Penicillamine-Related Lichenoid Dermatitis and Utility of Zinc Acetate in a Wilson Disease Patient with Hepatic Presentation, Anxiety and Spect Abnormalities

Abstract

Wilson's disease is an autosomal recessive disorder of hepatic copper metabolism with consequent copper accumulation and toxicity in many tissues and consequent hepatic, neurologic and psychiatric disorders. We report a case of Wilson's disease with chronic liver disease; moreover, in our patient, presenting also with high levels of state anxiety without depression, 99mTc-ECD-SPECT showed cortical hypoperfusion in frontal lobes, more marked on the left frontal lobe. During the follow-up of our patient, penicillamine was interrupted after the appearance of a lichenoid dermatitis, and zinc acetate permitted to continue the successful treatment of the patient without side-effects. In our case the therapy with zinc acetate represented an effective treatment for a Wilson's disease patient in which penicillamine-related side effects appeared. The safety of the zinc acetate allowed us to avoid other potentially toxic chelating drugs; this observation is in line with the growing evidence on the efficacy of the drug in the treatment of Wilson's disease. Since most of Wilson's disease penicillamine-treated patients do not seem to develop this skin lesion, it could be conceivable that a specific genetic factor is involved in drug response. Further studies are needed for a better clarification of Wilson's disease therapy, and in particular to differentiate specific therapies for different Wilson's disease phenotypes.

Cutaneous lichenoid drug eruptions: A narrative review evaluating demographics, clinical features and culprit medications

“…Two cases (0.62%) for each of the following drugs were identified: Thioridazine, leflunomide, ibuprofen, sildenafil and ursodeoxycholic acid 124–133 . Single cases of a large number of additional drugs were identified as listed in Table S1 33,77,86,134–169 …”

Advances in Treatment of Wilson Disease

“…Penicillamine related pemphigus lichen planus and painful stomatitis have also been described. [68][69][70][71][72][73][74][75] Trientine. The need for an alternative to penicillamine arose with reports of penicillamine-induced nephropathy and renal failure in some patients with WD.…”

Psychiatric comorbidity in Wilson’s disease

“…Psychiatric symptoms/disorder Occurrence/topic Ala et al (2005) depression onset Ara ujo-de-Freitas et al 2014depression onset Aravind et al (2009) mania onset Bidaki et al (2012) psychosis onset Carr and McDonnell (1986) adjustment disorder onset Chakraborty et al (2015) depression onset Chan et al (2005) depression/personality disorder/suicide attempt onset Chand and Murthy (2006) mania onset Crumley (1990) depression onset Davis and Borde (1993) catatonia onset Grover et al 2014psychosis treatment Juki c et al 2006psychosis onset Keller et al (1999) mania onset Kenar and Menteseoglu (2014) mania in course Kontaxakis et al (1988) psychosis onset Krishnakumar and Riyaz (2005) depression onset Krsti c et al 2014psychosis onset Kulaksizoglu and Polat (2003) mania treatment Kumawat et al (2008) obsessive-compulsive disorder onset Leggio et al (2007) anxiety in course Lin et al (2006) Attention Deficit Hyperactivity Disorder (ADHD) onset Lingam et al (1987) conduct disorder onset Litwin, Chabik, and Członkowska (2013) depression treatment Litwin et al (2016) mania treatment Loganathan et al (2008) mania in course Machado et al (2008) mania onset Matarazzo (2002) psychosis onset McDonald and Lake (1995) psychosis treatment McDonnell and Esmonde (1999) bipolar disorder onset Millard et al (2015) irritability/suicidal ideation onset Mitra et al (2014) mania in course Modai et al (1985) schizophrenia-like psychosis treatment M€ uller (1999) psychosis/change in mood/aggressive behaviour/suicide attempt onset Nayak et al 2012catatonia onset Nazariah et al 2011mania onset Negro and Louz a Neto (1995) depr...…”

“…A number of case reports focused on bipolar disorder in the course of WD, presenting after discontinuance of penicillamine (Kenar & Menteseoglu, 2014), or improving with chelation only (Mitra, Ray, & Roy, 2014), or responsive to lithium (Loganathan et al, 2008;Rich & Lajoie, 2012;Rybakowski, Litwin, Chlopocka-Wozniak, & Czlonkowska, 2013). Rarely, it was reported in the course of WD as an obsessive-compulsive disorder (Sahu, Singhi, & Malhotra, 2013), behaviour disorder ( € Ozcan & Selimo glu 2009), and anxiety (Leggio et al, 2007).…”