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Background <p>Trigeminal autonomic cephalalgias (TACs) are rare headache disorders characterized by short, frequent attacks of unilateral pain accompanied by ipsilateral cranial autonomic features. Among TAC subtypes, paroxysmal hemicrania (PH) is distinguished by a robust response to indomethacin. When patients present with atypical phenotypic features or incomplete indomethacin responsiveness, diagnostic certainty decreases, the differential diagnosis broadens—including cervicogenic headache with autonomic features and short-lasting unilateral neuralgiform headache attacks (SUNHA)—and management becomes more complex.</p> Case presentation <p>A 35-year-old man presented with a 12-month history of very frequent short-lasting unilateral headache attacks (25–30 episodes/day, 2–5&#xa0;min each) associated with tearing, nasal obstruction, periorbital edema, and restlessness. The phenotype was atypical because pain lateralization alternated between sides. Brain MRI/MRA/MRV were unremarkable, whereas cervical spine MRI showed bilateral paraspinal muscular signal abnormality consistent with sustained tonic contraction (described radiologically as "spasm") and early multilevel spondylotic change without major neural compression. Previous trials of celecoxib (200&#xa0;mg twice daily for four weeks) and pregabalin (150&#xa0;mg twice daily for six weeks) were ineffective. Indomethacin 150&#xa0;mg/day produced only partial improvement (~ 20% reduction in attack frequency) after one month; dose escalation to 225&#xa0;mg/day (75&#xa0;mg three times daily) was not pursued because of emerging gastrointestinal intolerance. Because coexisting cervical symptoms and examination findings—including reduced cervical range of motion predominantly in right rotation, left lateral flexion, and flexion, and bilateral paraspinal muscular signal abnormality on MRI—suggested a possible cervical pain contribution, an eight-week adjunctive physiotherapy program emphasizing cervical mobilization and deep neck flexor strengthening was introduced while indomethacin was continued. Following this multimodal approach, attack burden decreased substantially, with residual mild episodes reported only 2–3 times per month at follow-up. The physiotherapy program was maintained as a home exercise regimen beyond the supervised phase.</p> Conclusions <p>This case describes an atypical TAC-like presentation with features overlapping PH, SUNHA, and cervicogenic headache. The incomplete indomethacin responsiveness, alternating laterality, and marked improvement following adjunctive physiotherapy suggest a possible cervical contribution to symptom persistence, but causality cannot be established from a single case, and spontaneous remission cannot be excluded. In selected patients with atypical TAC phenotypes, careful reassessment of coexisting cervical dysfunction may be reasonable as part of individualized multimodal management.</p>

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Adjunctive physiotherapy in a patient with a trigeminal autonomic cephalalgia phenotype and cervical dysfunction

  • 期刊论文

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  • 发表日期：

    2026年4月28日

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  • Adel Ibrahim Azzam,
  • Saad Ghanem,
  • Taha Ibrahim Aladrosy,
  • Ahmed Ali Hassan Ali

摘要

Background

Trigeminal autonomic cephalalgias (TACs) are rare headache disorders characterized by short, frequent attacks of unilateral pain accompanied by ipsilateral cranial autonomic features. Among TAC subtypes, paroxysmal hemicrania (PH) is distinguished by a robust response to indomethacin. When patients present with atypical phenotypic features or incomplete indomethacin responsiveness, diagnostic certainty decreases, the differential diagnosis broadens—including cervicogenic headache with autonomic features and short-lasting unilateral neuralgiform headache attacks (SUNHA)—and management becomes more complex.

Case presentation

A 35-year-old man presented with a 12-month history of very frequent short-lasting unilateral headache attacks (25–30 episodes/day, 2–5 min each) associated with tearing, nasal obstruction, periorbital edema, and restlessness. The phenotype was atypical because pain lateralization alternated between sides. Brain MRI/MRA/MRV were unremarkable, whereas cervical spine MRI showed bilateral paraspinal muscular signal abnormality consistent with sustained tonic contraction (described radiologically as "spasm") and early multilevel spondylotic change without major neural compression. Previous trials of celecoxib (200 mg twice daily for four weeks) and pregabalin (150 mg twice daily for six weeks) were ineffective. Indomethacin 150 mg/day produced only partial improvement (~ 20% reduction in attack frequency) after one month; dose escalation to 225 mg/day (75 mg three times daily) was not pursued because of emerging gastrointestinal intolerance. Because coexisting cervical symptoms and examination findings—including reduced cervical range of motion predominantly in right rotation, left lateral flexion, and flexion, and bilateral paraspinal muscular signal abnormality on MRI—suggested a possible cervical pain contribution, an eight-week adjunctive physiotherapy program emphasizing cervical mobilization and deep neck flexor strengthening was introduced while indomethacin was continued. Following this multimodal approach, attack burden decreased substantially, with residual mild episodes reported only 2–3 times per month at follow-up. The physiotherapy program was maintained as a home exercise regimen beyond the supervised phase.

Conclusions

This case describes an atypical TAC-like presentation with features overlapping PH, SUNHA, and cervicogenic headache. The incomplete indomethacin responsiveness, alternating laterality, and marked improvement following adjunctive physiotherapy suggest a possible cervical contribution to symptom persistence, but causality cannot be established from a single case, and spontaneous remission cannot be excluded. In selected patients with atypical TAC phenotypes, careful reassessment of coexisting cervical dysfunction may be reasonable as part of individualized multimodal management.