(Chest. 2002;121:291-292.)
© 2002
American College of Chest Physicians
Varicosities of the Valleculae*
An Unusual Cause of Hemoptysis?
Richard Booton, MRCP and
Badie K. Jacob, MD, FCCP
*
From the Department of Respiratory Services, Bradford Royal Infirmary, Bradford Hospitals NHS Trust, Bradford, West Yorkshire, UK.
Correspondence to: Badie K. Jacob, MD, FCCP, Consultant Chest Physician and Chief of Respiratory Services, Bradford Royal Infirmary, Bradford Hospitals NHS Trust, Duckworth Lane, Bradford, West Yorkshire BD9 6RJ, UK
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Abstract
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Hemoptysis is a common respiratory symptom causing a great deal of
anxiety. The cause is often apparent following a clinical history,
upper-airway examination, bronchoscopy, and CT scanning of the thorax.
We present a case of massive hemoptysis, the etiology of which was not
readily apparent despite this conventional approach. Vallecular
hemorrhage has been previously reported but is usually minor unless
associated with surgical trauma, and can be readily missed if not aware
of the possibility. We speculate about the etiology and mechanism for
recurrent hemorrhage.
Key Words: hemoptysis • valleculae • varicosities
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Introduction
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Severe
or life-threatening hemoptysis requires a thorough and timely
evaluation. The presence of concomitant chest disease may delay this
evaluation, compromising the abilities to deliver definitive treatment,
and to reduce morbidity and mortality in patients who rebleed. In
addition, unrecognized or rare causes of massive hemoptysis may have a
similar effect. We review a case of an elderly man who presented with
substantial hemoptysis without an apparent source despite conventional
management. Delays in diagnosis in this setting not only cause
considerable patient anxiety and morbidity but should also prompt us to
consider rare conditions.
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Case Report
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A 72-year-old man with COPD presented in July 1998 with a short
history of cough and dyspnea succeeded by frank hemoptysis and blood
clots. There was no history of weight loss or night sweats. He was a
current smoker of two ounces of tobacco per week for 60 years. Clinical
examination confirmed airflow limitation without evidence of clubbing
or lymphadenopathy or additional respiratory signs. Arterial oxygen
saturation on room air was 91%. Initial hematologic, biochemical, and
clotting profile findings were normal. Chest radiography demonstrated
overinflation, prominent pulmonary arteries, and patchy inflammatory
change in the right upper zone. Sputum culture findings demonstrated a
scanty growth of Haemophilus influenzae, with smear and
culture negativity for acid-fast bacilli (AFB) on three samples. No
evidence of malignancy was seen on cytologic examination.
Flexible fiberoptic bronchoscopy was undertaken, which demonstrated no
evidence of active bleeding and no mucosal lesion. A subsequent CT scan
of the thorax noted consolidation in the right upper lobe but without
evidence of bronchiectasis. BAL fluid was smear-negative for AFB. His
hemoptysis resolved after 1 week, and he was subsequently discharged.
At review 1 month later, no further hemoptysis had been encountered.
Lavage fluid revealed AFB after 3 weeks of culture, and he was
prescribed rifampicin, isoniazid, and pyrazinamide. Over the next 2
weeks, he was admitted to the hospital as an emergency patient on two
occasions with further, profuse, free-flowing hemoptysis (2 g/dL
reduction in hemoglobin); on both occasions, coagulation and
biochemical parameters were otherwise normal. An ear, nose, and throat
review revealed no active bleeding or significant pathology on
examination of the postnasal space or indirect laryngoscopy. He
underwent further bronchoscopic examinations (on each hospital
readmission); the findings of both were normal. However, on retracting
the bronchoscope on his last examination, a mucosal lesion was seen in
the oropharynx at the root of the tongue. A subsequent examination
under anesthesia of the pharynx and larynx demonstrated a normal
postnasal space and larynx. Valleculae varicosities were seen
bilaterally at the base of the tongue, and diathermy was performed
successfully. Since this time, there have been no further episodes of
hemoptysis. His lavage cultures identified Mycobacterium
avium-intracellulare resistant to all initiated agents, and his
antituberculous chemotherapy was discontinued. He remains well with
resolution of the right upper lobe consolidation and no CT evidence on
follow-up of bronchiectasis.
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Discussion
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Hemoptysis is one of the most alarming symptoms frequently
heralding the recognition of serious disease. It generally relates to
blood originating distal to the vocal cords, but in some instances
hemorrhage arising from the pharynx will cause diagnostic confusion.
Most cases can be identified by a combination of bronchoscopy and CT
scanning of the thorax.1
In a retrospective analysis of
208 patients presenting with hemoptysis over a 15-year period,
bronchiectasis (20%), lung cancer (19%), bronchitis (18%), and
pneumonia (16%) were responsible for the majority of episodes of
hemoptysis.1
The severity of hemoptysis in our patient was
not in keeping with an infectious etiology and evidence of an
underlying structural abnormality was lacking. Only one specimen of
M avium-intracellulare was cultured. Subsequent
successive negative culture findings (despite resistance to all drugs
used) suggest environmental contamination and clinical irrelevance. No
fresh blood was seen in the region of the valleculae during endoscopic
examination, though this was usually performed 2 to 3 days following
the cessation of bleeding. The extensive negative search for a bleeding
source coupled with its cessation following diathermy is suggestive
that the origin of bleeding was from these varicosities.
The valleculae are a little known, but definite, anatomic entity of the
oropharynx, representing pockets lying bilaterally between the
epiglottis and tongue and formally referred to as the valleculae
epiglottica. To our knowledge, there is only one previous
report2
in the literature of spontaneous hemorrhage from
the root of the tongue; reports of massive hemorrhage from the
valleculae have so far been secondary to surgical trauma.3
Wetherill and Ganghi,2
as in this case, reported abnormal
and tortuous vessels in a patient with an infective exacerbation of
bronchitis.
The published literature provides sparse insight into the etiology of
oropharyngeal varicosities. Increasing age is regarded as an important
factor and most are commonly sublingual; varicosities of the lip and
buccal mucosa are seen less frequently.4
Vallecular
anomalies are not listed as a prominent area for oropharyngeal
abnormality, although one may speculate that this relates to the
inaccessibility of the region to easy inspection.
The venous drainage of the pharynx and larynx may explain the origins
of oropharyngeal varicosities, particularly in patients with chronic
chest disease. A venous plexus eventually tributary to either the
internal jugular or brachiocephalic veins drains the pharyngeal and
laryngeal structures. Chronic elevations of right-heart pressure may
predispose to variceal formation. Factors influencing variceal size and
wall tension may be responsible for episodes of acute hemorrhage,
perhaps hypoxia. It is well recognized, in esophageal varices, that
portal pressure reflects intravariceal pressure,5
and that
the likelihood of hemorrhage relates to four factors: (1) pressure
within the varix, (2) tension on the variceal wall, (3) variceal size,
and (4) severity of liver disease.6
It is evident that the origin of large-volume hemoptysis is not always
readily apparent even after extensive investigations. In this patient,
no gross structural lung damage existed, confirmed by high-resolution
CT scanning and repeated direct visualization of the endobronchial
anatomy with a flexible fiberoptic scope. Consequently, and with the
aid of time, we are confident that the varices seen within the
oropharynx represent the sole reason for his hemoptysis, perhaps
supported by its cessation following variceal diathermy. We highlight
the valleculae as a region worthy of thorough inspection in order that
considerable morbidity can be avoided.
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Footnotes
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Abbreviation:
AFB = acid-fast bacilli
Received for publication January 25, 2001.
Accepted for publication June 19, 2001.
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References
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Hirshberg, B, Biran, I, Glazer, M, et al (1997) Hemoptysis: etiology, evaluation, and outcome in a tertiary referral hospital. Chest 112,440-444[Abstract/Free Full Text]
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Wetherill, JH, Ganghi, AG (1967) Haemorrhage from base of tongue. BMJ 3,784
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Premachandra, DJ, Prinsley, PR, McRae, D (1991) Massive hemorrhage from the vallecula: a diagnostic difficulty; case report. Eur J Surg 157,297-298[Medline]
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Neville, BW, Damm, DD, Allen, MC, et al (1995) Oral and maxillofacial pathology 1st ed. ,13-14 W.B. Saunders Philadelphia, PA.
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Dawson, J, Gertsch, P, Mosimann, F (1985) Endoscopic variceal pressure measurements: response to isosorbide dinitrate. Gut 26,843-847[Abstract/Free Full Text]
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Jalan, R, Hayes, PC (2000) UK guidelines on the management of variceal hemorrhage in cirrhotic patients. Gut 46(suppl),iii2-iii3
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Abstract
Introduction
