A 44-year-old man presented with painful swelling of wrists and ankles, severe pain at both tibiae, clubbing of fingers and toes and arthritis in wrist and ankle joints. The chest roentgenogram showed consolidation of the right lower lobe, whereas plain roentgenograms revealed solid periosteal reaction at both tibiae. CT and bronchoscopy confirmed the presence of adenocarcinoma of the right lower lobe. Moreover, mycobacterium of tuberculosis was isolated by culture of the patient's sputum

A Watanabe

Alexandra Adamidou

David Armstrong

Dimitrios Karamitsos

George Ntaios

M Marinez-Levin

Murat Karkuca

NA Dacosta

S Cicėnas

X Vandemergel

English

PubMed

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BioMed CentralCases Journal
ssOpen AcceCase Report
Hypertrophic pulmonary osteoarthropathy secondary to bronchial 
adenocarcinoma and coexisting pulmonary tuberculosis: a case 
report
George Ntaios*, Alexandra Adamidou and Dimitrios Karamitsos
Address: First Propedeutic Department of Internal Medicine, AHEPA Hospital, Aristotle University, Thessaloniki, Greece
Email: George Ntaios* - ntaiosgeorge@yahoo.gr; Alexandra Adamidou - aadam@yahoo.gr; Dimitrios Karamitsos - dkar@yahoo.gr
* Corresponding author    
Abstract
A 44-year-old man presented with painful swelling of wrists and ankles, severe pain at both tibiae,
clubbing of fingers and toes and arthritis in wrist and ankle joints. The chest roentgenogram showed
consolidation of the right lower lobe, whereas plain roentgenograms revealed solid periosteal
reaction at both tibiae. CT and bronchoscopy confirmed the presence of adenocarcinoma of the
right lower lobe. Moreover, mycobacterium of tuberculosis was isolated by culture of the patient's
sputum.
Our patient received antituberculous treatment and soon he underwent surgical excision of the
tumour and subsequent chemotherapy. Ten months later, he returned with metastatic lesions in
the brain and the adrenals. A few days later, he died.
The patient suffered from bronchial adenocarcinoma as well as pulmonary tuberculosis. As a
complication of these two coexisting conditions, the patient developed hypertrophic pulmonary
osteoarthropathy.
Introduction
Hypertrophic pulmonary osteoarthropathy (HPOA) or
else Pierre-Marie-Bamberger syndrome, constitutes a clin-
ical syndrome characterized by clubbing of fingers and
toes, arthritis and periostosis of the long distal bones. The
lesions are usually bilateral and symmetric causing swell-
ing and extreme pain of the affected limbs. It may occur as
a primary condition, which is familial and affects mainly
males [1]. However, in most instances, it occurs secondar-
ily to conditions characterized by arteriovenous shunt like
lung carcinoma, mesothelioma, pulmonary tuberculosis,
congenital cyanotic heart disease, hepatic and colorectal
carcinoma, inflammatory bowel disease, cirrhosis, pul-
monary fibrosis and empyema [2,3]. The coexistence of
two different conditions which both lead to hypertrophic
osteoarthropathy is very rare. In this case report, we
present the case of a 44-year-old man who developed
hypertrophic osteoarthropathy due to bronchial carci-
noma and coexisting pulmonary tuberculosis.
Case report
A 44-year-old man was admitted in our clinic due to high
fever and painful swelling of wrists and ankles for four
months prior to admission. He also complained of severe
pain at his tibiae. He was a heavy smoker (50 pack-years)
and he denied any chronic illness. At admission, clinical
examination revealed a severely ill patient who was una-
ble to walk due to extreme leg pain. He was febrile (39°C)
Published: 7 October 2008
Cases Journal 2008, 1:221 doi:10.1186/1757-1626-1-221
Received: 18 August 2008
Accepted: 7 October 2008
This article is available from: http://www.casesjournal.com/content/1/1/221
© 2008 Ntaios et al; licensee BioMed Central Ltd. 
This is an Open Access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/2.0), 
which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.Page 1 of 3
(page number not for citation purposes)
Cases Journal 2008, 1:221 http://www.casesjournal.com/content/1/1/221and arthritis of wrists and ankles could be easily noticed.
Moreover, clubbing of the fingers was evident (figure 1).
Erythrocyte sedimentation rate and C-reactive protein
were significantly increased (65 mm/hr and 8.6 mg/dl
respectively) but leukocyte count was within normal val-
ues. The chest roentgenogram revealed consolidation of
the right lower lobe. He was initially treated with broad
spectrum antibiotics for three days without any clinical
improvement. Meanwhile, computer tomography of the
chest was performed, which revealed a solitary mass in the
right lower lobe accompanied by a single mediastinal
lymph node. There was no pleural effusion. Soon, bron-
choscopy was performed which confirmed the presence of
adenocarcinoma of the right lower lobe. In the meantime,
mycobacterium of tuberculosis was isolated by sputum
culture. Hence, our patient suffered from bronchial aden-
ocarcinoma as well as pulmonary tuberculosis. As a com-
plication of these two coexisting conditions, he developed
hypertrophic pulmonary osteoarthropathy. Indeed, plain
roentgenograms revealed a solid reaction of the perios-
teum of both tibiae. These findings were further con-
firmed by bone scintigraphy.
Our patient received antituberculous treatment and soon
he underwent surgical excision of the tumour and subse-
quent chemotherapy. Ten months later, he returned with
metastatic lesions of the brain and adrenals. A few days
later, he died.
Discussion
Coexistence of tuberculosis and lung cancer is fairly rare.
However, there are different estimates about the preva-
lence of this combination. Watanabe et al suggested that it
reaches 2.1% of patients with lung cancer [4]. This result
was further confirmed by Cicėnas and Vencevičius [5].
However, Dacosta and Kinare identified this combination
in 13.1% of patients with lung cancer [6].
Cicėnas and Vencevičius also indicated that epidermoid
carcinoma is the lung tumour which is mainly associated
with tuberculosis [5]. On the contrary, bronchial adeno-
carcinoma, which was the case in our patient, was diag-
nosed only in 21.7% of cases.
Although the association of HPOA with bronchial carci-
noma and pulmonary tuberculosis is well known, the
exact pathogenetic mechanism of the syndrome is still
unclear. It was suggested that in cases of lung diseases,
congenital cyanotic heart diseases and liver cirrhosis, meg-
akaryocytes bypass the pulmonary circulation through
arteriovenous shunting and escape fragmentation. Then,
these unfragmented megakaryocytes stimulate the pro-
duction of platelet derived growth factor (PDGF) and vas-
cular endothelial growth factor (VEGF) by endothelial
cells, resulting in angiogenesis and endothelial hyperpla-
sia which is manifested as clubbing of fingers and toes [7].
Summarizing, we present a rare case of HPOA due to coex-
istence of bronchial adenocarcinoma and pulmonary
tuberculosis. The presence of a readily recognised cause of
HPOA should not prevent the clinical doctor from exclud-
ing other potential coexisting pathologic conditions that
could also contribute to HPOA.
Consent
Written informed consent was obtained from the patient
for publication of this case report and any accompanying
images. A copy of the written consent is available for
review by the Editor-in-Chief of the <<Cases JournalŬ.
Competing interests
The authors declare that they have no competing interests.
Authors' contributions
GN, AA and DK were the treating physicians. GN drafted
the manuscript. All authors read and approved the final
manuscript.
References
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Hypertrophic pulmonary osteoarthropathy secondary to bronchial adenocarcinoma and coexisting pulmonary tuberculosis: a case report

Crossref

Decaux G: Review on hypertrophic osteoarthropathy and digital clubbing. Rev Med Brux

Erturk Engin, Capkin Erhan, Akyazi Hikmet, Ozden Gonca, Tosun Mehmet: Primary hypertrophic osteoarthropathy (pachydermoperiostosis): a case report. Rheumatol Int

Hypertrophic osteoarthropathy. In Rheumatology 3rd edition. Edited by: Hochberg

Krinare SG: Association of lung carcinoma and tuberculosis.

Vencevičius V: Lung cancer in patients with tuberculosis.

Wright Gary: Hypertrophic pulmonary osteoarthropathy (HPOA) (Pierre Marie-Bamberger syndrome): two cases presenting as acute inflammatory arthritis. Description and review of the literature. Rheumatol Int

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Hypertrophic pulmonary osteoarthropathy secondary to bronchial adenocarcinoma and coexisting pulmonary tuberculosis: a case report

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