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Osteochondroma of scapula complicated long-term cough in an adult female.

Byline: Kun-Chi. Wu, Chung-Hua. Chu, Dah-Ching. Ding

Cough caused by scapular osteochondroma has never been reported. This study reports a case of long-term coughing caused by osteochondroma growing over the right scapula. A 44-year-old Asian female presented complaining a persistent cough for 2 months. Physical examination revealed no specific abnormality and clear and symmetrical breathing. A lesion revealed on X-rays, and computer tomography was diagnosed as an osteochondroma of the medial border of the scapula and the tumor caused the rib cage deformity. Following a preoperative examination, the patient underwent open surgery for lesion removal. Histological examination confirmed the clinical diagnosis of osteochondroma. Clinical examination 3 months later demonstrated a healed wound without cough and no recurrence was noted. Cough caused by the mass effect of osteochondroma is rare. The pathophysiology of nonproductive cough might cause by enlarging tumor in the lung field and caused a small part of alveoli atelectasis. Despite its rarity, osteochondroma should be considered in the differential diagnosis of any adult presenting with chronic coughing.

INTRODUCTION

Osteochondroma is the most common benign tumor of the cartilage and bone. [sup][1],[2] Osteochondroma is a cartilage-capped outgrowth on the external surface of bone. [sup][3] Osteochondroma usually grows from childhood to adolescence, and growth ceases once bone maturity is reached, and the epiphyseal plates close. Osteochondroma occurs in 3% of the general population, and in 85-90% of cases is solitary (nonhereditary) while in 10-15% of cases it is multiple (hereditary, autosomal dominant). [sup][4] Solitary osteochondroma tends to locate over the metaphysis of the long bone, such as the femur and humerus, but has also been reported over the flat bone, like the ventral surface of scapular. [sup][5],[6] Some investigations have described how scapular osteochondroma results in "winged scapular," but mass effect caused by scapula osteochondroma was never reported. [sup][7],[8],[9] Here, we report a rare case with scapular osteochondroma complicated with coughing.

Case Report

A 44-year-old Asian female complained of long-term nonproductive cough lasting more than 2 years and became more severe in recent 2 months. She denied chest pain, back pain, fever, rhinorrhea, sore throat, heartburn, and shortness of breath or other symptoms. She also denied being a smoker. She did not receive any pharmacological or physical therapy. No trauma or allergy was noted, and nor was there any relevant family history.

Physical examination identified no specific abnormality together with clear and symmetric breathing. Chest X-ray revealed no obvious lung lesion, through a large radiodense mass was noted over the right scapular area [Figure 1]a and b. The tumor was compressed the rib cage and caused rib cage deformity [Figure 1]b. Whole body bone scan [Figure 1]c and chest computer tomography (CT), [Figure 1]d identified a focal area with increased technetium-99m diphosphonate scintigram uptake over the right scapula compatible with right scapular mass and a large cauliflower-like osteosclerotic lesion exophytic from the inner area of the right scapular body. The transverse view of chest CT also revealed the tumor located closely to the rib cage [Figure 1]e. Surgical intervention was planned based on the preoperative diagnosis of right scapular osteochondroma.{Figure 1}

An open surgery was performed under general anesthesia. The incision was made along the medial border of the scapula. The dissection of the trapezius and dentatus muscles exposed the lesion. The tumor compressed the rib cage was noted. The lesion was then totally excised from the healthy scapular base, removing the entire cartilaginous cap. An approximately, 7 cm x 6 cm x 5 cm bony mass was excused from over the medial aspect of the right scapula [Figure 2]a. The histological investigation confirmed the clinical diagnosis of osteochondroma [Figure 2]b.{Figure 2}

The patient was discharged 4 days postsurgery. The patient was prescribed oral analgesic with aceclofenac 100 mg twice daily with an arm sling for 2 weeks. A follow-up in the outpatient department was arranged 3 months later and found a healed wound, no cough or local pain, and no recurrence.

DISCUSSION

Osteochondroma is most common benign skeletal tumor, [sup][1],[2] and occurs in 3% of the general population, [sup][3] involves the scapular in 3-4% of cases (mostly occurring on the long bone epiphysis of extremities), and is indicated in 14.4% of all scapular tumors. [sup][8] Ventral surface involvement was most common in all scapular osteochondroma. [sup][5],[10]

The etiologies of long-term cough (lasting more than 8 weeks [sup][11] ) most commonly include asthma induced by allergens, chronic upper airway cough syndrome (postnasal drip), gastroesophageal reflux disease, chronic obstructive pulmonary disease, and even pulmonary tuberculosis. [sup][12],[13] Nevertheless, cough caused by the mass effect of osteochondroma is rare. The pathophysiology of nonproductive cough might cause by enlarging tumor in the lung field and caused a small part of alveoli atelectasis.

Osteochondroma usually forms from childhood or adolescent and grows slowly. Osteochondroma is an outgrowth of the growth plate that consists of bone and cartilage, resulting in a cartilaginous cap with enchondral bone. [sup][1],[2],[4] If the cartilaginous cap is more than 2 cm thick, malignant transformation should be highly suspected. Malignant change is rare being reported in just 1% of cases and is combined with severe pain. [sup][2] In addition, the growth of osteochondroma in adulthood should be considered malignant as in the present case. However, the mass of the patient in this study was located incidentally after chronic cough had developed for 2 years. The patient did not notice the lesion during maturation or even into middle age.

Osteochondroma is usually painless, but the most common complaints in cases of scapular osteochonodroma resulting from mass effect include pain, limited range of motion, impingement mechanism, nerve compression, and friction-related bursitis [sup][14],[15] with swing scapular even being described in some case reports. [sup][7],[8],[9] However, chronic cough owing to osteochondroma has not been seen in previous studies. We hypothesize that the possible mechanism of cough involved a mass over the ventral side of the scapula compressing and irritating the rib, thus indirectly resulting in limited expansion of the lung, accompanied by a chronic cough.

A plain radiograph is the main diagnostic method, and is usually diagnosed incidentally. [sup][4] CT scan provides the anatomic characteristic of the lesion and a surgical plan. Magnetic resonance imaging can clarify cartilage growth on the bone surface more easily to rule out malignancy. [sup][16] Most osteochondroma do not need surgery, but in the trunk or in adulthood even symptom-free masses require surgery. The recommended choice of treatment is an open surgery, with no postoperative complications if the glenohumeral joint is reserved. [sup][5],[6],[15] Tumor relapse is not common, except where resected tumor margin is involved. [sup][6]

CONCLUSIONS

This case study outlines a rare presentation of chronic cough resulting from osteochondroma growing over the scapula. The case report provides an important differential diagnosis of long-term cough in adulthood.

ACKNOWLEDGMENT

Ted Knoy is appreciated for his editorial assistance.

References

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5. Frost NL, Parada SA, Manoso MW, Arrington E, Benfanti P. Scapular osteochondromas treated with surgical excision. Orthopedics 2010;33:804.

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8. Esenkaya I. Pseudowinging of the scapula due to subscapular osteochondroma. Orthopedics 2005; 28:171-2.

9. Orth P, Anagnostakos K, Fritsch E, Kohn D, Madry H. Static winging of the scapula caused by osteochondroma in adults: A case series. J Med Case Rep 2012;6:363.

10. Kwon OS, Kelly JI. Delayed presentation of osteochondroma on the ventral surface of the scapula. Int J Shoulder Surg 2012;6:61-3.

11. Vertigan AE, Theodoros DG, Gibson PG, Winkworth AL. Review series: Chronic cough: Behaviour modification therapies for chronic cough. Chron Respir Dis 2007; 4:89-97.

12. Irwin RS, Madison JM. The diagnosis and treatment of cough. N Engl J Med 2000;343:1715-21.

13. Irwin RS, Baumann MH, Bolser DC, Boulet LP, Braman SS, Brightling CE, et al. Diagnosis and management of cough executive summary: ACCP evidence-based clinical practice guidelines. Chest 2006;129 (1 Suppl):1S-23.

14. Krieg JC, Buckwalter JA, Peterson KK, el-Khoury GY, Robinson RA. Extensive growth of an osteochondroma in a skeletally mature patient. A case report. J Bone Joint Surg Am 1995;77:269-73.

15. Fageir MM, Edwards MR, Addison AK. The surgical management of osteochondroma on the ventral surface of the scapula. J Pediatr Orthop B 2009;18:304-7.

16. Mohsen MS, Moosa NK, Kumar P. Osteochondroma of the scapula associated with winging and large bursa formation. Med Princ Pract 2006;15:387-90.
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Title Annotation:Case Report
Author:Wu, Kun-Chi; Chu, Chung-Hua; Ding, Dah-Ching
Publication:Journal of Medical Sciences
Article Type:Case study
Date:Jul 1, 2015
Words:1573
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