Pulmonary hamartoma is the most common benign tumor of the lung. We report  the case of a healthy, 46-year-old woman with an incidentally discovered  chondromatous hamartoma, which has been excised thorascopically. She  developed hamartoma within a year

Dubajić Snezana

Gatarić Zorica

Jaković Radoslav

Lazović Biljana

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37Case reportswww�onk�ns�ac�rs/Archive	 Vol	19,	No�	1–2,	July	2011Pulmonary hamartoma - case report and review of literatureBiljana	Lazović¹,	Radoslav	Jaković²,	Snezana	Dubajić¹,	Zorica	Gatarić¹sUMMARyPulmonary hamartoma is the most common benign tumor of the lung. We report the case of a healthy, 46-year-old woman with an incidentally discovered chondromatous hamartoma, which has been excised thorascopically. She developed hamartoma within a year.Key worsd: Hamartoma; Lung NeoplasmsINTRODUCTION Pulmonary	 hamartomas	 (PH)	 also	 known	 as	 mesenchymomas,	 can	 be	parenchymal	(80%)	or	endobronchial	(10–20%)�	Parenchymal	lesions	are	usually	an	incidental	finding	and	range	in	size	from	1	cm	to	8	cm	in	various	series�	The	endobronchial	tumors	usually	present	with	new	onset	of	respira-tory	symptoms,	which	are	most	commonly	recurrent	chest	infections	or	hemoptysis�	Pulmonary	hamartoma	are	the	most	common	form	of	benign	lung	tumors	with	an	incidence	of	between	0�025%	–	0�32%	according	to	different	necropsy	studies	(1-3)�	We	report	a	case	of	accidentally	found	pul-monary	hamartoma	in	previously	healthy	woman	who	was	admitted	to	emer-gency	room	because	of	strong	pain	in	upper	back	on	the	day	of	admission�CAse RePORT A	46-yers-old	female,	a	worker	by	occupation	and	a	chronic	smoker,	presented	in	emergency	room	with	a	strong	pain	in	upper	back�	She	felt	it suddenly	in	working	place�	On	admission,	physical	examination	and	laboratory	 investigations	 were	 normal�	 Per	 anamnesis,	 obtained	 data	indicated	a	history	of	chronic	gastritis�	She	was	an	average	built,	denied	any	significant	loss	in	weight	and	fever�	Plain	chest	radiogram	showed	a	solitary	nodular	opacity	of	1�8�x1�8	cm	in	diameter	(Figure	1)�               Figure 1. Chest X-ray and CT evaluation showed a solitary nodule in left upper lobe  Figure 1. Chest X-ray and CT evaluation showed a solitary nodule in left upper lobeBecause	of	respiratory	infection	a	year	before,	patient	had	chest	X	ray	and	the	finding	was	normal�	To	find	out	the	origin	of	pulmonary	change	the	 patient	 was	 examined	 by	 abdominal	 ultrasonography,	 computed	tomography	of	chest	and	abdomen,	gastroscopy,	colonoscopy,	breast	ultrasonography,	bronchoscopy,	and	gynecology�	During	clinical	evalua-tion,	pain	in	upper	back	disappeared	after	application	of	NSAID�Gastroscopy	 confirmed	 the	 presence	 of	 chronic	 gastritis�	 Breast	 ultra-sonography	revealed	hypoehogenic	small	nodule	in	left	upper	quadrant,	8mm	in	diameter�	The	results	of	mammography	excluded	of	any	sus-pected	neoplasm�	In	the	sample	obtained	by	bronchoaspiration,	we	found	staphylococcus	coagulase	negative	species	sensitive	to	cephalosporin,	penicillin,	and	tetracycline�	The	evaluation	of	cytological	analyses	showed	moderate	population	of	squamous	cell	layered	epithelium,	mostly	single	and	without	cell	nuclear	atypia,	but	still	present	in	smaller	individual	small	groups	with	enlarged	nuclei,	and	mild	to	moderate	anisonucleosis-	the	finding	suspected	of	malignancy�	Patient	was	sent	to	the	thoracic	surgery	for	 further	 diagnostic	 procedures�	 Thorascopic	 excision	 of	 the	 sample	contained	islands	of	cartilage,	fat,	fibromyxoid	stroma,	and	narrow	spaces	lined	by	respiratory	epithelium�	Histopathological	finding	of	the	specimen	confirmed	the	presence	of	chondroid	hamartoma�	After	the	procedure,	patient	was	feeling	well�	She	was	advised	for	regular	checkups�	 DIsCUssIONPulmonary	hamartoma	was	first	described	by	Albrecht	in	1904�	Pulmonary	hamartoma	was	thought	to	arise	from	embryologic	rests	that	were	present	in	fetal	life	but	generally	did	not	become	visible	until	adulthood�	However,	a	cytogenetic	analyses	of	the	pulmonary	hamartomas	show	an	abnormal	karyotype	and	reveal	recombinations	between	chromosomal	bands	6p21	and	14q24	(4),	thus	supporting	the	opinion	that	a	hamartoma	of	the	lung	is	a	true	neoplasm�	However,	the	nosogenesis	of	PH	remains	unclear�Pulmonary	hamartomas	are	usually	found	in	adults	with	a	peak	incidence	in	the	sixth	decade�	There	is	a	male	preponderance,	the	male:female	ratio	being	2:1	to	3:1�	Pulmonary	hamartomas	are	often	asymptomatic	and	they	are	typically	discovered	as	an	incidental	coin	lesion	on	a	routine	chest	radiograph�	Radiologically,	hamartomas	account	for	7%	to	14%	of	pulmonary	coin	lesions	(5)�	PH	can	occur	in	all	parts	of	lung,	but	most	often,	they	are	found	in	the	periphery	and	rarely	near	the	hilar	parts�	On	chest	radiographs,	pulmonary	hamartomas	characteristically	appear	as	well-defined,	solitary	pulmonary	nodules�	They	may	show	varying	pat-terns	of	calcification,	including	an	irregular	popcorn,	stippled,	or	curvilin-ear	pattern,	or	even	a	combination	of	all	3	patterns�	When	calcification	or	fat	is	detected	in	a	well-circumscribed	peripheral	lung	tumor,	a	diagnosis	of	 hamartoma	 can	 confidently	 be	 made�	 However,	 the	 characteristic	calcification	is	seen	in	only	approximately	15%	of	patients�	Calcification	in	a	pulmonary	nodule	on	imaging	indicates	a	high	probability	that	the	lesion	is	benign�	However,	not	all	calcified	pulmonary	nodules	are	benign	and	the	differential	diagnosis	includes	a	primary	central	lung	carcinoid,	metastasis,	and	a	primary	bronchogenic	carcinoma	(6)�Serial	 chest	 PHs	 radiographs	 may	 demonstrate	 slow	 growth�	 Rapid	growth	has	rarely	been	reported�	This	feature	may	make	the	differentiation	of	a	hamartoma	from	a	bronchogenic	carcinoma	difficult	(7)�Arch	Oncol	2011;19(1-2):37-8�UDC:	616�24-006�03:615�07	DOI:	10�2298/AOO1102037L	1Department	of	pulmology,	Clinical	Hospital	Center	Zemun,	Belgrade,	Serbia,	2Thoracic	Surgery,	Clinical	Centre	of	Serbia,	Belgrade,	SerbiaCorrespondence	to:	Biljana	Lazović,	Milutina	Milankovića	122,	11070	Belgrade,	Serbialazovic.biljana@gmail.com Received:	26�09�2010	Provisionally	accepted:	08�10�2010		Accepted:	28�10�2010	© 2011, Oncology Institute of Vojvodina, Sremska Kamenica38Case reportswww�onk�ns�ac�rs/Archive	 Vol	19,	No�	1–2,	July	2011Today,	even	with	the	advancement	in	medical	therapy,	pulmonary	resec-tion	remains	the	most	important	treat	measure	of	patients	with	pulmonary	hamartoma�	Controversy,	however,	still	exists	about	the	indication	and	timing	of	surgery	(8)�	Since	most	pulmonary	hamartomas	s	are	nonex-panding	or	slowly	growing	neoplasms,	some	authors	believe	that	surgery	is	necessary	only	when	expansion	is	recorded	in	young	or	middle	aged	patients	or	accompanying	obvious	pulmonary	symptoms�	According	to	Gui	and	associates	(9),	the	pulmonary	hamartoma	has	the	tendency	of	expansion	or	recurrence,	and	chronic	inflammatory	stimulation	of	local	position	may	contribute	to	the	development	of	malignancy�	Therefore,	when	a	solitary	pulmonary	lesion	is	more	than	2�5	cm	or	the	possibility	of	malignancy	cannot	be	excluded,	surgical	resection	is	deserved	to	be	performed	and	should	be	mandatory�	Our	opinion	is	that	pulmonary	hamartoma	is	indicated	for	surgery	in	case	of:	(1)	a	solitary	pulmonary	lesion	with	diameter	above	2�5	cm;	(2)	it	is	an	overweight	psychic	burden	for	a	patient;	(3)	having	the	tendency	of	expansion	or	recurrence;	(4)	pulmonary	symptoms	unresponsive	to	drug	treatment;	and	(5)	the	lesion	cannot	be	differentiated	from	malignancy�In	case	of	our	patient	initial	diagnosis	of	metastatic	disease	was	indicated	by	radiological	finding	of	solitary	lung	nodule,	which	rapidly	developed	only	one	year	after	the	normal	X-ray	finding�	As	bronchoaspiration	find-ings	referred	to	malignancy,	surgical	treatment	was	the	only	solution	to	establish	diagnose�	The	case	of	our	patient	implies	that	hamartoma	can	be	expressed	in	very	short	time,	within	a	year�Conflict of interestWe	declare	no	conflicts	of	interest�ReFeReNCes  1	 Salminen	US�	Pulmonary	hamartoma�	A	clinical	study	of	77	cases	in	a	21-year	period	and	review	of	literature�	Eur J Cardiothorac Surg�	1990;4(1):15-8�  2	 Cosio	BG,	Villena	V,	Echave-Sustaeta	J,	de	Miguel	E,	Alfaro	J,	Hernandez	L,	et	al�	Endobronchial	hamartoma� Chest� 2002;122(1):202-5�  3	 Hansen	CP ,	Holtveg	H,	Francis	D�	Pulmonary	hamartoma�	J Thorac Cardiovasc Surg�	1992;104:676-8  4	 Johansson	M,	Dietrich	C,	Mandahl	N,	Hambraeus	G,	Johansson	L,	Clausen	PP ,	et	al�	Recombinations	of	chromosomal	bands	6p21	and	14q24	characterize	pulmonary	hamartomas�	Br J Cancer. 1993,	67:1236-41�  5	 Wiatrowska	BA,	Yazdi	HM,	Matzinger	FR,	MacDonald	LL�	Fine	needle	aspiration	biopsy	of	pulmonary	hamartomas�	Radiologic,	cytologic	and	immunocytochemical	study	of	15	cases�	Acta Cytol�	1995;39:1167-74�  6	 Grewal	RG,	Austin	JH�	CT	demonstration	of	calcification	in	carcinoma	of	the	lung�	J Comput Assist Tomogr�	1994;18:867-71�  7	 Murray	J,	Kielkowski	D,	Leiman	G�	The	prevalence	and	age	distribution	of	peripheral	pulmonary	hamartomas	in	adult	males:	an	autopsy-based	study�	S Afr Med J�	1991;79:247-9�  8	 Lien	YC,	Hsu	HS,	Li	WY,	Wu	YC,	Hsu	WH,	Wang	LS,	et	al�	Pulmonary	hamartoma�	J Chin Med Assoc�	2004;67:21-6�  9	 Guo	W,	Zhao	YP ,	Jiang	YG,	Wang	RW,	Ma	Z�	Surgical	treatment	and	outcome	of	pulmonary	hamartoma:	a	retrospective	study	of	20-year	experience�	J Exp Clin Can Res�	2008,27:8�

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Pulmonary hamartoma: Case report and review of literature

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Pulmonary hamartoma: Case report and review of literature

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