Skip to main content

Advertisement

Log in

A rare manifestation of cricopharyngeal myopathy presenting with dysphagia in sarcoidosis

  • Short Communication
  • Published:
Rheumatology International Aims and scope Submit manuscript

Abstract

Sarcoidosis is a systemic inflammatory granulomatous disease that affects multiple organs in the body; however, dysphagia is a relatively rare manifestation at early stages. Dysphagia in sarcoidosis is attributed to many mechanisms, such as mediastinal lymphadenopathy, esophageal or laryngeal involvement, cranial neuropathy, and brainstem infiltration. In this article, we report an extremely rare case with sarcoidosis who presented with dysphagia due to isolated cricopharyngeal myopathy. The 75-year-old woman presented with slowly progressive swallowing difficulty and videofluorography showed insufficient opening of the upper esophageal sphincter. On presentation, she had no cranial nerve or central nervous system impairments. A cricopharyngeal myotomy was performed, and histopathological study revealed a significant inflammatory change with non-necrotizing granulomas within the muscle tissue. We concluded that this was a very rare case of sarcoidosis presenting with localized cricopharyngeal myopathy. Postoperatively, a contracture of the esophageal entrance was successfully released and the dysphagia was alleviated.

This is a preview of subscription content, log in via an institution to check access.

Access this article

Price excludes VAT (USA)
Tax calculation will be finalised during checkout.

Instant access to the full article PDF.

Fig. 1
Fig. 2
Fig. 3
Fig. 4

References

  1. Cappell MS (1995) Endoscopic, radiographic, and manometric findings in dysphagia associated with sarcoid due to extrinsic esophageal compression from subcarinal lymphadenopathy. Am J Gastroenterol 90:489–492

    PubMed  CAS  Google Scholar 

  2. Geissinger BW, Sharkey MF, Criss DG, Wu WC (1996) Reversible esophageal motility disorder in a patient with sarcoidosis. Am J Gastroenterol 91:1423–1426

    PubMed  CAS  Google Scholar 

  3. Bredenoord AJ, Jafari J, Kadri S, Simcock DE, Sifrim D, Preston SL (2011) Case report: achalasia-like dysmotility secondary to oesophageal involvement of sarcoidosis. Gut 60:153–155

    Article  PubMed  Google Scholar 

  4. Bower JS, Belen JE, Weg JG, Dantzker DR (1980) Manifestations and treatment of laryngeal sarcoidosis. Am Rev Respir Dis 122:325–332

    PubMed  CAS  Google Scholar 

  5. Mayerhoff RM, Pitman MJ (2010) Atypical and disparate presentations of laryngeal sarcoidosis. Ann Otol Rhinol Laryngol 119:667–671

    PubMed  Google Scholar 

  6. Castroagudin JF, Gonzalez-Quintela A, Moldes J, Forteza J, Barrio E (1999) Acute reversible dysphagia and dysphonia as initial manifestations of sarcoidosis. Hepatogastroenterology 46:2414–2418

    PubMed  CAS  Google Scholar 

  7. Fahoum F, Drory VE, Issakov J, Neufeld MY (2009) Neurosarcoidosis presenting as Guillain-Barré-like syndrome. A case report and review of the literature. J Clin Neuromuscul Dis 11:35–43

    Article  PubMed  Google Scholar 

  8. Zuniga G, Ropper AH, Frank J (1991) Sarcoid peripheral neuropathy. Neurology 41:1558–1561

    Article  PubMed  CAS  Google Scholar 

  9. Bos MM, Overeem S, van Engelen BG, Scheffer H, van den Elzen C, Ter Laak H, Lammens M, Schelhaas HJ, Zwarts MJ (2006) A case of neuromuscular mimicry. Neuromuscul Disord 16:510–513

    Article  PubMed  Google Scholar 

  10. Delaney P (1977) Neurologic manifestations in sarcoidosis: review of the literature, with a report of 23 cases. Ann Intern Med 87:336–345

    PubMed  CAS  Google Scholar 

  11. Winterbauer RH (1983) Disease of uncertain etiology, sarcoidosis. In: Isselbacher KJ, Adams RD, Braunwald E (eds) Harrison’s principles of internal medicine. McGraw-Hill, New York, pp 1248–1253

    Google Scholar 

  12. Utz JP, Limper AH, Kalra S, Specks U, Scott JP, Vuk-Pavlovic Z, Schroeder DR (2003) Etanercept for the treatment of stage II and III progressive pulmonary sarcoidosis. Chest 124:177–185

    Article  PubMed  CAS  Google Scholar 

  13. Prayson RA (1999) Granulomatous myositis. Clinicopathologic study of 12 cases. Am J Clin Pathol 112:63–68

    PubMed  CAS  Google Scholar 

  14. Wolfe SM, Pinals RS, Aelion JA, Goodman RE (1987) Myopathy in sarcoidosis: clinical and pathologic study of four cases and review of the literature. Semin Arthritis Rheum 16:300–306

    Article  PubMed  CAS  Google Scholar 

  15. Fayad F, Lioté F, Berenbaum F, Orcel P, Bardin T (2006) Muscle involvement in sarcoidosis: a retrospective and followup studies. J Rheumatol 33:98–103

    PubMed  Google Scholar 

  16. Otake S, Banno T, Ohba S, Noda M, Yamamoto M (1990) Muscular sarcoidosis: findings at MR imaging. Radiology 176:145–148

    PubMed  CAS  Google Scholar 

  17. Le Roux K, Streichenberger N, Vial C, Petiot P, Feasson L, Bouhour F, Ninet J, Lachenal F, Broussolle C, Sève P (2007) Granulomatous myositis: a clinical study of thirteen cases. Muscle Nerve 35:171–177

    Article  PubMed  Google Scholar 

  18. Liem IH, Drent M, Antevska E, Lamers RJ, Heidendal GA (1998) Intense muscle uptake of gallium-67 in a patient with sarcoidosis. J Nucl Med 39:1605–1607

    PubMed  CAS  Google Scholar 

  19. Kaplan S (1951) Paralysis of deglutition, a post-poliomyelitis complication treated by section of the cricopharyngeal muscle. Ann Surg 133:572–573

    PubMed  CAS  Google Scholar 

  20. Poirier NC, Bonavina L, Taillefer R et al (1997) Cricopharyngeal myotomy for neurogenic oropharyngeal dysphagia. J Thorac Cardiovasc Surg 113:233–240

    Article  PubMed  CAS  Google Scholar 

  21. Shin T, Tsuda K, Takagi S (1999) Surgical treatment for dysphagia of neuromuscular origin. Folia Phoniatr Logop 51:213–219

    Article  PubMed  CAS  Google Scholar 

  22. Kelly JH (2000) Management of upper esophageal sphincter disorders: indications and complications of myotomy. Am J Med 108(Suppl 4a):43S–46S

    Article  PubMed  Google Scholar 

Download references

Author information

Authors and Affiliations

Authors

Corresponding author

Correspondence to Masamitsu Hyodo.

Rights and permissions

Reprints and permissions

About this article

Cite this article

Nishikubo, K., Hyodo, M., Kawakami, M. et al. A rare manifestation of cricopharyngeal myopathy presenting with dysphagia in sarcoidosis. Rheumatol Int 33, 1089–1092 (2013). https://doi.org/10.1007/s00296-011-2242-y

Download citation

  • Received:

  • Accepted:

  • Published:

  • Issue Date:

  • DOI: https://doi.org/10.1007/s00296-011-2242-y

Keywords

Navigation