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. 2022 May 17;107(6):e2258-e2266.
doi: 10.1210/clinem/dgac120.

Neuropathic-like Pain in Fibrous Dysplasia/McCune-Albright Syndrome

Tiahna L Spencer  1 , Laura Watts  2 , Anushka Soni  3 , Rafael Pinedo-Villanueva  3 , Anne-Marie Heegaard  4 , Alison M Boyce  5 , M Kassim Javaid  3
Affiliations

Neuropathic-like Pain in Fibrous Dysplasia/McCune-Albright Syndrome

Tiahna L Spencer et al. J Clin Endocrinol Metab. .

Abstract

Context: Pain is a major symptom in adults with fibrous dysplasia/McCune-Albright syndrome (FD/MAS) and response to current treatments, including bisphosphonates and standard analgesics (nonsteroidal anti-inflammatory drugs and opiates) is unpredictable. No studies have explored whether the type of pain is variable in this patient group.

Objective: To determine the frequency of neuropathic-like pain in patients with FD/MAS.

Design: Retrospective, dual registry study.

Setting: Community.

Patients: FD/MAS online registries: the US-based Familial Dysautonomia Foundation (FDF) and the UK-based Rare and Undiagnosed Diseases (RUDY) study.

Intervention: Subjects completed questionnaires to evaluate the presence of features of neuropathic-like pain (painDETECT) and the impact on sleep quality (Pittsburgh Sleep Quality Index) and mental health (Hospital Anxiety and Depression Scale). Descriptive statistics were used to characterize the prevalence and associated burden of neuropathic-like pain.

Main outcome measures: Incidence of neuropathic, nociceptive, and unclear pain.

Results: Of 249 participants, one third experienced neuropathic-like pain. This group had statistically significantly (P < 0.001) worse mental well-being and sleep in comparison to those with predominately nociceptive pain.

Conclusions: Neuropathic-like pain is common in patients with FD/MAS and associated with worse quality of life. Evaluation of pain in patients with FD/MAS should include assessment of neuropathic-like pain to guide personalized approaches to treatment and inform future research.

Keywords: McCune-Albright syndrome; fibrous dysplasia; mental health; neuropathic-like pain; nociceptive pain; quality of life.

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Figures

Figure 1.
Figure 1.
Distribution of current, average and strongest pain severity in 249 participants with fibrous dysplasia/McCune-Albright syndrome. Each line describes the smoothed probability estimate of the frequency distribution for reported current, average, and strongest pain during the last past 4 weeks from the painDETECT questionnaire.
Figure 2.
Figure 2.
Proportions of nociceptive, unclear, and neuropathic-like pain for each severity of strongest pain strength during the past 4 weeks. Within each level of strongest pain during the past 4 weeks from 1 to 10, the proportion of individuals within in each pain with nociceptive, unclear, or neuropathic is shown as defined by the painDETECT questionnaire (33)
See this image and copyright information in PMC

References

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