# Dysphagia Secondary to Myotonic Dystrophy Unveiled in a Case of Destructive Spondylitis With Synovitis, Acne, Pustulosis, Hyperostosis, Osteitis (SAPHO) Syndrome Presenting As Torticollis

**Authors:** Yoshinori Ishikawa, Takashi Kobayashi, Ryo Shoji, Naohisa Miyakoshi

PMC · DOI: 10.7759/cureus.54271 · Cureus · 2024-02-15

## TL;DR

A rare case shows how myotonic dystrophy can cause post-surgery swallowing issues in a patient with SAPHO syndrome and cervical spine problems.

## Contribution

Highlights the under-recognized risk of myotonic dystrophy in patients with SAPHO syndrome undergoing spinal surgery.

## Key findings

- Postoperative dysphagia occurred in a patient with SAPHO syndrome and undiagnosed myotonic dystrophy.
- Delayed diagnosis of myotonic dystrophy complicated surgical and postoperative management.
- Surgeons should consider myotonic dystrophy in patients with complex spinal conditions and unexplained neurological symptoms.

## Abstract

To report an instructive case involving destructive spondylitis and synovitis-acne-pustulosis-hyperostosis-osteitis (SAPHO) syndrome, presenting with torticollis and postoperative dysphagia without hoarseness, attributed to hidden myotonic dystrophy (DM).

A 51-year-old male patient with a cervical deformity, who was previously managed conservatively for a metastatic tumor, underwent reconstruction surgery and subsequently experienced postoperative dysphagia. The presence of destructive spondylitis with torticollis, warranting prompt assessment to prevent paralysis, adds complexity to the delayed identification of DM. Given the rarity of DM, peculiar neurological symptoms and other systemic comorbidities did not lead to a preoperative diagnosis without prior knowledge.

The patient’s dysphagia induced respiratory arrest and required reintubation. Challenges in extubation and ventilator weaning arose due to hypercapnia. Superimposed COVID-19 infection elongated the duration of intubation. Extubation failed due to aspiration pneumonia and required a tracheotomy. Despite laryngeal elevation and preservation of the relaxation of the oesophageal entrance, the sensation and movement of the tracheopharynx were disturbed. The patient exhibited an oropharyngeal propulsive disorder, predominantly indicative of motor neuron disease. The patient’s mother stated that his brother had been hospitalized for a long time after abdominal surgery. Finally, the patient was diagnosed with DM, which is known to cause post-anesthetic dysphagia.

Recognizing the existence of severe destructive cervical spondylitis associated with SAPHO is crucial. Although DM is not very common, it is not classified as extremely rare. Therefore, surgeons should be mindful of the potential risks associated with general anesthesia in patients with DM. The complexity of preoperative conditions may hinder an accurate diagnosis. Recognizing and establishing preoperative expectations can assist surgeons in preventing complications, even if complex spinal surgery is required for patients with DM.

## Linked entities

- **Diseases:** myotonic dystrophy (MONDO:0016107), SAPHO syndrome (MONDO:0019266), torticollis (MONDO:0008583), aspiration pneumonia (MONDO:0000265)

## Full-text entities

- **Diseases:** paralysis (MESH:D010243), Acne, Pustulosis, Hyperostosis, Osteitis (SAPHO) Syndrome (MESH:D010000), COVID-19 infection (MESH:D000086382), Torticollis (MESH:D014103), tumor (MESH:D009369), oropharyngeal propulsive disorder (MESH:D009959), DM (MESH:D009223), metastatic (MESH:D000092182), cervical deformity (MESH:D002575), hypercapnia (MESH:D006935), aspiration pneumonia (MESH:D011015), hoarseness (MESH:D006685), Synovitis (MESH:D013585), motor neuron disease (MESH:D016472), Dysphagia (MESH:D003680), respiratory arrest (MESH:D012131), Destructive Spondylitis (MESH:D013166)
- **Species:** Homo sapiens (human, species) [taxon 9606]

## Full text

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## References

14 references — full list in the complete paper: https://tomesphere.com/paper/PMC10944548/full.md

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Source: https://tomesphere.com/paper/PMC10944548