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Laryngeal manifestation of gout: a case report of a subglottic gout tophus
Auris Nasus Larynx 28 (2001) 265– 267 www.elsevier.com/locate/anl
Laryngeal manifestation of gout: a case report of a subglottic gout tophus Walter Habermann a,*, Karl Kiesler a, Andreas Eherer b, Alfred Beham c, Gerhard Friedrich a b
a Department of ENT, Uni6ersity of Graz, Medical School, Auenbruggerplatz 20, A-8036 Graz, Austria Department of Internal Medicine, Uni6ersity of Graz, Medical School, Auenbruggerplatz 20, A-8036 Graz, Austria c Department of Pathology, Uni6ersity of Graz, Medical School, Auenbruggerplatz 20, A-8036 Graz, Austria
Received 14 July 2000; received in revised form 21 November 2000; accepted 24 November 2000
Abstract A subglottic tophaceous deposition of urate crystals is a rare finding. We report on a case of a male Caucasian who had a moderate dysphonia without any further laryngeal symptoms. The laryngoscopy revealed a hemispheric lesion on the left subglottic region. An excision biopsy was performed, and the histopathological examination of the dissected specimen showed a tophus. Diagnostic and therapeutic strategies are discussed. © 2001 Elsevier Science Ireland Ltd. All rights reserved. Keywords: Dysphonia; Gout; Larynx
1. Introduction Gout includes a heterogeneous group of disorders: hyperuricemia; attacks of acute typically mono-articular inflammatory arthritis; tophaceous deposition of urate crystals in and around joints; interstitial deposition of urate crystals in renal parenchyma; and urolithiasis [1]. The main biochemical feature, hyperuricemia, is a prerequisite for gout. Hyperuricemia is defined as a plasma urate concentration above 7.0 mg/dl, resulting from an increase in the production of urate, or a decrease in the excretion of uric acid, or a combination of both processes. Uric acid is the final breakdown product of purine degradation in humans. Urates, the ionic form of uric acid, predominate in plasma, extracellular fluids, and synovial fluid. Factors that induce the dissolving of uric acid and the shedding of previously formed crystals, include stress, trauma, infection, surgery, starvation, weight reduction, hyperalimentation, alcohol and medication. * Corresponding author. Tel.: + 43-316-3853465; fax: +43-3163853869. E-mail address: w – [email protected] (W. Habermann).
One important feature of gout is an acute attack of monarticular arthritis. Gouty arthritis primarily involves peripheral joints, and in addition, periarticular sites such as fascies, tendon insertions, or other tendosynovia can be affected. To our knowledge, nine cases of laryngeal manifestation of gout have been reported in the English or German literature published in North America or Europe, and only two of them were located close to the arytenoid cartilage [2,3]. We report the diagnosis and treatment of a case of gout tophus located at the cricoarytenoid joint.
2. Case report A 50 year old male Caucasian had been hospitalized due to a subglottic tumorous lesion. This lesion was discovered during a routine ENT examination when the patient presented for a candidiasis of the tongue. The patient had reported that he had had a moderate dysphonia for one year. The voice disorder did not bother him, and no investigation was intended by the patient himself.
0385-8146/01/$ - see front matter © 2001 Elsevier Science Ireland Ltd. All rights reserved. PII: S 0 3 8 5 - 8 1 4 6 ( 0 0 ) 0 0 1 1 9 - X
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W. Habermann et al. / Auris Nasus Larynx 28 (2001) 265–267
Laryngoscopy revealed a hemispherical expansion, with a smooth surface on the left subglottic region, below the vocal process (Fig. 1). Stroboscopically, the vocal fold showed no pathologic finding. An ultrasound scan of the neck and a barium swallow identified no pathologies. Routine laboratory values were normal, and the only pathologic finding was hyperuricemia (9.42 mg/dl). This hyperuricemia was detected several years ago, when the patient suffered from acute arthritis of the first metacarpophalangeal joint of the right hand. Since then, he has been treated with an uricosuricum. A microlaryngoscopic laser resection of the lesion was performed with the intention of performing an excision biopsy (Fig. 2). Histopathological examination of the dissected specimen (6 ×2× 7 mm in size) showed a central confluence containing some urate crystals (due to the fixation of the tissue with formalin, the majority of the crystals had been dissolved) that was surrounded by infiltrating lymphatic cells and some giant cells of the foreign-body type (Figs. 3 and 4). The surface of the lesion was covered by respiratory epithelium (Fig. 3). The post-operative period was uneventful, and the patient regained a good voice quality within the following fortnight. Two years after the operation, no pathologies of the larynx related to gout were observed.
Fig. 2. Laser resection of the lesion.
described in two cases by Garrod in 1863 and Goodman et al. in 1976 [2,3]. As mentioned the deposition of urate is mostly located in the tendosynovia of a joint. With regards to the laryngeal localisation of the reported cases, it is
3. Discussion As discussed in the literature by Guttenplan et al. in 1991 and Lefkovits in 1965 laryngeal manifestations of gout is not as rare as previously thought [4,5]. We agree that the small number of cases noted compared to the larger number of expected cases is due to the expression of a non-specific symptom, hoarseness. A tophaceous deposition at the arytenoid cartilage has only been
Fig. 1. Microlaryngoscopic picture of the subglottic lesion at the left side.
Fig. 3. Histopathological picture of the dissected specimen with respiratory epithelium covering the surface (*confluence of urate crystals), magnification 20 ×, HE staining.
W. Habermann et al. / Auris Nasus Larynx 28 (2001) 265–267
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ogy [5]. The endolaryngeal laser surgery enables us to resect especially the smaller lesions of unknown histopathology according to the principles of excision biopsy. The therapy of gout depends on the kind of gout [1]. Acute gouty arthritis is treated with colchicine, indomethacine, and other non-steroidal anti-inflammatory agents. The treatment of hyperuricemia should keep uric acid at below 7 mg/dl. In addition to medical treatment, a low purine diet, reduction of weight and alcohol consumption are recommended.
4. Conclusion
Fig. 4. Histopathological picture of the dissected specimen: magnification 40 × , HE staining. (*confluence of urate crystals; giant cells of the foreign body type).
surprising that a gout tophus was associated with the cricoarytenoid joint in only two cases, while in eight other cases, it was associated with other locations distant to a joint [4]. According to Goodman et al. in 1976 and Gacek et al. in 1999, a gout arthritis of the cricoarytenoid joint can be suspected due to specific features of the case history in combination with an impaired mobility of the vocal folds, i.e. incomplete closure of the posterior glottis, and reduced vocal fold abduction [2,6]. In the case reported herein, vocal fold mobility was completely unimpaired. Considering the many different ethiologies of impaired vocal fold mobility, we do not think that there are specific laryngoscopic findings that may lead to the diagnosis of gouty arthritis clinically. The diagnosis of a laryngeal tophaceous gout manifestation is based on biopsy and subsequent histopathol-
.
The diagnosis of a gout tophus at the cricoarytenoid joint should be based on a biopsy because no laryngoscopic findings are specific for a gout tophus. The resection of the tophus and subsequent histopathological workup are the preconditions required for the establishment of a diagnosis. The resection of the tophus is also the local therapy that must be followed by adequate medical treatment and diet to retain a purine metabolism balance.
References [1] Wortmann RL. Gout and other disorders of purine metabolism. In: Isselbacher KJ, Braunwald E, Wilson JD, Martin JB, Fauci AS, Kasper DL, editors. Harrison’s Principles of Internal Medicine, 13th ed. New York: McGraw-Hill, 1994:2079 –88. [2] Goodman M, Montgomery W, Minette L. Pathologic findings in gouty cricoarytenoid arthritis. Arch Otolaryngol 1976;102:27 – 9. [3] Garrod AB. Nature and Treatment of Gout and Rheumatic Gout, second ed. London: Walton and Maberly, 1863:1982 – 3. [4] Guttenplan MD, Hendrix RA, Townsend MJ, Balsara G. Laryngeal manifestations of gout. Ann Otol Rhinol Laryngol 1991;100:899 – 902. [5] Lefkovits AM. Gouty involvement of the larynx: report of a case and review of the literature. Arthrit Rheum 1965;8:1019 –26. [6] Gacek RR, Gacek MR, Montgomery WW. Evidence for laryngeal paralysis in cricoarytenoid joint arthritis. Laryngoscope 1999;109:279 – 83.
Laryngeal manifestation of gout: a case report of a subglottic gout tophus Walter Habermann a,*, Karl Kiesler a, Andreas Eherer b, Alfred Beham c, Gerhard Friedrich a b
a Department of ENT, Uni6ersity of Graz, Medical School, Auenbruggerplatz 20, A-8036 Graz, Austria Department of Internal Medicine, Uni6ersity of Graz, Medical School, Auenbruggerplatz 20, A-8036 Graz, Austria c Department of Pathology, Uni6ersity of Graz, Medical School, Auenbruggerplatz 20, A-8036 Graz, Austria
Received 14 July 2000; received in revised form 21 November 2000; accepted 24 November 2000
Abstract A subglottic tophaceous deposition of urate crystals is a rare finding. We report on a case of a male Caucasian who had a moderate dysphonia without any further laryngeal symptoms. The laryngoscopy revealed a hemispheric lesion on the left subglottic region. An excision biopsy was performed, and the histopathological examination of the dissected specimen showed a tophus. Diagnostic and therapeutic strategies are discussed. © 2001 Elsevier Science Ireland Ltd. All rights reserved. Keywords: Dysphonia; Gout; Larynx
1. Introduction Gout includes a heterogeneous group of disorders: hyperuricemia; attacks of acute typically mono-articular inflammatory arthritis; tophaceous deposition of urate crystals in and around joints; interstitial deposition of urate crystals in renal parenchyma; and urolithiasis [1]. The main biochemical feature, hyperuricemia, is a prerequisite for gout. Hyperuricemia is defined as a plasma urate concentration above 7.0 mg/dl, resulting from an increase in the production of urate, or a decrease in the excretion of uric acid, or a combination of both processes. Uric acid is the final breakdown product of purine degradation in humans. Urates, the ionic form of uric acid, predominate in plasma, extracellular fluids, and synovial fluid. Factors that induce the dissolving of uric acid and the shedding of previously formed crystals, include stress, trauma, infection, surgery, starvation, weight reduction, hyperalimentation, alcohol and medication. * Corresponding author. Tel.: + 43-316-3853465; fax: +43-3163853869. E-mail address: w – [email protected] (W. Habermann).
One important feature of gout is an acute attack of monarticular arthritis. Gouty arthritis primarily involves peripheral joints, and in addition, periarticular sites such as fascies, tendon insertions, or other tendosynovia can be affected. To our knowledge, nine cases of laryngeal manifestation of gout have been reported in the English or German literature published in North America or Europe, and only two of them were located close to the arytenoid cartilage [2,3]. We report the diagnosis and treatment of a case of gout tophus located at the cricoarytenoid joint.
2. Case report A 50 year old male Caucasian had been hospitalized due to a subglottic tumorous lesion. This lesion was discovered during a routine ENT examination when the patient presented for a candidiasis of the tongue. The patient had reported that he had had a moderate dysphonia for one year. The voice disorder did not bother him, and no investigation was intended by the patient himself.
0385-8146/01/$ - see front matter © 2001 Elsevier Science Ireland Ltd. All rights reserved. PII: S 0 3 8 5 - 8 1 4 6 ( 0 0 ) 0 0 1 1 9 - X
266
W. Habermann et al. / Auris Nasus Larynx 28 (2001) 265–267
Laryngoscopy revealed a hemispherical expansion, with a smooth surface on the left subglottic region, below the vocal process (Fig. 1). Stroboscopically, the vocal fold showed no pathologic finding. An ultrasound scan of the neck and a barium swallow identified no pathologies. Routine laboratory values were normal, and the only pathologic finding was hyperuricemia (9.42 mg/dl). This hyperuricemia was detected several years ago, when the patient suffered from acute arthritis of the first metacarpophalangeal joint of the right hand. Since then, he has been treated with an uricosuricum. A microlaryngoscopic laser resection of the lesion was performed with the intention of performing an excision biopsy (Fig. 2). Histopathological examination of the dissected specimen (6 ×2× 7 mm in size) showed a central confluence containing some urate crystals (due to the fixation of the tissue with formalin, the majority of the crystals had been dissolved) that was surrounded by infiltrating lymphatic cells and some giant cells of the foreign-body type (Figs. 3 and 4). The surface of the lesion was covered by respiratory epithelium (Fig. 3). The post-operative period was uneventful, and the patient regained a good voice quality within the following fortnight. Two years after the operation, no pathologies of the larynx related to gout were observed.
Fig. 2. Laser resection of the lesion.
described in two cases by Garrod in 1863 and Goodman et al. in 1976 [2,3]. As mentioned the deposition of urate is mostly located in the tendosynovia of a joint. With regards to the laryngeal localisation of the reported cases, it is
3. Discussion As discussed in the literature by Guttenplan et al. in 1991 and Lefkovits in 1965 laryngeal manifestations of gout is not as rare as previously thought [4,5]. We agree that the small number of cases noted compared to the larger number of expected cases is due to the expression of a non-specific symptom, hoarseness. A tophaceous deposition at the arytenoid cartilage has only been
Fig. 1. Microlaryngoscopic picture of the subglottic lesion at the left side.
Fig. 3. Histopathological picture of the dissected specimen with respiratory epithelium covering the surface (*confluence of urate crystals), magnification 20 ×, HE staining.
W. Habermann et al. / Auris Nasus Larynx 28 (2001) 265–267
267
ogy [5]. The endolaryngeal laser surgery enables us to resect especially the smaller lesions of unknown histopathology according to the principles of excision biopsy. The therapy of gout depends on the kind of gout [1]. Acute gouty arthritis is treated with colchicine, indomethacine, and other non-steroidal anti-inflammatory agents. The treatment of hyperuricemia should keep uric acid at below 7 mg/dl. In addition to medical treatment, a low purine diet, reduction of weight and alcohol consumption are recommended.
4. Conclusion
Fig. 4. Histopathological picture of the dissected specimen: magnification 40 × , HE staining. (*confluence of urate crystals; giant cells of the foreign body type).
surprising that a gout tophus was associated with the cricoarytenoid joint in only two cases, while in eight other cases, it was associated with other locations distant to a joint [4]. According to Goodman et al. in 1976 and Gacek et al. in 1999, a gout arthritis of the cricoarytenoid joint can be suspected due to specific features of the case history in combination with an impaired mobility of the vocal folds, i.e. incomplete closure of the posterior glottis, and reduced vocal fold abduction [2,6]. In the case reported herein, vocal fold mobility was completely unimpaired. Considering the many different ethiologies of impaired vocal fold mobility, we do not think that there are specific laryngoscopic findings that may lead to the diagnosis of gouty arthritis clinically. The diagnosis of a laryngeal tophaceous gout manifestation is based on biopsy and subsequent histopathol-
.
The diagnosis of a gout tophus at the cricoarytenoid joint should be based on a biopsy because no laryngoscopic findings are specific for a gout tophus. The resection of the tophus and subsequent histopathological workup are the preconditions required for the establishment of a diagnosis. The resection of the tophus is also the local therapy that must be followed by adequate medical treatment and diet to retain a purine metabolism balance.
References [1] Wortmann RL. Gout and other disorders of purine metabolism. In: Isselbacher KJ, Braunwald E, Wilson JD, Martin JB, Fauci AS, Kasper DL, editors. Harrison’s Principles of Internal Medicine, 13th ed. New York: McGraw-Hill, 1994:2079 –88. [2] Goodman M, Montgomery W, Minette L. Pathologic findings in gouty cricoarytenoid arthritis. Arch Otolaryngol 1976;102:27 – 9. [3] Garrod AB. Nature and Treatment of Gout and Rheumatic Gout, second ed. London: Walton and Maberly, 1863:1982 – 3. [4] Guttenplan MD, Hendrix RA, Townsend MJ, Balsara G. Laryngeal manifestations of gout. Ann Otol Rhinol Laryngol 1991;100:899 – 902. [5] Lefkovits AM. Gouty involvement of the larynx: report of a case and review of the literature. Arthrit Rheum 1965;8:1019 –26. [6] Gacek RR, Gacek MR, Montgomery WW. Evidence for laryngeal paralysis in cricoarytenoid joint arthritis. Laryngoscope 1999;109:279 – 83.