o u r linage, and the flmctions of teaching and research should naturall)' follow. Some e l a b o r a t i o n m a y be ira Order: T h e clinical

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pathologist must be available when needed b)" his clinical colleagues. This ineans around-the-clock availability, at least b)' phone contact, and optimall)' one melnber of the group should be available to come to the hospital. This means availability and visibilit)' within tile hospital, including clinical areas on weekends a n d holida)'s and after hours. As so mauy physicians have found, once our routine availability is established, it will be unlikely to be abused by o u r clinical colleagues. A listing of areas in which the clinically consulting pathologist can critically participate in patient care decisions is virtually infinite----operatingroom consultation on questionable surgical cases as well as frozen sections, managelnent and monitoring of problematic transfilsion cases, interpretation of spinal fluid bacterial stains, monitoring of 9therapeutic drug parameters, recommendations for antibiotic therap)', case finding, and diagnosis of previously unsuspected hematologic or helnostatic disorders are merely a few of weekly, if not daily, activities in which the pathologist in o u r llospital actively participates. It does not take long with this type of laboratory medicine pnactice before tile pathologist is socially included as a clinical physician by the hosptial lnedical staff. I lence, the pathologist is not isolated witlfin his own speciahy, but will have the enthusiastic support of the entire lnedical staff. T h e establishment o f an accurate, reliable, timely, and appropriate clinical laboratory service is a necessary first step, bt, t it is only a first step in the practice of laboratory lnedicine. It is tile necessary foundation u p o n which the active clinical consuhative practice of the laboratory physician lnnst be based. It ma)" bc necessary to lengthen the training program, but for reasons other than adding a )'ear of "clinical exposure." l have seen no correlation between thc active clinical participation of pathologists a n d the p r e s e n c e o f a c l i n i c a l i n t e r n s h i p ira t h e i r t r a i n i n g background. Pathologists who are inost clinically oriented often have had straight pathology training, a n d man)' pathologists wllo are restricted to "in-the-laboratory" practice have had excellent rotating ilaternships. "Fire answer would seem to rest with training as a clinical consultant as part of the pathology residency. In other words, the direc-