Case Report
Lymphoid papillary hyperplasia of palatine tonsils: A rare pediatric case
from Turkey
Demet Altun
a,*
, Hasan Huseyin Arslan
b, Haldun Umudum
c, Gokcan Okur
d, Yusuf Ersal
eaDepartment of Pediatrics, Etimesgut Military Hospital, Ankara, Turkey
bDepartment of Otorhinolaryngology, Etimesgut Military Hospital, Ankara, Turkey
c
Department of Pathology, Ufuk University Medical Faculty, Ankara, Turkey d
Department of Radyology, Etimesgut Military Hospital, Ankara, Turkey e
Department of Pathology, Eskisehir Military Hospital, Eskisehir, Turkey
1. Introduction
Lymphoid papillary hyperplasia (LPH) is a rare benign lesion of the palatine tonsils. On examination, numerous papillary struc-tures are observed on the surface of these tonsils. This view is often clinically considered a malignant lesion or papillomatosis. The pathogenesis, together with the clinical situation and etiology of this rare disorder, is uncertain. In some cases, individuals of the same family are reported to share an autosomal dominant distribution. LPH is described most frequently in Asian and Japanese populations [1] and only sporadically in the Western literature[2,3]. This abnormality has not been well documented among the Turkish population. Herein, we report a 13-year-old Turkish girl with this rare condition.
2. Case report
A 13-year-old girl presented to the pediatric department of our hospital, complaining of repeated tonsillitis associated with a
foreign-body sensation in the throat and dysphagia over the past five years, with progressive worsening for the previous three months. Her past medical history was unremarkable, and her family members denied any family medical history. Physical examination showed that the largest of the tonsils was entirely covered by about 0.2 cm of numerous papillary structures. There were no accompanying signs of lymphadenopathy or infection. The clinical impression was papillomatosis. No other clinical or laboratory abnormalities were found. Surgical excision was performed, with specimens fixed in formalin. Macroscopically, the right and left lateral palatine tonsils were measured and averaged 3.5 2 1 cm, with an elastic texture, gray–white coloration, and a friable papillary surface (Fig. 1A). Numerous tiny papilliform configurations, which were grossly similar to papillomatosis, occurred in the upper respiratory tract, covering the entire available surface of the tonsils. There was no hemorrhage, necrosis, or infiltration grossly present. Histopatho-logical examination of the palatine tonsils revealed arboreal lesions with finger-like papillary projections on their surfaces. These were composed of remarkable germinal centers of follicular lymphoid hyperplasia covered by stratified squamous epithelium (Fig. 1B). Several reactive lymphoid follicles were located in the core of the papillary projections (Fig. 1C). Some areas showed small bacteria collections on the parakeratotic squamous epithelium (Fig. 1D). The histologic appearance of the tissue was suggestive of a diagnosis of LPH. The postoperative period was uneventful, and
International Journal of Pediatric Otorhinolaryngology Extra 10 (2015) 25–27
A R T I C L E I N F O
Article history:
Received 6 December 2014
Received in revised form 17 January 2015 Accepted 19 January 2015
Keywords: Child
Lymphoid papillary hyperplasia Tonsil
Turkish
A B S T R A C T
Lymphoid papillary hyperplasia (LPH) is a rare abnormality of the tonsils with a tendency to affect young Asian females. We report a case of this rare condition in a 13-year-old girl. To our best knowledge, this case is the only pediatric Turkish case of LPH reported in the literature. The significance of recognizing this abnormality lies in its clinical appearance, given the fact that despite the clinical characteristics suggesting a diagnosis of epithelial papilloma or even malignancy, this process is benign, non-neoplastic, can readily be differentiated from other neoplastic lesions by histologic examination, and can easily be cured by tonsillectomy.
ß2015 Elsevier Ireland Ltd. All rights reserved.
* Corresponding author at: Etimesgut Military Hospital Pediatric Department, 06100 Etimesgut, Ankara, Turkey. Tel.: +90 3122491011x3623;
fax: +90 3122444977; mobile: +90 5474914749.
E-mail addresses:[email protected](D. Altun),[email protected]
(H.H. Arslan),[email protected](H. Umudum),[email protected](G. Okur),
[email protected](Y. Ersal).
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http://dx.doi.org/10.1016/j.pedex.2015.01.002
the patient was subsequently discharged. There has been no evidence of recurrence of disease on follow-up for two years. Patient consent form was obtained from the patient with her family. 3. Discussion
LPH is an uncommon abnormality of the palatine tonsils, with the clinical presentation of both a benign and a malignant tumor
[1–3], such as oral squamous papilloma or lymphoid polyposis, both of which could produce severe pharyngeal obstruction, as preoperatively indicated by the clinicians in the present case. However, based on the microscopic examination, this lesion revealed rich lymphoid tissue with intense follicular hyperplasia with excessive increase of the germinal center, without mono-morphic lymphoid hyperplasia, and the covering mucosa showed only mild hyperplasia without atypia. Therefore, it could be readily identified as a benign, tumor-like lesion and could be easily distinguished from other neoplastic lesions.
This condition has been known since as early as 1896, when Roberts reported an interesting case of LPH that was clinically described as resembling a papilloma of the tonsil[3]. Since then, LPH has been most commonly reported in Asian populations
[1]. Since the first report of papillary hypertrophy of the tonsils by Ogino and Matsui in 1924, approximately three dozen cases of this disease have been documented in the Japanese literature
[3]. However, the current case, to the best of our knowledge, represents the only case reported in the literature in recent years of LPH in a Turkish patient. Furthermore, there have only been four documented examples of this disease in non-Asian individuals to date[3].
The age and sex distribution of previously reported cases reveals females to be slightly more commonly affected than males, with an age range of 2–54 years[3], but the most recently reported
cases show a fairly female predominance, especially in young girls
[1]. This is not surprising, given that symptoms of obstruction related to hyperplasia of the pharyngeal tonsils are common in children because of the small size of the nasopharynx in this age group[3], as in our case. However, lymphoid hyperplasia may also affect adults, probably due to local dysfunction of the epithelial structures[4]. Most reported LPH presents with involvement of the bilateral tonsils. However, the papillary hyperplasia was more significant on the right tonsil than the left tonsil in our patient. Ming et al. presented a patient in whom only the right lateral palatine tonsil was affected[5].
The etiology of LPH remains enigmatic [1–3]. Some of the suggested causative factors, as reviewed in the old archives by Dias et al.[3], include repeated inflammatory stimulation, hormonal influence, neoplasia, and congenital deformity with autosomal dominant inheritance. An interesting finding was first documented in 1980 by Enomoto et al. [1], who described a lesion called papillary hypertrophy of the tonsils affecting a young female child with a complaint of an obstructive feeling in the throat; unexpectedly, this disease was found in 13 members of her family pedigree. Genetic analysis showed that this lesion was transmitted by an autosomal dominant gene. However, in the current case, the patient denied any family medical history of detected hypertrophy of the palatine tonsil. It was impossible to determine whether any other evidence of abnormal hormonal stimuli were potentially contributory to this lesion in our patient. Otherwise, repeated inflammatory stimulation may be a more likely potential factor contributing to the development of this disease[3]. It has been established that in chronic tonsillitis, after repeated antigenic stimulation, lymphoid follicle hyperplasia takes place proportion-ally in the tonsils to a hypertrophic degree[3]. Therefore, LPH may be the result of excessively persistent antigenic stimulation on the tonsils. The T-lymphocyte-mediated immune response may play a
[(Fig._1)TD$FIG]
Fig. 1. (A) Grossly, the surface of the palatine tonsils was enclosed by numerous different-sized small papilliform projections. (B) Histopathologic examination revealed finger-like papillary projections with reactive lymphoid follicles, which were surrounded by squamous epithelium (H&E 20). (C) Several reactive lymphoid follicles were located in the core of papillary projections (H&E 40). (D) Some areas showed small bacteria collections on the parakeratotic squamous epithelium (H&E 100).
D. Altun et al. / International Journal of Pediatric Otorhinolaryngology Extra 10 (2015) 25–27 26
critical role in this process[3]. However, which exact regulatory mechanism is involved is not wholly clear.
Conflict of interest
The authors report no conflict of interest to disclose. Appendix A. Supplementary data
Supplementary data associated with this article can be found, in the online version, atdoi:10.1016/j.pedex.2015.01.002.
References
[1]T. Enomoto, K. Matsui, T. Tabata, Ann. Otol. Rhinol. Laryngol. 89 (1980) 132–134.
[2]J. Carrillo-Farga, F. Abbud-Neme, E. Deutsch, Am. J. Surg. Pathol. 7 (1983) 579–582.
[3]E.P. Dias, S.E. Alfaro, S.C. De Piro, Oral Surg. Oral Med. Oral Pathol. Oral Radiol. Endod. 95 (2003) 77–79.
[4]C.A. Mogoanta, E. Ionita, D. Pirici, M. Mitroi, F. Anghelina, S. Ciolofan, E. Patru, Rom. J. Morphol. Embryol. 49 (2008) 381–386.
[5]M. Zhao, J. Yu, C. Li, Int. J. Clin. Exp. Pathol. 6 (2013) 1957–1960.
