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Tracheobronchomalacia due to amyloidosis in a patient with rheumatoid arthritis.

Turkstra F, Rinkel RN, Biermann H, van der Valk P, Voskuyl AE - Clin. Rheumatol. (2008)

Bottom Line: In this case report, we describe a patient with longstanding rheumatoid arthritis who developed tracheobronchomalacia with fatal outcome.Despite negative antemortem biopsies of abdominal fat and tongue, amyloid was found postmortem in the trachea and appeared to be associated with tracheobronchomalacia.

View Article: PubMed Central - PubMed

Affiliation: Department of Rheumatology, VU University Medical Center, Amsterdam, the Netherlands. f.turkstra@janvanbreemen.nl

ABSTRACT
In this case report, we describe a patient with longstanding rheumatoid arthritis who developed tracheobronchomalacia with fatal outcome. Despite negative antemortem biopsies of abdominal fat and tongue, amyloid was found postmortem in the trachea and appeared to be associated with tracheobronchomalacia.

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Amyloidosis of the trachea in Congo red
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Fig1: Amyloidosis of the trachea in Congo red

Mentions: The patient was a 69-year-old female with seropositive erosive RA, who had not been treated with disease-modifying antirheumatic drugs since 1984. In March 2005, she presented to another hospital because of progressive dyspnea of several hours, due to a bronchopneumonia. After 2 days, she developed a stridor. Otolaryngological examination results showed diffuse swelling of the tongue, pharynx and neck, and an extensive tracheobronchomalacia. Treatment consisted of intubation, antibiotics, diuretic therapy, and corticosteroids. She was admitted to our Intensive Care Unit because of progressive swelling and respiratory insufficiency due to tracheobronchomalacia. Physical examination results revealed extensive swelling and a remarkable protrusion of the tongue. Typical rheumatoid joint deformities and rheumatoid nodules were present but no active arthritis. Laboratory investigations revealed a normocytic anemia and a renal insufficiency. The measured creatinine clearance was 12 ml/min, with a proteinuria of 0.35 g/24 h. Computed tomography scan of thorax and neck provided no explanation for the tracheobronchomalacia. Clinical suspicion of amyloidosis was not confirmed by biopsies of abdominal fat tissue and of the tongue. Ultimately, the patient succumbed to multiple organ failure. Autopsy revealed AA amyloid in the trachea (Fig. 1), spleen, liver, and perivascularly in the kidneys.Fig. 1


Tracheobronchomalacia due to amyloidosis in a patient with rheumatoid arthritis.

Turkstra F, Rinkel RN, Biermann H, van der Valk P, Voskuyl AE - Clin. Rheumatol. (2008)

Amyloidosis of the trachea in Congo red
© Copyright Policy
Related In: Results  -  Collection

Show All Figures
getmorefigures.php?uid=PMC2367392&req=5

Fig1: Amyloidosis of the trachea in Congo red
Mentions: The patient was a 69-year-old female with seropositive erosive RA, who had not been treated with disease-modifying antirheumatic drugs since 1984. In March 2005, she presented to another hospital because of progressive dyspnea of several hours, due to a bronchopneumonia. After 2 days, she developed a stridor. Otolaryngological examination results showed diffuse swelling of the tongue, pharynx and neck, and an extensive tracheobronchomalacia. Treatment consisted of intubation, antibiotics, diuretic therapy, and corticosteroids. She was admitted to our Intensive Care Unit because of progressive swelling and respiratory insufficiency due to tracheobronchomalacia. Physical examination results revealed extensive swelling and a remarkable protrusion of the tongue. Typical rheumatoid joint deformities and rheumatoid nodules were present but no active arthritis. Laboratory investigations revealed a normocytic anemia and a renal insufficiency. The measured creatinine clearance was 12 ml/min, with a proteinuria of 0.35 g/24 h. Computed tomography scan of thorax and neck provided no explanation for the tracheobronchomalacia. Clinical suspicion of amyloidosis was not confirmed by biopsies of abdominal fat tissue and of the tongue. Ultimately, the patient succumbed to multiple organ failure. Autopsy revealed AA amyloid in the trachea (Fig. 1), spleen, liver, and perivascularly in the kidneys.Fig. 1

Bottom Line: In this case report, we describe a patient with longstanding rheumatoid arthritis who developed tracheobronchomalacia with fatal outcome.Despite negative antemortem biopsies of abdominal fat and tongue, amyloid was found postmortem in the trachea and appeared to be associated with tracheobronchomalacia.

View Article: PubMed Central - PubMed

Affiliation: Department of Rheumatology, VU University Medical Center, Amsterdam, the Netherlands. f.turkstra@janvanbreemen.nl

ABSTRACT
In this case report, we describe a patient with longstanding rheumatoid arthritis who developed tracheobronchomalacia with fatal outcome. Despite negative antemortem biopsies of abdominal fat and tongue, amyloid was found postmortem in the trachea and appeared to be associated with tracheobronchomalacia.

Show MeSH
Related in: MedlinePlus