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Extremely grotesque somatic delusions in a patient of delusional disorder and its response to risperidone treatment
Available online at www.sciencedirect.com
General Hospital Psychiatry 31 (2009) 185 – 186
Case Reports
Extremely grotesque somatic delusions in a patient of delusional disorder and its response to risperidone treatment Takaki Akahane, M.D., Hiroshi Hayashi, M.D., Ph.D.⁎, Haruyoshi Suzuki, M.D., Shinobu Kawakatsu, M.D., Ph.D., Koichi Otani, M.D., Ph.D. Department of Psychiatry, Yamagata University School of Medicine, Yamagata 990-9585, Japan Received 3 June 2008; accepted 11 July 2008
Abstract A 54-year-old man with delusional disorder, somatic type (DDST) showed extremely grotesque somatic delusions, i.e., the presence of another lower jaw with teeth and tongue accompanied by annoying sensations. These somatic delusions responded favorably to treatment with risperidone 2–3 mg/day. The single photon emission computed tomography taken when he had marked somatic delusions showed hypoperfusion in the temporal and parietal lobes. The present report suggests that delusion of reduplication of body parts, which has been generally associated with organic brain diseases, is also observed in DDST. This report also supports prior observations on the efficacy of risperidone for DDST and the association between this disorder and hypoperfusion in the temporal and parietal lobes. © 2009 Elsevier Inc. All rights reserved. Keywords: Delusional disorder; Risperidone; Somatic delusion; SPECT
1. Introduction Delusional disorder, somatic type (DDST), also known as monosymptomatic hypochondriacal psychosis (MHP), is characterized by somatic or hypochondriacal delusions [1]. The most common delusions in DDST are that he or she emits a foul odor from the skin, mouth, rectum or vagina; that there is an infestation of insects on or in the skin; that there is an internal parasite; that certain parts of the body are definitely misshapen or ugly or that parts of the body are not functioning [2]. Knowledge of aetiology in DDST is scanty, because so little modern research has been conducted [1]. Some researchers including us have suggested that DDST has some association with hypoperfusion in the temporal and parietal lobes [3–5]. As to treatment, Munro has recommended pimozide as the first choice drug for DDST [1]. However, recent reports suggest the efficacy of risperidone [4,6] that has a more benign side effect profile than pimozide [7]. We describe the case of a patient with DDST, who showed extremely grotesque somatic delusions accompanied by ⁎ Corresponding author. Tel.: +81 23 628 5322; fax: +81 23 628 5325. E-mail address: [email protected] (H. Hayashi). 0163-8343/$ – see front matter © 2009 Elsevier Inc. All rights reserved. doi:10.1016/j.genhosppsych.2008.07.004
hypoperfusion in the temporal and parietal lobes, and responded favourably to risperidone treatment. 2. Case report The case was a 54-year-old man. He had no past history or family history of psychiatric disorders. His social and occupational histories were quite normal. In August of 2005, he felt that “something has stuck between under front teeth.” From September, he felt that “there is another lower jaw with teeth between the real upper jaw and real lower jaw, and there is another tongue between the false lower jaw and the real lower jaw”; “the teeth on the false lower jaw are growing steadily”; “I try to cut the false teeth off with the real teeth, but the false teeth do not stop growing”; “the false teeth melt into holes in the false lower jaw, but later grow again from those holes”; “something like spaghetti is coming into and going out from the holes” and “the false lower jaw rolls up and is coming into the throat.” Because of these annoying sensations, he had mild depressive symptoms such as depressed mood, decrease in appetite, restlessness and fatigue. Despite these symptoms, he was able to continue working. In October, he visited our out patient clinic. He had the grotesque somatic delusions and mild depressive symptoms
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T. Akahane et al. / General Hospital Psychiatry 31 (2009) 185–186
Fig. 1. The first SPECT taken on Day 13.
but no other psychiatric symptom. His orientation and memory were intact. Hallucinations, disorganized speech, disorganized or catatonic behavior, affective flattening, alogia and avolition were not observed. He was given the diagnosis of DDST according to Diagnostic and Statistical Manual of Mental Disorders, Fourth Edition [2]. Sulpiride 200 mg/day was administered for 2 weeks, but there was no clinical improvement. Therefore, on Day 14 sulpiride was switched to risperidone 2 mg/day. On Day 31, there was apparent improvement, and he said that, “the sizes of the false lower jaw and teeth are half of those before.” From that day, the dose of risperidone was increased to 3 mg/day. On Day 44, there was no depressive symptom. The somatic delusions were markedly improved, and he noted that “the false lower jaw is withdrawing, and there is no tooth growing or spaghetti coming in and going out,” “I have recovered 90%.” There was no side effect of risperidone. On Day 107, he left for other city for a reason related to work. His magnetic resonance imaging and electroencephalography were within normal limits. The single photon emission computed tomography (SPECT) using Xe-133 was also performed twice with the patient's consent. The first SPECT taken on Day 13 showed reduced regional cerebral blood flow in the temporal and parietal lobes (Fig. 1). The second SPECT taken on Day 94 when there were only minor symptoms remaining continued to show hypoperfusion in the same regions. 3. Discussion In the present case, organic brain diseases were ruled out based on clinical and laboratory examinations. Schizophrenia was also ruled out because of lack of psychotic symptoms suggestive of this disorder other than marked somatic delusions. Therefore, it is reasonable to consider that he was suffering from DDST. The present case complained that there were another lower jaw with teeth and tongue in his mouth, i.e., delusion
of reduplication of body parts. He also complained of extremely grotesque and annoying sensations on the false lower jaw and teeth. In general, delusion of reduplication of body parts has been associated with organic brain diseases [8]. To our knowledge, this form of delusion has never been reported previously in DDST or MHP. The somatic delusions in this case started to improve within two weeks of risperidone treatment and were markedly improved after 1 month of the treatment. Risperidone caused no side effects. Therefore, this report supports prior observations on the efficacy of risperidone for DDST [4,6]. Risperidone may be preferable to pimozide recommended by Munro [1], because of its lower side effect profile than pimozide [7]. In fact, in the case reported by Narumoto et al. [4] pimozide induced acute dystonia necessitating discontinuation of the drug, while risperidone caused no definite side effect. The first SPECT taken when he had marked somatic delusions was consistent with prior observations on the association between DDST and hypoperfusion in the temporal and parietal lobes [3–5]. The essentially unchanged SPECT after near-remission suggests that the hypoperfusion in these regions may be a stable trait in some DDST patients or perhaps unrelated to the disorder. Alternatively, there may be a time lag between SPECT normalization and clinical recovery in some DDST patients. In fact, Hashioka et al. [9] have reported on a depressed patient with Cotard's delusion in whom the SPECT abnormality disappeared several months after clinical recovery. References [1] Munro A. Persistent delusional symptoms and disorders. In: Gelder MG, Lopez-Ibor JJ, Andreasen N, editors. New Oxford Textbook of Psychiatry. Oxford, UK: Oxford University Press; 2000. p. 651–76. [2] American Psychiatric Association. Diagnostic and Statistical Manual of Mental Disorders. 4th ed. Washington (DC): American Psychiatric Association; 1994. [3] Hayashi H, Oshino S, Ishikawa J, Kawakatsu S, Otani K. Paroxetine treatment of delusional disorder, somatic type. Hum Psychopharmacol 2004;19:351–2. [4] Narumoto J, Ueda H, Tsuchida H, et al. Regional cerebral blood flow changes in a patient with delusional parasitosis before and after successful treatment with risperidone: a case report. Prog NeuroPsychopharmacol Biol Psychiatry 2006;30:737–40. [5] Wada T, Kawakatsu S, Komatani A, Okuyama N, Otani K. Possible association between delusional disorder, somatic type and reduced regional cerebral blood flow. Prog Neuro-Psychopharmacol Biol Psychiatry 1999;23:352–71. [6] Kitamura H. A case of somatic delusional disorder that responded to treatment with risperidone. Psychiatry Clin Neurosci 1997;51:337. [7] Bruggeman R, Van der Linden C, Buitelaar JK, et al. Risperidone versus pimozide in Tourette's disorder: a comparative double-blind parallelgroup study. J Clin Psychiatry 2001;62:50–6. [8] Weinstein EA, Kahn RL, Malitz S, Rozanski J. Delusional reduplication of parts of the body. Brain 1954;77:45–60. [9] Hashioka S, Monji A, Sasaki M, et al. A patient with Cotard syndrome who showed an improvement in single photon emission computed tomography findings after successful treatment with antidepressants. Clin Neuropharmacol 2002;25:276–9.
General Hospital Psychiatry 31 (2009) 185 – 186
Case Reports
Extremely grotesque somatic delusions in a patient of delusional disorder and its response to risperidone treatment Takaki Akahane, M.D., Hiroshi Hayashi, M.D., Ph.D.⁎, Haruyoshi Suzuki, M.D., Shinobu Kawakatsu, M.D., Ph.D., Koichi Otani, M.D., Ph.D. Department of Psychiatry, Yamagata University School of Medicine, Yamagata 990-9585, Japan Received 3 June 2008; accepted 11 July 2008
Abstract A 54-year-old man with delusional disorder, somatic type (DDST) showed extremely grotesque somatic delusions, i.e., the presence of another lower jaw with teeth and tongue accompanied by annoying sensations. These somatic delusions responded favorably to treatment with risperidone 2–3 mg/day. The single photon emission computed tomography taken when he had marked somatic delusions showed hypoperfusion in the temporal and parietal lobes. The present report suggests that delusion of reduplication of body parts, which has been generally associated with organic brain diseases, is also observed in DDST. This report also supports prior observations on the efficacy of risperidone for DDST and the association between this disorder and hypoperfusion in the temporal and parietal lobes. © 2009 Elsevier Inc. All rights reserved. Keywords: Delusional disorder; Risperidone; Somatic delusion; SPECT
1. Introduction Delusional disorder, somatic type (DDST), also known as monosymptomatic hypochondriacal psychosis (MHP), is characterized by somatic or hypochondriacal delusions [1]. The most common delusions in DDST are that he or she emits a foul odor from the skin, mouth, rectum or vagina; that there is an infestation of insects on or in the skin; that there is an internal parasite; that certain parts of the body are definitely misshapen or ugly or that parts of the body are not functioning [2]. Knowledge of aetiology in DDST is scanty, because so little modern research has been conducted [1]. Some researchers including us have suggested that DDST has some association with hypoperfusion in the temporal and parietal lobes [3–5]. As to treatment, Munro has recommended pimozide as the first choice drug for DDST [1]. However, recent reports suggest the efficacy of risperidone [4,6] that has a more benign side effect profile than pimozide [7]. We describe the case of a patient with DDST, who showed extremely grotesque somatic delusions accompanied by ⁎ Corresponding author. Tel.: +81 23 628 5322; fax: +81 23 628 5325. E-mail address: [email protected] (H. Hayashi). 0163-8343/$ – see front matter © 2009 Elsevier Inc. All rights reserved. doi:10.1016/j.genhosppsych.2008.07.004
hypoperfusion in the temporal and parietal lobes, and responded favourably to risperidone treatment. 2. Case report The case was a 54-year-old man. He had no past history or family history of psychiatric disorders. His social and occupational histories were quite normal. In August of 2005, he felt that “something has stuck between under front teeth.” From September, he felt that “there is another lower jaw with teeth between the real upper jaw and real lower jaw, and there is another tongue between the false lower jaw and the real lower jaw”; “the teeth on the false lower jaw are growing steadily”; “I try to cut the false teeth off with the real teeth, but the false teeth do not stop growing”; “the false teeth melt into holes in the false lower jaw, but later grow again from those holes”; “something like spaghetti is coming into and going out from the holes” and “the false lower jaw rolls up and is coming into the throat.” Because of these annoying sensations, he had mild depressive symptoms such as depressed mood, decrease in appetite, restlessness and fatigue. Despite these symptoms, he was able to continue working. In October, he visited our out patient clinic. He had the grotesque somatic delusions and mild depressive symptoms
186
T. Akahane et al. / General Hospital Psychiatry 31 (2009) 185–186
Fig. 1. The first SPECT taken on Day 13.
but no other psychiatric symptom. His orientation and memory were intact. Hallucinations, disorganized speech, disorganized or catatonic behavior, affective flattening, alogia and avolition were not observed. He was given the diagnosis of DDST according to Diagnostic and Statistical Manual of Mental Disorders, Fourth Edition [2]. Sulpiride 200 mg/day was administered for 2 weeks, but there was no clinical improvement. Therefore, on Day 14 sulpiride was switched to risperidone 2 mg/day. On Day 31, there was apparent improvement, and he said that, “the sizes of the false lower jaw and teeth are half of those before.” From that day, the dose of risperidone was increased to 3 mg/day. On Day 44, there was no depressive symptom. The somatic delusions were markedly improved, and he noted that “the false lower jaw is withdrawing, and there is no tooth growing or spaghetti coming in and going out,” “I have recovered 90%.” There was no side effect of risperidone. On Day 107, he left for other city for a reason related to work. His magnetic resonance imaging and electroencephalography were within normal limits. The single photon emission computed tomography (SPECT) using Xe-133 was also performed twice with the patient's consent. The first SPECT taken on Day 13 showed reduced regional cerebral blood flow in the temporal and parietal lobes (Fig. 1). The second SPECT taken on Day 94 when there were only minor symptoms remaining continued to show hypoperfusion in the same regions. 3. Discussion In the present case, organic brain diseases were ruled out based on clinical and laboratory examinations. Schizophrenia was also ruled out because of lack of psychotic symptoms suggestive of this disorder other than marked somatic delusions. Therefore, it is reasonable to consider that he was suffering from DDST. The present case complained that there were another lower jaw with teeth and tongue in his mouth, i.e., delusion
of reduplication of body parts. He also complained of extremely grotesque and annoying sensations on the false lower jaw and teeth. In general, delusion of reduplication of body parts has been associated with organic brain diseases [8]. To our knowledge, this form of delusion has never been reported previously in DDST or MHP. The somatic delusions in this case started to improve within two weeks of risperidone treatment and were markedly improved after 1 month of the treatment. Risperidone caused no side effects. Therefore, this report supports prior observations on the efficacy of risperidone for DDST [4,6]. Risperidone may be preferable to pimozide recommended by Munro [1], because of its lower side effect profile than pimozide [7]. In fact, in the case reported by Narumoto et al. [4] pimozide induced acute dystonia necessitating discontinuation of the drug, while risperidone caused no definite side effect. The first SPECT taken when he had marked somatic delusions was consistent with prior observations on the association between DDST and hypoperfusion in the temporal and parietal lobes [3–5]. The essentially unchanged SPECT after near-remission suggests that the hypoperfusion in these regions may be a stable trait in some DDST patients or perhaps unrelated to the disorder. Alternatively, there may be a time lag between SPECT normalization and clinical recovery in some DDST patients. In fact, Hashioka et al. [9] have reported on a depressed patient with Cotard's delusion in whom the SPECT abnormality disappeared several months after clinical recovery. References [1] Munro A. Persistent delusional symptoms and disorders. In: Gelder MG, Lopez-Ibor JJ, Andreasen N, editors. New Oxford Textbook of Psychiatry. Oxford, UK: Oxford University Press; 2000. p. 651–76. [2] American Psychiatric Association. Diagnostic and Statistical Manual of Mental Disorders. 4th ed. Washington (DC): American Psychiatric Association; 1994. [3] Hayashi H, Oshino S, Ishikawa J, Kawakatsu S, Otani K. Paroxetine treatment of delusional disorder, somatic type. Hum Psychopharmacol 2004;19:351–2. [4] Narumoto J, Ueda H, Tsuchida H, et al. Regional cerebral blood flow changes in a patient with delusional parasitosis before and after successful treatment with risperidone: a case report. Prog NeuroPsychopharmacol Biol Psychiatry 2006;30:737–40. [5] Wada T, Kawakatsu S, Komatani A, Okuyama N, Otani K. Possible association between delusional disorder, somatic type and reduced regional cerebral blood flow. Prog Neuro-Psychopharmacol Biol Psychiatry 1999;23:352–71. [6] Kitamura H. A case of somatic delusional disorder that responded to treatment with risperidone. Psychiatry Clin Neurosci 1997;51:337. [7] Bruggeman R, Van der Linden C, Buitelaar JK, et al. Risperidone versus pimozide in Tourette's disorder: a comparative double-blind parallelgroup study. J Clin Psychiatry 2001;62:50–6. [8] Weinstein EA, Kahn RL, Malitz S, Rozanski J. Delusional reduplication of parts of the body. Brain 1954;77:45–60. [9] Hashioka S, Monji A, Sasaki M, et al. A patient with Cotard syndrome who showed an improvement in single photon emission computed tomography findings after successful treatment with antidepressants. Clin Neuropharmacol 2002;25:276–9.