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Presented at the United States and Canadian Academy of Anatomy Annual Meeting, March 25–31, 2000, New Orleans, Louisiana.
David E Kardon M.D.1, Bruce M Wenig M.D.2, Dennis K Heffner M.D.1 and Lester D R Thompson M.D.1
Lymphangiomatous polyps are aberrant amiable tumors of the tonsils.
Twenty-six cases of lymphangiomatous polyps diagnosed amidst 1980 and 1999 were retrieved from the files of the Otorhinolaryngic-Head and Abutting Bump Registry of the Armed Forces Institute of Pathology. Hematoxylin and eosin-stained slides were advised to characterize the histologic actualization of these tumors. Immunohistochemical stains were performed on 15 cases. Analytic aftereffect abstracts were obtained. Results: The patients included 13 males and 13 females, ages 3 to 63 years (mean, 25.2 years). Patients accomplished dysphagia, abscessed throat, and the awareness of a accumulation in the throat. Affection were present from a few weeks to years. The tonsillar masses were unilateral in all cases. Clinically, the lesions were frequently mistaken for a bump (n = 18 patients). Grossly, all of the lesions were polypoid and abstinent 0.5 to 3.8 cm (mean, 1.6 cm). Histologically, the polyps were covered by squamous epithelium assuming capricious epithelial hyperplasia, dyskeratosis, and lymphocytic epitheliotropism. The masses showed a appropriate submucosal admeasurement of baby to medium-sized, endothelial-lined, lymph-vascular channels defective actualization of malignancy. Collagen, bland muscle, and adipose tissue were present in the stroma. Intravascular proteinaceous aqueous and lymphocytes were noted. Immunohistochemical allegation accepted the endothelial agent of the vascular admeasurement and a alloyed lymphoid population. The cogwheel analysis included fibroepithelial polyp, lymphangioma, boyish angiofibroma, and squamous papilloma. In all patients with follow-up, complete surgical abatement was alleviative (mean follow-up, 5.4 years; range, 1 mo to 14 years).
We detail the analytic and pathologic actualization of tonsillar lymphangiomatous polyps. These tumors are aberrant and may clinically be mistaken for a cancerous neoplasm. The appropriate histologic actualization should acquiesce for its actual analysis and adverse from agnate actualization tonsillar lesions.
Hamartoma, Immunohistochemistry, Lymphangioma, Lymphangiomatous polyp, Pediatric, Polyps, Tonsil
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Benign tumors or tumor-like lesions of the palatine tonsil are beneath accepted than malignancies. Squamous papillomas annual for the majority of the amiable lesions, admitting vascular tumors are reportedly attenuate (1). We articular ten well-documented cases of lymphangiomatous tumors of the tonsil (MEDLINE 1966–1999) (1, 2, 3, 4, 5, 6, 7), with a distinct case address as aboriginal as 1910 (8) (summarized in Table 1). Because of the abnormal analytic and pathologic actualization of these polyps, pathologists and clinicians akin may accept adversity in classifying them correctly. Therefore, we call the clinical, histopathologic, and immunohistochemical actualization of a alternation of 26 lymphangiomatous polyps (LAPs), which are amiable lymph-vascular proliferations with capricious degrees of fibrous, adipose, and lymphoid components.
Twenty-six cases of lymphangiomatous polyps were articular in the files of the Otorhinolaryngic-Head and Abutting Anatomy Registry of the Armed Forces Institute of Anatomy from 1980 to 1999, called from a absolute of 1389 (1.9%) tonsillar amiable and cancerous tumors. Two of the cases in our alternation accept been ahead appear (3). The accent in the abstract acclimated to analyze this commodity is capricious and includes variants of the agreement "polypoid," "lymphangiomatous," and "hamartomatous." Materials aural the Institute's files were supplemented by a analysis of accommodating demographics, affection at presentation, accessible and surgical anatomy reports, and advice acquired from accounting questionnaires or articulate advice with the alleviative physician(s). Fifteen cases were acquired from noncombatant sources, including university medical centers and adopted contributors, and 11 cases were acquired from aggressive hospitals. This analytic analysis was conducted in accordance and acquiescence with all statutes, directives, and guidelines of the Code of Federal Regulations, Title 45, Allotment 46, and the Department of Defense Directive 3216.2, apropos to animal capacity in research.
We advised the hematoxylin and eosin-stained (H&E) slides and evaluated the histologic features. Lesional fibrosis was graded on a calibration from 0 to 3 based on the bulk and blazon of collagen. Although the analysis of tonsillar LAP does not crave accessory stains, we performed a array of immunohistochemical stains on formalin-fixed, paraffin-embedded, 4-m-thick tissue sections to characterize the antigenic contour of the entity. Immunohistochemical stains were performed on a distinct block in 15 cases application the connected avidin-biotin-peroxidase adjustment as has been ahead declared (9). The antibodies and titers acclimated are abbreviated in Table 2. Standard absolute controls were acclimated throughout, with accustomed serum acclimated as the abrogating control.
The 13 males and 13 females ranged in age from 3 to 63 years, with a beggarly age at presentation of 25.2 years (median, 25.5 years). There was no aberration in age at presentation amidst females (25.0 years) and males (25.5 years). Eight of the patients were advised pediatric or boyish (less than 21 years).
Twenty patients presented clinically with a tonsillar mass. Patients additionally presented with dysphagia, abscessed throat, and/or adversity swallowing. Two patients presented with affection of tonsillitis alone. In all cases the LAP was unilateral, afterwards a ancillary predilection. One accommodating presented with the accumulation in his nasopharynx, arising from the adenoids rather than the faucial tonsils.
Fifteen patients were advised by excisional biopsy of the accumulation and 11 by tonsillectomy. No added analysis was rendered for this bane in any of the patients. Aftereffect advice was accessible in 23 cases. We did not access added aftereffect for the three adopted patients, two of which were ahead appear (3). There was no tonsillar aberancy or development of alternate ache in any accommodating during the aftereffect aeon that ranged from 1 mo to 14 years (mean, 5.4 years). All patients were animate afterwards affirmation of ache at aftermost aftereffect with the barring of one accommodating who died of altered causes afterwards 5.8 years.
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The tumors ranged in greatest ambit from 0.5 cm to 3.8 cm (mean, 1.6 cm). They were declared as polypoid (Fig. 1) with a bland white, tan, or chicken cut surface. The masses assorted from absorptive to abutting and fibrous. Sixteen of the lesions were clearly pedunculated or polypoid, admitting the actual ten were sessile to unremarkable.
Histologically, the above apparent epithelium was either squamous or respiratory, and in a few cases was hyperplastic or dyskeratotic (although afterwards dysplasia). Three cases showed a arresting nested epitheliotropism, in which baby lymphocytes (round nuclei and abridged nuclear chromatin) were abiding into angled intramucosal spaces (Fig. 2). The attendance of lymphocytes aural tonsillar epithelium is normal. The lymphocytes are usually abiding in an affectionate amalgamation of alone lymphocytes percolating amidst the epithelial beef of the catacomb apparent epithelium basic a archetypal "lymphoepithelium." Therefore, the accumulation of detached pockets of lymphocytes in LAP is unusual, and to the authors' ability not ahead described. The acceptation and analysis of this epitheliotropism is unclear.
LAPs are composed of a admeasurement of submucosal aggrandized aqueous vascular channels and capricious amounts of coarse affiliation tissue. In accordance with the arresting appearance, the majority of the tumors were pedunculated polypoid masses arising from the apparent of the tonsil (Fig. 1) and showed no aggression of the basal stroma. Four of the lesions had a added papillary architecture, accompanying with the lymphoid and edematous stroma. None of the cases continued into the surrounding pharyngeal tissues.
The vascular apparatus were thin-walled and usually independent proteinaceous aqueous (n = 19 cases) and lymphocytes (n = 20 cases). In a few cases, the lymphocytes were abiding into the argosy in such as way as to accomplish the channels difficult to analyze on accepted H&E stain (Fig. 3). A stromal adipose tissue basic was present in nine cases (Fig. 4). Fibrosis was arresting (2 to 3 ) in 18 cases, basal (1 ) in four, and absent in the actual four.
Factor VIII–related antigen was absolute in the endothelium and subendothelium of the aqueous channels in all (n = 15) cases activated (Fig. 3). The argosy were acknowledging with anti-CD31 in 13 cases, anti-CD34 in six cases, and one case was abrogating for both markers. Bland beef actin was present in the walls of the aggrandized aqueous argosy in all of the cases tested. The hematologic markers (CD45RB [LCA], CD3 [T-cell] [Fig. 3], and CD20 [L26, B-cell]) were bidding in the lymphoid beef in an accepted arrangement of administration for tonsillar tissue in all cases. Lymphocytes aural the vascular channels and aural the epithelium of the polyps were predominantly CD3 immunoreactive, alike admitting there was a polymorphous B-cell and T-cell immunophenotype.
The arch and abutting are the best accepted anatomic regions for lymphangiomatous lesions, accounting for over 90% of all lymphangiomas (10). Best appear in the bark and subcutaneous tissues, but added sites accommodate the larynx, parotid gland, mouth, and argot (11). The tonsil is a beneath accepted armpit for the development of lymphangiomatous tumors, and their allocation in this area is confusing. In the aboriginal allotment of the 20th century, histologically identical lesions of the tonsil were appear by a cardinal of altered names, including angiomas (12), angiofibromas, or fibroangiomas (13). Still others accept had adversity classifying their cases specifically, and accept called them fibrolipomas afterwards the stromal components, or accept accustomed them a added anecdotic diagnosis, such as "polypoid bump absolute fibroadipose tissue." (3)
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Consequently, the accurate accident of these lesions is difficult to accurately appraise from the literature. As such, we allotment the opinions of others (1, 3) and accept lymphangiomatous tonsillar tumors accept a college accident than is reported. Furthermore, they are apparently added accepted than added angiomatous lesions in this location. In our series, lymphangiomatous polyps accounted for 1.9% of all tonsillar tumors apparent in appointment during the aforementioned aeon of review. In a antecedent abstraction (14), lymphangiomatous lesions represented 8% of all amiable tonsillar tumors, admitting hemangiomas and fibromas represented 2 and 3%, respectively.
We adopt the appellation lymphangiomatous polyp rather than lymphangioma because the pathogenesis of these lesions is still unclear. As acclaimed in the abstract (5), the aqueous channels of LAPs are frequently dilated, but about are not as arresting as in the archetypal lymphangioma. In addition, the stromal apparatus are frequently added abounding than the vessels. We accede with the affirmation that these lesions are best acceptable hamartomatous (5, 6, 15) because they abide of a accidental admeasurement of elements that are commonly begin in the tonsil. These tumors occasionally become ample and account adverse affection and in these circumstances, accept about been present for years—up to 30 years in one appear case (6).
Histologically, four of our lesions were papillary and apparent a abutting affinity to the ahead appear "lymphoid papillary hyperplasia" or "papillary lymphoid polyp" of the tonsil (4, 16). This attenuate commodity occurs alone in children. Although some of our cases were papillary, they did not accept the arresting lymphoid follicles appropriate of that diagnosis, and instead had added arresting aqueous spaces (Fig. 5). LAPs are afar from the tonsillar parenchyma, admitting papillary hyperplasia blends with the basal lymphoid stroma. Moreover, 18 of our cases did not action in pediatric-aged patients. Accustomed that these two lesions accommodate agnate apparatus in differing architectures and proportions, it is absolutely accessible they may represent a forme fruste of the aforementioned commodity forth a adorning arc.
As ahead noted, immunohistochemistry is not bare to definitively analyze LAP. However, in the absorption of accurately documenting the immunoprofile of these lesions in a ample series, we activated a array of commercially accessible immunoantibodies. As expected, the aggrandized aqueous argosy accommodate at atomic a attenuate bank of bland muscle, and are analogously acknowledging with Factor VIII–related antigen. CD31 is added frequently acknowledging than CD34, although this aberration in staining did not associate with added actualization of the tumors. The endothelial markers may be of use in highlighting the argosy in cases that accept abounding intraluminal lymphocytes accretion the channels to the point they may be difficult to recognize. Notably, the endothelial markers were non-reactive in the intramucosal spaces that amidst the nested epitheliotropic lymphocytes. The hematologic markers showed the accepted arrangement of administration for B-cells and T cells. Best of the lymphocytes, including the lymphocytes, were of T-cell agent (Fig. 2). The lymphocyte analysis was that of accustomed complete lymphocytes and as such should not be abashed with the agnate clearing into epithelium, alleged "Pautrier's microabscesses," which may be apparent in cancerous T-cell lymphoma.
The cogwheel diagnoses of accidental pathologists included boyish angiofibroma, fibroepithelial polyps, papilloma, and lymphangioma. It is important to analyze LAP from boyish angiofibroma as the closing bane is usually advised added aggressively to anticipate accessible recurrence. Clinically, angiofibromas about action in the nasopharynx of boyish males, generally attaining a ample size, with all-encompassing advance and alike cartilage erosion, and presenting with epistaxis due to the affluent claret supply. Although one of our cases of LAP occurred in the nasopharynx, it developed in a 28-year-old changeable afterwards epistaxis and did not access into the surrounding structures. Histologically, the stroma of angiofibromas is added cellular, composed of stellate and beefy cells, and contains staghorn-like attenuate belted vascular channels. LAPs usually accept a almost paucicellular coarse accomplishments and abounding added lymphocytes. Squamous papilloma is usually an exophytic apparent epithelial proliferation, abiding in assorted layers, not advancing the basal stroma, and defective a aqueous and lymphocytic component. When lymphangiomas are described, they usually accommodate broadly aggrandized vascular channels with luminal proteinaceous aqueous and lymphocytes. Our cases of LAP independent close coarse affiliation tissue to a capricious degree, a affluent lymphocyte investment, and a cardinal independent adipocytes. These apparatus are the accustomed capacity of this site, except abiding in a altered pattern, and so we accept they added acceptable represent a hamartomatous admeasurement rather than actuality a archetypal lymphangioma.
Lymphangiomatous polyps of the tonsil are amiable tumors that best frequently present as accumulation lesions and are composed of aggrandized aqueous channels amidst a fibrous, lymphoid, and/or adipose stroma. They are best acceptable hamartomatous proliferations, and as such are convalescent by simple surgical excision. Afterwards surgical resection, there were no incidences of recurrence. They (LAPs) accept a assorted analysis and may display papillary growth. LAPs are apparently not as attenuate as appear in the literature, and accounted for about 2% of all tonsillar neoplasms in our study.
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The authors acknowledge Ms. Serena Lei for her analytical analysis of the article.
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