Table 1.
Articles identified from PubMed literature search using terms “Gaucher Disease” and “Fatigue”
| Reference | Language | Patients (N) | Fatigue assessed? | Fatigue tool | Data description |
|---|---|---|---|---|---|
| Verderese et al., 1993 [13] | English | 12 | Yes | 5 questions | All patients reported chronic fatigue as a pervasive problem at baseline; all perceived improvement at 4 months after starting ERT |
| Niederau et al., 1994 [14] | English | 5 | Yes | None mentioned | Case reports of 5 patients with GD receiving ERT for 12–18 months; all reported marked reductions in fatigue within a few weeks after starting ERT |
| Gagnon et al., 1998 [15] | English | 24 | Yes | Questionnaire; fatigue either yes/no | National Gaucher Foundation GD screening program; fatigue was the most commonly reported symptom (79.4 % of all respondents; only 24 patients had confirmed GD) |
| Hayes et al., 1998 [11] | English | 16 | Yes | Unspecified | Patients asked open-ended questions about chronic fatigue which were based on components of several unspecified instruments; 88 % reported being easily fatigued |
| Niederau et al., 1998 [16] | English | 1 | Yes | None mentioned | Case report of elderly patient with GD receiving ERT for 30 months; fatigue decreased within several months of starting ERT |
| Masek et al., 1999 [17] | English | 25 | Yes | SF-36 | SF-36 administered before and after initiation of ERT; vitality domain (a measure of energy and fatigue) was improved significantly at 6, 12, 18, and 24 months vs. pretherapy |
| Khan et al., 2000 [18] | English | 1 | Yes | None mentioned | Case report of diagnosis of adolescent patient with GD; fatigue mentioned as a presenting and persisting symptom |
| Chou et al., 2004 [19] | English | 1 | No | — | Case report of diagnosis of adolescent patient with GD |
| Tsai et al., 2008 [20] | English | 7 | Muscle fatigue only | — | Evaluation of myopathy in GD; 3 patients developed insidious, nonprogressive muscle weakness with easy muscle fatigue; other measures of fatigue not assessed |
| Shapiro et al., 2009 [21] | English | — | — | — | Describes clinical trial in patients with Tay-Sachs disease; not relevant to analysis of fatigue in GD |
| Samuels et al., 2012 [22] | English | 12 | Yes | FACIT-F | “Additional concerns” of fatigue evaluated at baseline and following acupuncture treatment; mean scores on fatigue-specific scale of FACIT-F increased following acupuncture (27.6 vs. 35.9; P = 0.008) |
| Wyatt et al., 2012 [23] | English | 134 | Yes | FSS | All patients receiving ERT; no longitudinal data; found no association between fatigue and time on ERT (P = 0.57) |
| Elstein et al., 2015 [24] | English | 38 | No | None | Patients received velaglucerase alfa in an extension trial; reports of fatigue were collected as adverse events and as infusion-associated reactions |
| Stirnemann et al., 2015 [25] | English | 99 | Yes | None mentioned | Retrospective collection of data on patients’ characteristics, treatment, and clinical and biological parameters; fatigue was reported in 8 % of patients |
| Dulgar et al., 2016 [26] | English | 1 | Yes | None mentioned | Case report of 1 patient with tuberculosis and untreated GD; fatigue mentioned as symptom |
| Niederau et al., 2001 [27] | German | — | Unknown | Unknown | German recommendations for diagnosis and treatment of GD; fatigue mentioned as common symptom |
| Schaison et al., 2002 [28] | French | 108 | Yes | Unknown | Fatigue alleviated by ERT |
| Juhász et al., 2012 [29] | Hungarian | 2 | Unknown | — | Case report of 2 patients diagnosed in late adulthood; fatigue mentioned as GD sign/symptom |
| Hansen et al., 2015 [30] | Danish | 1 | Yes | Unknown | Case report of a 10-year-old girl with GD; extreme fatigue over several years was reported |
ERT enzyme-replacement therapy, FACIT-F Functional Assessment of Chronic Illness Therapy–Fatigue, FSS Fatigue Severity Scale, GD Gaucher disease, SF-36 36-item Short Form Health Survey