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Journal of Pediatric Surgery (2009) 44, E21–E23

Endobronchial chondroid hamartoma in an infantVishesh Jain a, Prabhud Goel a, Dinesh Kumar b, Aashu Seith b, Chitra Sarkar c,Sushil Kabra d, Sandeep Agarwala a,⁎

aDepartment of Pediatric Surgery, All India Institute of Medical Sciences, New Delhi 110029, IndiabDepartment of Radiodiagnosis, All India Institute of Medical Sciences, New Delhi 110029, IndiacDepartment of Pathology, All India Institute of Medical Sciences, New Delhi 110029, IndiadDepartment of Pediatrics, All India Institute of Medical Sciences, New Delhi 110029, India

Received 26 March 2009; revised 9 June 2009; accepted 10 June 2009

0d

Key words:Endobronchialhamartoma;

Chondroid hamartoma;Infant

Abstract Endobronchial tumors in infants are uncommon. The clinical and radiologic findings andmanagement of a rare case of endobronchial chondroid hamartoma in an infant is presented along with areview of the literature.© 2009 Elsevier Inc. All rights reserved.

Hamartomas are the most common benign tumor of thelung in adults; however, they are occasionally reported inchildren as well. Most hamartomas are intraparenchymal,whereas endobronchial hamartomas are less common. Theoccurrence of an endobronchial hamartoma in an infant isvery rare. We describe the clinical presentation and manage-ment of this rare tumor in an infant.

1. Case report

A three-and-a-half-month-old boy presented with ahistory of repeated respiratory distress since birth. Theantenatal period was uneventful. The child was a full-terminfant delivered by normal abdominal delivery. The patientdeveloped respiratory distress soon after birth and requiredmechanical ventilatory support for 6 days. Thereafter, the

⁎ Corresponding author. Fax: +91 11 26588641.E-mail address: sandpagr@hotmail.com (S. Agarwala).

022-3468/$ – see front matter © 2009 Elsevier Inc. All rights reserved.oi:10.1016/j.jpedsurg.2009.06.011

child did well and was discharged on the 14th day of life.Soon after discharge, he developed tachypnea with moderatesubcostal retractions and was brought to our hospital. Onphysical examination, the trachea was central in position, butair entry was decreased on the right side. The chestradiograph revealed a hyperinflated right lung with nomediastinal shift (Fig. 1A). Contrast-enhanced chest com-puted tomographic scan showed a solid mass lesion withinthe right intermediate bronchus with a hyperinflated rightupper lobe and collapsed right lower lobe (Fig. 1B and C).The left bronchus was normal. Flexible bronchoscopyshowed a mass lesion in right intermediate bronchus.Cytologic examination of the bronchial aspirate wasinconclusive. Right thoracotomy was performed andrevealed collapsed middle and lower lobes with a hyperin-flated upper lobe. Bronchotomy of the right bronchusintermedius revealed the presence of a whitish firmintraluminal mass with a wide base, just distal to the rightupper lobe opening. Right middle and lower lobectomy wasperformed. The histopathologic examination of the lesionwas reported as a chondroid hamartoma (Fig. 1D). Thepatient had an uneventful recovery and was discharged on

Fig. 1 A, Chest radiograph showing hyperinflated lung with no mediastinal shift. B and C, Contrast-enhanced chest computed tomographicscan showing a solid mass lesion within the right intermediate bronchus with hyperinflated right upper lobe and collapsed right lower lobe. D,Photomicrographs of histologic section from the endobronchial mass showing disorganized lobules of cartilage, smooth muscle, and dilatedglands of bronchial type (H&E, ×100).

E22 V. Jain et al.

the eighth postoperative day. On follow-up examination, thechild remains asymptomatic.

2. Discussion

Primary pulmonary neoplasms are unusual in the pediatricage group, and nearly one fourth of these are benign [1].Inflammatory pseudotumor is the most common benigntumor in children followed by hamartoma. Endobronchialneoplasms make up a very small fraction of these benignneoplasm with bronchial adenoma being the most commonendobronchial tumor in children. Other benign endobron-chial neoplasms reported are hamartomas, hemangioma,papillomas, leiomyomas, mucous gland tumors, and inflam-matory pseudotumor [2].

The term hamartoma was coined in 1904 by Albrecht[3] to describe certain tumorlike malformations resultingfrom a presumed error in development of tissues normallypresent in the involved organ. In 1934, Goldsworthy [4]applied this term to benign lung tumors composed of acombination of adipose tissue and cartilage. Pulmonaryhamartomas can be parenchymal or endobronchial inlocation. Endobronchial hamartomas are extremely rare inchildren and have a low frequency in all the reported seriesin adults. In the largest review of pulmonary hamartomas in

adults (n = 215), only 1.4% of hamartomas had anendobronchial location; the remainder are located withinthe parenchyma [5]. Other adult series estimate theincidence of endobronchial hamartomas between 10% and20% of all lung hamartomas [6-8]. As compared toparenchymal hamartomas, most of the endobronchialhamartomas are symptomatic.

Our patient presented with lower respiratory tractinfection with atelectasis of the involved lobe. Atelectasisor hyperinflation results from complete or partial occlusionof the airway and may be because of a foreign body, mucousplug, mucosal web, cysts, or tumors. Because of the rarity ofendobronchial tumors, it is infrequently considered in thedifferential diagnosis. Treatment options are diverse. If thediagnosis of hamartoma is confirmed and the tumor isaccessible, endoscopy and excision of tumor using ND:YAGlaser, cryosurgery, electrosurgery snare, electrocoagulation,or forceps resection is preferred. If endoscopic resection isnot possible, then bronchotomy and excision of tumor,bronchoplasty or lobectomy, and pneumonectomy have beendone. A thorough review of literature revealed this to be theyoungest reported case of endobronchial hamartoma. Onlyone previous case report of endobronchial hamartoma in aninfant 8 months of age who underwent endoscopic resectionwas found [9]. Endobronchial hamartoma, although rare,should be considered in the differential diagnosis of anendobronchial mass in children.

E23Endobronchial chandroid hamartoma in an infant

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