KBB ve BBC Dergisi 17 (1):16-18, 2009
Angioleiomyoma of the Inferior Turbinate & A Short Review
Alt Konka Anjiyoleiyomyoması ve Literatürün Kısaca Gözden Geçirilmesi
Ganapathy DHANASEKAR FRCS, FRCS [ORLHNS], Archie MALCOLM FRC Path, Stuart David THOMPSON MD, FRCS [ORL]
Departments of Otolaryngology/Head & Neck Surgery & Histopathology Royal Shrewsbury Hospital, Shrewsbury
ABSTRACT
We report a very rare case of angioleiomyoma arising from the inferior turbinate. We discuss the presentation, management and also a review of the lite-rature.
Keywords
Angioleiomyoma, nasal cavity, inferior turbinate
ÖZET
Alt konkadan köken alan çok nadir bir anjiyoleiomyom olgusunu sunuyoruz. Olgunun başvuru şeklini, tedavisini tartıştık ve literatürü gözden geçir-dik.
Anahtar Sözcükler
Angioleiomyoma, nasal boşluk, alt konka
Çalıșmanın Dergiye Ulaștığı Tarih: 02.07.2008 Çalıșmanın Basıma Kabul Edildiği Tarih: 22.10.2008
≈≈
Correspondence
Mr. Ganapathy DHANASEKAR, FRCS
6, Woodhayes Croft Wolverhampton WV10 8PP U.K. Tel: 0044-1902 861022
Angioleiomyoma of the Inferior Turbinate & A Short Review 17
Turkiye Klinikleri J Int Med Sci 2008, 4 17
IN TRO DUC TI ON
e i om yo ma is a ra re, usu ally slow-gro wing, be nign smo oth musc le tu mo ur. It is di vi ded in to two gro -ups: non-vas cu lar and vas cu lar (an gi o le i om yo mas, an-gi om yo mas, vas cu lar le i om yo ma). An an-gi o le i om yo mas [AL] are ra rely se en in the he ad and neck are a.1 Only
abo ut ten ca ses ha ve be en re por ted in the na sal ca vity so far2 ma inly at tri bu ted to the pa u city of smo oth musc le at
this si te3 AL usu ally ori gi na te from the smo oth musc le in
the walls of vas cu lar chan nels.4-7 So me aut hors ha ve
sug-ges ted it is a kind of ha mar to ma,6,7a vas cu lar mal for ma
-ti on,5or one sta ge in a pro cess of smo oth musc le pro li
fe-ra ti on from ha e man gi o ma to so lid le i om yo ma.5 Mo ri mo
-to8clas si fi ed vas cu lar le i om yo mas in to so lid, ca ver no us
and ve no us types.AL usu ally de ve lop in the lo wer ex tre -mi ti es and pre sent as so li tary, small, pa in ful cu ta ne o us mass of the so lid type.7,8 Only 8.5-10% of AL ari se in the
he ad and neck are a and they usu ally pre sent as a pa in less mass of ve no us or ca ver no us type.5,7,8
CA SE RE PORT
A 74 ye ar old gent le man pre sen ted to our ENT de-part ment with a his tory of re cur rent epis ta xis from his left nos tril. The re was no na sal obs truc ti on, as so ci a ted pa in or ot her na sal and si nus symptoms.
On exa mi na ti on the re was a lar ge poly po i dal le si on ari sing from the left in fe ri or tur bi na te. Midd le me a -ti, post na sal spa ce and right nos tril exa mi na ti on was nor mal. He was lis ted for an ur gent exa mi na ti on un der ana est he tic of the no se and ex ci si on bi opsy of the le si on. At ope ra ti on a soft, oe de ma to us mass was se en ari -sing from the in fe ri or tur bi na te. The le si on was comp le tely ex ci sed un der en dos co pic con trol. The pos t-o pe ra ti ve pe ri t-od was une vent ful.
His to pat ho logy
The tu mo ur sho wed nu me ro us thick wal led ve ins with the smo oth musc le of the ves sel walls split ting out the ad ja cent tis su e. The re was fo cal myxo id chan ge with so me fat cells. The re was no sig ni fi cant ple o morp hism or mi to ses. [Figure 1, 2]
DIS CUS SI ON
AL, a be nign tu mo ur with vas cu lar and smo oth musc le muscom po nents is ra re in the he ad and nemusck are a. Hamusc hi -su ga et al.7in the ir study of 562 ca ses of AL, they fo und
only 48 ca ses (8%) in the he ad and neck. Only fi ve of the
-se ca -ses we re fo und in the na sal ca vity. Ma e sa ka et al.9in
1966 re por ted the first ca se. To da te only ten na sal ca vity AL’s ha ve be en re por ted in the li te ra tu re.
AL usu ally pre sent in the midd le aged as most of the ob ser va ti ons ha ve be en in the fifth and sixth de ca de. The -re ap pe ars to be fe ma le p-re pon de ran ce as eight out of the ten ca ses we re wo men.10The ro le of sex ste ro id re cep tors
in the de ve lop ment of the se tu mo urs has not be en comp -le tely elu ci da ted. The pre va -len ce of fe ma -le ca ses of AL and the in cre a sed pa in du ring preg nancy and the mens -tru al cycle in the se ca ses at se ve ral si tes7, 11 sug gest that Figure 2. A lesional blood vessel showing the very thick layer of smooth mus-cle spilling out into adjacent tissue (bottom of the photo) [H&E x 50] Figure 1. Underneath the respiratory mucosa of the nose there are numer-ous thick walled blood vessels with smooth muscle extending into adjacent tissue. [H&E x 20]
KBB ve BBC Dergisi 17 (1):16-18, 2009 18
pro ges te ro ne re cep tors inf lu en ce na sal AL de ve lop ment. Pro ges te ro ne re cep tor ac ti vity usu ally de pends on oes tro -gen.11 Ma ri o ni et al.12in the ir ca se fo und it to be only pro
-ges te ro ne-re cep tor po si ti ve and oes tro gen-re cep tor ne ga ti ve on im mu no his toc he mi cal analy sis. They sug ges -ted that the growth of AL may be hor mo ne-de pen dent.
The com mon pre sen ting symptoms of na sal ca vity AL’s are fa ci al pa in or he a dac he, na sal obs truc ti on and epis ta xis.2,13 AL’s de ve lop mostly from the midd le or
in-fe ri or tur bi na te.2,14-18 The na sal ves ti bu le9,19-23has be en
the si te of ori gin in six ca ses, pa ra na sal si nu ses24-27in
fo ur ca ses and the na sal sep tum17,28 in two ca ses.
The dif fe ren ti al di ag no sis of AL inc lu des he man gi o ma, fib rom yo ma, le i om yob las to ma, an gi om yo li po -ma and vas cu lar le i om yo sar co -ma. Con ven ti o nal light mic ros copy af ter ha e ma toxy lin-eo sin sta i ning sho uld be re in for ced by im mu no his toc he mi cal in ves ti ga ti ons. Im-mu no his toc he mi cal sta i ning for smo oth Im-musc le ac tin, des min and vi men tin can pro vi de ad di ti o nal in for ma ti -on to help cla rify the di ag no sis.
Ima ging in the form of MRI or CT scan is ne e ded only if the le si on is cli ni cally lar ge. MR ima ging is helpful in dif fe ren ti a ting ne op las tic tis su e from inf lam ma -tory chan ges such as si nu si tis or mu co ce le and al so gi ves mo re ac cu ra te in for ma ti on on the re la ti ons hip of the tu mo ur with blo od ves sels and du ra. CT scans de li -ne a tes bo -ne ero si ons cle arly. Sin ce the le si ons are usu-ally of mo de ra te vas cu la ri sa ti on an gi og raphy and pre-ope ra ti ve em bo li za ti on are not ro u ti nely re qu i red.
Sur gery in the form of lo cal ex ci si on using a trans -na sal ap pro ach is usu ally ade qu a te for small le si ons as se en in our ca se.
CONC LU SI ON
AL’s of the in fe ri or tur bi na te are ra re tu mo urs. They can pre sent as pa in less mas ses with epis ta xis. They can be comp le tely ex ci sed with no evi den ce of re cur ren ce.
1. Soames JV, Moore UJ, Ord RA, McElroy JH. Angiomyoma of the tempo-ral region. Br J Otempo-ral Maxillofac Surg 1989;27:229-35.
2. Khan MHZ, Jones AS, Haqqani MT. Angioleiomyoma of the nasal cavity – report of a case and review of the literature. J Laryngol Otol 1994;108: 244-6.
3. Batsakis JG. Tumors of the Head and Neck: Clinical and pathological con-siderations. 2nded. Baltimore: Williams & Wilkins; 1979. p.354-6.
4. Shout AP. Solitary cutaneous and subcutaneous Leiomyoma. Am J Cancer 1937;29:435-69.
5. Duhig JT, Ayer JP. Vascular Leiomyoma: a study of sixty-one cases. Arch Dermatol 1959;79:32-41.
6. Magner D, Hill DP. Encapsulated Angiomyoma of the skin and subcuta-neous tissue. Am J Clin Pathol 1961;35:137-41.
7. Hachisuga T, Hashimoto H, Enjoji M. Angioleiomyoma: a clinical reapp-raisal of 562 cases. Cancer 1984;54:126-30.
8. Morimoto N. Angiomyoma (vascular leiomyoma): a clinico-pathologic study. Med J Kagoshima Univ 1973;24:663-83.
9. Maesaka A, Keyaki Y, Nakahashi T. Nasal angioleiomyoma and leiomyo-sarcoma: report of 2 cases. Otologia (Fukuoka) 1966;12:42-7. 10. Nall AV, Stringer SP, Baughman RA. Vascular Leiomyoma of the superior
turbinate: First reported case. Head and Neck 1997;19(1):63-7. 11. Di Tommaso L, Scarpellini F, Salvi F, Ragazzini T, Foschini MP.
Proges-terone receptor expression in orbital cavernous haemangioma. Virchow’s Arch 2000;436:284-8.
12. Marioni G, Marchese- Ragona R, Fernandez S, Bruzon J, Marino F, Staf-fieri A. Progesterone receptor expression in Angioleiomyoma of the nasal cavity. Acta Otolaryngol 2002;122:408-12.
13. Ragbeer MS, Stone J. Vascular Leiomyoma of the nasal cavity: report of a case and review of the literature. J Oral Maxillofac Surg 1990;48:1113-7. 14. McCaffrey TV, McDonald TJ, Unni KK. Leiomyoma of the nasal cavity.
Report of a case. J Laryngol Otol 1978;92: 817-9.
15. Hanna GS, Akosa AB, Ali MH. Vascular leiomyoma of the inferior turbi-nate- report of a case and a review of the literature. J Laryngol Otol 1988;102:1159-60.
16. Wolfowitz BL, Schmaman A. smooth muscle tumours of the upper respi-ratory tract. S Afr Med J 1973;47:1189-91.
17. Trott MS, Gewirtz A, Lavertu P, Wood BG, Sebek BA. Sinonasal leiom-yomas. Otolaryngol Head Neck Surg 1994;111:660-4.
18. Murono S, Ohmura T, Sugimori S, Furukawa M. Vascular Leiomyoma with abundant adipose cells of the nasal cavity. Am J Otolaryngol 1998;19:50-3. 19. Fu Y-S, Perzin KH. Nonepithelial tumours of the nasal cavity, paranasal si-nuses and nasopharynx: A clinicopathologic study: IV. Smooth muscle tu-mours (leiomyoma, leiomyosarcoma). Cancer 1975;35:1300-8. 20. LijovetzkyG, Zaarura S, Gay I. Leiomyoma of the nasal cavity: Report of
a case. J Laryngol Otol 1985;99:197-200.
21. Chiantelli A, Papini M, Bocci N, Ghilardi PL, Casani A, Cagno MC. Le-iomyoma of the nose. A case report. Acta Otorhinolaryngol Belg 1989; 43:339-42.
22. Nam HK, Kaufman MW, Wolff AP. Pathologic quiz case 2. Leiomyoma of the nasal vestibule. Arch Otolaryngol Head Neck Surg 1989;115:244-5, 247. 23. Sawada Y. Angioleiomyoma of the nasal cavity. J Oral Maxillofac Surg
1990;48:1100-1.
24. Schwartzman J. Leiomyoangioma of paranasal sinuses: Case report. Laryn-goscope 1973;83:1856-8.
25. Zijlker TD, Visser R. A vascular Leiomyoma of the ethmoid. Report of a case. Rhinology 1989;27:129-35.
26. Harcourt JP, Gallimore AP. Leiomyoma of the paranasal sinuses. J Laryn-gol Otol 1993;107:740-1.
27. LaBruna A, Reagan B, Papageorge A. Leiomyoma of the maxillary sinus. A diagnostic dilemma. Otolaryngol Head Neck Surg 1995;112:595-8. 28. Barr GD, More TAR, McCallum HM. Leiomyoma of the nasal septum. J
Laryngol Otol 1990;104:891-3.
