https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6171510/?fbclid=IwAR3stQcRkNnJ-g5GEC1PkigHCupQO2kJZbmaCKeoAZjAaTaYAHCQEsYFO9M

Reframing delusional infestation: perspectives on unresolved puzzles (is it DI or MD--Morgellons Disease )

Psychol Res Behav Manag. 2018; 11: 425–432.
Published online 2018 Oct 1. doi: 10.2147/PRBM.S166720
PMCID: PMC6171510
PMID: 30319296
Reframing delusional infestation: perspectives on unresolved puzzles
Jianbo Lai,1,2,3 Zhe Xu,4,5 Yi Xu,1,2,3 and Shaohua Hu1,2,3
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Abstract
Delusional infestation (DI), a debilitating psychocutaneous condition, featured as a false fixed belief of being infested accompanied by somatosensory abnormality, behavior alteration, and cognitive impairment. Although management of primary causes and pharmacotherapy with antipsychotics and/or antidepressants can help to alleviate symptoms in most patients, the underlying etiology of DI still remains unclear. Morgellons disease (MD), characterized by the presence of cutaneous filaments projected from or embedded in skin, is also a polemic issue because of its relationship with spirochetal infection. This review aims to discuss the following topics that currently confuse our understandings of DI: 1) the relationship of real/sham “infestation” with DI/MD; 2) behavior alterations, such as self-inflicted trauma; 3) neuroimaging abnormality and disturbance in neurotransmitter systems; and 4) impaired insight in patients with this disease. In discussion, we try to propose a multifactorial approach to the final diagnosis of DI/MD. Future studies exploring the neurobiological etiology of DI/MD are warranted.

Keywords: delusional infestation, Morgellons disease, behavior, neurotransmitter, insight
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Introduction
Delusional infestation (DI) is an uncommon, intricate psychocutaneous condition.1 Against available medical evidence, patients with DI have a strong conviction that they are infested with little animals or less frequently inanimate matter.1,2 Meanwhile, patients always complain of abnormal skin sensations, such as stinging, biting, and crawling, which were ascribed to the “infestation”. The symptoms of DI can occur as primary, or more commonly, secondary to diverse medical conditions, such as neuropsychiatric diseases, nutrient deficiency, psychotropic medications, infections, intoxication, tumors, and metabolic disturbance.3 Etiology-dependent management and antipsychotics/antidepressants have been reported to be therapeutically effective.1,3,4

Worldwide retrospective researches and case reports have painted an inexplicit epidemiological picture of DI.5–7 In clinical practice, DI might be underdiagnosed as patients are always reluctant to psychiatric referral and prefer to visit dermatologists, microbiologists, and general practitioners. The prevalence of appropriately 80 cases per million was reported in private practices, while much less cases were presented and identified in public health services (appropriately 5.5 cases per million).8 Middle-aged to elderly women, especially those with inadequate social contact, are more likely to be afflicted.5–8 The duration of illness can be less than 1 year or as long as three decades.3 However, the final diagnosis and proper management of DI are always delayed.

To date, there are still many puzzles hindering our in-depth understanding of somatosensory abnormality, behavior alteration, and cognitive impairment in DI. 1) Are symptoms associated with DI simply delusional? Can real infestation cause DI-like symptoms? 2) How to unscramble the behavior pattern in DI patients, such as self-inflicted skin trauma? 3) Antipsychotics and/or antidepressants are generally effective in managing symptoms of DI. This phenomenon indicates disturbance in neurotransmitter systems according to the pharmacological actions of agents. Moreover, recent advances in neuroimaging researches of DI patients need to be updated, including brain anatomical and functional abnormalities. 4) Patients with DI commonly lost their insight of disease nature. Whether their insight can be restored after improvement in symptoms? A better characterizing of aforementioned puzzles is of significant importance in clinical practice and fundamental research.

Herein, we start with expounding the current knowledge of above puzzles. In the “Discussion” section, we try to propose a multifactorial approach, which helps to facilitate the diagnosis of DI/Morgellons disease (MD).

Sham or real infestation
This question should be prudently answered, as it directly determines the nature of DI. To date, a great many of animate pathogens have been blamed, including all kinds of arthropods, worms, bacteria, and fungi.8 These pathogens are always described as small and vivid. In contrast, inanimate matter, long and thin, such as fibers, threads, hair, and the like, is less frequently reported.9 A comprehensive review of different “pathogens” is listed in Figure 1.10–23 The specific type of alleged pathogen by certain patient could be affected by one’s own knowledge, life experience, and living environment. These pathogens might be wrapped with containers, referred to as the “specimen” or “matchbox” sign,3 and taken to clinic or hospital as an evidence of infestation. For most of the time, however, microscopy or even skin biopsy fails to find out the alleged pathogenic agent. In addition, the abnormal skin sensations, such as stinging, crawling, biting, and pinching, intensify patients’ belief of infestation. In DI patients, abnormal activation of an itch pathway from the skin to the central nervous system is suspected.16 Dysfunction of interoception, improper processing, and misinterpretation of perceived sensations contribute to the formation of tactile or even visual hallucinations. Of note, it seems that delusions are always infestation oriented and, apart from infestation, the individuals’ function well in other life scenes.

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Figure 1
An atlas of alleged pathogens in patients with delusional infestation.

Notes: In the animate group, schematic diagrams of black mold, lice, dust mite, Gongylonema pulchrum, scabies mite, worm, ant, flea, Leishmania, gnats, tick, fly, strepsiptera, spider, and bacteria are listed. In the inanimate group, fiber, thread, hair, and sand are listed.

The closest diagnosis to DI in the Diagnostic and Statistical Manual of Mental Disorders, the fifth edition (DSM-5), is somatic-type delusional disorder.24 However, the diagnosis of delusional disorder should be exclusive, that is, the disturbance cannot be better explained by any substance, underlying mental comorbidities and psychical conditions.24 Within this framework, most DI cases reported in literatures are secondary, not primary, and therefore could not be diagnosed as somatic-type delusional disorder. Whatever, we should first determine whether the belief is delusional or not.

MD, characterized as embedded or protruding filaments in skin lesions, was once considered as a variation of DI by some researchers.3,9 However, other researchers have claimed an infectious process underlying this disease. The presentation of specimen, which is further confirmed as cutaneous filaments, may not be delusional in some cases. They reported positive tests of Borrelia spirochetes in patients with MD.22,23 In these studies, the researchers found that patients with MD shared similar systematic symptoms (eg, skin irritation, fatigue, joint pain, cardiac complications, and cognitive deficits) to those with tick-borne illnesses, for example, Lyme disease.25 They reported that the major components of MD cutaneous filaments were keratin and collagen, which were produced by keratinocytes and fibroblasts.25 The formation of these filaments could be resulted from skin proliferation in underlying infectious process. Treatment of MD patients seems to be more challenging than those with DI patients.26,27 Therefore, MD seems more likely to be a discrete entity, rather than a variation of DI. Some studies against the infectious cause of MD have also been published. In the Pearson study, for instance, researchers did not found any link between MD and Lyme disease.28 The histopathologic abnormality of skin samples was most likely to be solar elastosis, and most of the materials presented by the patients were composed of cellulose, compatible with cotton fibers.28 However, the Pearson study selected patients via a retrospective review of medical records. The inclusion criteria in this study did not strictly require the presentation of embedded or protruding skin fibers. The patients were a heterogeneous group, and the fiber analysis was conducted in a small number of patients.25,28

Notably, neither MD nor DI is listed in DSM-5. Tests for Lyme disease are insensitive, which complicate the diagnosis. The use of techniques, such as immunostaining and PCR, to directly detect spirochetal pathogens in skin is needed. Moreover, the skin lesions caused by self-mutilation behaviors may result in secondary infection, which further complicates this condition. Based on current findings, MD is not a subtype of DI and its interplay with spirochetal infection needs further investigations.

Alterations in behavioral characteristics
Health-related quality of life was significantly reduced in DI patients. Individuals without this condition can hardly imagine the encounters of afflicted patients. These patients always isolate themselves and present altered behavioral characteristics, which can be typically classified into two patterns, outward and inward. A common explanation for these behaviors is to find out and eradicate the putative pathogens. Another reason could be a way to release anxiety and stress. However, negative medical findings and vain self-inflicted attempts, eventually result in a vicious circle, which promotes the persistence of disease and increases the severity of symptoms.

The outward pattern
1) Patients repetitively seek help from many doctors of different specialties and are prescribed with antibiotic, virucide, pesticide, or other anti-infectives.15,29 The effect of these medications is always unsatisfactory and sometimes detrimental, causing skin irritation and abnormal sensations similar to skin complaints of DI, as well as nausea, headache, diarrhea, fatigue, and even elevated liver enzymes.15 2) In cases of DI by proxy, especially patients with children living together, precautionary protection of children from excessive cleansing is particularly important.30 Similar condition is that pet owners hold a fixed, but false belief that their pets have been infested and need veterinary treatments.31,32 3) A increased risk of drug abuse was observed in DI patients, which would worsen their conditions.5 However, the causes for this phenomenon is unclear and may relate to physical or psychiatric comorbidities. In the outward pattern, patients focus on the surroundings rather than their own body.

The inward pattern
1) Self-inflicted skin trauma caused by mechanical force or fingernails is thought to target the causative pathogen and relieve skin discomfort.12,33 2) Self-therapy with different chemicals or physical strategies aims to kill or flush away the pathogens from the body.3,34 3) Repetitive and intensive self-cleaning with an obsessive trait, such as frequent changing of clothes and hair washing, reflects excessive fear of contamination.35 4) In addition, comorbidity with psychiatric diseases or depression, secondary to DI, is associated with increased proneness to suicidal ideations and attempts.36 These inward behaviors are exhaustive, frustrated, and even detrimental and are generally more convenient to be implemented than outward behaviors.

The abnormal behaviors in DI patients are associated with their loss of disease insight. The skin lesions secondary to self-mutilation, eg, ulcerations, erosions, and pigmentations, can worsen the condition and sometimes were perceived as evidence of infestation.3,35 Apparently, these patients have difficulties in decision making and risk evaluation of their behaviors.37 In general, abnormal behavioral patterns, accompanied with personality alteration, are adaptive to the anxiety and fear caused by uncomfortable skin sensations and hallucinations. Alterations in behaviors, in turn, may also consolidate the skin discomfort.

Updates on the neural correlates of DI
To date, the neural mechanisms of DI remain largely unknown. Many DI cases are secondary to various medical conditions, which make the issue more complicated. The role of genetic vulnerability and neurodevelopmental or neuroimmune aberration has been inadequately investigated. However, disturbance in brain neurotransmitter systems is evident since many cases of DI respond well to antipsychotic or antidepressant treatment. Recent neuroimaging findings of DI also need to be updated.

Imbalance in neurotransmitter systems
A comprehensive review of drugs that can elicit or otherwise treat DI is presented in Table 1.13,38–60 Accordingly, several lines of dysfunction in neurotransmitter systems are revealed. Except for the well-known hyperactivity in dopamine system, dysfunction in serotonergic neurotransmission and adrenergic neurotransmission, as well as the histamine and gamma-aminobutyric acid circuits, should also be noted. These findings indicate a complicated biological underpinning of DI. First, different neurotransmitter pathways are involved and provide possible explanations for the diverse symptom spectrums of DI, eg, tactile hallucination, delusion, behavior, affect, and cognitive alterations. Second, these neurotransmitter pathways are interconnected and their interactions may contribute to the onset of DI. Third, not only neurotransmitter receptors but also their transporters and various enzymes appear to be neuropharmacological targets of drugs listed in Table 1. Therefore, the pharmacotherapy for DI patients should be established on the symptomatic features, past treatment responses, and adverse effects of medications.

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Dear RM Agents and Readers,

I agree with some of this study but am hoping they start looking at what is causing the chronic fatigue because therein lies the answer to why doctors have to prescribe amphetamines or folks take it upon themselves to do the same or harder drugs in order to move out of the bed.

Fix the chronic fatigue (go to the root of that) and then you are getting somewhere folks. Albeit this does start to remove the MD group from just the nutty label to a condition associated with tic born diseases that are not easily known because most of the tests suck so you end up still telling folks that are physically ill that they are crazy.

Now can we look at the hydrogen sulfide link; you know the one where it stays in the body of those with the tic illness's and causes chronic fatigue, you know that one!

So I am hoping for an intravenous approach to my own therapy as the liquid glutathione and the glutathione cream together are working but the pain is horrendous (true we just went through a full moon)as it is pushing out. I've actually had black oil coming out of my hands and coming from under my arm area. I've always been the type to pull the bandaid off quickly. I expect it will hurt quite a bit after the intravenous treatment but I'll be able to get on with life after that, it won't keep going and going and going.

Here is that article on the association of hydrogen sulfide to chronic fatigue: https://phoenixrising.me/research-2/hydrogen-sulfide-a-breakthrough-in-mecfs/hydrogen-sulfide-and-chronic-fatigue-syndrome-the-originator-speaks

The way I see it the black oil that Morgies speak of is hydrogen sulfide.

Many Blessings,
CrystalRiver